EPA/600/J-93/470 November 1993 Critical Reviews in Toxicology, 23(3):283-335 (1993) Developmental and Reproductive Toxicity of Dioxins and Related Compounds: Cross- Species Comparisons Richard E. Peterson,3-"' H. Michael Theobald,3 and Gary L. Kimmelc •School of Pharmacy and "Environmental Toxicology Center, University of Wisconsin, Madison, Wl; and "U.S. Environmental Protection Agency, Washington, D.C. •Address all correspondence to: Dr. Richard E. Peterson, School of Pharmacy, University of Wisconsin, 425 N. Charter Street, Madison, WI 53706 ABSTRACT: Developmental toxicity to TCDD-like congeners in fish, birds, and mammals, arid reproductive toxicity in mammals are reviewed. In fish and bird species, the developmental lesions observed are species dependent, but any given species responds similarly to different TCDD-like congeners. DeveloDmentaLtoxicito in fish resembles "blue sac disease," whereas structural malformations can occur in atJtdS-i5rr?TJircLslKcies. In mammals, developmental toxicity includes decreased growth, structural malformations, functional artSEflSanNi and prenatal mortality. At relatively low exposure levels, structural malformations are noicnrnrmiD.irwmawfljgiiAB species. In contrast, functional alterations are the most sensitive signs of developmental TOxicity. These include effects on the male reproductive system and male reproductive behavior in rats, and neurobehavioral effects in monkeys. Human infants exposed during the Yusho and Yu-Cheng episodes, and monkeys and mice exposed perinatally to TCDD developed an ectodermal dysplasia syndrome that includes toxicity to the skin and teeth. Toxicity to the central nervous system in monkey and human infants is a potential part of the ectodermal dysplasia syndrome. Decreases in spermatogenesis and the ability to conceive and carry a pregnancy to term are the most sensitive signs of reproductive toxicity in male and female mammals, respectively. KEY WORDS: perinatal exposure, structural malformations, functional alterations, antiestrogenicity, sexual differentiation, Ah receptor. I. INTRODUCTION 2,3,7,8 - Tetrachlorodibenzo - p - dioxin (TCDD) is one of 75 possible chlorinated di- benzo-/?-dioxin (CDD) congeners. It is one of the most potent of the CDDs..hrominated_dihenzo- p-dioxins (BDDs), chldriii&tdC feHtKKEAurans (CDFs), brominated diOenz'otvirans'ClDFs), polychlorinated biphenyte" ^PCffil/ThBdiDoly- brominated biphenyls (PBBS^/ Ailuil.WCDD serves as the prototype congener for investigating the toxicity elicited by these classes of chemicals. Developmental and reproductive toxicity is gen- erally believed to be caused by the parent com- pound. There is no evidence that TCDD metab- olites are involved. The toxic potency of TCDD is due to the number and position of chlorine substitutions on the dibenzo-p-dioxin molecule. CDD congeners with decreased lateral (2,3,7, and 8) or increased nonlateral chlorine and bro- mine substituents are less potent than TCDD; however, most of these congeners will produce toxicity, and the pattern of responses within an- imals of the same species, strain, sex, and age Printed on Recycled Paper 283 ------- will generally be similar to that of TCDD.2 3 PCB congeners with zero or one ortho chlorine, two para chlorines, and at least two meta chlorines can assume a coplanar conformation sterically similar to TCDD and also produce a pattern of toxic responses similar to that of TCDD. In con- trast, PCB congeners with two or more ortho chlorines cannot assume a coplanar conformation and do not resemble TCDD in toxicity.1'3 CDD and CDF congeners chlorinated in the lateral positions, as compared with those lacking chlorines in the 2,3,7, and 8 positions, are pref- erentially bioaccumulated by fish, reptiles, birds, and mammals.4"6 Furthermore, coplanar PCBs and/or monoorf/io-chlorine-substituted analogs of the coplanar PCBs bioaccumulate in fish, wildlife, and humans.7"11 This is of concern be- cause the combined effects of the lateral-substi- tuted CDD, BDD, CDF, BDF, PCB, and PBB congeners that act through an aryl hydrocarbon (Ah) receptor mechanism have the potential to decrease feral fish and wildlife populations sec- ondary to developmental and reproductive tox- jcjtv 5,12-14 Humans are not exempt from the de- velopmental and reproductive effects of complex halogenated aromatic hydrocarbon mixtures. Such mixtures that contain both TCDD-like congeners and nonTCDD-like congeners have been impli- cated in causing the developmental and repro- ductive toxicity in the Yusho and Yu-Cheng poi- soning incidents in Japan and Taiwan.15"17 Thus, exposure to TCDD-like congeners is a health con- cern for humans, domesticated animals, fish, and wildlife, although the relative contributions of TCDD- and nonTCDD-like congeners are not known in some exposure situations. A mechanism of action that CDD, BDD, CDF, BDF, PCB, and PBB congeners substituted in the lateral positions have in common is that they bind to the Ah receptor, which then binds to a translocating protein that carries the activated TCDD receptor complex into the nucleus. These activated TCDD receptor complexes bind to spe- cific sequences of DNA referred to as dioxin- response elements (DREs), resulting in altera- tions in gene transcription. There is evidence that this Ah receptor mechanism is involved in the antiestrogenic action of TCDD and in its ability to produce the structural malformations cleft pal- ate and hydronephrosis in mice. However, its role in producing other signs of developmental and reproductive toxicity is less firmly established or not established. This leaves open the possibility for some of these TCDD effects to not be Ah receptor mediated. The information in this review has been or- ganized into sections on developmental toxicity, reproductive toxicity in males, and reproductive toxicity in females. However, it is important to emphasize that developmental and reproductive toxicity, particularly in females, can be interre- lated. Therefore, the reader should view the sec- tion subheadings as topic indexes that indicate where most of the information on a particular endpoint is concentrated. The endpoints de- scribed within each subheading are not intended to be thought of as being independent of end- points described in other sections. For example, the effects of TCDD on the actions of reproduc- tive hormones, peptides, and steroids can be in- volved in reproductive dysfunction as well as developmental toxicity. II. DEVELOPMENTAL TOXICITY The manifestations of developmental toxicity to TCDD have been divided into three categories for convenience in assessing the data base with respect to an Ah receptor-mediated response. These categories include death/growth/clinical signs, structural malformations, and functional alterations. Exposure-related effects on death/ growth/clinical signs are described for fish, birds, laboratory mammals, and humans along with structure activity results that are consistent with, but do not prove, an Ah receptor-mediated mech- anism. Structural malformations, particularly cleft palate formation and hydronephrosis, occur in mice. However, in other mammalian species, postnatal functional alterations, some of which may be irreversible, are the most sensitive ad- verse developmental effects of TCDD-like con- geners. These include effects on the male repro- ductive system of rats and object-learning behavior in monkeys. 284 ------- DATE DUE A. Death/Growth/Clinical Signs 1. Fish Early life stages of fish appear to be more sensitive to TCDD-induced mortality than adults. This is suggested by the LD50 of TCDD in rain- bow trout sac fry (0.4 |Ag/kg egg weight) being 25 times less than that in juvenile rainbow trout (10 |xg/kg body weight).13'18 The significance of this finding is that early life stage mortality caused by high concentrations of TCDD-Iike congeners in fish eggs may pose the greatest risk to feral fish populations.5-13 Cooper19 reviewed the de- velopmental toxicity of CDDs and CDFs in fish and Cook et al.5 discussed components of an aquatic ecological risk assessment for TCDD in fish. The reader is referred to this literature for more in-depth coverage than is presented here. TCDD is directly toxic to early life stages of fish. This has been demonstrated for Japanese medaka, pike, rainbow trout, and lake trout ex- posed as fertilized eggs to graded concentrations of waterborne TCDD. In these species, TCDD causes an overt toxicity syndrome characterized by edema, hemorrhages, and arrested growth and development culminating in death. 13>14'20~23 His- topathological evaluation of lake trout embryos and sac fry has shown this syndrome to be es- sentially identical to that of blue sac disease.21'23 Following egg exposure to TCDD, signs of tox- icity are not detected in medaka until after the liver rudiment forms,22 and in lake trout, toxicity is first detected approximately 1 week prior to hatching, but becomes fully manifest during the sac fry stage.14-23 Among all fish species inves- tigated thus far, lake trout are the most sensitive to TCDD developmental toxicity. Following ex- posure of fertilized lake trout eggs to graded wa- terborne concentrations of TCDD, the NOAEL for sac fry mortality is 34 pg TCDD/g egg, the LOAEL is 55 pg TCDD/g egg, and the egg TCDD concentration that causes 50% mortality above control at swim up (LD50) is 65 pg TCDD/g egg.14 Thus, TCDD is a potent developmental toxicant in fish and the effect is not secondary to maternal toxicity. The Ah receptor has not been identified in early life stages of fish; however, it is assumed NOV 1 5 3JG8 Is — in .25 n- la re s- ~ IS W a t )r o/ ie d 1C /- >- 'S iV e The Library Store #47-0106 £.. oiras Bird embryos also are more sensitive to TCDD toxicity than adults. The LD50 of TCDD in the chicken embryo (0.25 (xg/kg egg weight) is 100 to 200 times less than the TCDD dose that causes mortality in adult chickens (25 to 50 jig/ kg body weight).29-30 The LD50 of TCDD injected into fertilized ring-necked pheasant eggs (1.1 to 1.8 (xg/kg egg weight) is 14 to 23 times less than the TCDD dose that causes 75% mortality in ring- necked hen pheasants (25 (J-g/kg body weight).31 Among bird species, most of the develop- mental toxicity research has been done on chick- ens. Injection of TCDD or its approximate iso- stereomers into fertilized chicken eggs causes a toxicity syndrome in the embryo characterized by pericardial and subcutaneous edema, liver le- sions, inhibition of lymphoid development in the thymus and bursa of Fabricius, microophthalmia, beak deformities, cardiovascular malformations, and mortality.32"40 On the other hand, injection of a coplanar PCB into fertilized turkey eggs at a dose high enough to cause microophthalmia, beak deformities, and embryo mortality did not 285 ------- produce liver lesions, edema, or thymic hypo- plasia, all hallmark signs of TCDD toxicity in the chicken embryo.36 This disparity in signs of TCDD embryotoxicity among bird species is not unique to the turkey and chicken. In fertilized eggs of ring-necked pheasants and eastern blue- birds, injection of TCDD produces embryo mor- tality, but all of the other signs of toxicity seen in the chicken embryo are absent, including car- diovascular malformations3MM2 Thus, in bird embryos, the signs of toxicity elicited by TCDD and its approximate isostereomers are highly spe- cies dependent; the only toxic effect common to all bird species is embryo mortality. There is evidence in chicken embryos that the Ah receptor may be involved in producing developmental toxicity. The Ah receptor has been detected in chicken embryos36'43 and the rank or- der potency of PCB congeners for producing chicken embryo mortality is 3,3',4,4',5-PCB > 3,3',4,4'-TCB > 3,3',4,4',5,5'-HCB > 2,3,3',4,4'-PCB > 2,3,4,4',5-PCB, with 2,2',4,5'-TCB, 2,2',4,4',5,5'-HCB and 2,2',3,3',6,6'-HCB being inactive, and this rank order is similar to that for a classic Ah receptor- mediated response in the chicken embryo, i.e., cytochrome P-4501A1 induction.35-37-44 Inas- much as the nonsteroidal anti-inflammatory drug benoxoprofen suppresses 3,3',4,4'-TCB-in- duced toxicity in the chicken embryo without altering its ability to induce microsomal enzyme activity,45 induction of cytochrome P-4501A1 and toxicity are part of a pleiotropic response linked to the Ah receptor, but these effects are not other- wise causally related. Also, for 3,3',4,4'-TCB, 3,3',4,4',5,5'-HCB, and TCDD, there is a marked dissociation of the dose-response rela- tionship for lethality and enzyme induction in the chicken embryo.35 A decrease in activity of uroporphyrinogen decarboxylase (URO-D) and an increase in ac- cumulation of uroporphyrins are effects that are readily produced by exposure of cultured chicken embryo liver cells to TCDD, 3,3' ,4,4'-TCB, and other PCBs.46-^8 Coplanar PCB congeners are more potent inhibitors of URO-D activity in cul- tured chicken embryo liver cells than are non- coplanar PCB congeners,49 suggesting an Ah re- ceptor-mediated mechanism. Unlike the results in cultured cells, however, a lethal dose of TCDD (6 nmol/egg) does not affect URO-D activity or cause any increase in accumulation of uropor- phyrins in chicken embryos.35 Thus, TCDD-in- duced lethality in chicken embryos is not asso- ciated with effects of TCDD on URO-D activity, even though a decrease in URO-D activity might be expected to occur if a sufficient dose of TCDD could be reached without being lethal. The chicken embryo heart is a target organ for TCDD and other halogenated aromatic hydro- carbons that act via an Ah receptor mechanism. The classic sign of chick embryo toxicity in- volving the heart is pericardial edema. However, TCDD has other effects on the chick embryo heart that are less well known. These include its ability to produce cardiovascular malformations and to increase cardiac release of arachidonic acid metabolites. When fertilized chicken eggs are in- jected with graded doses of TCDD, cardiovas- cular malformations are produced, including ven- tricular septal defects, aortic arch anomalies, and conotruncal malformations. Approximately 1.6 pmol TCDD/egg (9 ng/kg egg, assuming a 55-g egg weight) causes cardiovascular malformations in 46% of treated embryos vs. 29% of control embryos.3233 The cardiovascular malformation response may be unique to the chicken embryo because in fertilized ring-necked pheasant and eastern bluebird eggs injected with TCDD the incidence of such malformations is not in- creased.31-41-42 In the chicken embryo heart, arachidonic acid metabolism is stimulated by TCDD, resulting in increased formation of prostaglandins.50 Dose- response relationships for the release of immu- noreactive PGE2, PGF2a, and TXB2 from chick embryonic heart are biphasic, with an apparent maximally effective dose of 100 pmol TCDD/ egg. When the egg TCDD dose is increased fur- ther, release of these prostaglandins tends to de- cline toward levels in control hearts. Biphasic dose-response curves for cardiac PGE2 release also were obtained with 3,3',4,4'-TCB and 3,3',4,4',5,5'-HCB.50 The thymus and bursa of Fabricius are other TCDD target organs in the chicken embryo. TCDD, 3,3',4,4'-TCB, and 3,3',4,4'-TCAOB cause dose-related decreases in lymphoid development of both of these im- mune system organs.38"40 Cultured thymus an- lage from chick embryos are 100 times more 286 ------- sensitive to the inhibitory effect of TCDD on lymphoid development than cultured thymus an- lage from turkey and duck embryos.38 This sug- gests that the reason thymic atrophy was not seen in turkey embryos at egg doses of 3,3',4,4'-TCB that were overtly toxic36 was not because the turkey embryo thymus was incapable of respond- ing to 3,3',4,4'-TCB, rather turkey embryos ap- pear to be more sensitive to the lethal rather than the immunotoxic effect of this coplanar PCB. Within the same bird species, the signs of developmental toxicity elicited by TCDD and its approximate isostereomers are similar. In the chicken embryo, TCDD, 3,3',4,4',5-PCB, 3,3',4,4'-TCB, and 3,3',4,4',5,5'-HCB all cause pericardial and subcutaneous edema, liver le- sions, microopthalmia, beak deformities, and mortality; and TCDD, 3,3',4,4'-TCB and 3,3',4,4'-TCAOB inhibit lymphoid develop- ment.32-37"39 In pheasant embryos, an altogether different pattern of responses is seen. Neverthe- less, TCDD-like congeners, TCDD, and 3,3',4,4'-TCB, injected into fertilized pheasant eggs, produce the same pheasant embryo-specific pattern. This pattern consists of embryo mortality in the absence of ederna, liver lesions, thymic hypoplasia, and structural malformations.31-51 The lethal potency of TCDD and its approx- imate isostereomers in embryos of different bird species varies widely. The chicken embryo is an outlier in that it is by far the most sensitive of all bird species to TCDD. Turkey, ring-necked pheasant, mallard duck, domestic duck, domestic goose, golden-eye, herring gull, black-headed gull, and eastern bluebird embryos are consid- erably less sensitive to the embryo-lethal effect of TCDD and TCDD-like congeners.31-36-41-42-51-52 TCDD is 4 to 7 times more potent in causing embryo mortality in chicken than pheasant em- bryos, and 3,3',4,4'-TCB is 20 to 100 times more potent in chicken than in turkey embryos.30-31-36 In chicken embryos, an egg dose of 4 jig/kg 3,3',4,4'-TCB increased embryo mortality, whereas an egg dose of 100 jig/kg of the same coplanar PCB had no embryotoxic effect in pheasants and mallard ducks, and a dose of 1000 (xg/kg egg had no effect on embryo mortality in domestic ducks, domestic geese, golden eye, her- ring gulls, and black-headed gulls.51-53 In contrast to the above-mentioned species differences, the potency of 3,3',4,4'-TCB in causing embryo mortality among different strains of chickens is quite similar with the LD50 in six different strains, varying less than fourfold.53 Graded doses of TCDD have been admin- istered to fertilized eastern bluebird and ring- necked pheasant eggs for the purpose of deter- mining the LOAEL and NOAEL for embryo- toxicity. Mortality was the most sensitive embryotoxic effect in both species. For eastern bluebirds, the LOAEL was 10,000 pg TCDD/g egg and the NOAEL was 1000 pg TCDD/g egg.42 For ring-necked pheasants, the LOAEL was 1000 pg TCDD/g egg and the NOAEL was 100 pg TCDD/g egg. The LD50 for embryo mortality in the ring-necked pheasant is 1354 pg TCDD/g egg when the dose is injected into the egg albumin and 2182 pg TCDD/g egg when the dose is in- jected into the egg yolk.31 In contrast, for chick- ens, the LD50 for embryo mortality is 240 pg TCDD/g egg.30 3. Laboratory Mammals When exposed to TCDD during adulthood, laboratory mammals display wide differences in the LD50 of TCDD. It is interesting to note, how- ever, that when exposure occurs during prenatal development, the potency of TCDD tends to be similar across species. The LD50 of TCDD in adult hamsters, 1157 to 5051 (xg/kg, makes adult hamsters three orders of magnitude more resistant to TCDD-induced lethality than are adult guinea pigs.54,55 yet, a maternal dose of 18 jig TCDD/ kg can increase the incidence of prenatal mor- tality in the hamster embryo/fetus. Because this dose is only 12-fold larger than the 1.5 jig TCDD/ kg dose that increases the incidence of prenatal mortality in the guinea pig, the hamster embryo/ fetus approaches other rodent species in its sen- sitivity to TCDD-induced lethality.56-57 Thus, the magnitude of the species differences in lethal potency of TCDD is affected by the timing of TCDD exposure during the life history of the animal. Exposure to TCDD during pregnancy causes prenatal mortality in the monkey, guinea pig, rabbit, rat, hamster, and mouse (Table 1). Given a particular dosage regimen, the response is dose 287 ------- TABLE 1 Relationship between Maternal Toxicity and Prenatal Mortality in Laboratory Mammals Exposed to TCDD during Gestation Species/strain Monkey/rhesus Guinea pig/Hartley Rabbit/New Zealand Rat/Wistar Rat/Sprague-Dawley Hamster/Golden Syrian Mouse/CD-1 Daily TCDD dose (,tg/kg/day) O1 0.1 0.25 0.5 1 0' 0.125 0.25 0.5 1 1 2 4 0' 0.03 0.125 0.5 2 8 O1 25 50 100 200 400 Cumulative TCDD dose (jig/kg) 0° 0.2 1 5 0' 0.15 1.5 0 1 2.5 5 10 0 1.25 2.5 5 10 10 20 40 0 0.3 1.25 5 20 80 Oh 1.5 3 6 18 0 250 500 1000 2000 4000 Overt maternal toxicity Percent prenatal mortality" 25 25 81 100 7 12 42 22 100 3 1 2 9 8 36= Ref. 74 57 79 78 25 21 15 41 * 95= 100= 58 7 6 13 14 87 97 62 57 76 • Decreased body weight gain or marked edema compared to vehicle-dosed controls. A (+) or (-) indicates the presence or absence of an effect. b Percentage of absorptions plus late gestational deaths relative to all implantations. A (+) or (-) is given to indicate the presence of absence of an effect. 