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PubMed1993

Neonatal treatment of male monkeys with a gonadotropin-releasing hormone agonist alters differentiation of central nervous system centers that regulate sexual and skeletal development.

Male rhesus monkeys treated continuously with a GnRH agonist for the first 4 months of postnatal life exhibited a delay in the onset of puberty and an attenuated peripubertal rise in testosterone (T) secretion. The objectives of the current study were to determine whether these effects on sexual development were permanent and whether the hypothalamic-pituitary-testicular axis was functioning normally in these animals as adults. Neonatal GnRH agonist treatment delays but does not permanently block sexual maturation in male monkeys. Treated animals that did not show a pubertal rise in serum T during the breeding season of their 4th year exhibited a seasonal but subnormal elevation of serum T during the subsequent breeding season. Growth of the skeleton was diminished as evidenced by shorter adult crown-rump, tibia, and femur length and reduced bone mineral density of the humerus and lumbar spine. The magnitude of the serum LH and T response to iv pulses of GnRH [50 ng/kg body weight (BW)] and naloxone (1 mg/kg BW) did not differ between control and treated animals during the nonbreeding or breeding season at 6 yr of age. Conversely, treated animals showed a subnormal serum LH and T response to N-methyl-D,L-aspartic acid (5 mg/kg BW iv) during the nonbreeding season. These data suggest that adult monkeys treated neonatally with a GnRH agonist exhibit subnormal sensitivity of the central nervous system to one or more excitatory amino acids (e.g. aspartate or glutamate). Thus, abolishing neonatal activation of the pituitary-testicular axis with a GnRH agonist may permanently alter differentiation of central nervous system centers that are either involved in GnRH secretion or govern this process.

Animals↗
PubMed1988

[Reproduction studies of carboplatin (II)--Intravenous administration to rats during the period of fetal organogenesis].

Carboplatin, an oncostatic drug, was administered intravenously to pregnant Crj: CD (Sprague-Dawley) rats from day 7 through 17 of gestation at dose levels of 1, 2 and 4 mg/kg/day. The summarized results obtained are as follows: 1. Carboplatin 4 mg/kg suppressed the maternal body weight gains from day 13 through 20 of gestation. 2. Uterine weights were reduced in F0 dams at term at carboplatin 4 mg/kg. 3. Carboplatin 4 mg/kg brought the inhibition of fetal growth accompanied by the lowered values in fetal weights, crown-rump distances and tail lengths. Furthermore, the elevated incidences of unossified 5th and 6th sternum , as well as retarded ossification of sacrococcygeal vertebrae were also noted in this dose level. 4. The birth rate was reduced in neonates (F1) at carboplatin 4 mg/kg. 5. Body weight gains in male F1 rats were suppressed at carboplatin 4 mg/kg from 4 to 8 weeks of age. 6. Carboplatin 4 mg/kg decreased the brain weights on an absolute basis in female F1 rats, but failed to affect their postnatal differentiations, early behavioral developments, learning ability, motor activity or emotional development. 7. Reproductive ability in F1 rats of both sexes were not affected by carboplatin. 8. Influences on prenatal development were not apparently observed for F2 fetuses derived from F1 rats whose dams had ever received carboplatin during the organogenetic period. Based on these results, the no-effect dose level of carboplatin under the present experimental condition was estimated to be 2 mg/kg/day against dams and their offspring.

Animals↗
PubMed1990

[Reproductive and developmental toxicity studies of buspirone hydrochloride (I)--Oral administration to rats during the period of fetal organogenesis].

