A mechanistic approach to teratogenesis: analysis of caudal dysplasia syndrome (sacral agenesis)

Kaplan, Stanley A.

doi:10.1007/978-94-011-5910-4_11

Cited by 5 publications

(3 citation statements)

References 34 publications

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“…The lumbar segment was relatively spared whilst the sacral and coccygeal segments exhibited much marked inhibition of development. Earlier investigators have largely failed to note any change in the proximal segments possibly because of the impression that one gets from the clinical studies in which 'caudal regression' has been frequently recorded as the most typical malformation in diabetic pregnancy (Kaplan, 1979;Mills et al, 1979). What appears from this study is that it is aplasia rather than 'regression' that affects the caudal segment of the vertebral column in this experimental situation.…”

Section: Discussionmentioning

confidence: 57%

Effect of Streptozotocin Induced Maternal Diabetes on Fetal Skeletal Development in the Rat

Padmanabhan

Al-Zuhair

1989

Congenital Anomalies

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Alizarin red‐S stained skeletons of the fetuses of the streptozotocin induced (on days 2, 6 and 8 of gestation) diabetic and insulin and buffer treated control rats were examined on day 20 of gestation for malformations and hypoplasia. Hypoplasia of both the neurocranial and viscerocranial bones was significant in all 3 diabetic groups. Insulin treatment had a remarkable rescue effect on fetuses of the day 8 group; a moderately similar response was found in others. Inhibition of ossification of the scapula, humerus, radius and ulna was relatively less remarkable when compared to that of the homologous bones of the hind‐limbs. However the metacarpals were affected more than the metatarsals. Day 6 of gestation was particularly susceptible for retardation of sternal ossification. Agenesis, hemi‐ and double hemivertebrae were also observed in a few cases. Reduction in caudal sacral and coccygeal vertebral development was remarkable. Incidence of the 14th rib was highest in the day 6 group and insulin administration significantly reduced this abnormality. Meticulous metabolic control had beneficial effect over the growth retarding effects of maternal diabetes but did not completely prevent the malformations.

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Section: Discussionmentioning

confidence: 57%

Effect of Streptozotocin Induced Maternal Diabetes on Fetal Skeletal Development in the Rat

Padmanabhan

Al-Zuhair

1989

Congenital Anomalies

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“…If, as first claimed by Passarge & Lenz (1966) and since widely accepted (e.g., Kaplan 1979, Kalltn et al 1992, sirenomelia is sacral agenesis carried to its most severe degree, and even to be equated with it (e.g., Schwartz et al 1982); and if sacral agenesis is associated with diabetic pregnancy, it is to be expected that sirenomelia would also be associated with maternal diabetes. This supposition is explored below.…”

Section: The Caudal Regression Syndromementioning

confidence: 93%

Case reports of malformations associated with maternal diabetes: history and critique

Kalter

1993

Clinical Genetics

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“…The infants we describe present a variety of abnormalities which could result from a common teratogenic process. Caudal dysplasia can be produced experimentally in chickens by various teratogens, includine insulin (Kaplan 1979). The damaged embryos show caudal blisters or haematomata which displace mesodermal somites; when they resolve they result in caudal dysplasia.…”

Section: N G R O S E 1972) a R E T H E R Ementioning

confidence: 99%

Alcohol, Other Drugs and the Fetus

Poskitt¹,

Hensey²,

Smith³

1982

Develop Med Child Neuro

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SUMMARY The congenital abnormalities in five infants born to mothers who drank excessive amounts of alcohol during pregnancy are described. Four of the five mothers also took other drugs, known to potentiate the effects of alcohol in the adult. It is suggested that the severity of the abnormality in some of these infants was due to potentiation of the effects of alcohol by associated drugs. The poor prospect for normal family life among the children of alcoholic women seems to have improved little since it was discussed in a paper from Liverpool in 1899. RÉSUMÉ Les anomalies congénitales de cinq nourrissons nés de mères ayant consommé des quantités excessives d'alcool pendant la grossesse sont décrites. Quatre des cinq mères avaient pris en outre durant la grossesse des médications connues pour potentialiser les effects de l'alcool chez l'adulte. On peut penser que la gravité des anomalies chez certains des nourrissons est liée à la potentialisation des effects de l'alcool par des médications asociées. Le faible espoir d'une vie familiale normale chez les enfants de femme alcoolique ne semble guère s'etre amélioré depuis qu'il a été discuté dans un article de Liverpool en 1899. ZUSAMMENFASSUNG Bei fünf Säuglingen, deren Mütter in der Schwangerschaft excessive Mengen Alkohol tranken, werden Mißbildungen beschrieben. Vier der Mütter nahmen in der Schwangerschaft zusätzlich andere Drogen, die die Wirkung des Alkohols beim Erwachsenen verstärken. Man nimmt an, daß die Schwere der Mißbildungen bei einigen dieser Kinder durch die Potenzierung der Alkoholwirkung durch zusätzliche Drogen hervorgerufen wurde. Die schlechte Prognose für ein normales Familienleben bei Kindern alkoholsüchtiger Mütter hat sich seit 1899, als das Problem in einer Zeitschrift von Liverpool diskutiert wurde, wenig geändert. RESUMEN Se describen las anomalias congénitas de cuatro niños nacidos de madres que bebian cantidades excesivas de alcohol durante el embarazo. Cuatro de las cinco madres tomaban fármacos, cuya acción se sabe que potencia los efectos del alcohol en el adulto, durante la gestación. Se sugiere que la gravedad de las anomalias en algunos de estos ninños se debia a la potenciación de los efectos del alcohol por fármacos asociados. La pobre perspectiva de una vida familiar normal entre los niños de mujeres alcohólicas, no parece haber mejotado mucho desde que el tema fue discutido en una communicación de Liverpool en 1899.

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A mechanistic approach to teratogenesis: analysis of caudal dysplasia syndrome (sacral agenesis)

Cited by 5 publications

References 34 publications

Effect of Streptozotocin Induced Maternal Diabetes on Fetal Skeletal Development in the Rat

Effect of Streptozotocin Induced Maternal Diabetes on Fetal Skeletal Development in the Rat

Case reports of malformations associated with maternal diabetes: history and critique

Alcohol, Other Drugs and the Fetus

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