0 TCDD administered in a single or divided doses between gestational days 20 and 40. " Effects include thickening and reddening of the eyelids, weight loss, dryness and granularity of the skin, loss of hair, and, in some cases, anemia, purpura, and bleeding from the nose and mouth. • Single dose of TCDD administered on gestational day 14. ' TCDD administered daily on days 6-15 of gestation. = Significant at p <0.05. h Single dose of TCDD administered on gestational day 7 or 9. ' TCDD administered daily on days 7-16 of gestation. From Couture, L. A., Abbott, B. D., and Birnbaum, L. S., Teratology, 42, 619, 1990. 288 ------- related, and there appear to be species and/or strain differences in susceptibility to TCDD-in- duced prenatal mortality. The rank order of sus- ceptibility from the most sensitive to least sen- sitive species would appear to be monkey = guinea pig > rabbit = rat = hamster > mouse. However, an important caveat must be applied to the information presented in Table 1. This is that the time period during which exposure of the embryo/fetus to TCDD occurs is just as im- portant a determinant of prenatal mortality as is the dose of TCDD administered. This point is illustrated in the text that follows when prenatal mortality is described for different strains of mice. It is important to note that the concept of a critical time period for exposure makes the anal- ysis of lethality data in the embryo/fetus quali- tatively different from that which might be ap- plied to similar data in adult animals. For example, a common dosing regimen used in mice, rats, and rabbits (Table 1) is to administer 10 daily doses of TCDD to the pregnant dam on days ~6 to 15 of gestation. This dosing regimen is ex- pected to cover the critical period of early de- velopment that results in the greatest incidence of prenatal toxicity. However, in nearly all spe- cies of adult laboratory mammals, a single lethal dose of TCDD would be expected to produce a similar delayed onset death regardless of the age of the adult animal. Susceptibility to TCDD-in- duced prenatal mortality, in contrast, may be greatly dependent on the age of the embryo/fetus. In this case, multiple doses of TCDD that cover this critical period may result in prenatal mor- tality, whereas a single dose may miss the critical time and not result in prenatal mortality. The following paragraphs illustrate a type of analysis using an index of cumulative maternal dose similar to the type of analysis that might be applied to lethality data resulting from multiple dosing of adult animals. After presenting the re- sults of applying this type of analysis to prenatal mortality data from different species, the caveat of critical time dependence is applied to the data obtained by using different strains of mice. This then illustrates the importance of considering dosage regimen when evaluating published pre- natal mortality data. In regard to prenatal mor- tality, a difference of one gestational day may be critically important. We show that the form of analysis using cumulative maternal dose gives the greatest possible degree of species variation. In turn, different species seem to actually be more similar with respect to susceptibility to prenatal mortality than would be superficially suggested by this type of analysis. Using the cumulative dose data that are given in Table 1, there appears to be a 10- to 20-fold difference in the fetolethal potency of TCDD when the monkey/guinea pig is compared to the rabbit/ rat/hamster. In the CD-I mouse administered cu- mulative doses of TCDD on gestational days 7 to 16, not including day 6, it appears to require a daily dose of 200 u>g TCDD/kg to significantly increase prenatal mortality. Given a TCDD half- life of approximately 5.5 days in the pregnant dam,58 the pregnant CD-I mouse would be ex- posed to a maximal accumulated dose of ap- proximately 1200 (Jig TCDD/kg by the lowest dosage regimen that significantly increased pre- natal mortality. Therefore, by using the index of cumulative dose, the CD-I mouse would appear to be ~ 1200-fold less sensitive than the monkey/ guinea pig for TCDD-induced prenatal mortality. However, in NMRI mice administered TCDD only on day 6 of gestation, prenatal mortality begins to increase after a single dose of 45 |Jig TCDD/kg.59 The NMRI embryo/fetus is less sus- ceptible to TCDD-induced prenatal mortality when the TCDD is administered on later gesta- tional days up to day 15. Thus, there appears to be only about a 45-fold difference between the monkey/guinea pig and the NMRI mouse when the NMRI embryo/fetus is exposed specifically on day 6. In C57BL/6 mice, prenatal mortality is significantly increased after a single maternal dose of 24 (xg TCDD/kg given on gestational day 6.60 This mouse strain therefore is about 20- to 30-fold less sensitive to TCDD-induced prenatal mortality than is the monkey/guinea pig when exposed specifically on day 6. As with the NMRI mouse, there was little or no increase in prenatal mortality for the C57BL/6 strain when TCDD was administered to the pregnant dam on ges- tational days 8, 10, 12, or 14. The concept of a critical window for TCDD- induced lethality in the embryo/fetus suggests an explanation for the apparent insensitivity of the CD-I embryo/fetus exposed to cumulative doses of TCDD. It could very well be that the critical 289 ------- window for prenatal mortality in the mouse oc- curs on or before gestational day 6. If the embryo/ fetus is not exposed to TCDD by gestational day 6, much larger doses of TCDD are required to produce prenatal mortality. Given that exposure of the pregnant CD-I dams did not begin until gestational day 7, this interpretation is consistent with the ability of a single 24 jig TCDD/kg dose to increase the incidence of prenatal mortality when administered to pregnant C57BL/6 mice on gestational day 6, but not when administered on gestational days 8, 10, 12, or 14.60 Similarly, Neubert and Dillman59 found that the largest in- crease in prenatal mortality occurred when a sin- gle dose of TCDD was given on gestational day 6 compared to when the TCDD dose was ad- ministered on one of the gestational days 7 to 15. In addition, this would suggest that the CD-I embryo/fetus does not have quite the relative in- sensitivity to the lethal effects of TCDD com- pared to the embryo/fetus of other species that would be indicated by using the cumulative ma- ternal dose as the index of exposure. Human pregnancies also are affected by crit- ical periods during which the human embryo/ fetus is susceptible to particular forms of chem- ical-induced developmental toxicity. In the first 2 weeks of human pregnancy, the predominant adverse developmental response is prenatal mor- tality, although other manifestations of devel- opmental toxicity may occur during this period. The gestational period between weeks 2 through 8 is when the human embryo/fetus is most sus- ceptible to the occurrence of structural malfor- mations. Minor structural defects and postnatal functional alterations are the dominant adverse developmental effects that occur after 8 weeks of pregnancy. It should be noted that the concept of a critical window for prenatal mortality could potentially alter all of the species comparisons made pre- viously that were based on the cumulative ma- ternal doses shown in Table 1. If this turned out to be the case, then the true differences between species with respect to their susceptibility to TCDD-induced prenatal mortality could be sub- stantially less than those indicated by using the cumulative maternal dose. This, of course, would involve a comparison between species using only single doses of TCDD given during the critical time period for each species. At the present time, it does not seem possible to make such a com- parison from the information available in the lit- erature. Similar to fish and birds, the mammalian em- bryo/fetus is more sensitive to the lethal action of TCDD than is the adult. The maternal dose of TCDD that causes 58% fetal mortality in ham- sters is 64 to 280 times less than the LD50 of TCDD in adult hamsters.54'55-61 In Sprague-Daw- ley rats, the cumulative maternal dose of TCDD that causes 41 % prenatal mortality is 5 to 10 times less than the approximate LD50 of TCDD in adult rats of the same strain.62'63 In rhesus monkeys, the cumulative maternal TCDD dose that causes 81% prenatal mortality is 6 and 25 times less, respectively, than the lowest TCDD dose re- ported to cause mortality in 1-year-old and adult rhesus monkeys.64"66 A general finding in all nonprimate labora- tory mammals, with the possible exception of the hamster, is that TCDD-induced prenatal mortal- ity is most commonly associated with maternal toxicity that is not severe enough to result in maternal lethality. This is seen in Table 1 for the guinea pig, rabbit, rat, and mouse. In each spe- cies, the dose-response relationship for maternal toxicity, indicated by decreased maternal weight gain and/or marked subcutaneous edema of the dam, is essentially the same as that for increased prenatal mortality. What this means is that there may be an association between the fetolethal ef- fect of TCDD and maternal toxicity in all of these species. Even in the hamster, where maternal toxicity is far less severe, fetuses exhibit in- creases in neutrophilic metamylocytes and bands, whereas increases in leukocyte number and bands also are found in maternal blood.56>S7 More re- cently, in mice, it has been shown that TCDD exposure causes rupture of the embryo-maternal vascular barrier, which results in hemorrhage of fetal blood into the maternal circulation.67 It is not known whether these extraembryonic he- matologic changes are contributory to or coin- cidental with developmental toxicity in these spe- cies. However, their occurrence reinforces the concept that prenatal mortality can be associated with maternal toxicity. In rhesus monkeys, on the other hand, fewer data are available to make the association be- 290 ------- tween prenatal mortality and maternal toxicity. While only small numbers of monkeys have been studied to date, the results following dietary exposure to 25 ppt TCDD6869 and 50 ppt TCDD70"73 before and during pregnancy suggest that TCDD-induced prenatal mortality can occur in monkeys in the absence of overt toxic effects on the mother (see Section III. A. 1). In other stud- ies, developmental toxicity in monkeys exposed to a total cumulative maternal dose of 1 (xg TCDD/kg administered during the first trimester indicated a high incidence of prenatal mortal- ity.65'74 However, maternal toxicity occurred in some but not all of the mothers exposed. In these monkeys, 13 of 16 pregnancies resulted in pre- natal mortality. Within 20 to 147 days after abort- ing, 8 of the 13 females that had aborted showed signs of maternal toxicity and 3 of these monkeys died. Thus, the remaining 5 of 13 instances of prenatal mortality apparently occurred in the ab- sence of overt maternal toxicity. The results of these studies indicate that some levels of TCDD exposure can result in prenatal mortality in mon- keys even though overt toxicity seems absent in the mother. As is described in Section III.A.I, however, only limited attention has been given to female reproductive toxicity in general, and to the effects of maternal toxicity during pregnancy on fetal development in particular. Therefore, the relationship between maternal toxicity and pre- natal mortality in the monkey is not well charac- terized. The integrity of the embryo-maternal vascular barrier, for example, has not been evalu- ated after TCDD exposure. It is possible that TCDD-induced developmental toxicity results from actions exerted on the mother, embryo/ fetus, placenta, or any combination of these sites. In most laboratory mammals, gestational ex- posure to TCDD produces a characteristic pattern of fetotoxic responses that consist of thymic hy- poplasia, subcutaneous edema, decreased fetal growth, and prenatal mortality. In addition to these common fetotoxic effects are other effects of TCDD that are highly species specific. Ex- amples of the latter are cleft palate formation in the mouse and intestinal hemorrhage in the rat. Table 2 shows those maternal and fetal toxic re- sponses that are produced by gestational exposure to TCDD in various species of laboratory mam- mals. In the mouse, hydronephrosis is the most sensitive sign of prenatal toxicity, followed by cleft palate formation and atrophy of the thymus at higher doses, and by subcutaneous edema and mortality at maternally toxic doses.59'75"77 In the rat, TCDD prenatal toxicity is manifested by in- testinal hemorrhage, subcutaneous edema, de- creased fetal growth, and mortality.62-78 Struc- tural abnormalities do occur in the rat but only at relatively large doses.75 In the hamster fetus, hydronephrosis and renal congestion are the most sensitive effects, followed by subcutaneous edema and mortality at fetolethal doses.56-57 In the rab- bit, an increased incidence of extra ribs and pre- natal mortality is found,79 whereas in the guinea pig and rhesus monkey, prenatal mortality is seen.57-74 4. Structure-Activity Relationships in Laboratory Mammals The structure-activity relationship for devel- opmental toxicity in laboratory mammals is gen- erally similar to that for Ah receptor binding. Gestational treatment of rats with CDD conge- ners that do not bind the Ah receptor, 2-MCDD, 2,7-DCDD, 2,3-DCDD, or 1,2,3,4-TCDD, does not cause TCDD-like fetotoxic effects.78 On the other hand, hexachlorodibenzo-p-dioxin, which has intrinsic Ah receptor activity, produces fe- totoxic responses in rats that are essentially iden- tical to those of TCDD.80 Similarly, when ad- ministered to pregnant rhesus monkeys or CD-I mice, PCB congeners that act via an Ah receptor- mediated mechanism, 3,3',4,4'-TCB and 3,3',4,4',5,5'-HCB, cause the same type of fetotoxic effects as TCDD. In contrast, 4,4'-DCB, 3,3',5,5'-TCB, 2,2',4,4',5,5'-HCB, 2,2',4,4',6,6'-HCB, and 2,2',3,3',5,5'-HCB, which essentially have no or very weak affinity for the Ah receptor, do not produce a TCDD- like pattern of prenatal toxicity in mice.65-81"83 Thus, most structure-activity results for overt fetotoxic effects of the halogenated aromatic hy- drocarbons are consistent with an Ah receptor- mediated mechanism. Nevertheless, one finding that stands out as being inconsistent is that 2,2',3,3',4,4'-HCB, which has a very weak, if any, affinity for binding to the Ah receptor, causes the same pattern of fetotoxic effects in mice as does TCDD.81 291 ------- TABLE 2 Developmental Toxicity Following Gestational Exposure to 2,3,7,8-TCDD Treatment Sacrifice Species/strain Mice/C57BL/6N Mice/C57BL76N Mice/C57BL76N Mice/C57BL76N Mice/C57BL/6N Mice/C57BL/6J Mice/C57BL/6J Dally dose* 0, 1 , or 3 (ig/kg 0, 12, 17, or 22 ^g/kg 0,3, or 12 (ig/kg 0 or 3 (ig/kg 0, 6, 9, 12, 15, or 18 M-9/kg 0 or 3 M-g/kg (subcutaneous) 20 jig/kg days" 10, 10-13 10 11,10-13 10-13 10, 12 6-15 10 da 18 18 18 18 18 18 17 Maternal effects0 Mice/C57BL76J Mice/NMR NR 0, 0.5, 1, 2, or 4 y-g/kg 6-15 0.3, 3, 4, 5, or 9 ng/kg 6-15 18 18 Increase in liver-to-body weight ratio Increase in liver-to-body weight ratio Increase in liver-to-body weight ratio Increase in liver-to-body weight ratio and weight gain Increase in liver-to-body weight ratio Increase in liver-to-body weight ratio Increase in liver-to-body weight ratio NR Mice/CF-1 Mice/DBA Mice/DBA Mice/CD-1 Mice/CD-1 Rats/CD Rats Sprague-Dawley Rats/Sprague-Dawley 0, 0.001, 0.01, 0.1, or 1 .3 (ig/kg 0 or 3 M.g/kg (subcutaneous) 0, 0.5, 2, 4, or 8 ng'kg 0, 1 , or 3 (ig/kg (subcutaneous) 0, 25, 50, 100, 200, or 400 M-g/kg 0 or 0.5 M-g/kg 2 M-g/kg (subcutaneous) 0, 0.125, 0.5, or 2 jig/kg 0.03, 0.125, 0.5, 2, or 6-15 6-15 6-15 6-15 6-15 6-15 9-10 or 13-14 0-2 6-15 NR 18 18 17 17 20 20 21 None Increase in liver-to-body weight ratio Increase in liver-to-body weight ratio; decrease in thymus-to- body weight ratio None Increase in liver-to-body weight ratio None at 0.5 M.Q kg NR at 2 ng/kg Decrease in weight gain Decrease in weight gain; toxic- Rats/Wistar 0,0.125,0.25,0.5, 1,2, 4, 8, or 16 M-g/kg ity 5-14 21 Toxicity Guinea pigs/Hartley 0, 0.15, or 1.5 jig/kg 14 Hamsters/Golden 0, 1.5, 3, 6, or 18 mj/kg 7, 9 Syrian 58 Increase in mortality; toxicity 15 Increase in liver-to-body weight ratio Rabbits/New Zealand 0, 0.1, 0.25, 0.5, or 1 tig/kg 6-15 28 Decrease in weight gain; toxicity Embryo/fetal effects0 Ref. Increase in cleft palate 144 and hydronephrosis Increase in cleft palate 102 and hydronephrosis Increase in cleft palate 145 and hydronephrosis Increase in hydrone- 277 phrosis Increase in cleft palate 278 and hydronephrosis Increase in cleft palate 77 and kidney anomaly Increase in cleft palate, 273 hydronephrosis, and fetal body weight Increase in cleft palate, 149 hydronephrosis, and fetal body weight Increase in cleft palate 59 and fetal mortality; de- crease in fetal body weight Increase in cleft palate 276 and hydronephrosis Increase in cleft palate 77 and kidney anomaly Increase in cleft palate 149 and hydronephrosis Increase in cleft palate 77 and kidney anomaly Increase in cleft palate, 76 hydronephrosis, and fetal mortality — 77 Increase in kidney anomaly Decrease in fetal body 274 weight Increase in fetal mortal- 62 ity, resorptions, edema, and gastroin- testinal hemmorrhage Increase in fetal mortal- 78 ity, edema, and gas- trointestinal hemor- rhage; decrease in fetal weight Increase in fetal mortal- 56 ity Increased fetal mortality, 56 hydronephrosis, and renal congestion; de- creased thymus size Increase in fetal mortal- 79 ity and resorptions; extra ribs 292 ------- TABLE 2 (continued) Developmental Toxicity Following Gestational Exposure to 2,3,7,8-TCDD Species/strain Rhesus monkeys Monkeys/rhesus Dally dose* 0, 5, 25, 50, or 500 ppt 7 months before and during pregnancy 0,0.2," 1,d 1,»or 5" j Treatment days" Chronic 20-40 Sacrifice day* Maternal effects' Increase in mortality; toxicity Increase in mortality; toxicity Note: NR = Not reported. Embryo/fetal effects' Ret. Increase in fetal mortal- 71, ity 223 Increase in fetal mortal- 65 ity a Oral exposure unless otherwise noted. 6 All days adjusted to reflect plug day — gestational day 0. c Effects reported are only those that were statistically significant. d Cummulative dose divided into nine oral doses administered between days 20 and 40 of gestation; two to four monkeys/dose. 8 Three animals given single oral dose on either gestational days 25, 30, 35, or 40; 12 monkeys total. From Couture, L. A., Abbott, B. D., and Birnbaum, L. S., Teratology, 42, 619, 1990. 5. Humans In the Yusho and Yu-Cheng poisoning epi- sodes, developmental toxicity was reported in babies born to affected mothers who had con- sumed rice oil contaminated with PCBs, CDFs, and PCQs.15~17-84 In these incidents, it is essen- tially impossible to determine the contribution of TCDD-like vs. nonTCDD-like congeners to the fetal/neonatal toxicity. Nevertheless, high peri- natal mortality was observed among hyperpig- mented infants born to affected Yu-Cheng women, who themselves did not experience in- creased mortality.16 Thus, in humans, the devel- oping embryo/fetus may be more sensitive than the intoxicated mother to mortality caused by halogenated aromatic hydrocarbons. In most cases, women who had affected chil- dren in the Yusho and Yu-Cheng episodes had chloracne themselves.85 Based on this evidence, Rogan suggested that "exposure to amounts in- sufficient to produce some effect on the mother probably lessens the chance of telepathy consid- erably."85 In support of this interpretation, overt signs of halogenated aromatic hydrocarbon tox- icity were not observed in infants born to appar- ently unaffected mothers in the Seveso, Italy, and Times Beach, MO, TCDD incidents.86-87 In laboratory mammals, the studies sum- marized previously in Table 1 indicate an appar- ent association between prenatal mortality and maternal toxicity in nonprimate species. How- ever, some TCDD-exposed rhesus monkeys were not able to carry their pregnancies to term, even in the absence of any overt signs of maternal toxicity. This result in monkeys indicates that the relationship between maternal toxicity and any prenatal toxic effects on the human embryo/fetus must be cautiously defined. More data may be required to determine whether or not there is any association between overt maternal toxicity and embryo/fetal toxicity both in monkeys and hu- mans. Effects of chemical exposure on normal de- velopment of the human fetus can have four out- comes, depending on the dose and time during gestation when exposure occurs: (1) fetal death, (2) structural malformations, (3) organ system dysfunction, and (4) growth retardation. In the Yusho and/or Yu-Cheng incidents, all of these outcomes were found.15-17'84 Increased prenatal mortality and low birth weight suggesting fetal growth retardation were observed in affected Yusho and Yu-Cheng women. 