Buspirone hydrochloride (buspirone), an anxiolytic drug, was administered orally to pregnant Crj: CD (Sprague-Dawley) rats from day 7 through 17 of gestation at dose levels of 2, 12 and 75 mg/kg/day. The summarized results obtained are as follows: 1. Decreased activity was observed for F0 dams at buspirone 75 mg/kg. Further, the suppression of maternal body weight gains accompanied by the reduction of food consumption was shown during the administration period at the same dose level. 2. Liver weights were increased in F0 dams at term at buspirone 12 mg/kg and higher. Besides, brain, pituitary, adrenal and ovarian weights were increased in F0 dams at term at buspirone 75 mg/kg. 3. Buspirone 75 mg/kg brought the inhibition of fetal growth followed by the lowered values in fetal weights, crown-rump distances and tail lengths. Furthermore, the elevated incidence of skeletal abnormalities such as nodular and wavy ribs and unossified 5th and 6th sternum , as well as retarded ossification of cervical vertebrae, forelimbs and hindlimbs were also noted in this dose level. Also, the retarded ossification was observed at 12 mg/kg. 4. Buspirone failed to affect the parturition of F0 dams. 5. Buspirone did not function the viability of newborns (F1), and postnatal differentiations, learning ability, motility, motor activity or emotional development in F1 animals. 6. Body weight gains were depressed in female F1 rats from 4 to 9 weeks of age and food consumption was decreased in male F1 rats from 6 to 8 weeks of age at buspirone 75 mg/kg. 7. Buspirone 75 mg/kg produced suppressions of brain weights at 10 weeks of age in male and female F1 rats and lung weights at weaning in male F1 rats. Spleen weights were increased in female F1 rats at 10 weeks of age at the same dose level. However, buspirone failed to affect their reproductive ability. 8. F2 neonates derived from F1 rats whose dams had ever received buspirone during the period of fetal organogenesis showed no changes in observation items at birth. Based on these results, the no-effect dose level of oral buspirone under the present experimental condition was estimated to be 2 mg/kg/day against dams and their offspring.

Abnormalities, Drug-Induced↗
PubMed1994

[Reproductive and developmental toxicity studies of paclitaxel. (I)--Intravenous administration to rats prior to and in the early stages of pregnancy].

Paclitaxel, an antineoplastic agent, was administered intravenously to Crj: CD (SD) rats daily at dose levels of 0 (saline and vehicle), 0.1, 0.3 and 1.0 mg/kg for 63 days prior to mating and during the mating period in males, and for 14 days prior to mating and during the mating period as well as day 0 to day 7 of gestation in females. Results were as follows: 1. Body weight gains were shown a tendency to hasten in vehicle-treated male rats associated with the increased food consumption. However, the vehicle-treated group had no effect in the other parameters that were measured in this study when compared to the saline-treated group. 2. 1.0 mg/kg paclitaxel caused suppression of the body weight gains accompanied by the decreased food consumption in either male or female rats. 3. Adrenal and ovarian weights were decreased in 1.0 mg/kg dams at term. 4. The fertility indices in both sexes of 1.0 mg/kg were lower than the saline-treated group. However, the copulation indices in both sexes in 1.0 mg/kg rats were comparable to those of the saline-treated group. 5. Decreases in the number of corpora lutea, implantations and live fetuses or increases in the number of empty implantation sites and total embryo-fetal deaths were observed in 1.0 mg/kg dams. However, the fetal weights, crown-rump distances and tail lengths in live fetuses were not affected by paclitaxel treatment. Based on the reproductive and developmental indices, the no toxic-effect dose level of paclitaxel is 0.3 mg/kg/day for parent animals and their fetuses.

Adrenal Glands↗
PubMed2004

Development of preincubated chicken eggs following exposure to 50 Hz electromagnetic fields with 1.33-7.32 mT flux densities.

The effects of applying extremely low-frequency (50 Hz) electromagnetic fields (ELF-EMF) for 24 hr and different densities (1.33-7.32 mT) were examined on healthy, freshly fertilized white leghorn chicken eggs (55-65 g). Results showed no increase in the rate of abnormalities in exposed groups, but were only significant in 4.19, 5.32 and 5.86, 6.65 mT densities. Alizarin red S and alcian blue 8GX staining showed some embryos with extra ribs, defects in ribs and vertebrae, anuria and abnormal beaks. Study of egg weight, after 9 days of incubation, showed no significant differences between control, sham-exposed and experimental groups. Analysis of crown-rump, beak-occipital length and weight of embryo, showed significant decrease in weight at 4.39 and 5.52 mT intensities, comparing with control and sham-exposed groups. These results revealed that 50 Hz electromagnetic fields can even induce developmental alterations in preincubated chick embryos and confirm that its strength could be a determinant factor for the embryonic response to extremely low frequency EMFs (window effects) prior to incubation.