16-84-88~92 A struc- tural malformation, rocker bottom heel, was ob- served in Yusho infants.84 Organ dysfunctions involving the central nervous system (CNS), which were characterized by delays in attaining developmental milestones and neurobehavioral abnormalities, were reported in Yu-Cheng chil- dren exposed transplacentally.92-93 293 ------- Organs and tissues that originate from embry- onic ectoderm are well-known targets for toxicity following exposure to TCDD-like halogenated aromatic hydrocarbons. For example, treatment of adult monkeys with TCDD results in effects involving the skin, meibomian glands, and nails.70 Similarly, a hallmark sign of fetal/neonatal tox- icity in the Yusho and Yu-Cheng episodes was an ectodermal dysplasia syndrome, which is characterized by hyperpigmentation of the skin and mucous membranes, hyperpigmentation and deformation of finger and toe nails, hypersecre- tion of the meibomian glands, conjunctivitis, gin- gival hyperplasia, presence of erupted teeth in newborn infants, and altered eruption of per- manent teeth, missing permanent teeth, and ab- normally shaped tooth roots.15-17'84-88'91-92'94-96 Accelerated tooth eruption has been observed in newborn mice exposed to TCDD by lactation,97 as well as in the human infants mentioned pre- viously. In addition, other effects have been re- ported in the Yusho and Yu-Cheng exposed in- fants that resemble effects observed following TCDD exposure in adult monkeys. These include subcutaneous edema of the face and eye- lids.70-84-89'98 Also, larger and wider fontanels, and abnormal lung auscultation were found in the human infants.84'89'92 The similarities between certain effects reported in human infants exposed during the Yusho and Yu-Cheng incidents, as well as adult monkeys and neonatal mice exposed to TCDD, enhance the probability that certain effects reported in the human infants were caused by the TCDD-like PCB and CDF congeners in the contaminated rice oil ingested by the mothers of these infants. Chloracne is the most often cited effect of TCDD exposure involving the skin in adult hu- mans. This effect has an animal correlate in the hairless mouse and can be studied by using a mouse teratoma cell line in tissue culture.3 Never- theless, it has rarely been explicitly recognized in the TCDD literature that the nervous system, like the skin, is derived from embryonic ecto- derm.99 As is described in Section II.C.2, neuro- behavioral effects occur following transplacental and neonatal exposure to TCDD-like congeners in mice, as well as transplacental exposure to TCDD in monkeys. In addition, some of the Yu- Cheng children that were exposed transplacen- tally to PCBs, PCDFs, and PCQs have affected a clinical impression of developmental delay or psychomotor delay including impairment of in- tellectual development.92-93 Because there is a clustering of effects due to TCDD-induced tox- icity in organs derived from ectoderm, it is rea- sonable to speculate that direct effects of TCDD- like congeners on the CNS are responsible for some of the neurobehavioral effects observed in these children. Effects of TCDD on EOF recep- tors are associated with certain aspects of the ectodermal dysplasia syndrome, such as hyper- keratinization of the skin100 and accelerated tooth eruption.97 Decreased autophosphorylation of the EOF receptor in human placentas is associated with decreased birth weight in infants born to exposed mothers 4 years after the initial Yu-Cheng exposure incident.101 This last result supports the earlier conclusion that careful study is needed to define the relationship between maternal toxicity, placental toxicity, and developmental toxicity in humans. In addition, further research is needed to characterize and elucidate the mechanisms by which TCDD affects the nervous system. B. Structural Malformations Developmental effects consisting of cleft pal- ate, hydronephrosis, and thymic hypoplasia are produced in mice following in utero exposure to halogenated dibenzo-/?-dioxin, dibenzofuran, and biphenyl and naphthalene congeners, which bind stereospecifically to the Ah receptor.102-105 Of these effects in the mouse, cleft palate is less responsive than hydronephrosis inasmuch as the latter is induced in the absence of cleft palate.60 Both responses can be induced at TCDD doses that are not otherwise overtly toxic.75 The po- tency of TCDD for producing teratogenesis in the mouse is clearly evident when one considers that only 0.0003% of a maternally administered dose can be isolated from the fetal palatal shelves or kidneys. More specifically, a maternal TCDD dose of 30 M-g/kg administered on gestational day 11 results in a tissue concentration of 0.65 pg TCDD/mg in the palatal shelves 3 days after dos- ing, and the same tissue concentration of TCDD is present in the kidneys at that time.106 294 ------- Susceptibility to the developmental toxicity of TCDD in mice depends on two factors: the genotype of the fetus and the stage of develop- ment at the time of exposure. Because mouse strains that produce Ah receptors with relatively high affinity for TCDD respond to lower doses of TCDD than mouse strains that produce rela- tively low-affinity Ah receptors,107'108 the Ah re- ceptor is thought to mediate the developmental effects of TCDD.3 Thus, one genetically encoded parameter that determines the responsiveness of different mouse strains is the Ah receptor protein itself. The differences that exist between mouse strains with respect to developmental respon- siveness to these chemicals are not absolute be- cause all strains, including those with Ah recep- tors of relatively low affinity, respond when exposed to sufficiently large doses during the critical period of organogenesis.109 In the mouse, the peak times of fetal sensitivity vary slightly depending on which developmental effect is used as the endpoint. However, exposure between days 6 and 15 of gestation will produce teratogene- 60,75 In inbred strains of mice, the developmental response, characterized by altered cellular pro- liferation, metaplasia, and modified terminal dif- ferentiation of epithelial tissues,3 is extremely organ specific, occurring only in the palate, kid- ney, and thymus.109 Pharmacokinetic differences are not responsible for this high degree of tissue specificity, and Ah receptors are not found ex- clusively in the affected organs.110-111 Therefore, other factors intrinsic to the palate, kidney, and thymus appear to play a role along with the Ah receptors in these tissues in producing the struc- tural malformations. For various developmental effects, the time at which exposure occurs is im- portant and defines the critical period during which the toxicant must be present in order to produce the effect. This critical period differs among organs and tissues within organs. Differences exist between mammalian spe- cies with respect to susceptibility to the devel- opmental effects of TCDD. Although genetic dif- ferences between species or strains may affect absorption, biotransformation, and/or elimina- tion of TCDD by the maternal system and its transport across the placenta, such species dif- ferences do not account for the lack of cleft palate formation in species other than mice.109 Rather, the species differences in susceptibility to cleft palate formation appear to be due to differences in the interaction between TCDD and the devel- oping palatal shelves themselves. This is dem- onstrated by the occurrence of similar responses when palatal shelves from different species are exposed to TCDD in organ culture.106-112'113 The key difference is that, relative to the concentra- tion of TCDD that affects murine palatal shelves in culture, much higher concentrations of TCDD are required to elicit the same effects in cultured palatal shelves from other species (Table 3). As a result, it appears that differences in the res- ponsivity of palatal tissue to the effects of TCDD explain the absence of cleft palate in nonmurine species except at maternal doses that are fetotoxic and maternally toxic.75'109 In mice and hamsters, hydronephrosis can be elicited at TCDD doses that are neither fetotoxic nor maternally toxic,57 whereas thymic hypopla- sia is a fetal response to TCDD observed in vir- tually all laboratory mammalian species tested.114 Studies in humans have not clearly identified an association between TCDD exposure and struc- tural malformations.86-115"117 1. Cleft Palate a. Characterization of TCDD Effect Palatal shelves in the mouse originate as out- growths of the maxillary process. Eventually, they come to lie vertically within the oral cavity on both sides of the tongue. In order to form the barrier between the oral and nasal cavities, the shelves in the mouse must reorient themselves from a vertical direction to a horizontal direction. Once they come together horizontally, their me- dial aspects bring apposing epithelia into close contact.118-119 At this stage, the apposing medial edge epithelia of the separate palatal shelves each consist of an outer layer of periderm that overlays a strata of cuboidal shaped basal cells. These basal cells, in turn, rest on top of a continuous basal lamina. There is a sloughing of the outer periderm cells followed by the formation of junc- tions between the newly apposing basal epithelial 295 ------- TABLE 3 TCDD Responsiveness of Palatal Shelves from the Mouse, Rat, and Human in Organ Culture Molar concentration of TCDD Species Mouse Rat" Human" Prevention of the epithelium-to- mesenchyme transformation process LOEL EC100 1 x10-'3 1 x10-'° 5x1Q-11 5x10-11 1x10-a 1x10-" Cytotoxicity 1x10-10 1x10-7 1x10-7 • At the highest concentration tested, 60% of the palatal shelves failed to undergo the transformation process. b One of four shelves responded by failing to undergo the transformation process at 5x10-" M. From Birnbaum, L S., Biological Basis lor Risk Assessment of Dioxins and Related Compounds, Gallo, M. A., Scheuplein, R. J., and van der Heijden, C. A., Eds., Branbury Report 35, Cold Spring Harbor Laboratory Press, Cold Spring Harbor, NY, 1991, 51. cells that are left within the seam. The midline seam thus formed consists of two healthy-ap- pearing layers of basal cells (one on each side of the seam), whereas the outer periderm cells have already been shed. As the palatal shelf continues to grow, the bilayer seam, which itself grows at a slower rate, turns into a single layer of cells, and then breaks up into small islands of cells. Eventually, the basal lamina disappears, and the elongating former basal cells within the small islands extend filopodia into the adjacent con- nective tissue. During this process, the former basal cells lose epithelial characteristics and gain fibroblast-like features. Essentially, the above- described process makes the medial edge epithe- lium an ectoderm that retains the ability to trans- form into mesenchyme. Upon completion of this epithelial to mesenchyme transformation, the once separate and apposing palatal shelves are fused so that a single continuous tissue is formed.120'121 Cleft palate can result from a failure of the shelves to grow and come together, or a failure of the shelves to fuse once they are in close ap- position.122 TCDD and other Ah receptor agonists act by allowing the shelves to grow and make contact, but the subsequent process involving the epithelial-to-mesenchyme transformation does not occur. Therefore, a cleft is formed as the palatal shelves continue to grow without fusing. When TCDD is administered to pregnant mice on ges- tational days 6 to 12, the incidence of cleft palate formation increases with time. However, day 12 is a critical window, after which the incidence of cleft palate formation decreases. No cleft palates are formed when TCDD is administered on day 1460 Palatal shelves of the mouse, rat, and human can be removed from the fetus and placed into organ culture. Under these conditions, when the separate shelves are placed in an apposing con- dition in vitro, sloughing periderm cells are trap- ped within the seam. 12° Thus, due to the presence of these trapped dead cells, the fusion process was originally described as a process of pro- grammed cell death. 118'119>123 However, the newer idea, which presumes transformation of the basal epithelial cells into mesenchyme rather than their death, is believed to be valid under explant con- ditions in vitro, as well as in vivo.120 When ex- posed to TCDD as explants in vitro, the palatal shelves of the mouse, rat, and human all respond to TCDD in a similar way by not completing the fusion process. 106'112>113'124 There is death of the outer peridermal cells, after which, the epithelial- to-mesenchyme transformation of the basal epi- thelial cells does not occur. Instead, there is a differentiation of these basal cells into a stratified squamous epithelium such that they eventually 296 ------- resemble the squamous keratinizing oral cells within the tissue.125 Table 3 shows the lowest TCDD concentra- tion that prevents the epithelial-to-mesenchyme transformation process in isolated palatal shelves (LOEL), the TCDD concentration that produces a 100% maximal response (EC100), and the lowest concentration of TCDD that produces cytotox- icity. Palatal shelves of rats and humans respond to TCDD in a manner identical to the mouse; however, higher concentrations of TCDD are re- quired to prevent the epithelial-to-mesenchyme transformation process. The relative insensitivity of rat palatal shelves may explain the lack of cleft palates when fetal rats are exposed to nonmater- nally toxic doses of TCDD. Sensitivity of human palatal shelves to TCDD in vitro is similar to the rat. This suggests that exposure to maternally toxic and fetotoxic doses of TCDD would be required to cause cleft palate formation in hu- mans. A disruption in the normal spatial and tem- poral expression of EGF, TGF-a, TGF-pl, and TGF-fJ2 correlates with altered proliferation and differentiation in the medial region of the de- veloping palate resulting in a palatal cleft. Thus, the abnormal proliferation and differentiation of TCDD-exposed medial cells may be related to reduced expression of EGF and TGF-a. Also, decreased levels of immunohistochemically de- tectable TGF-pl could contribute to the contin- ued proliferation and altered differentiation of medial cells.126 It is important to note that EGF and TGF-a both exert their actions by binding to EGF receptors. The differentiation of basal cells to a stratified squamous epithelium, which re- sembles the keratinizing oral epithelium within the developing palate mentioned above, is similar to certain effects of TCDD that can be studied in cultured human keratinocytes. These effects in cultured human keratinocytes involve altered EGF binding to those cells. In addition, the Ah receptor is implicated in producing this re- sponse.100 Thus, the mechanisms by which TCDD produces a palatal cleft in the mouse may have similarities to the mechanisms by which TCDD produces other effects that are part of the ecto- dermal dysplasia syndrome. This is consistent with the description given by Fitchett and Hay,120 in that the medial edge epithelium within the developing palate is essentially an ectoderm that retains the ability to transform into mesenchymal cells. b. Evidence for an Ah Receptor Mechanism i. Genetic When wild-type C57BL/6 (AhbAhb) mice are crossed with DBA/2 (AhdAhd) mice that contain a mutation at the Ah locus, all of the heterozy- gous B6D2F1 progeny (AhbAhd) resemble the wild-type parent in that arylhydrocarbon hydrox- ylase (AHH) activity is inducible by TCDD and other halogenated aromatic hydrocarbons.127 Test crosses between the B6D2F1 progeny and each original parent strain, and other B6D2F1 progeny mice demonstrate that in the C57BL/6 and DBA/ 2 strains susceptibility to AHH induction segre- gates as a simple dominant trait in the backcross and F2 progeny. Thus, the trait of AHH induci- bility is expressed in progeny that contain the AhbAhb and AhbAhd genotypes, but is not ex- pressed in the AhdAhd progeny from these crosses. Certain other effects of TCDD, such as its bind- ing affinity for the hepatic Ah receptor,128 thymic atrophy,107 hepatic porphyria,129 and immuno- suppressive effects,130-131 have been shown in similar genetic crosses and test crosses to seg- regate with the Ah locus, which permits AHH induction. Thus, for these effects of TCDD, ge- netic evidence demonstrates an involvement of the Ah locus.3 Nebert's group was the first to relate devel- opmental toxicity to the Ah locus in mice.132-133 Subsequently, Poland and Glover107 administered a single 30-jig TCDD/kg dose to pregnant mice on gestational day 10. It was found that there was a 54% incidence of cleft palate in homo- zygous C57BL/6 (AhbAhb) fetuses, a 13% inci- dence in heterozygous B6D2F1 (C57BL/6 and DBA/2 hybrid, AhbAhd) fetuses, and only a 2% incidence in homozygous DBA/2 (AhdAhd) fe- tuses. This pattern of inheritance, in which the incidence of developmental toxicity in the het- erozygous Fl generation is intermediate between that of the homozygous parental strains, is con- sistent with the autosomal dominant pattern of inheritance described for AHH inducibility and 297 ------- the Ah locus,127 even if dominance is incomplete in the case of developmental toxicity. However, the pattern of inheritance for developmental tox- icity described when Poland and Glover107 crossed C57BL/6 and DBA/2 mice is not proof positive that the Ah locus is the only genetic locus that controls susceptibility to TCDD-induced devel- opmental toxicity in these mouse strains. To provide such proof, it is necessary to show genetic linkage between the susceptibility for de- velopmental toxicity and the Ah locus. The stan- dard of proof would be that developmental tox- icity and a particular allele at the Ah locus must always segregate together in genetic crosses be- cause if the loci are the same there can be no recombination between the loci. This is generally accomplished by demonstrating cosegregation between the two loci not only in crosses between the two homozygous parental strains, which in and of itself is insufficient proof of genetic link- age, but also in test crosses or backcrosses be- tween the heterozygous Fl hybrids with each homozygous parental strain. It was stated earlier in this section that certain effects of TCDD are well known to segregate with the Ah locus due to the results of appropriate crosses and backcrosses between responsive and nonresponsive mouse strains and their hybrid Fl progeny. With this standard of proof in mind, the evidence that specifically links develop- mental toxicity with the Ah locus is now de- scribed. It is intended that this information con- tain a considerable degree of detail. This is done so that the reader can independently determine whether or not the standard of proof has been satisfied by the evidence available. In order to strengthen their conclusion based on the results of simple crosses between C57BL/6 and DBA/2 mice, Poland and Glover107 planned to perform a backcross between the hybrid B6D2F1 and DBA/2. However, the low inci- dence of cleft palate in B6D2F1 mice would have required characterizing and phenotyping a pro- hibitively large number of fetuses. Alternatively, the backcross between B6D2F1 and C57BL/6 was considered in which AhbAhb and AhbAhd progeny would have been distinguished by the amount of high-affinity specific binding for TCDD in fetal liver. In this case, however, overlap between in- dividual mice would have made the results un- certain in some of the progeny. Therefore, it was not possible to obtain satisfactory results from either backcross. Instead, Poland and Glover107 examined the incidence of cleft palate in ten inbred strains of mice exposed to TCDD: five strains with high- affinity Ah receptors and five strains with low- affinity receptors. In the five latter strains, there was only a 0 to 3% incidence of cleft palate formation, whereas four of the five strains with high-affinity Ah receptors developed a 3=50% incidence. The one strain with high-affinity Ah receptors that did not follow the pattern, the CBA strain, also is resistant to cleft palate formation induced by glucocorticoids and may therefore be generally resistant to chemically induced cleft palate formation. Overall, these results indicate that cleft palate formation probably segregates with the Ah locus. The incidence of cleft palate formation was studied in fetuses from a cross between C57BL/6 and AKR/NBom mice administered 3,3',4,4'- TCAOB on gestational day 12.134 Although C57BL/6 mice are responsive for AHH induction and cleft palate formation, AKR mice are less responsive, requiring higher doses for both ef- fects. In a manner unlike the result of a cross between C57BL/6 and DBA/2, the incidence of cleft palate formation in the B6AKF1 progeny was <2%, showing that nonresponsiveness seg- regates as the dominant trait when C57BL/6 mice are crossed with AKR mice. Similarly, cleft pal- ate formation was virtually absent in the progeny of a backcross between AKR/NBom and B6AKF1, demonstrating dominance of the non- inducible trait. Although Ah phenotyping of the backcross progeny was not performed in this par- ticular study, Robinson et al.135 had previously evaluated segregation of the Ah locus in back- crosses between C57BL/6 and AKR/N mice. They found in these two strains that noninducibility for AHH activity segregates as the dominant trait. Thus, inducibility for cleft palate formation and AHH activity both segregate as dominant traits when C57BL/6 mice are crossed with DBA/2, but noninducibility is dominant for both traits when C57BL/6 mice are crossed with AKR/N. These results are consistent with the interpreta- tion that cleft palate induction probably segre- gates with the Ah locus. 298 ------- Like Poland and Glover,107 Hassoun et al.108 were unable to determine whether or not cleft palate formation segregates with the Ah locus in C57BL/6 and DBA/2 mice by performing simple backcrosses. Instead, they evaluated cosegrega- tion of the Ah locus and 2,3,7,8-TCDF-induced cleft palate formation using a series of recom- binant strains called BXD mice. These strains are fixed recombinants produced from an original cross between the two parental strains C57BL/6J and DBA/2J. Hybrid B6D2F1 mice were crossed to produce F2 progeny and these were strictly inbred by sister and brother matings into several parallel strains. The mice used in this study were from the F42 and F58 generations of inbreeding. It was found that the incidence of TCDF-induced cleft palate formation after matings within eight different BXD strains with high-affinity Ah re- ceptors is >85%. After similar matings with eight different BXD strains with low-affinity Ah re- ceptors, the incidence of TCDF-induced cleft pal- ate formation is <2%. These results of Hassoun et al.108 corroborate those of Poland and Glover107 and provide the best evidence currently available that cleft palate formation segregates with the Ah locus. Accordingly, the Ah locus and the Ah receptor are involved in the formation of palatal clefts that are induced by TCDD-like congeners. As additional evidence, stereospecific, high- affinity Ah receptors can be isolated from cytosol fractions prepared from embryonic palatal shelves. These receptors are present in palatal shelves of AhbAhb, C57BL/6 fetuses, but are not detectable in similar tissue from AhdAhd, AKR/J fetuses.136 However, the significance of this find- ing may be mitigated to some extent by the fol- lowing observation. In cytosols prepared from homogenates of whole embryo/fetal tissue (mi- nus head, limbs, tail, and viscera), the concen- tration of specific-binding TCDD receptors is 256 fmol/mg protein in C57BL/6 mice compared to a concentration of 21 fmol/mg protein in the less responsive DBA/2 strain, 15 fmol/mg protein in the less responsive AKR/J strain, and 19 fmol/ mg protein in the less responsive SWR/J strain. However, when embryonic tissue is cultured, the differences between the strains in receptor num- bers are less pronounced; and in the receptors isolated from cultured embryonic cells of differ- ent strains, there is only about a twofold differ- ence in the relative binding affinity for 3H-TCDD. The mechanistic reasons for the diminished de- gree of difference between responsive and less responsive mouse strains during embryonic cell culture are not known.137 The possible influence of maternal toxicity on cleft palate formation was evaluated by per- forming reciprocal blastocyst transfer experi- ments using the high-affinity Ah receptor NMRI and lower affinity Ah receptor DBA strains of mice.138 After administration of 30 fig TCDD/kg or 8 mg TCAOB/kg to pregnant dams on ges- tational day 12, 75 to 100% of all NMRI fetuses developed cleft palates. This was true whether the fetuses remained within the uterus of their natural mother or were transferred into the uterus of a DBA mouse. Under the same conditions, none of the 24 DBA fetuses transferred into an NMRI mother developed a cleft palate, even though 89% of their NMRI littermates were af- fected. Thus, these results, along with the pres- ence of Ah receptors in palatal shelves and re- sponsiveness of palatal shelves in organ culture to TCDD, indicate that cleft palate formation in mice is due to the direct effect of TCDD on the palatal shelf itself and is not secondary to ma- ternal toxicity. ii. Structure Activity Because the genetic evidence in mice indi- cates that the Ah receptor mediates TCDD-in- duced cleft palate formation and hydronephrosis (see Sections II.B.l.b.i and II.B.2.b.i), struc- ture-activity requirements based on Ah receptor- binding characteristics should predict the relative potencies of different agonists for producing cleft palate and hydronephrosis. Of the halogenated aromatic hydrocarbons, TCDD has the greatest affinity for binding to the Ah receptor and it is the most potent teratogen in inbred mouse strains. Table 4 shows the relative potencies for cleft palate induction and hydronephrosis in C57BL/6 mice for a number of TCDD-like congeners. Be- cause TCDD is the most potent, it is assigned a value of 1.000. When examined using probit analysis, the dose-response curve of each con- gener, compared to all of the others, did not deviate from parellelism. Therefore, the relative 299 ------- TABLE 4 Relative Teratogenic Potency of Halogenated Aromatic Hydrocarbon Congeners in C57BL/6 Mice Relative potency , TCDD/EDa, Congener) 1.000 0.235 0.100 0.095 0.049 0.026 0.010 0.005 . 0.004 0.0000287 1.000 0.444 0.333 0.057 0.021 0.074 0.049 0.009 0.018 0.0000894 Congener Cleft palate Hydronephrosis 2,3,7,8-TCDD 2,3,7,8-TBDD 2,3,7,8-TBDF 2,3,4,7,8-PeCDF 2,3,7,8-TCDF 1,2,3,7,8-PeCDF 1,2,3,4,7,8-HxCDF 2,3,4,7,8-PeBDF 1,2,3,7,8-PeBDF 2,3,4,5,3',4'-HxCB From References 102-104 and 109. potencies of the congeners are valid for any given incidence of cleft palate formation or hydrone- phrosis. The main finding, however, is that the rank order potency of the various congeners for producing these two developmental effects is generally similar to that for binding to the Ah receptor (Table 4), with the notable exception that the apparent binding affinities for the brom- inated dibenzofurans have not yet been reported. There are additional ligands for the Ah receptor that cause cleft palate formation in C57BL/6 mice at nonmaternally toxic doses, but they are not listed in the table. These include 3,3',4,4'- TCAOB,108 3,3',4,4'-tetrachlorobiphenyl,83 3,3',4,4',5,5'-hexachlorobiphenyl,82 and a mix- ture that contained 1,2,3,4,6,7- and 2,3,4,5,6,7- hexabromonaphthalenes.139 Also consistent with the structure-activity re- lationships for binding to the Ah receptor is the finding that a number of hexachlorobiphenyls do not induce cleft palate formation. These conge- ners either lack sufficient lateral substitution or are substituted in such a manner that they cannot achieve a planar conformation. Included in this category are the diortho- and tetraoAtfco-chlorine- substituted 2,2',3,3',5,5'-, 2,2',3,3',6,6'-, 2,2',4,4',5,5'-, and 2,2',4,4',6,6'-hexachloro- biphenyls.81 In addition, it is consistent with the structure-activity relationships that monoorffto- chlorine-substituted 2,3,4,5,3',4'-HCB is a weak teratogen. Its potency relative to that of TCDD varies from 3 x 10"5 to 9 x 10~5 for cleft palate formation, AHH induction, and hydronephrosis (Table 4).8 A result that would not be expected according to the structure-activity relationships for binding to the Ah receptor is that dio/t/zo-chlorine-sub- stituted 2,2',3,3',4,4'-hexachlorobiphenyl causes cleft palate formation and hydronephrosis in mice.81 However, another diorr/zo-chlorine-sub- stituted PCB congener, 2,2',4,4',5,5'-hexach- lorobiphenyl, also can cause hydronephrosis and is a very weak inducer of 7-ethoxyresorufin-O- deethylase (EROD) activity.140-141 It is consistent with the interpretation that 2,2',4,4',5,5'-hex- achlorbiphenyl is a partial Ah receptor agonist, that it can competitively displace TCDD from the murine hepatic cytosolic receptor, and, at large enough doses, that it can inhibit TCDD-induced cleft palate formation and immunotoxicity in C57BL/6 mice.140'141 These results suggest that PCB congeners do not have to be in a strictly planar configuration to cause teratogenesis. c. Species Differences Cleft palate is induced in rats only at mater- nally toxic TCDD doses that are associated with a high incidence of fetal lethality. Schwetz et al.80 reported an increased incidence of cleft palate after maternal administration of 100 (jug hexa- chlorodibenzo-/7-dioxin/kg/day on days 6 to 15 of gestation to Sprague-Dawley rats. Couture et al.142 also observed an increased incidence of cleft palate formation after a single dose of 300 jxg/ kg of 2,3,4,7,8-pentachlorodibenzofuran given to Fisher 344 rats. Similarly, cleft palate can be produced in fetal hamsters following maternally toxic and fetotoxic doses of TCDD.61 In monkeys, bifid uvula143 and bony defects in the hard palate65 were reported, but there were no corresponding soft tissue defects or clefts of the secondary palate. Cleft palates have not been reported in human fetuses of mothers accidentally exposed to TCDD or mixtures of PCBs and CDFs.17-115-117 Thus, sensitivity of the palate in mice to TCDD is unique. In other species, in- cluding humans, other forms of fetal toxicity oc- cur at doses lower than those required for cleft palate formation. 300 ------- 2. Hydronephrosis a. Characterization of TCDD Effect Hydronephrosis is the most sensitive devel- opmental response elicited by TCDD in mice. It is produced by maternal doses of TCDD too low to cause palatal clefting and is characterized as a progressive hydronephrosis preferentially oc- curring in the right kidney, which can be accom- panied by hydroureter and/or abnormal nephron development.77-102-144-147 Hyperplasia of the ur- eteric lumenal epithelium results in ureteric ob- struction. Therefore, the TCDD-induced kidney malformation in the mouse is a true hydrone- phrosis in that blockage of urine flow results in back pressure damaging or destroying the renal papilla.146 When dissected on gestational day 12 from control embryos, isolated ureters exposed to 1 x 10~10 M TCDD in vitro display evidence of epithelial cell hyperplasia.l48 This is significant in that it shows that the hydronephrosis response is due to the direct effect of TCDD on the ureteric epithelium. Embryonic cell proliferation within the ureter may be regulated by the actions of growth factors, including EOF.148 In control ur- eteric epithelia, the expression of EOF receptors decreases with advancing development, whereas after TCDD exposure, the rate of 3H-thymidine incorporation and EGF receptor numbers do not decline. Therefore, in TCDD-treated mice, there is a correlation between excessive proliferation of ureteric epithelial cells and increased expres- sion of EGF receptors. Other effects of TCDD on the developing kidney involve changes in extracellular matrix components and basal lamina.147 In TCDD-ex- posed fetal kidneys, extracellular matrix fibers are of a diameter consistent with type III collagen similar to such fibers in unexposed fetal kidneys. However, the abundance of these type III col- lagen fibers is reduced by TCDD treatment. In the developing kidney, these collagen fibers are associated with undifferentiated mesenchymal cells. Similarly, the expression of fibronectin, which also is associated with undifferentiated mesenchymal cells, is decreased by TCDD ex- posure. In the glomerular basement membrane, the distribution of laminin and type IV collagen is altered by TCDD exposure. These changes in the glomerular basement membrane may affect the functional integrity of the filtration barrier and could exacerbate the hydronephrosis and hydroureter. The proteins within the extracellular matrix and basal lamina that are altered by TCDD exposure, i.e., laminin, fibronectin, and colla- gen, are considered markers of a commitment to differentiate into epithelial structures. In the mouse embryo/fetus, TCDD exposure also blocks differentiation within the epithelium of the de- veloping palate. Although there are effects of TCDD exposure on EGF in the developing ureter, as well as the developing palate, the urinary sys- tem, unlike parts of the soft palate, is derived from mesoderm. Thus, it is important to note that the ectodermal dysplasia syndrome is intended to denote a clustering of effects that appear to in- volve ectoderm-derived organs. It is not intended to imply that all TCDD-induced developmental toxicity involves organs derived from ectoderm. b. Evidence for an Ah Receptor Mechanism \. Genetic With respect to involvement of the Ah locus in TCDD-induced hydronephrosis, very few ge- netic studies have been done. Prior to the dis- covery of the Ah locus, Courtney and Moore77 reported a 62% incidence of hydronephrosis in C57BL/6 mice exposed to a maternal TCDD dose of 3 (jLg/kg/day on days 6 to 15 of gestation, whereas the incidence in similarly exposed DBA/2 mice was only 26%. More recently, Silkworth et al.149 re- ported that when TCDD is administered on ges- tational days 6 to 15 the incidence of hydrone- phrosis is dose related. As the maternal dose of TCDD is increased from 0.5 to 4 (xg/kg/day, the incidence of hydronephrosis in C57BL/6 mice increases from 31 to 92%; in DBA/2 mice, the incidence varies from 5 to 37% over the same dose range. In DBA/2 mice, the incidence of hydronephrosis increases to 60% when the largest dose of TCDD administered is doubled to 8 jxg/ kg/day (but does not reach the 92% level seen in C57BL/6 mice at 4 ^.g TCDD/kg). Thus, the incidence of hydronephrosis is higher in the mouse strain that produces high-affinity Ah receptors 301 ------- (C57BL/6) compared to the strain (DBA/2) that produces Ah receptors having lower ligand-bind- ing affinity.150 The largest dose of TCDD used in these experiments resulted in hydronephrosis of the fetus without affecting the mean body weight or body weight gain of the dam. In the BXD strains,108 the incidence of 2,3,7,8,-TCDF- induced hydronephrosis is 34 to 48% in eight strains with high-affinity Ah receptors and 3 to 4% in eight strains with low-affinity Ah recep- tors. These results obtained in the BXD strains of mice provide the best evidence currently avail- able of an association between the ability of TCDD-like congeners to induce hydronephrosis and the wild-type Ahb allele. Accordingly, the Ah locus and the Ah receptor are involved in the hydronephrosis that is induced by TCDD-like congeners. ii. Structure Activity The rank order of potencies for various hal- ogenated aromatic hydrocarbon congeners to cause hydronephrosis in mice is consistent with the structure-activity requirements for binding to the Ah receptor (Table 4). This provides further evidence that the Ah receptor mediates the effects of these TCDD-like congeners on the developing mouse kidney. c. Species Differences Hydronephrosis has been reported after ad- ministration of low maternal doses of TCDD to rats and hamsters. Possibly due to the small num- bers of fetuses examined, the observed inci- dences of hydronephrosis in rats after exposure to cumulative maternal doses <2 (xg TCDD/kg have not been statistically significant.77-151 On the other hand, following a 1.5-^g TCDD/kg dose administered on gestational days 7 and 9, the incidence of hydronephrosis in hamster fetuses was 11 and 4.2%, respectively. This is in contrast to an incidence of <1% in control hamster fe- tuses. Therefore, hydronephrosis is one of the most sensitive indicators of prenatal toxicity57 in hamsters and mice. C. Postnatal Effects 1. Male Reproductive System of Rats Because TCDD can decrease plasma andro- gen concentrations and be transferred from mother to young in utero and during lactation,152'153 it is expected to have a great impact on the male re- productive system during early development.154 Testosterone and/or its metabolite DHT are es- sential prenatally and/or early postnatally for im- printing and development of accessory sex organs155-157 and for initiation of spermatogene- sis.158 In addition, aromatization of testosterone to 17p-estradiol within the CNS is required peri- natally for the imprinting of typical adult male patterns of reproductive behavior159 and LH se- cretion.160 Thus, normal development of male reproductive organs and imprinting of typical adult sexual behavior patterns require sufficient tes- tosterone be secreted by the fetal and neonatal testis at critical times in early development before and shortly after birth.161'162 If perinatal imprint- ing fails to occur in the accessory sex organs of a neonatal male rat, the results may be that these organs will not develop a normal trophic response to androgenic stimulation and will not function normally as the animal becomes sexually mature. a. Perinatal Androgen Deficiency To determine if in utero and lactational ex- posure to TCDD produces a perinatal androgenic deficiency, Mably et al.154J63 dosed pregnant rats with 1.0 (xg TCDD/kg on day 15 of gestation. Plasma testosterone concentrations were greater in control male than in control female fetuses on days 17 to 21 of gestation, particularly during the prenatal testosterone surge (days 17 to 19). On days 18 to 21 of gestation, TCDD exposure reduced the magnitude of this sex-based differ- ence. Postnatally, plasma testosterone concentra- tions peaked 2 h after birth in control males, whereas in TCDD-exposed males, the peak did not occur until 4 h after birth and was only half as large. Thus, in male rats, perinatal exposure to TCDD can produce both prenatal and early postnatal androgenic deficiencies. 302 ------- b. Overt Toxicity Assessment To determine how the male reproductive sys- tem is affected by in utero and lactational TCDD exposure. Mably et al.154'163-165 treated pregnant rats with a single oral dose of TCDD (0.064, 0.16, 0.4, or 1.0 (xg/kg) or vehicle on day 15 of gestation (day 0 = sperm positive). Day 15 was chosen because most organogenesis in the fetus is complete by this time and the hypothalamic/ pituitary/testis axis is just beginning to func- tion.166"168 The pups were weaned 21 days after birth, and the consequences of this single, ma- ternal TCDD exposure for the male offspring were characterized at various stages of postnatal sexual development. Mably et al.163 found that TCDD treatment had no effect on daily feed intake during preg- nancy and the first 10 days after delivery, nor did it have any effect on the body weight of dams on day 20 of gestation or on days 1, 7, 14, or 21 postpartum. Treating dams with graded doses of TCDD on day 15 of gestation had no effect on gestation index, length of gestation, or litter size. Except for an 8% decrease at the highest maternal dose, TCDD had no effect on live birth index. Neither the 4-day nor 21-day survival in- dex was significantly affected by TCDD. In all dosage groups, the number of dead offspring was equally distributed between males and females, and of the females that failed to deliver litters, none were pregnant. Signs of overt toxicity among the offspring were limited to the above-men- tioned 8% decrease in live birth index (highest dose only), the initial 10 to 15% decreases in body weight (two highest doses), and the initial 10 to 20% decreases in feed intake (measured for males only, two highest doses). The latter two effects disappeared by early adulthood, after which the body weights of the maternally ex- posed and nonexposed rats were similar. No male or female offspring with gross external malfor- mations were found. c. Androgenic Status Androgenic status of the male offspring, which includes such parameters as plasma an- drogen concentrations and androgen-dependent structures and functions, was reduced by a single maternal TCDD dose as low as 0.16 (xg/kg. Ano- genital distance, which is dependent both on cir- culating androgen concentrations and androgenic responsiveness,169 was reduced in 1- and 4-day- old male pups, even when slight decreases in body length were considered. Testis descent, an androgen-mediated development event that nor- mally occurs in rats between 20 and 25 days of age,170 was delayed =sl.7 days. For accessory sex organs of an adult male rat to grow normally and respond fully to andro- gens, there is a critical period that starts before birth and lasts until sexual maturity during which adequate concentrations of androgens are nec- essary. 