Animals↗
PubMed2000

Disproportional skeletal growth and markedly decreased bone mineral content in growth hormone receptor -/- mice.

Growth hormone (GH) is important for skeletal growth as well as for a normal bone metabolism in adults. The skeletal growth and adult bone metabolism was studied in mice with an inactivated growth hormone receptor (GHR) gene. The lengths of femur, tibia, and crown-rump were, as expected, decreased in GHR-/- mice. Unexpectedly, GHR-/- mice displayed disproportional skeletal growth reflected by decreased femur/crown-rump and femur/tibia ratios. GHR-/- mice demonstrated decreased width of the growth plates in the long bones and disturbed ossification of the proximal tibial epiphysis. Furthermore, the area bone mineral density (BMD) as well as the bone mineral content (BMC)/body weight were markedly decreased in GHR-/- mice. The decrease in BMC in GHR-/- mice was not due to decreased trabecular volumetric BMD but to a decreased cross-sectional cortical bone area In conclusion, GHR-/- mice demonstrate disproportional skeletal growth and markedly decreased bone mineral content.

Animals↗
PubMed1997

Development of Meckel's cartilage in the symphyseal region in man.

BACKGROUND: The aim of this work is to clarify the aspects which are at present most controversial about the development of the anterior segments of Meckel's cartilage, such as the role of and determination of the area that is incorporated in the development of the human mandible. METHODS: Light microscope studies were done on 25 embryos and human fetuses from the collection of the Institute of Embryology at the University Complutense of Madrid and the Department of Morphological Science from the University of Granada. Specimen length was between 18 and 125 mm crown-rump. RESULTS: During the embryonic period, Meckel's cartilages were placed in the midline of the mandibular arch but fusion was not observed between them. Ossification of Meckel's cartilage begins at the end of the embryonic period and is completed in the fetal period and the portion that participates in mandibular formation is determined. This segment extends from the mental foramen to near the midline of the mandible. In this region, on the dorsal surface of the symphysis, cartilaginous nodules that originate from Meckel's cartilage are isolated. CONCLUSIONS: The ventral portions of Meckel's cartilage do not fuse in the midline of the mandibular arch. These present endo- and perichondral ossification and the section from the mental foramen to near the midline (mandibular symphysis) participates in mandibular formation. The ventral ends of Meckel's cartilage, i.e., the ends nearest the midline, do not ossify and remain isolated on the dorsal surface of the fetal mandibular symphysis.

Cartilage↗
PubMed1983

The effects of low dosages of trichloroethylene on chick development.

This study reports the effects of low dosages of trichloroethylene (TCE) (1-25 mumol/egg) on chick development when embryos were exposed directly to TCE on days 1 and 2 and examined at day 14 of embryogenesis. The results indicate that in terms of embryotoxicity, growth defects and morphological anomalies, the treated embryos differed significantly from the controls. The low doses of TCE tested produced 50% mortalities. Survivors in all instances exhibited these developmental defects: evisceration; subcutaneous edema; light pigmentation of the epidermis; beak malformations; club foot and patchy feathering. The incidence of these malformations varied depending on the day of exposure to TCE except for embryos treated with 1 mumol. Irrespective of the day of injection or concentration, the highest proportion of malformations were those of light pigmentation and edema. Neither controls (uninjected embryos, O) nor sham controls (mineral oil injected embryos OMO) exhibited any malformations. Differences in measurements of crown-rump (CR), leg, wing, toe and beak lengths between treated embryos and controls were significant at the P less than 0.05 level of confidence. However, there was no significant difference between the weights of livers of treated embryos and controls nor was any pathology noted for the livers. The embryotoxicity of TCE is described by comparing the average percentage of deaths caused by all concentrations of TCE injected on both days 1 and 2 of embryogenesis. For TCE injected embryos this average was 2.5 times higher than the average for sham controls and 6 times higher than for uninjected controls.

Abnormalities, Drug-Induced↗
PubMed1979

Foot length--a new and potentially useful measurement in the neonate.

The foot length, occipito-frontal head circumference (OFC), crown-rump, and crown-heel length (CHL) of 123 neonates of gestational ages 26-42 weeks, were measured between 12 hours and 5 days. A gauge, designed and constructed at St Mary's Hospital, Manchester, was used to measure foot length. In term babies (37-42 weeks) who were of weights appropriate for gestational age (AGA) the scatter about the mean of foot length measurements was small (coefficient of variation = 4.5%) compared with birthweight (coefficient of variation = 12.0%). The wide range of foot length measurements in babies of different gestational ages prevented maturity being accurately estimated. The mean birthweight of term light-for-dates (LFD) babies was 30.9% lower than term AGA babies, whereas the mean foot length, OFC, and body length of LFD babies was reduced by only 4.2-8.8%. There was a positive linear correlation between foot length and other indices of body size in LFD and AGA babies of all gestational ages. However, in premature babies (less than 37 weeks) the correlation between foot length and birthweight (r = 0.95) and foot length and CHL (r = 0.96) was pronounced. The 95% confidence limits of the regression lines were +/- 327 g and +/- 2.3 cm respectively. Birthweight and CHL of premature babies can therefore be estimated from a measurement of foot length that is performed simply and rapidly. Measurements of foot length are valuable in premature babies who are too ill at birth for conventional anthropometric measurements to be made, and in whom such measurements cannot be carried out subsequently because of the encumbrance of the incubator and intensive care apparatus. Drug dosages and intravenous fluid requirements based on body weight or surface area can be indirectly calculated from a measurement of foot length.

Anthropometry↗
PubMed1987

[Arterial and neural pedicles of the anorectal region in the human embryo and fetus. Inductor role in normal and pathological development].

To gain a better understanding of the development of ano-rectal malformations, a study of the arterial supply of the ano-rectal region was made in 26 human embryos and fetuses. The development of the ano-rectum was studied in 20 embryos, varying in length from 3 mm to 40 mm (Crown-Rump: 22-60 days), and 6 using Born's method reconstruction; 10 fetuses aged from 3 to 7-8 months were used for a topographic study of the ano-rectal region. 40 fetuses, the majority frozen after expulsion were injected immediately. The fetuses obtained were of ages corresponding to 12-39 weeks of amenorrhea. A staged histologic study after selective micropaque injections of the inferior mesenteric artery was carried out to demonstrate the arterial parietal distribution in the ano-rectum. Smith silver impregnation method, emphasizes the connections between the rectal arteries and extrinsic and intrinsic nerves. The subdivision of the cloaca is constituted by the folding of the ano-rectal and uro-genital parts and by the thickening of the dorsal cloacal epithelium, explained perhaps by the precocious development of arterial supply. The superior rectal artery is predominant and supplies the whole of the anorectum, eventually penetrating the wall of the anal canal. The well-developed median sacral artery is surrounded by a nervous tissue which reaches the M. Levator Ani. The sympathetic nerve fibers follow the arteries until the submucous layer. The development of the intramural plexus of the rectum is independent of the arterial supply.(ABSTRACT TRUNCATED AT 250 WORDS)

Anal Canal↗
PubMedDate not supplied

Growth of the body in early fetuses studied by multivariate analysis.