155-157,171,172 To determine if perinatal TCDD exposure affects postnatal growth of the accessory sex organs, one rat from each litter was sacrificed at 32, 49, 63, and 120 days of age, corresponding to juvenile, pubertal, post- pubertal, and mature stages of sexual develop- ment, respectively. At each developmental stage, dose-related decreases in seminal vesicle and ventral prostate weights were found. These de- creases could not be explained by decreases in body weight. There were trends (although not statistically significant) for plasma testosterone and DHT concentrations to be decreased at these times, whereas plasma LH concentrations were gener- ally unaffected. An exception was a 95% de- crease in plasma LH concentration on postnatal day 32 caused by a maternal TCDD dose of 1.0 M-g/kg. The lowest maternal TCDD dose to affect a parameter of androgenic status was the lowest dose tested — 0.064 (jig/kg. This dose resulted in a significantly depressed ventral prostate weight at 32 days of age. When ventral prostate weight was indexed to body weight, however, 0.16 n,g TCDD/kg was the lowest dose that caused a sig- nificant reduction in relative ventral prostate weight. Although there was no effect on the body weight of male pups at this dose, 0.16 jig TCDD/kg caused a consistent pattern of effects that indicated a depression of androgenic status. The reductions in seminal vesicle and ventral prostate weights may be due to modest reductions in plasma androgen concentrations and/or andro- gen responsiveness caused by incomplete peri- natal imprinting of the accessory sex organs.163 303 ------- Collectively, these results demonstrate that in utero and lactational TCDD exposure decreases androgenic status of male rats from the fetal stage into adulthood. Table 5 summarizes these ef- fects.154-163 d. Spermatogenesis Mably et al.154-165 found that decreased sper- matogenesis was among the most sensitive re- sponses of the male rat reproductive system to perinatal TCDD exposure. Testis and epididymis weights and indices of spermatogenesis were de- termined on postnatal days 32, 49, 63, and 120. Perinatal TCDD exposure caused dose-related decreases in testis and epididymis weights. Weights of the caudal portion of the epididymis where mature sperm are stored prior to ejacula- tion were decreased the most, by approximately 45%. The number of sperm per cauda epididymis was decreased by 75 and 65% on days 63 and 120, respectively, and appeared to be the most sensitive effect of perinatal TCDD exposure on the male reproductive system. Daily sperm pro- duction was decreased by «43% at puberty, day 49, but the decrease was only 29% at sexual maturity, day 120. Seminiferous tubule diameter was decreased at all four developmental stages. Each effect of TCDD was dose related, and in all cases a significant decrease was seen in re- sponse to the lowest maternal TCDD dose tested, 0.064 u,g/kg, during at least one stage of sexual development. In general, the magnitude of the decreases recovered with time, although not com- pletely, thus suggesting that perinatal TCDD ex- posure delays sexual maturation. These results are summarized in Table 6.154'165 Severe pre- and/or postweaning undernutri- tion can affect the reproductive system of adult male rodents, including decreased spermatoge- nesis.173-176 At the two highest maternal TCDD doses, the feed consumption and body weight of male offspring were decreased; even so, the weight decreases did not exceed 21% of the con- trol values.163 However, reductions in sex organ weights, epididymal sperm reserves, and sper- matogenesis occurred at the two lowest maternal TCDD doses, neither of which reduced feed in- take or body weight of the male offspring. Thus, TABLE 5 Effects of In Utero and Lactational TCDD Exposure on Indices of Androgenic Status Index Anogenital distance Time to testis descent Plasma testosterone concentration Plasma 5a-dihydrotestosterone concentration Plasma LH concentration Absolute seminal vesicle weight Relative seminal vesicle weight* Absolute ventral prostate weight Relative ventral prostate weight0 Note: NS = not statistically significant. Lowest effective maternal dose TCDD/kg)- Maximum effect" 0.1 6 (days 1 and 4) 0.16 NS NS 1 .0 (day 32) 0.16 (days 32 and 63) 0.1 6 (day 63) 0.064 (day 32) 0.1 6 (days 32 and 63) 21 % decrease 1.7-day delay 69% decrease 59% decrease 95% decrease 56% decrease 50% decrease 60% decrease 53% decrease (day 1) (day 32) (day 49) (day 32) (day 49) (day 49) (day 32) (day 32) 8 The lowest dose of TCDD (given on day 15 of gestation) that caused a significant (p <0.05) effect in the male offspring and the day or days at which this dose caused such an effect are shown. b The magnitude of the greatest change seen in response to maternal dosing with 1.0 M-9/kQ TCDD and the day at which this effect was seen are shown. c Weight of organ divided by body weight of rat. From Mably, T. A., Moore, R. W., Bjerke, D. L, and Peterson, R. E., Biological Basis for Risk Assessment of Dioxins and Related Compounds, Gallo, M. A., Scheuplein, R. J., and van der Heijden, C. A., Eds., Branbury Report 35, Cold Spring Harbor Laboratory Press, Cold Spring Harbor, NY, 1991, 69. 304 ------- TABLE 6 Effects of In Utero and Lactational TCDD Exposure on Indices of Spermatogenic Function and Reproductive Capability Lowest effective maternal dose (,ig TCDD/kg)- 0.40 (day 32) 0.064 (days 49, 120) 0.064 (days 63, 120) 0.064 (days 63, 120) 0.064 (days 63, 120) 0.064 (days 32, 49, 120) 0.40 (day 32) NS NS NS NS Index Testis weight Epididymis weight Cauda epididymis weight Sperm per cauda epididymis Daily sperm production rate Seminiferous tubule diameter Plasma FSH concentration Leptotene spermatocyte: Sertoli cell ratio Sperm motility; percentage abnormal sperm Fertility Gestation index; litter size; live birth index; pup survival Note: NS = not statistically significant. • The lowest dose of TCDD (given on day 15 of gestation) that caused a significant (p <0.05) effect in the male offspring and the day or days at which this dose caused such an effect are shown. b The magnitude of the greatest change seen in response to maternal dosing with 1.0 jj-g/kg TCDD and the day on which this effect was seen are shown. From Mably, T. A., Moore, R. W., Bjerke, D. L, and Peterson, R. E., Biological Basis for Risk Assessment of Dioxins and Related Compounds, Gallo, M. A., Scheuplein, R. J., and van der Heijden, C. A., Eds., Branbury Report 35, Cold Spring Harbor Laboratory Press, Cold Spring Harbor, NY, 1991, 69. Maximum effect" 17% decrease (day 32) 35% decrease (day 32) 53% decrease (day 63) 75% decrease (day 63) 43% decrease (day 49) 15% decrease (day 32) 15% decrease (day 32) No dose-related effects No dose-related effects 22% decrease (day 70) No dose-related effects undernutrition cannot account for these repro- ductive system effects, including the decreases in spermatogenesis observed at the lower mater- nal TCDD doses.163-165 Since FSH and testosterone are essential for quantitatively normal spermatogenesis,158 an al- ternative explanation for the decreases in daily sperm production is a decrease in FSH and/or testosterone levels. In rats, the length of one sper- matogenic cycle is 58 days,177"179 so the de- creases in plasma FSH concentrations in 32-day- old male offspring could contribute to the re- ductions of spermatogenesis when the rats were 49 and 63 days of age. However, the modest depressant effect of perinatal TCDD exposure on plasma FSH concentrations was transitory; no effect was found on plasma FSH levels when the offspring were 49, 63, and 120 days old. It was concluded that reduced spermatogenesis in 120- day-old male rats, perinatally exposed to TCDD, was not due to decreases in plasma FSH levels when the animals were 49 to 120 days of age.165 Plasma testosterone concentrations in the same rats were reduced =s69% by perinatal TCDD exposure, yet intratesticular testosterone concen- trations must be reduced by at least 80% in rats before spermatogenesis is impaired.180 Based on the magnitude of the reductions in plasma an- drogen concentrations, it was concluded that cor- responding reductions in testicular testosterone production in perinatal TCDD-exposed offspring would probably not be severe enough to impair spermatogenesis.163>16S In normal rats, daily sperm production does not reach a maximum until 100 to 125 days of age,181 but in rats perinatally exposed to TCDD, it takes longer for sperm production to reach the adult level. Furthermore, the length of the delay is directly related to maternal TCDD dose,165 and if the dose is high enough, the reduction in sper- matogenesis may be permanent. This is sug- gested by a maternal TCDD dose of 1.0 \igfkg that decreased the daily sperm production in male rat offspring that were 300 days of age.182 Be- 305 ------- cause the mechanism by which perinatal TCDD exposure decreases spermatogenesis in adulthood is unknown, it is unclear whether the irreversible effect at the largest maternal dose, 1 M-g/kg, which results in depressed feed consumption and de- creased body weight, is caused by the same mechanism as that at smaller maternal doses, which do not result in undernutrition and from which the male offspring may eventually re- cover. A key observation for postulating mecha- nisms by which perinatal TCDD exposure re- duces spermatogenesis in adulthood is the finding that the ratio of leptotene spermatocytes per Ser- toli cell in the testes of 49-, 63-, and 120-day- old rats is not affected by in utero and lactational TCDD exposure, even though daily sperm pro- duction is reduced.165 Because Sertoli cells pro- vide spermatogenic cells with functional and structural support183 and the upper limit of daily sperm production in adult rats is directly depen- dent on the number of Sertoli cells per testis,184 three possible mechanisms for the decrease in daily sperm production may be involved: TCDD could (1) increase the degeneration of cells in- termediate in development between leptotene spermatocytes and terminal stage spermatids (the cell type used to calculate daily sperm produc- tion); (2) decrease post-leptotene spermatocyte cell division (meiosis); and/or (3) decrease the number of Sertoli cells per testis.185 Elucidating the mechanism by which perinatal TCDD ex- posure decreases spermatogenesis is important because it is one of the most sensitive responses of the male reproductive system to TCDD. e. Epididymis The epididymis has two functions: in prox- imal regions, spermatozoa mature gaining the ca- pacity for motility and fertility; in distal regions, mature sperm are stored before ejaculation.186 Mably et al.154-165 found that motility and mor- phology of sperm taken from the cauda epidi- dymis on postnatal days 63 and 120 were unaf- fected by perinatal TCDD exposure. Thus, no effect of TCDD on epididymal function was de- tected. The dose-dependent reduction in epi- didymis and cauda epididymis weights in post- pubertal rats 63 and 120 days of age can be accounted for, in part, by decreased sperm pro- duction. However, in immature males, 32 and 49 days of age, where sperm are not present in the epididymis, the decrease in weights of epi- didymal tissue cannot be explained by effects on sperm production. Because epididymal growth is androgen dependent, a TCDD-induced androgen deficiency and/or decrease in androgren respon- siveness of the epididymis could account for de- creased size of the organ.187-188 f. Reproductive Capability To assess reproductive capability, male rats born to dams given TCDD (0.064, 0.16, 0.40, or 1.0 (Jig/kg) or vehicle on day 15 of gestation were mated with control virgin females when the males were —70 days of age.154-165 The fertility index of the males is defined as the number of males impregnating females divided by the num- ber of males mated. The two highest maternal TCDD doses decreased the fertility index of the male offspring by 11 and 22%, respectively. However, these decreases were not statistically significant, and at lower doses, the fertility index was not reduced. The gestation index, defined as the percentage of control dams mated with TCDD-exposed males that delivered at least one live offspring, also was not affected by perinatal TCDD exposure. With respect to progeny of these matings, there was no effect on litter size, live birth index, or 21-day survival index. When peri- natal TCDD-exposed males were mated again at 120 days of age, there was no effect on any of these same parameters. Thus, despite pro- nounced reductions in cauda epididymal sperm reserves, when the TCDD-treated males were mated, perinatal TCDD exposure had little or no effect on fertility of male rats or on the survival and growth of their offspring. These results are summarized in Table 6.154-165 Because rats produce and ejaculate 10 times more sperm than are necessary for normal fertility and litter size,189'190 the absence of a reduction in fertility of male rats exposed perinatally to TCDD is not inconsistent with the substantial reductions in testicular spermatogenesis and ep- ididymal sperm reserves. In contrast, reproduc- 306 ------- tive efficiency in human males is very low; the number of sperm per ejaculate is close to that required for fertility.191 Thus, measures of fer- tility using rats are not appropriate for low-dose extrapolation in humans.192 A percent reduction in daily sperm production in humans, similar in magnitude to that observed in rats,154-165 may re- duce fertility in men. g. Sexual Differentiation of the CNS Sexual differentiation of the CNS is depen- dent on the presence of androgens during early development. In rats, the critical period of sexual differentiation extends from late fetal life through the first week of postnatal life.161 In the absence of adequate circulating levels of testicular andro- gen during this time, adult rats display high levels of feminine sexual behavior (e.g., lordosis), low levels of masculine sexual behavior, and a cyclic (i.e., feminine) pattern of LH secretion when castrated and primed with ovarian steroids.160-194 In contrast, perinatal androgen exposure of rats will result in the masculinization of sexually di- morphic neural parameters, including reproduc- tive behaviors, regulation of LH secretion, and several morphological indices.193'194 The mech- anism by which androgens cause sexual differ- entiation of the CNS is not completely under- stood. In the rat, it appears that ITp-estradiol, formed by the aromatization of testosterone within the CNS, is one of the principal active steroids responsible for mediating sexual differentia- tion;195 however, androgens also are involved. i. Demasculinization of Sexual Behavior Mably et al.154-164 assessed sexually dimor- phic functions in male rats born to dams given graded doses of TCDD or vehicle on day 15 of gestation. Masculine sexual behavior was as- sessed in male offspring at 60, 75, and 115 days of age by placing a male rat in a cage with a receptive control female and observing the first ejaculatory series and subsequent postejaculatory interval (Table 7). The number of mounts and intromissions (mounts with vaginal penetration) before ejaculation were increased by a maternal TCDD dose of 1.0 M-g/kg. The same males ex- hibited 12- and 11-fold increases in mount and intromission latencies, respectively, and a 2-fold increase in ejaculation latency. All latency effects were dose related and significant at a maternal TCDD dose as low as 0.064 |xg/kg (intromission latency) and 0.16 n-g/kg (mount and ejaculation latencies). Copulatory rates (number of mounts + intromissions/time from first mount to eja- culation) were decreased to <43% of the control rate. This effect on copulatory rates was dose related, and a statistically significant effect was observed at maternal TCDD doses as low as 0.16 M-g/kg. Postejaculatory intervals were increased 35% above the control interval, and a statistically significant effect was observed at maternal doses of TCDD as low as 0.40 |xg/kg. Collectively, these results demonstrate that perinatal TCDD exposure demasculinizes sexual behavior. Since perinatal exposure to a maternal TCDD dose of 1.0 ng/kg has no effect on the open field locomotor activity of adult male rats,196 the in- creased mount, intromission, and ejaculation la- tencies appear to be specific for these masculine sexual behaviors, not secondary to a depressant effect of TCDD on motor activity. Postpubertal plasma testosterone and DHT concentrations in littermates of the rats evaluated for masculine sexual behavior were as low as 56 and 62%, respectively, of control.154'163 However, plasma testosterone concentrations that were only 33% of control were still sufficient to masculinize the sexual behavior of adult male rats.197 Therefore, the modest reductions in adult plasma androgen concentrations following perinatal TCDD expo- sure were not of sufficient magnitude to demas- culinize sexual behavior. Reductions in perinatal androgenic stimula- tion can inhibit penile development and subse- quent sensitivity to sexual stimulation in adult- hood.198-199 Therefore, the demasculinization of sexual behavior could, to some extent, be sec- ondary to decreased androgen-dependent penile development. However, perinatal TCDD expo- sure had no effect on gross appearance of the rat penis. In addition, TCDD-exposed males exhib- ited deficits in such masculine sexual behaviors as mount latency and postejaculatory interval, which do not depend on stimulation of the penis for expression.200 Thus, although some effects of 307 ------- TABLE 7 Effects of In Utero and Lactational TCDD Exposure on Indices of Sexual Behavior and Regulation of LH Secretion in Adulthood Index Masculine sexual behavior Mount latency Intromission latency Ejaculatory latency Number of mounts Number of intromissions Copulatory rate (mounts plus intromissions/minute) Postejaculatory interval Feminine sexual behavior1 Lordosis quotient8 Lordosis intensity score Regulation of LH secretion LH surge Lowest effective maternal dose (^.g/kg TCDD)' 0.16 0.064 0.16 0.064 1.0 0.16 0.40 0.16 0.40 0.40 Maximum effect" 1200% 1100% 97% 130% 38% 43% increase increase increase increase increase decrease 35% increase 300% 50% increase increase 460% increase' • The lowest dose of TCDD (given on day 15 of gestation) that caused a significant (p <0.05) effect in the male offspring is shown. b The magnitude of the greatest change seen in response to maternal dosing with 1.0 |xg/ kg TCDD is shown (average of three trials for masculine behavior and two for feminine). c Measured when the rats were ~60, 75, and 115 days of age. " Feminine sexual behavior was measured following castration, estrogen priming, and progesterone administration. The rats were 170-184 days old. ' Number of times lordosis was displayed in response to a mount divided by the number of times each rat was mounted times 100. 1 Because control males do not secrete LH in response to progesterone, this percentage was calculated by comparing peak plasma LH concentrations in TCDD-exposed rats with plasma LH concentrations in control males at the same time after progesterone was administered. From Mably, T. A., Moore, R. W., Bjerke, D. L., and Peterson, R. E., Biological Basis for Risk Assessment of Dioxins and Related Compounds, Gallo, M. A., Scheuplein, R. J., and van der Heijden, C. A., Eds., Branbury Report 35, Cold Spring Harbor Laboratory Press, Cold Spring Harbor, NY, 1991, 69. TCDD, such as decreased copulatory rate and prolonged latency until ejaculation, could be due to reduced sensitivity of the penis to sexual stim- ulation, the 12-fold increase in mount latency and increase in postejaculatory interval could not be explained by this mechanism. ii. Feminization of Sexual Behavior Mabley et al.154-164 determined if the potential of adult male rats to display feminine sexual be- havior was altered by perinatal TCDD exposure. Male offspring of dams treated on day 15 of gestation with various doses of TCDD up to 1 (xg/kg or vehicle were castrated at ~ 120 days of age and beginning at —160 days of age were injected weekly for 3 weeks with 17p-estradiol ben/oate, followed 42 h later by progesterone. At 4 to 6 h after the progesterone injection on weeks 2 and 3, the male was placed in a cage with a sexually excited control stud male. The frequency of lordosis in response to being mounted by the stud male was increased from 18% (control) to 54% by the highest maternal TCDD dose, 1.0 jig/kg (Table 7). Lordosis in- tensity scored after Hardy and DeBold201 as a (1) for light lordosis, (2) for moderate lordosis, and (3) for a full spinal dorsoflexion was in- creased in male rats by perinatal TCDD exposure. 