The growth of the body was studied in 30 human fetuses ranged from 10 to 22 weeks of gestation. The fetuses were fixed by immersion in 4 percent formaldehyde and the following dimensions were studied: a) lengths: arm, forearm, hand, thigh, leg, foot and crown-rump (sitting height), b) perimeters: head, thorax and abdomen. A covariance matrix was calculated from natural logarithms of all measurements. The relative growth of these measurement was computed by multivariate allometry using a principal components analysis (PCA). All characters were positively correlated with the first principal component which accounted for 94.65 per cent of the total variance. Considering the different measurements in the sequence of the increasing growth rates no one was considered to increase in isometric relationship. PCA showed that the following measurements grew with negative allometry: head perimeter, C-R length, thoracic perimeter, length of the forearm and abdominal perimeter. On the other hand, the following lengths grew with positive allometry: hand, foot, thigh, arm and leg. In conclusion, during the first two trimesters of prenatal life the growth of the body is allometrical. Limbs increase with greater growth rates than the perimeters of the body cavities.

Body Height↗
PubMed2005

Evidence of genetic regulation of fetal longitudinal growth.

BACKGROUND: Genetic as well as environmental factors are important determinants of fetal growth but there have been few studies of the influence of paternal factors on fetal growth. AIM: To study the influence of paternal anthropometry on detailed measurements of offspring at birth. DESIGN: A prospective cohort study involving biochemistry, and anthropometry, of mothers and fathers at 28 weeks gestation, and detailed anthropometry of children within 24 h of birth. SUBJECTS: 567 White Caucasian singleton, non-diabetic, full term pregnancies recruited from central Exeter, UK. RESULTS: Paternal height, but not paternal BMI, was correlated with birth weight (r = 0.19) and with birth length (r = 0.33). This was independent of potential confounders and maternal height. All measurements of fetal skeletal growth including crown-rump, knee-heel and head circumference were associated with paternal height. Maternal height showed similar correlations with birth weight (r = 0.18) and birth length (r = 0.26). Maternal BMI was correlated with birth weight (r = 0.27) and birth length (r = 0.15). In a multifactorial analysis 38% of the variance in fetal height could be explained by gestation, sex, paternal height, maternal height, maternal glucose, maternal BMI, parity and maternal smoking. CONCLUSION: Paternal height has an independent influence on size at birth. This predominantly influences length and skeletal growth of the baby. In contrast to maternal obesity the degree of paternal obesity does not influence birth weight. This work suggests that there is genetic regulation of skeletal growth while the maternal environment predominantly alters the adiposity of the fetus.

Adolescent↗
PubMed1987

Temporal relationship between fetal bovine skeletal growth and circulating hormonal levels.

Skeletal growth and serum hormonal levels in bovine fetuses were studied cross-sectionally from late first trimester to late third trimester of gestation (corresponding approximately to 75-275 days of age, and a crown-rump (CR) 5-105 cm/in size). Measurements of tibial and femoral lengths showed that bone growth proceeds at a 30% faster rate in fetuses of 50-105 cm CR, coincident with the appearance and exponential growth (y = 0.000207 e 0.12522 x; y, dried weights of ossification in grams and x, CR in cm) of a secondary center of ossification in the epiphysis. During this period there is an increase in the proportion of [3H]-thymidine-labeled nuclei (measured by autoradiography) in the proliferative zone of the epiphyseal growth plate and a progressive hypertrophy of chondrocytes in the epiphysis; in serum there is a rise in alkaline phosphatase activity, a rise in the calcium, and a decrease in the phosphorus concentrations. Cellularity (nuclei/area) and the proportion of [3H]-thymidine labeled nuclei in epiphyseal cartilage decline during the period of 15-105 cm CR, except at 25-45 cm CR when both parameters of chondrocyte growth have transiently stabilized. [35S]-Sulfate and [3H]-proline incorporation (cpm/100 micrograms DNA) in epiphyseal cartilage also decline initially during 10-25 cm CR, then attain a stable level during 25-50 cm CR; subsequently, [35S]-sulfate incorporation gradually increases and [3H]-proline incorporation remains at a constant level. The proportion of [3H]-thymidine-labeled nuclei in the epiphyseal growth plate also declines in early gestation, then becomes stabilized at 20-50 cm CR. The whole growth plate thickness varies during gestation and is maximal during 20-50 cm CR. The proliferative zone attains maximal thickness at 20-50 cm CR while the hypertrophied and degenerative zone has maximal thickness at 40-80 cm CR. Gestational changes of hormone levels were quantitated in fetal serum. Glucocorticoids and thyroxine were measured by radioimmunoassay; somatomedinlike bioactivity was measured as the capacity of a serum sample to stimulate [3H]-thymidine incorporation in chondrocytes compared to that of a control serum. Temporally related to the changes occurring in the skeletal tissues, there is a high serum level of glucocorticoids at 10-20 cm CR when the cartilaginous activities are declining, a peak level of serum somatomedinlike bioactivity at 20-50 cm CR when cartilage growth and metabolism become stabilized, and a rise of thyroxine level after 45 cm CR during which time there is an increasing rate of bone formation.(ABSTRACT TRUNCATED AT 400 WORDS)

Animals↗
PubMed1975

[Normal prenatal growth of the golden hamster (author's transl)].

In the golden hamster (Mesocricetus auratus) the gestation period and the loculus size of the gravid uterus from day 6 to day 15.5 (parturition) of gestation as well as the weight, width (umbilcus-black) and lenght (crown-rump) of the embryos from day 9 to the parturition were measured. The results obtained were as follows: 1. The gestation period of the golden hamster was 15 days and 15 +/- 3 hours post coitum. 2. The average values of the length (in the direction of uterine long axis) and width (mesome-trial-antimesometrial axis) in the loculus of the gravid uterus were 0.39 cm and 0.56 cm at 6 days, and 2.42 cm and 1.74 cm at 15.5 days (partiurition), respectively. 3. The average values of the crown-rump measurements, width (umblicus-back) and weight in the hamster embryos were 0.42 cm, 0.24 cm and 0.026 g at 9 days, and 2.40 cm, 1.39 cm and 2.272 g at 15.5 days (parturition), respectively. 4. The loculus size of the gravid uterus, the crown-rump measurements and width (umblicus-back) of the embryos in the period from day 13 to day 14 of gestation were markedly increased in comparison with any other period, and the weight increase of the embryos occurred rapidly from day 13 of gestation. 5. The shape of the loculus during the gestation was ovoid (mesometrial-antimesometrial axis) until the end of 10 days converged to the spherical form and thereafter changed gradually to the ovoid from in the direction of uterine long axis contraly to the previous days.

Animals↗
PubMed2001

Patterns of body and visceral growth in human prenates with clefts of the lip and palate.

OBJECTIVE: To address the hypothesis that human prenates with birth defects such as clefts of the lip and palate show growth patterns in which body size and visceral growth are aberrant for prenatal age. METHODS: Body size and organ weight measurements were derived from a cross-sectional population sample of 167 legally donated human embryos that were representative of key periods in visceral organogenesis and growth. Measurements included those of overall body size (i.e., length and weight) and weights of major organs. Data were analyzed for two comparative (age-matched) groups consisting of 120 typical-for-age "control" and 47 specimens showing the three typical cleft types. Organ weight data for the entire cleft group and for each of the three cleft subgroups were compared with those of the control group. RESULTS: Cleft specimens showed crown-rump body sizes that were within one standard deviation of the control group. Among the entire cleft sample, each of the organ weight values was generally consistent with the organ weights observed for the age-matched control group. When organ weights for the cleft group differed from the controls, with one exception, some organs showed either increased or decreased organ weights. One remarkable exception was with lung weights, which were reduced for each of the prenates across the three cleft types. Among the three cleft groups, fetuses with cleft lip and palate showed the highest percentage of organ weight discrepancies (i.e., smaller than expected) as compared with the group with cleft palate, which showed the lowest. CONCLUSIONS; Organ weight aberrancies were observable (whether increased or reduced weights), and such aberrancies were not apparent until approximately 24 weeks (fertilization age).