308 ------- The effects on lordosis behavior in males were dose related and significant at maternal TCDD doses as low as 0.16 ^g/kg (increased lordotic frequency) and 0.40 |xg/kg (increased lordotic intensity). Together they indicate a feminization of sexual behavior in these animals. Although severe undernutrition from 5 to 45 days after birth potentiates the display of lordosis behavior in adult male rats,202 the increased frequency of lor- dotic behavior was seen at a maternal TCDD dose of 0.16 |xg/kg that had no effect on feed intake or body weight. It was concluded that perinatal TCDD exposure feminizes sexual behavior in adult male rats independent of undernutrition. iii. Feminization of LH Secretion Regulation The effect of perinatal TCDD exposure on regulation of LH secretion by ovarian steroids was determined in male offspring at ~270 days of age. There is normally a distinct sexual di- morphism to this response. In rats castrated as adults, estrogen-primed females greatly increase their plasma LH concentrations when injected with progesterone, whereas similarly treated males fail to respond.203 Progesterone had little effect on plasma LH concentrations in estrogen- primed control males, but significant increases were seen in males exposed to maternal TCDD doses as low as 0.40 ng/kg. Thus, perinatal TCDD exposure increases pituitary and/or hypothalamic responsiveness of male rats to ovarian steroids in adulthood, indicating that regulation of LH se- cretion is feminized. Table 7 summarizes sexual behavior and LH secretion results.154-164 iv. Comparison to Other Ah Receptor- Mediated Responses The induction of hepatic cytochrome P-4501A1 and its associated EROD activity is an extremely sensitive Ah receptor-mediated re- sponse to TCDD exposure. Yet, in 120-day-old male rats that had been exposed to TCDD peri- natally, alterations in sexual behavior, LH secre- tion, and spermatogenesis were observed when induction of hepatic EROD activity could no longer be detected. 154-163~165 These results suggest that TCDD affects sexual behavior, gonado- trophic function, and spermatogenesis when vir- tually no TCDD remains in the body, and that the demasculinization and feminization of sexual behavior, feminization of LH secretion, and re- duced spermatogenesis caused by in utero and lactational exposure to TCDD is such that a con- tinuing presence of TCDD is not required to pro- duce these effects in adulthood.164-165 v. Possible Mechanisms and Significance The most plausible explanation for the de- masculinization and feminization of sexual be- havior and feminization of LH secretion is that perinatal exposure to TCDD impairs sexual dif- ferentiation of the CNS. Undernutrition, altered locomotor activity, reduced sensitivity of the penis to sexual stimulation, and modest reductions in adult plasma androgen concentrations of the male offspring cannot account for these effects.164 On the other hand, exposure of the developing brain to testosterone, conversion of testosterone into 17(i-estradiol within the brain, and events initi- ated by the binding of 17(3-estradiol to its recep- tor are all critical for sexual differentiation of the CNS. If TCDD interferes with any of these pro- cesses during late gestation and/or early neonatal life, it could irreversibly demasculinize and fem- inize sexual behavior204^206 and feminize the reg- ulation of LH secretion207-208 in male rats in adult- hood. Treatment of dams on day 15 of gestation with 1.0 n-g TCDD/kg significantly decreases plasma testosterone concentrations in male rat fetuses on days 18 and 20 of gestation and in male rat pups 2 h postpartum.163 Thus, the ability of maternal TCDD exposure to reduce prenatal and early postnatal plasma testosterone concen- trations may account, in part, for the impaired sexual differentiation of male rats exposed peri- natally to TCDD. Other mechanisms that may potentially contribute to the TCDD-induced im- pairment in CNS sexual differentiation are a de- crease in the formation of 17(3-estradiol from tes- tosterone within the CNS, which is independent of the decrease in plasma testosterone concen- trations, and/or a reduction in responsiveness of the CNS to estrogen during the critical period of 309 ------- sexual differentiation. The latter mechanism is consistent with the Ah receptor-mediated anties- trogen action of TCDD, which is described in Section III. A.3 for rat and mouse uterus and for estrogen-responsive MCF-7 and Hepa IclcV cells. In utero and/or lactational exposure to TCDD may cause similar effects in other animal species, including nonhuman primates,209"211 in which sexual differentiation is under androgenic con- trol. Although social factors may account for much of the variation in the sexually dimorphic behavior in humans, there is evidence that pre- natal androgenization influences both the sexual differentiation of such behavior and the brain hypothalamic structure.212"214 2. Neurobehavior Because differentiated tissues derived from ectoderm, i.e., skin, conjunctiva, nails, and teeth, are sites of action of halogenated aromatic hy- drocarbons in transplacentally exposed human in- fants, another highly differentiated tissue derived from ectoderm, the CNS, should be considered a potential site of TCDD action. In support of this possibility is the fact that sexual differentia- tion of the CNS of adult male rats is irreversibly altered in a dose-related fashion by perinatal ex- posure to TCDD.154 164 As is shown later, in mice transplacentally exposed to 3,3' ,4,4'-TCB, mon- keys perinatally exposed to TCDD, and children transplacentally exposed to a mixture of PCBs, CDFs, and PCQs in the Yu-Cheng incident, the CNS is affected by TCDD and other halogenated aromatic hydrocarbons. Thus, functional CNS al- terations, which may or may not be irreversible, are observed following perinatal exposure to these chemicals. Ah receptors have been identified in rat brain110 but may be associated with glial cells rather than neurons.110'215 Following administra- tion of 14C-TCDD in the rat, the highest concen- trations TCDD-derived radioactivity are found in the hypothalamus and pituitary. Much lower con- centrations are found in the cerebral cortex and cerebellum.216 In another study, the Ah receptor was not detected in whole rat or mouse brain, but was detected in the cerebrum of the hamster and cerebrum and cerebellum of the guinea pig.217 Ah receptors appear to be absent in the human frontal cortex.215 310 a. Mice CD-I mice exposed transplacentally to 3,3',4,4'-TCB at a maternal oral dose of 32 mg/ kg administered on days 10 to 16 of gestation exhibited neurobehavioral, neuropathological, and neurochemical alterations in adulthood.218'220 The neurobehavioral effects consisted of circling, head bobbing, hyperactivity, impaired forelimb grip strength, impaired ability to traverse a wire rod, impaired visual placement responding, and im- paired learning of a one-way avoidance task.218 The brain pathology in adult mice exhibiting this syndrome consisted, in part, of alterations in syn- apses of the nucleus accumbens.219 This sug- gested that in utero exposure to 3,3',4,4'-TCB may interfere with synaptogenesis of dopami- nergic systems. In support of this possibility, Agrawal et al.220 found that adult mice transpla- centally exposed to 3,3',4,4'-TCB had decreased dopamine levels and decreased dopamine recep- tor binding in the corpus striatum, both of which were associated with elevated levels of motor activity. It was concluded that transplacental ex- posure to 3,3',4,4'-TCB in mice may perma- nently alter development of striatal synapses in the brain. Eriksson et al.221 examined the neurobehav- ioral effects of 3,3',4,4'-TCB in NMRI mice exposed to a single oral dose of 0.41 or 41 mg/ kg on postnatal day 10. Following sacrifice of the mice on day 17, muscarinic receptor concen- trations in the brain were significantly decreased, at both dose levels. This effect was shown to occur in the hippocampus but not in the cortex. More recently, NMRI mice were exposed to the same two doses of 3,3',4,4'-TCB similarly ad- ministered on postnatal day 10.222 At 4 months of age, the effects of PCB on locomotor activity were assessed. At both dose levels, abnormal activity patterns were exhibited in that the treated mice were significantly less active than controls at the onset of testing, but were more active than controls at the end of the test period. This pattern of effects can be interpreted as a failure to ha- bituate to the test apparatus. In contrast to the previous results with CD-I mice, circling or head bobbing activities were not observed in these ani- mals. Upon sacrifice after the activity testing was complete, a small but statistically significant in- crease (as opposed to the decrease found after sacrifice on postnatal day 17) in the muscarinic ------- receptor concentration of the hippocampus was found in animals from the high-dose group. These results suggest that the neurochemical effects of 3,3',4,4'-TCB are complex. Cholinergeric as well as dopaminergic systems in the brain are in- volved. Of all the developmental and reproductive endpoints reported in this section for laboratory animals, the only ones that have not yet been demonstrated to occur following perinatal ex- posure to TCDD are the above-mentioned neuro- toxic effects in mice. These have only been stud- ied following perinatal exposure to 3,3'4,4'-TCB. In addition, there is no evidence yet to show that (1) among inbred mouse strains, having low- and high-affinity Ah receptors, susceptibility to 3,3',4,4'-TCB-induced neurotoxicity segregates with the Ah locus, or (2) the rank order binding affinity of congeners for the Ah receptor corre- lates with their rank order potency for causing these neurotoxic effects in mice. The rapid me- tabolism of 3,3',4,4'-TCB compared to the rel- atively slow metabolism of TCDD in mice causes some uncertainty about the potential involvement of the Ah receptor in 3,3',4,4'-TCB-induced neurotoxicity. Contributing to this uncertainty is the hypothesis that 3,3',4,4'-TCB may produce CNS effects by being converted to a hydroxylated metabolite that is neurotoxic. While there is no evidence for or against this hypothesis, there also is no evidence for or against the Ah receptor- mechanism hypothesis of 3,3',4,4'-TCB neuro- toxicity. Further research is needed to test these hypotheses. In so doing, it should become ap- parent whether 3,3' ,4,4'-TCB-induced neurotox- icity effects are relevant to TCDD-induced de- velopmental toxicity. b. Monkeys Schantz and Bowman69 and Bowman et al.223 conducted a series of studies on the long-term behavioral effects of perinatal TCDD exposure in monkeys. Because these were the first studies to evaluate the behavioral teratology of TCDD, monkeys exposed to TCDD via the mother during gestation and lactation were screened on a broad selection of behavioral tests at various stages of development.223 At the doses studied (5 or 25 ppt in the maternal diet), TCDD did not affect reflex development, visual exploration, locomotor ac- tivity, or fine motor control in any consistent manner.68 However, the perinatal TCDD expo- sure did produce a specific, replicable deficit in cognitive function.69 TCDD-exposed offspring were impaired on object learning, but were un- impaired on spatial learning. TCDD exposure also produced changes in the social interactions of mother-infant dyads.224 TCDD-exposed infants spent more time in close physical contact with their mothers. The pattern of effects was similar to that seen in lead-exposed infants and suggested that mothers were providing increased care to the TCDD-exposed infants.224 c. Humans The intellectual and behavioral development of Yu-Cheng children transplacentally exposed to PCBs, CDFs, and PCQs was studied through 1985 by Rogan et al.92 In Yu-Cheng children, matched to unexposed children of similar age, area of residence, and socioeconomic status, there was a clinical impression of developmental or psychomotor delay in 12 (10%) Yu-Cheng chil- dren compared with 3 (3%) control children, and of a speech problem in 8 (7%) Yu-Cheng children vs. 3 (3%) control children. Also, except for verbal IQ on the Wechsler Intelligence Scale for Children, Yu-Cheng children scored lower than control children on three developmental and cog- nitive tests.92 Neurobehavioral data on Yu-Cheng children obtained after 1985 show that the intel- lectual development of these children continues to lag somewhat behind that of matched control children.93 As measured from 1985 through 1990, this effect persists at least up to the age of 7 years, and it occurs in children born long after the initial Yu-Cheng exposure.279 Also, Yu-Cheng children are rated by their parents and teachers to have a higher activity level, more health, habit, and behavioral problems, and to have a temper- amental clustering closer to that of a "difficult child."93 It is concluded that, in humans, trans- placental exposure to halogenated aromatic hy- drocarbons can affect CNS function postnatally. However, which congeners, TCDD-like vs. nonTCDD-like, are responsible for the neurotox- icity is unknown. 311 ------- Further research on the mechanism of these postnatal neurobehavioral effects, dose-response relationships, and reversibility of the alterations is needed before the role of TCDD-like congeners vs. nonTCDD-like congeners in causing this tox- icity can be understood. Mechanisms that re- spond uniquely to TCDD-like congeners may not necessarily be involved inasmuch as three lightly chlorinated, ortho-substituted PCB congeners, 2,4,4'-TCB, 2,2',4,4'-TCB, and 2,2',5,5'-TCB, were detected in monkey brain following dietary exposure to Aroclor 1016 and appear to be re- sponsible for decreasing dopamine concentra- tions in the caudate, putamen, substantia nigra, and hypothalamus of these animals.225 These nonplanar PCB congeners are believed to cause these effects by acting through a mechanism that does not involve the Ah receptor. On the other hand, the results presented for mice and monkeys suggest that TCDD-like congeners could be in- volved in producing the observed postnatal neu- robehavioral effects in humans. D. Cross-Species Comparison of Effect Levels TCDD exposure levels that cause a variety of developmental effects in different species are summarized for fish in Table 8, birds in Table 9, and mammals in Table 10. Fertilized lake trout eggs and Japanese medaka eggs were exposed to different waterborne concentrations of 3H-TCDD. Estimates of the amount of TCDD in these eggs were then made from measurement of the TCDD- derived radioactivity within them. Fertilized rain- bow trout, chicken, ring-necked pheasant, and eastern blue bird eggs were injected directly with the indicated doses of TCDD. Thus, the doses of TCDD given in Tables 8 and 9 for all fish and bird species represent TCDD egg burdens in which a significant portion of the dose may be present within the yolk of the egg rather than the devel- oping embryo. Mammalian embryo/fetuses, on the other hand, were exposed via administration of TCDD to the pregnant female. Therefore, the doses given in Table 10 are maternal TCDD doses in which a significant portion of the dose may be retained by the mother and never actually reach the em- bryo/fetus. In some studies, pregnant rats and rhesus monkeys were exposed to TCDD on a chronic or subchronic basis, respectively. The doses given in Table 10 for these particular stud- ies represent the calculated maternal body bur- dens at the time of conception. In rats, the du- ration of chronic exposure was much longer than the whole body elimination half-life for TCDD in rats. Therefore, the body burden of TCDD given for the rat is 92.8% of the calculated steady- state body burden. In rhesus monkeys, the half- life for whole body elimination of TCDD is longer than was the duration of exposure prior to con- ception. Therefore, the steady-state body burdens that would be expected for rhesus monkeys ex- posed to the different levels of dietary TCDD TABLE 8 Cross-Species Comparison of NOAELS and LOAELS for TCDD Developmental Toxicity in Fish Species Lake trout Japanese medaka Rainbow trout Lake trout Rainbow trout Japanese medaka Effect Sac fry mortality Lesions8 Sac fry mortality Sac fry mortality Sac fry mortality Sac fry mortality Lesions8 Exposure Static waterborne Static waterborne Single injection Static waterborne Static waterborne Single injection Static waterborne Egg Dose (ng/kg) 34 <100 194 40 55 291 300 Effect level NOAEL NOAEL NOAEL LOAEL LOAEL LOAEL LOAEL Ref. 14 22 275 23 14 275 22 Consist of a spectrum of effects, including hemorrhage in various areas, pericardial edema, collapse of the yolk sphere, cessation of blood flow throughout the animal, and embryo mortality. 312 ------- TABLE 9 Cross-Species Comparison of NOAELS and LOAELS for TCDD Developmental Toxicity in Birds Species Ring-necked pheasant Eastern bluebird Chicken Chicken Ring-necked pheasant Ring-necked pheasant Ring-necked pheasant Eastern bluebird Effect Embryo mortality Embryo mortality Exposure Single injection Single injection Egg Dose (rig/kg) Effect level Cardiac malformations Single injection Embryo mortality Single injection Embryo mortality Single injection Embryo mortality Single injection Embryo mortality Single injection Embryo mortality Single injection Ref. 100 1,000 9a 240 1,000 1,354" 2,182C 10,000 NOAEL NOAEL LOAEL La*, LOAEL LD50 LD50 LOAEL 31 41 42 32 33 30 31 31 31 41 Chi-square analysis of the data in Table 1 of Cheung et al.32 demonstrated that the incidence of cardiac malformations in all embryos examined at dose levels of 1.6 pmol/egg or greater are significantly (p <0.05) increased compared to the incidence in the control group designated "all examined." As- suming a 55-g egg weight, 1.6 pmol/egg corresponds to a TCDD egg burden of 9 ng/kg. Injected into the egg albumin. Injected into the egg yolk. intake are approximately 3 times greater than the maternal body burdens estimated at the time of conception (Table 10). Both in rats and rhesus monkeys, the maternal body burdens are calcu- lated using a one-compartment open model, as- suming 86.1% bioavailability for TCDD. The bioavailability used for TCDD was determined in rats.226 Because no estimate for TCDD bio- availability has been reported in rhesus monkeys, the same 86.1% value was used. The whole body elimination half-life used for TCDD in the rat is 23.7 days.226 McNulty et al.227 estimated a half- life of approximately 1 year for TCDD elimi- nation from adipose tissue in the rhesus monkey; for calculation of the body burdens estimated in Table 9, this half-life was rounded to 400 days for whole body elimination. The maternal body burden given for chronic exposure in the rat was calculated from the data of Murray et al.228 The maternal body burdens given for subchronic ex- posure in the rhesus monkey were calculated from data obtained from Bowman,229 which included the daily dietary TCDD exposure level for each pregnant female used in the studies reported by Bowman et al.68'223 and Schantz and Bowman.69 Bowman's results indicate that the range of TCDD half-lives in these monkeys was 200 to 600 days, which is consistent with the results of McNulty et al.227 The body burdens estimated for rhesus monkeys used in these studies are averages based on the average daily TCDD consumption of all pregnant females used at a particular level of maternal TCDD exposure. As summarized in Table 8, lake trout and rainbow trout sac fry and Japanese medaka em- bryos are similarly affected by a spectrum of lesions, which includes hemorrhage, edema, col- lapse of the yolk sac, cessation of blood flow, and embryo mortality. Estimates of the NOAEL and LOAEL are given in the table for the ap- pearance of these lesions in Japanese medaka embryos and for embryo mortality in the two trout species. Fertilized lake trout eggs and Jap- anese medaka eggs were exposed to various TCDD concentrations dissolved in static water, and fertilized rainbow trout eggs were injected directly with TCDD; however, the egg doses given in Table 8 represent the concentration of TCDD within the eggs themselves. Therefore, the dif- ferent NOAELs and LOAELs for developmental toxicity in different fish species probably repre- sent species differences in