Body Weight↗
PubMed1991

Brief communication: Morphometric data for adult lion-tailed macaques (Macaca silenus).

Basic morphometric data were collected from 22 adult lion-tailed macaques (M. silenus) of both sexes. M. silenus is a rare primate species from which adequate morphometric data have not heretofore been available for comparative purposes. Data collected include measures of gross body size (weight; crown-rump and rump-heel length), and for males, measures of secondary sexual characteristics (canine tooth and testes size). Degree of sexual dimorphism was marked, with males significantly larger and heavier than females. The three body size measures were correlated for males but not for females. There was substantial variation among individual males in secondary sex characteristics measurements. The data indicate than lion-tailed macaque morphometrics are consonant with the general pattern of positive allometry for body size and sexual dimorphism characteristic of the primate order.

Animals↗
PubMed1986

Growth patterns of rat body, brain, and cerebellum in fetal alcohol syndrome.

Pregnant Sprague-Dawley rats were offered protein- and vitamin-enriched, nutritionally balanced, liquid ethanol diet, providing 35% of calories as ethanol, beginning on day 6 of gestation. Control animals were pair-fed identical liquid diet except that maltose-dextrins were substituted for ethanol. At birth the pups of both ethanol exposed and pair-fed mothers were surrogate-fostered by normal dams. Plasma concentrations of ethanol and thyroxine were determined in dams of experimental and control groups. Thyroxine concentrations were significantly reduced in ethanol-fed dams compared with pair-fed. Pups of both groups were killed at 0-1, 5, 7, 10, 14, 21, 28, 35, and 42 postnatal days. The crown-rump, crown-tail lengths, and weights of body, brain, and cerebellum were significantly reduced in ethanol-exposed pups compared with controls at all time sequences studied.

Animals↗
PubMed2005

Early fetal akinesia deformation sequence: a case report with unusual autoptic features.

In this paper we report a case of early onset fetal akinesia, with unusual pathological findings. This is a product of medical abortion of young, healthy, unrelated parents. The mother's obstetrical history revealed two previous early miscarriages and a suspicion of FADS in the second previous gestation. At 17 weeks of gestation, an ultrasound examination disclosed absence of fetal movements, fixed extended knees and deformation of the feet. Amniocentesis showed a normal 46, XX karyotype. Hydrops fetalis and multiple skin webs (pterygia), which are usually present in cases of early fetal akinesia, were absent. A diagnosis of arthrogryposis was made and the pregnancy was terminated at 17 weeks of gestation. Postmortem examination was performed according to the necropsy technique suggested by Langley. Thus, body weight and external measurement, including crown-rump, crown-heel, foot lengths, head, thorax and abdominal circumferences were estimated and compared with standard values for assessment of fetal growth. External dysmorphic features were evaluated prior to the evisceration. On internal examination the location and shape of every organ was evaluated. Every organ, skin, muscles from different parts of the body, the brain and spinal cord were sampled and histologically examined. External examination revealed a female fetus with marked muscular hypoplasia of upper and lower extremities with thin arms and legs and multiple joint contractures of lower extremities. The face showed a flattened nose, micrognatia, hypertelorism, cleft palate and low-set ears. There was also a small nuchal fold. The abdomen was distended with a very thin and almost transparent wall. Histologically, muscles were characterized by severe fibrosis with fatty infiltration and by moderate variability in diameter of muscle fibers. The spinal cord disclosed a paucity of anterior horn motor neurons. We suggest multiple pterygium as a diagnosis. Lethal multiple pterygium syndrome (LMPS) is only a symptom and the precise diagnosis is more likely to be spinal atrophy. We, moreover believe that the paucity of spinal motoneurons could be due to the anomalies of programmed death during fetal development and the consequence of genetic defects.

Abnormalities, Multiple↗