susceptibility to TCDD- induced developmental toxicity rather than dif- ferences in the method of TCDD exposure. Of the three fish species, lake trout sac fry are the most sensitive to TCDD-induced mortality. How- 313 ------- ever, based on the LOAELs shown in Table 8, the difference in susceptibility between fish spe- cies may be less than tenfold. Based on the LOAELs shown in Table 9, the sensitivity of different bird species to TCDD- induced embryo mortality varies more than 40- fold. The chicken embryo is more susceptible to TCDD-induced mortality than are embryos of the ring-necked pheasant and eastern blue bird. In addition, chicken embryos are highly sensitive to the formation of TCDD-induced structural de- fects in the heart and aortic arch. The incidence of cardiac malformations in the chicken embryo is increased at an egg exposure level as low as 9 ng TCDD/kg egg. However, such cardiac mal- formations have not been found in any other bird species examined. Table 10 summarizes the levels of TCDD exposure that cause certain structural malfor- mations, functional alterations, and prenatal mor- tality in the embryo/fetus of different mammalian species. Based on the LOAELs given in Table 10, functional alterations in learning behavior and the male reproductive system occur at lower TCDD doses than those required to produce structural malformations. Although TCDD-in- duced developmental toxicity has been exten- sively studied in mice and rats, the LOAELs in Table 10 indicate that the embryo/fetus of rodent species is generally not as sensitive to TCDD- TABLE 10 Cross-Species Comparison of NOAELS and LOAELS for TCDO Developmental Toxicity in Mammals Species Monkey Rat Rat Mouse Mouse Monkey Rat Monkey Rabbit Rat Rabbit Rat Mouse Guinea pig Hamster Mouse Hamster Mouse Effect Prenatal mortality Prenatal mortality Prenatal mortality Hydronephrosis Cleft palate Object learning Male reproductive Prenatal mortality Extra ribs Fetal growth Prenatal mortality Prenatal mortality Hydronephrosis Prenatal mortality Thymic hypoplasia Cleft palate Prenatal mortality Prenatal mortality Exposure Multiple dose 1 ng/kg/day Multiple dose Multiple dose Multiple dose 0.126 ng/kg/day Single dose 0.642 ng/kg/day Multiple dose Multiple dose Multiple dose Multiple dose 10 ng/kg/day Multiple dose Multiple dose Single dose Single dose Multiple dose Single dose Single dose Maternal dose 22 ng/kg, 9 x , gd 20-40 27 ng/kg,a chronic 30 ng/kg/day, gd 6-15 100 ng/kg/day, gd 6-15 300 ng/kg/day, gd 6-15 19 ng/kg,a subchronic 64 ng/kg, gd 15 97 ng/kg,a subchronic 111 ng/kg, 9 x, gd 20-40 100 ng/kg/day, gd 6-15 125 ng/kg/day, gd 6-15 250 ng/kg/day, gd 6-15 270 ng/kg," chronic 500 ng/kg/day, gd 6-15 500 ng/kg/day, gd 6-15 1,500 ng/kg, gd 14 1,500 ng/kg, gd 7 or 9 3,000 ng/kg/day, gd 6-15 18,000 ng/kg/day, gd 7 or 9 24,000 ng/kg/day, gd 6 Effect level NOAEL NOAEL NOAEL NOAEL NOAEL LOAEL LOAEL LOAEL LOAEL LOAEL LOAEL LOAEL LOAEL LOAEL LOAEL LOAEL LOAEL LOAEL LOAEL LOAEL Ref. 74 228 62 276 59 69 163-165 69 74 79 62 79 228 62 149 57 57 77 61 60 Note: gd = gestational day. Maternal body burdens of TCDD at the time of conception were calculated by assuming a one-compartment open model and a half-life for whole body TCDD elimination of 400 days in the monkey227 and 23.7 days in the rat.226 A bioavailability of 86.1% was used in the monkey and rat.226 The daily dietary exposure levels in rhesus monkeys were approximately 5 and 25 ppt at the NOAEL and LOAEL doses, respectively. Rhesus monkeys were exposed to these levels of TCDD for 7 months prior to conception. At this time (0.525 half- lives), the accumulated amount of TCDD in rhesus monkeys was 30.5% of the calculated steady-state level. Rats were exposed to the indicated daily doses of TCDD for a period of 90 days (3.8 half-lives) prior to conception. At this time, the accumulated amount of TCDD in rats was 92.8% of the calculated steady-state level. 314 ------- induced prenatal mortality as is the embryo/fetus of the rhesus monkey. The sensitivity of the em- bryo/fetus to TCDD-induced prenatal mortality in different mammalian species varies approxi- mately 240-fold. This is in contrast to the 1000- to 5000-fold variation in the LD50 of TCDD when adult animals of these same species are exposed. The agreement between studies with respect to the LOAEL in Table 9 for prenatal mortality in rats and monkeys is particularly striking. The 500-ng/kg dose of TCDD on gestational days 6 to 15 that caused prenatal mortality in rats62 agrees with the maternal TCDD body burden of 270 ng/kg calculated from the chronic exposure228 to within a factor of 2. Similarly, the TCDD dose of 111 ng/kg that was given to rhesus monkeys 9 times during the first trimester of pregnancy74 agrees with the maternal body burden of 97 ng/kg that increased prenatal mortality in rhesus mon- keys following subchronic dietary exposure.69 I. REPRODUCTIVE TOXICITY A. Female 1. Reproductive Function/Fertility TCDD and its approximate isostereomers have been shown to affect female reproductive endpoints in a variety of animal studies. Among the effects reported are reduced fertility, reduced litter size, and effects on the female gonads and menstrual/estrous cycle. These studies are re- viewed next. Other TCDD effects on pregnancy maintenance, embryo/fetotoxicity, and postnatal development were covered in Section II. The study by Murray et al.228 used a multi- generational approach to examine the reproduc- tive effects of exposure of male and female rats over three generations to relatively low levels of TCDD (0, 0.001, 0.01, and 0.1 |Ag/kg/day). There was variation in the fertility index in both the control and the exposed groups, and a lower than desirable number of impregnated animals in the exposed groups. Even so, the results showed ex- posure-related effects on fertility, an increased time between first cohabitation and delivery, and a decrease in litter size. The effects on fertility and litter size were observed at 0.1 jxg/kg/day in the F0 generation and at 0.01 jxg/kg/day in the FI and F2 generations. Additionally, in a 13-week exposure to 1 to 2 (xg/kg/day of TCDD in non- pregnant female rats, Kociba et al.230 reported anovulation and signs of ovarian dysfunction, as well as suppression of the estrous cycle. How- ever, at exposures of 0.001 to 0.01 |xg/kg/day in a 2-year study, Kociba et al.231 reported no effects on the female reproductive system. Allen and colleagues reported on the effects of TCDD on reproduction in the monkey.70"73 In a series of studies, female rhesus monkeys were fed 50 or 500 ppt TCDD for ^9 months. Females exposed to 500 ppt showed obvious clinical signs of TCDD toxicity and lost weight throughout the study. Five of the eight monkeys died within 1 year after exposure was initiated. Following 7 months of exposure to 500 ppt TCDD, seven of the eight females were bred to unexposed males. The remaining monkey showed such severe signs of TCDD toxicity that she was not bred due to her debilitated state. Of the seven females that were evaluated for their reproductive capabili- ties, only three were able to conceive and, of these, only one was able to carry her infant to term.72 When females exposed to 50 ppt TCDD in the diet were bred at 7 months, two of eight females did not conceive and four of six that did conceive could not carry their pregnancies to term. One monkey delivered a stillborn infant and only one conception resulted in a live birth.73 As de- scribed in an abstracted summary, these results at 50 and 500 ppt TCDD were compared to a group of monkeys given a dietary exposure to PBB (0.3 ppm, Firemaster FF-1) in which seven of seven exposed females were able to conceive, five gave birth to live, normal infants and one gave birth to a stillborn infant.71 Although the effects at 500 ppt TCDD may be associated with significant maternal toxicity, this would not ap- pear to be the case at the lower dose. After 50 ppt TCDD, there were no overt effects on ma- ternal health, but the ability to conceive and maintain pregnancy was reduced.71 In a similar series of experiments, female rhesus monkeys were fed diets that contained 0, 5, and 25 ppt TCDD.68 69 Reproductive function was not altered in the 5 ppt group since, after 7 months of dietary exposure to TCDD, seven of 315 ------- eight females mated to unexposed males were able to conceive. Six of these females gave birth to viable infants at term and one gave birth to a stillborn infant. This was not significantly dif- ferent from the results of the control group, which was fed a normal diet that contained no TCDD. All seven of the monkeys in this control group were able to conceive and gave birth to viable infants. The 25-ppt dietary exposure level, how- ever, did affect reproductive function. Only one of the eight females in this group that was mated gave birth to a viable infant. As in the 50 ppt group from earlier studies, there were no serious health problems exhibited by any females ex- posed to 0, 5, or 25 ppt TCDD. Therefore, the results in the 25- and 50-ppt groups suggest that maternal exposure to TCDD, before and during pregnancy, can result in fetal mortality without producing overt toxic effects in the mother. McNulty74 examined the effect of a TCDD exposure during the first trimester of pregnancy (gestational age, 25 to 40 days) in the rhesus monkey. At a total dose of 1 fxg/kg given in nine divided doses, three of four pregnancies ended in abortion, and two of these abortions occurred in animals that displayed no maternal toxicity. At a total dose of 0.2 |Ag/kg, one of four preg- nancies ended in abortion. This did not appear different from the control population, but the low number of animals per group did not permit sta- tistical analysis. McNulty74 also administered single l-(JLg/kg doses of TCDD on gestational day 25, 30, 35, or 40. The number of animals per group was limited to three, but it appeared that the most sensitive periods were the earlier pe- riods, days 25 and 30, and that both maternal toxicity and fetotoxicity were reduced when TCDD was given on later gestational days. For all days at which a single l-|o,g TCDD/kg dose was given (gestational day 25, 30, 35, or 40), 10 of 12 pregnancies terminated in abortion. Thus, for 16 monkeys given 1 u.g TCDD/kg in single or divided doses between days 25 and 40 of preg- nancy, there were only 3 normal births.65-74 In conclusion, the primary effects of TCDD on female reproduction appear to be decreased fertility, inability to maintain pregnancy for the full gestational period, and, in the rat, decreased litter size. In a few studies, some signs of ovarian dysfunction, such as anovulation and suppression of the estrous cycle, have been reported.71-72230 Unfortunately, the amount of attention that has been given to the female reproductive system, especially in the nonpregnant state, has been lim- ited. In addition, there is little information about how TCDD toxicity involving the mother and/or placenta may affect fetal development. 2. Alterations in Hormone Levels The potential for TCDD to alter circulating female hormone levels has been examined, but only to a very limited extent. In monkeys fed a diet that contained 500 ppt TCDD for =£9 months, the length of the menstrual cycle and the intensity and duration of menstruation were not appreci- ably affected by TCDD exposure.72 However, there was a decrease in serum estradiol and pro- gesterone concentrations in five of the eight ex- posed monkeys, and in two of these animals, the reduced steroid concentrations were consistent with anovulatory menstrual cycles. In summary form, Allen et al.71 described the effects of di- etary exposure of female monkeys to 50 ppt TCDD. After 6 months of exposure to this lower dietary level of TCDD, there was no effect on the serum estradiol and progesterone concentra- tions of these monkeys. Thus, the presence of these hormonal alterations is dependent on the level of dietary TCDD exposure. Shiverick and Muther232 reported that there was no change in circulating levels of estradiol in the rat after ex- posure to 1 (Jtg/kg/day TCDD on gestational days 4 to 15. Taking all of these results together, the effect of TCDD exposure on circulating female hormone levels may depend both on species and level of exposure. It appears that any significant effect is only seen at relatively high exposure levels, although more research needs to be done to examine the effects of TCDD on female hor- mones. 3. Antiestrogenic Action a. In Vivo Estrogens are necessary for normal uterine development and for maintenance of the adult 316 ------- uterus. The cyclic production of estrogens par- tially regulates the cyclic production of FSH and LH that results in the estrous cycling of female mammals. In addition, estrogens are necessary for the maintenance of pregnancy. Any effect that causes a decrease in circulating or target cell es- trogen levels can alter normal hormonal balance and action. Early experimental results in rats indicated that chronic exposure to TCDD can cause suppression or inhibition of the estrous cycle.230 Rhesus monkeys chronically exposed to TCDD, on the other hand, are affected by hormonal ir- regularities in their menstrual cycles.70'72 Al- though the authors of these studies in rats and monkeys did not attribute the effects of TCDD exposure to an antiestrogenic action, it seems reasonable to posit from more recent information that such an antiestrogenic action may have been responsible. In rhesus monkeys, the severity of the TCDD- associated reproductive alterations was correlated with decreased plasma levels of estrogen and pro- gesterone.72 Thus, one possible mechanism for these effects would be increased metabolism of estrogen and progesterone due to induction by TCDD of hepatic microsomal enzymes and/or a decrease in the rate at which these steroids are synthesized. On the other hand, serum concen- trations of 17(3-estradiol are not significantly af- fected when TCDD is administered to pregnant rats.232 Thus, an alternative mechanism for TCDD-associated reproductive dysfunction could involve effects of TCDD on gonadal tissue itself, such as a decrease in its responsiveness to estro- gen. In support of this latter mechanism, the administration of TCDD to CD-1 mice results in a decreased number of cytosolic and nuclear es- trogen receptors in hepatocytes and uterine cells. Although TCDD treatment induces hepatic cyto- chrome P-450 levels in these animals, it has no effect on serum concentrations of IVfJ-estra- diol.233 This indicates that the antiestrogenic ef- fect of TCDD in CD-1 mice is not caused by a decrease in circulating levels of estrogen. Effects of estrogen on the uterus include a cyclic increase in uterine weight, increased ac- tivity of the enzyme peroxidase, and an increase in the tissue concentration of progesterone re- ceptors. Antiestrogenic effects of TCDD admin- istration to female rats include a decrease in uter- ine weight, a decrease in uterine peroxidase activity, and a decrease in the concentration of progesterone receptors in the uterus.234 In addi- tion, when TCDD and 17(i-estradiol are coad- ministered to the same female rat, the antiestro- genic action of TCDD diminishes or prevents 17p-estradiol-induced increases in uterine weight, peroxidase activity, progesterone receptor con- centration, and expression of EGF receptor mRNA 234,235 similarly in mice, TCDD admin- istration decreases uterine weight and antago- nizes the ability of 17p-estradiol to increase uter- ine weight.236 The ability of TCDD to antagonize the effects of exogenously administered estrogen in the rat is dependent on the age of the animal. In 21-day- old rats, TCDD does not affect 17(i-estradiol- induced increases in uterine weight of proges- terone receptor concentration. On the other hand, in 28-day-old intact rats and 70-day-old ovariec- tomized rats, both of these 17p-estradiol-me- diated responses are attenuated by TCDD.234 Pre- viously, it was reported that TCDD administration does not alter the dose-dependent increase in uter- ine weight due to exogenously administered es- trone in sexually immature rats.237 The later work by Safe et al.234 suggest that this apparent lack of an antiestrogenic effect of TCDD may have been due to the young age of the rats used. b. In Vitro Both TCDD and progesterone can cause a decrease in the nuclear estrogen receptor con- centration in rat uterine strips. However, the ef- fect of progesterone is inhibited by actinomycin D, cycloheximide, and puromycin, whereas the effect of TCDD is inhibited only by actinomycin D. The reasons why the TCDD-induced decrease in nuclear estrogen receptors is blocked by a tran- scription inhibitor, but not by protein synthesis inhibitors, are not known. However, these results indicate that TCDD and progesterone decrease the nuclear estrogen receptor concentration via different mechanisms.238 In addition, the anties- trogenic actions of TCDD can be demonstrated in cell culture, and two prominent mechanisms could potentially be involved: (1) increased me- 317 ------- tabolism of estrogen due to Ah receptor-mediated enzyme induction; and (2) a downregulation of estrogen receptors within the target cell. In MCF-7 cells, which are estrogen-respon- sive cells derived from a human breast adeno- carcinoma, antiestrogenic effects caused by the addition of TCDD to the culture medium include a reduction of the 17p-estradiol-induced secre- tion of a 160-, 52-, and 34-kDa protein.239 The last two proteins are believed to be procathepsin D and cathepsin D, respectively. In addition, treatment of MCF-7 cells with TCDD suppresses the 17(3-estradiol-enhanced secretion of tissue plasminogen activator (tPA) and inhibits estro- gen-dependent postconfluent cell prolifera- tion.240'241 Thus, cultured MCF-7 cells have sev- eral estrogen-dependent responses that are inhibited by TCDD; this characteristic makes them a useful model system for studying the antiestro- genic actions of dioxin. In cultured MCF-7 cells, TCDD treatment in- duces AHH activity, the hallmark response of Ah receptor binding, and increases hydroxylation of 17p-estradiol at the C-2, C-4, C-6ct, and C-15a positions. It turns out that the particular cyto- chrome P-450 that catalyzes the C-2, C-15a, and C-6a hydroxylations of 17(3-estradiol is cyto- chrome P-4501A1, which is identical to AHH.243 TCDD treatment also results in reduced levels of occupied nuclear estrogen receptors.244 In MCF-7 cells, these results indicate that the antiestrogenic effect of TCDD could result from (1) the in- creased metabolism of estrogens due to Ah re- ceptor-mediated enzyme induction, and/or (2) a decreased number of estrogen receptors in the nucleus. Safe's group has published TCDD-con- centration response information for both the TCDD-induced decrease in occupied nuclear es- trogen receptors245 and the induction of AHH and EROD activities in MCF-7 cells.244 In addition, they have reported that TCDD causes a decreased number of cytosolic and nuclear estrogen recep- tors in Hepa Iclc7 cells, which is a mouse hep- atoma cell line.246 Independent analysis of the data suggests that the EC50 values for these ef- fects are not dissimilar enough to distinguish be- tween the proposed mechanisms. Instead, it ap- pears as though TCDD induces the enzymes AHH and EROD over the same concentration range that it causes a decreased concentration of oc- cupied nuclear estrogen receptors in MCF-7 cells. In Hepa Iclc7 cells, the lowest concentration used was 10 pM. Although exposure to 10 pM TCDD resulted in a statistically significant down- regulation of estrogen receptors, Israel and Whitlock247 reported that this concentration is the approximate EC50 for the induction of cyto- chrome P-4501A1 mRNA and enzyme activity in these cells. Therefore, in Hepa Iclc7 cells, as well as in MCF-7 cells, it would appear that the TCDD concentrations required to produce en- zyme induction and reduction in occupied nuclear estrogen receptor levels are not dissimilar enough to distinguish between the two proposed mech- anisms. More recently, Safe's group used an analog of TCDD, i.e., MCDF, that inhibits the 17(3- estradiol-induced secretion of the 34-, 52-, and 160-kDa proteins and downregulates estrogen re- ceptors in MCF-7 cells. These effects occurred at concentrations of MCDF for which there was no detectable induction of EROD activity.248 In addition, it has been stated that the downregu- lation of estrogen receptors in Hepa Iclc7 cells can be detected as early as 1 h after exposure of the cell cultures to 10 nM TCDD.246 This time is slightly less than the 2 h required by Israel and Whitlock247 to detect an increase in cytochrome P-4501A1 mRNA levels after exposure of Hepa Iclc7 cells to 10 pM TCDD. After exposure of Hepa Iclc7 cells to a maximally inducing con- centration of 1 nM TCDD, however, there are significant increases in the cellular concentration of cytochrome P-4501A1 mRNA after 1 h, whereas the induction of AHH activity takes slightly longer.247 Gierthy et al.240 reported that exposure of MCF-7 cells to 1 nM TCDD caused suppression of the 17(3-estradiol-induced secretion of tPA. This effect of TCDD, however, occurred in the absence of any measurable decrease in the whole cell concentration of estrogen receptors. Gier- thy's group pretreated their cultures with serum- free medium, which was done to reduce cell pro- liferation and maximize the cellular content of estrogen receptors. The disparity between this result of Gierthy et al.,240 which suggests no ef- fect of TCDD on the estrogen receptor content of MCF-7 cells, and the contrary results of Safe's group in this same cell line remains largely unex- 318 ------- plained. Overall, it appears as though no obvious distinction between the two proposed mecha- nisms can be made at the present time. Therefore, it seems that the antiestrogenic effect of TCDD results from both the increased metabolism of estrogen and a decreased number of estrogen re- ceptors. It is important to note that TCDD does not compete with radiolabeled estrogens or pro- gesterone for binding to estrogen or progesterone receptors, and that these steroids do not bind to the Ah receptor or compete with radiolabeled TCDD for binding.238'249 c. Evidence for an Ah Receptor Mechanism \. Ah Receptor Mutants Although the antiestrogenic effects of TCDD may be caused by either a decreased number of occupied nuclear estrogen receptors and/or the increased metabolism of estrogen, there is evi- dence that the antiestrogenic effect of TCDD is mediated by the Ah receptor. Thus, the anties- trogenic effects of TCDD in cultured cells appear to involve an Ah receptor-mediated alteration in the transcription of genes. This is indicated by studies using wild-type Hepa Iclc7 cells and mu- tant Hepa IclcV cells in culture.246 In wild-type cells, TCDD reduces the number of nuclear es- trogen receptors and this response can be inhib- ited by cycloheximide and actinomycin D. How- ever, in class 1 mutants, which have relatively low Ah receptor levels, TCDD has only a small effect. Similarly, in class 2 mutants, which have a defect in the accumulation of transcriptionally active nuclear Ah receptors, there was no effect of TCDD on the number of nuclear estrogen re- ceptors. Taken together, these results indicate that the downregulation of estrogen receptors in Hepa IclcV cells involves an Ah receptor-mediated ef- fect on gene transcription. As previously noted, TCDD induces cytochrome P-4501A1 mRNA transcription and enzyme activity in Hepa Iclc? cells.247 This effect also is Ah receptor me- diated.127 ii. Structure-Activity Relationships In Vivo Relative potencies of halogenated aromatic hydrocarbon congeners as inhibitors of uterine peroxidase activity in the rat are similar to their relative Ah receptor-binding affinities.250 Only limited relative potency information is available for the reduction of hepatic and uterine estrogen receptor concentrations per se by these sub- stances in rats. TCDD and 1,2,3,7,8-PeCDD both exhibit high affinity for the Ah receptor. At an 80-n,g/kg dose of either of these two substances, hepatic estrogen receptor concentrations are re- duced 42 and 41%, whereas uterine estrogen re- ceptor concentrations are reduced 53 and 49% by TCDD and 1,2,3,7,8-PeCDD, respectively. On the other hand, 1,3,7,8-TCDD and 1,2,4,7,8- PeCDD bind less avidly to the Ah receptor. At a 400-jig/kg dose of either of these two sub- stances, hepatic estrogen receptor concentrations are reduced 36 and 40%, whereas uterine estro- gen receptor concentrations are reduced 21 and 24% by 1,3,7,8-TCDD and 1,2,4,7,8-PeCDD, respectively.249 Because the potency of these con- geners for reducing estrogen receptor concentra- tions correlates with their Ah receptor-binding affinities, these in vivo results provide evidence that the antiestrogenic effect of TCDD is me- diated by the Ah receptor. iii. Genetic Evidence Consistent with the interpretation based on structure-activity relationships, there is a greater reduction in the number of hepatic estrogen re- ceptors when AhbAhb C57BL/6 mice are exposed to TCDD than when AhdAhd DBA/2 mice are similarly exposed.251 To date, however, the anti- estrogenic effects have not been studied in the progeny of test crosses between AhbAhb and AhdAhd mouse strains that, respectively, produce Ah receptors with high or low binding affinity for TCDD. Therefore, the potential segregation of the antiestrogenic effects of TCDD with the Ah locus has not been verified by the results of genetic crosses. iv. Structure-Activity Relationships In Vitro The Ah receptor is detectable in MCF-7 cells, and AHH as well as EROD activities are both inducible in these cells.245 The relative abilities of TCDD and other CDD, CDF, and PCB con- 319 ------- curs perinatally rather than in adulthood. To il- lustrate this sensitivity, a single maternal TCDD dose of 0.16 (xg/kg given on day 15 of gestation affects several endpoints in male offspring that collectively indicate a deficit in androgenic sta- tus.254-263 These include decreases in the weights of accessory sex organs such as the ventral pros- tate and epididymis and cauda epididymis, as well as a decreased anogenital distance and in- creased time of testis descent. In adult male rats exposed perinatally to TCDD, the effects on sex- ual behavior included increases in the number of mounts preceding ejaculation and intromission latency.254'265 These effects were produced by single TCDD doses as low as 0.064 fig/kg given on day 15 of gestation. When exposure to TCDD occurs in adulthood on the other hand, relatively large doses in the overtly toxic range are required to cause decreases in ventral prostate and caput epididymis weight.230-256 Kociba et al.230 reported that accessory sex organ weights are decreased in rats following exposure to 1 (xg TCDD/kg/ day, 5 days per week for 13 weeks. Using the parameters for TCDD half-life and bioavailabil- ity in the rat determined by Rose et al.,226 this dosage regimen results in a TCDD body burden of approximately 20 |xg/kg at the end of the dos- ing period. This body burden is similar to the ED50 of 15 |xg/kg determined by Moore et al.256 for some adverse effects of TCDD, including decreased plasma androgen concentrations and accessory sex organ weights in adult male rats. In addition, it is at least 100 times greater than doses of TCDD that decreased androgenic status after perinatal exposure. In male rats, TCDD exposure results in de- creased spermatogenesis. This effect occurs after exposure in adulthood to single doses of TCDD as low as 3 jxg/kg,260 whereas male rats exposed perinatally to only 0.064 (xg TCDD/kg given on day 15 of gestation are similarly affected in adult- hood.254-265 However, a comparison of the TCDD doses in adult rats that decrease plasma testos- terone levels and accessory sex organ weights with that which decreases spermatogenesis (15 vs. 3 (xg/kg) suggests that decreases in plasma an- drogen concentrations and/or androgen respon- siveness in TCDD-treated adult male rats may not completely explain the effects of TCDD on spermatogenesis. Similarly, the reduction in plasma testosterone concentration in perinatally exposed male rats may be insufficient to explain the effects of TCDD exposure on spermatogen- esis.180-265 In adult rats, the most sensitive toxic re- sponses to TCDD have been observed following long-term, low-level exposure. In a three-gen- eration reproduction study, Murray et al.228 re- ported that dietary administration of TCDD at doses as low as 0.01 |xg/kg/day significantly af- fected reproductive capacity in female rats, with no effects seen at 0.001 |xg/kg/day (NOAEL). The same NOAEL was found in a 2-year chronic toxicity and oncogenicity study in which an in- creased incidence of certain types of neoplasms was altered among rats given TCDD doses of 0.01 or 0.1 (xg/kg/day.231 Based on the phar- macokinetics of TCDD in the rat,226 the steady- state body burden of TCDD in these rats that were chronically dosed (>90 days) with either 0.01 or 0.001 jxg TCDD/kg/day is approximately 0.29 (xg/kg (LOAEL) and 0.029 p,g/kg (NOAEL), respectively. Yet, Mably et al.254-263-265 found that a single TCDD dose of 0.064 (xg/kg given on day 15 of gestation produced a number of statis- tically significant effects on the reproductive sys- tem of male rat offspring. Because 0.064 jxg TCDD/kg was the lowest dose tested, a NOAEL for developmental male reproductive toxicity, de- fined as the lowest dose used that had no statis- tically significant effect, could not be determined by Mably et al.254-263-265 It is concluded that de- velopmental effects on spermatogenesis occur at a maternal TCDD dose that is lower than any dose previously shown to produce toxicity in rats. IV. SUMMARY The potential for dioxins and related com- pounds to cause reproductive and developmental toxicity has been recognized for many years. Re- cent laboratory studies have broadened our knowledge in this area and suggest that altered development may be among the most sensitive TCDD endpoints. A substantial portion of the literature reviewed was published after 1985 and a special effort has been made to view the de- velopmental toxicity of TCDD in light of the Ah receptor model of TCDD. On the other hand, 322 ------- there is very little, if any, information available that can be used to relate the reproductive toxicity of TCDD to this receptor model. We chose to structure this review into sec- tions on developmental toxicity and male and female reproductive toxicity, but we recognize and want to emphasize that developmental and reproductive events are interrelated at all levels of biological complexity. Therefore, the reader should not view the section subheadings within each of these divisions as defining discrete end- points that are exclusive of other endpoints. For example, effects of TCDD on circulating levels of sex hormones and/or on responsiveness to sex hormones may be translated into reproductive dysfunction if exposure occurs in adulthood as well as abnormal development of sexual behavior if exposure occurs perinatally. Likewise, even though organ structure and growth are considered separate manifestations in developmental toxicity that are associated with perinatal exposure to TCDD, the normal development of an organ is dependent on normal growth processes, and in- hibition of prenatal growth can significantly dis- rupt the structural integrity of an organ system. Given the current data base, developmental toxicity endpoints tend to be observed at lower TCDD exposure levels than are endpoints of male and female reproductive toxicity. The lowest ef- fective TCDD egg burden for causing develop- mental toxicity in fish and birds and the lowest effective maternal TCDD body burden for pro- ducing a wide range of developmental responses in mammals are summarized in Tables 8,9, and 10, respectively. These results indicate that a wide variety of developmental events, crossing three vertebrate classes and several species within each class, can be perturbed, thus suggesting that TCDD has the potential to disrupt a large number of critical developmental events at specific de- velopmental stages. In addition, only transient exposure to relatively low levels of TCDD at critical times may be all that is necessary to cause irreversible disruptions in organ system or func- tion. Higher TCDD exposure levels cause em- bryo/fetal mortality, and this lethal effect of TCDD-like congeners clearly poses a danger to populations of the most sensitive native fish and wildlife species. Because developmental toxicity following exposure to TCDD-like congeners occurs in fish, birds, and mammals, it is likely to occur in man. Certain effects in human infants exposed to a complex mixture of PCBs, CDFs, and PCQs in the Yusho and Yu-Cheng poisoning episodes were probably caused by the combined exposure to those PCB and CDF congeners that are Ah re- ceptor agonists. The effects observed in human infants perinatally exposed to this complex mix- ture were similar to those reported in newborn mice and adult monkeys exposed only to TCDD, and this comparability of effects increases the probability that the outcomes of exposure in Yusho and Yu-Cheng children were due to the TCDD- like congeners in the contaminated rice oil in- gested by their mothers. Most significant is a clustering of effects in organs derived from ec- toderm, a syndrome referred to as ectodermal dysplasia. Included in this syndrome are effects on the skin, nails, and meibomian glands that have occurred in both adult monkeys exposed to TCDD and in the Yusho and Yu-Cheng infants exposed transplacentally to PCB-, CDF-, and PCQ-contaminated rice oils. In addition, accel- erated tooth eruption has been reported both in human infants affected by the Yusho and Yu- Cheng exposures and in neonatal mice exposed to TCDD. The CNS also is derived from ecto- derm, and the occurrence of functional neuro- toxic effects due to perinatal TCDD exposure in monkeys and rats suggests that brain function as a site of action of TCDD may be involved in the ectodermal dysplasia syndrome. Yu-Cheng chil- dren transplacentally exposed to PCB-, CDF-, and PCQ-contaminated rice oil have produced a clinical impression of developmental delay and psychomotor delay during developmental and cognitive tests, whereas monkeys perinatally ex- posed to TCDD are affected by a deficit in cog- nitive function. The concept that the ectodermal dysplasia syndrome in the Yusho and Yu-Cheng infants may be caused by the combination of PCB and CDF congeners in the rice oil that are Ah receptor agonists, but are less potent that TCDD, is consistent with structure-activity results for various developmental endpoints in different spe- cies of fish, birds, and mammals. These studies indicate that while there is variability between species in the profile of developmental responses elicited by TCDD, essentially all TCDD-like PCB, CDD, and CDF congeners that have Ah receptor affinity and intrinsic activity produce the 323 ------- same pattern of developmental effects within a given vertebrate species if a sufficiently high dose of the congener is given. In mammals, postnatal functional alterations involving learning behavior and the developing male reproductive system appear to be the de- velopmental events most sensitive to perinatal dioxin exposure. A maternal TCDD body burden of 19 ng/kg in the rhesus monkey at the time of conception is associated with impaired object learning in the offspring when they are tested at approximately 14 months of age. In the rat, a single maternal dose of TCDD as low as 64 ng/ kg on day 15 of gestation results in a reduction in spermatogenesis and alteration in masculine sexual behavior of the male offspring in adult- hood. In birds and fish, structural malformations and embryo mortality are the 'most sensitive ef- fects observed after direct injection of TCDD into fertilized eggs. In chicken embryos, a 17% in- crease in cardiovascular malformations was pro- duced by an egg TCDD dose as low as 9 ng/kg. In lake trout sac fry, the lowest TCDD egg burden to increase mortality was 40 and 55 ng/kg/egg. In mammals, alterations in structural end- points and diminished prenatal viability and growth begin to predominate during gestation at maternal TCDD body burdens and/or daily TCDD doses that are above 100 ng/kg. The incidence of a structural malformation that consists of extra ribs in rabbits is increased at a maternal TCDD dose during gestation of 100 ng/kg/day. Prenatal mortality in monkeys has been observed where the maternal body burden at the onset of preg- nancy is estimated to be 111 ng/kg, a level that produces no signs of overt maternal toxicity. In rats, fetal growth is inhibited at a maternal TCDD dose of 125 ng/kg/day during gestation. In mice, hydronephrosis can be elicited at a maternal TCDD dose of 500 ng/kg/day administered on gestational days 6 to 15, and in hamsters the same response can be caused by a single 1500 ng/kg dose of TCDD administered to the dam on either gestational day 7 or 9. These doses of TCDD that cause hydronephrosis in mice and hamsters are not maternally toxic. In the mouse, cleft pal- ate is produced at a maternal TCDD dose of 3000 ng/kg/day administered on gestational days 6 to 15. Prenatal mortality in the mouse results from a single maternal dose of 24,000 ng/kg admin- istered on gestational day 6, however, larger doses of TCDD are required to produce prenatal mor- tality in the mouse when administered on later gestational days. A general finding in fish, bird, and mammalian species is that the embryo or fetus is more sensitive to TCDD-induced mor- tality than the adult. Thus, the timing of TCDD exposure during the life history of an animal can greatly influence its susceptibility to overt dioxin toxicity. With respect to male and female reproductive endpoints, there are clear effects following dioxin exposure of the adult animal. Such reproductive effects generally occur at TCDD body burdens that are higher than those required to cause the more sensitive developmental endpoints. For ex- ample, TCDD exposure of the adult male causes reduced testis and accessory sex organ weights, abnormal testis structure, decreased spermato- genesis, reduced fertility, decreased testicular testosterone synthesis, reduced plasma androgen concentrations, and altered regulation of pituitary LH secretion. However, these effects only appear at TCDD exposure levels that are overtly toxic to the animal. In the more limited studies focus- ing on female reproduction, the primary effects include decreased fertility, inability to maintain pregnancy, and, in the rat, decreased litter size. Signs of ovarian dysfunction and alterations in hormone levels also have been reported. Exposure of female mice and rats to TCDD has an antiestrogenic effect. The dose of TCDD required to produce this response is generally higher than that needed to cause the most sen- sitive signs of developmental toxicity in these species. More specifically, hydronephrosis and cleft palate in mice and reductions in spermato- genesis in rats occur at maternal doses of TCDD that are far less than those needed to exert an antiestrogenic effect when young adult female mice and rats are exposed to dioxin. The precise mechanism of the antiestrogenic effect of TCDD is not fully understood. It may be caused by both a decrease in estrogen receptor number and/or by an increase in cytochrome P-4501A1-mediated estrogen metabolism within the target cell. The antiestrogenic action of TCDD is significant in that it may provide insight not only into the cause of certain female reproductive effects, but also developmental effects, such as the feminization of sexual behavior in male rats. 324 ------- The most convincing evidence for establish- ing a role of the Ah receptor in causing a partic- ular TCDD endpoint is to show that genetic link- age exists between the expression of that endpoint and a particular allele at the Ah locus. Of all the developmental and reproductive effects of TCDD, such genetic linkage has been demonstrated only for structural malformations in mice and anties- trogenicity in Hepa IclcV cells. Structure-activ- ity relationships can be used to support genetic evidence where it exists, or in the absence of genetic evidence, to indicate a probable role for Ah receptor involvement. The very limited amount of structure-activity type evidence avail- able for embryo/fetal mortality due to TCDD- like congeners in fish, birds, and mammals sug- gests that this effect also may involve the Ah receptor. For other developmental and reproduc- tive effects of TCDD, there have been no genetic assessments or relative potency evaluations of congeners for Ah receptor involvement. Al- though it remains possible that an Ah receptor mechanism will eventually be demonstrated for many more of these effects, it also is feasible that the Ah receptor mechanism may not be in- volved in all of them. ACKNOWLEDGMENTS The authors acknowledge Dr. Barbara D. Abbott, Dr. Linda S. Birnbaum, Mr. Donald L. Bjerke, Dr. Peter L. Defur, Dr. John F. Gierthy, Dr. Claude L. Hughes, Dr. Robert W. Moore, Dr. Stephen Safe, Dr. Susan L. Schantz, and Dr. Hugh M. Tilson for reviewing an earlier version of this publication. This work was supported in part by NIH grant ESO1332. 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