Is There a “Fetal Hydantoin Syndrome?”

Apt, Leonard; Gaffney, William L.

doi:10.1016/0002-9394(77)90693-6

Cited by 9 publications

(2 citation statements)

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“…On the other hand, it is difficult to explain how such a wide variety of malformations could be due to a single cause. Indeed, it has been suggested that the individual variability in the pattern of malformations is so great as to render the syndrome diagnostically useless [Apt and Gaffney, 1977;Janz, 1982a,b].…”

Section: Introductionmentioning

confidence: 99%

Variable patterns of malformation in the mouse fetal hydantoin syndrome

Finnell

Chernoff

1984

Am. J. Med. Genet.

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Controversy over the existence of a fetal hydantoin syndrome continues in medical literature despite numerous recent clinical studies describing additional patients with a characteristic pattern of abnormalities. Resistance to its acceptance as a clinical entity seems to stem from the variability of the component malformations seen in this syndrome. To examine this variability in a controlled experimental situation, we utilized data obtained in previously reported studies of a mouse model of the fetal hydantoin syndrome. In the mouse, prenatally exposed fetuses had congenital anomalies similar to those observed in the human syndrome. In terms of overall frequency of malformation there were no differences among three inbred mouse strains. However, when considering the individual rates of the 11 most common malformations, considerable differences were noted among the strains. These strain differences in the pattern of malformations appear to be related to genotypic differences in susceptibility to specific malformations. These results provide one possible explanation for the variability observed in the human fetal hydantoin syndrome.

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Section: Introductionmentioning

confidence: 99%

Variable patterns of malformation in the mouse fetal hydantoin syndrome

Finnell

Chernoff

1984

Am. J. Med. Genet.

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“…Until more data are obtained from controlled studies, using unbiased observation techniques, on the possible causal effect of PB or PHT on fetal teratogenicity or maturation, there is justification for the doubts about the very existence of these antiepileptic drug syndromes (117,(145)(146)(147)(148). Thus there is no reason to avoid or favor any of these drugs (except for diones) during pregnancy.…”

Section: Teratogenecity and Maturationmentioning

confidence: 99%

Side effects of phenobarbital and phenytoin during long-term treatment of epilepsy

Livanainen

Savolainen

1983

Acta Neurologica Scandinavica

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A B S T R A C T Phenobarbital and phenytoin have good antiepileptic effect, but clinically significant untoward effects occur during their long-term use. Phenobarbital may cause hyperactivity, behavioral problems, sedation, and even dementia; these effects are dose related to some extent. Side effects of phenytoin include sedation, a cerebellar syndrome, phenytoin encephalopathy, psychosis, locomotor dysfunction, hyperkinesia, megaloblastic anemia, decreased serum folate level, decreased bone mineral content, liver disease, IgA deficiency, gingival hyperplasia, and a lupus-like hypersensitivity syndrome. Especially susceptible to the neurotoxic effects of phenytoin are epileptic children with severe brain damage who are on multiple drugs. In those children, balance disturbance may develop and be followed by gradual loss of locomotion. Among 131 mentally retarded epileptic patients, phenytoin intoxication occurred in 73 (56 YG), of whom 18 experienced persistent loss of locomotion. There is experimental evidence that the toxic action of phenytoin lies at the cellular level, predominantly in the cerebellum. Many experts avoid the long-term use of phenytoin because of its insidious and potentially dangerous side effects. Phenobarbital (PB) and phenytoin (PHT) are classic antiepileptic drugs, but insidous and severe untoward effects have been associated with their use. These effects may include impaired mental capacity, disturbed body balance, and even loss of locomotion, which may jeopardize the effective control of epileptic seizures. Therefore, physicians are confused as to whether these drugs should be used at all; if so, then they want to know how the drug therapy should be arranged. This report surveys the clinical neurotoxicity and other side effects of PB and PHT during long-term treatment of epilepsy, including their possible mechanisms and significance in clinical practice, and offers quidelines for the management of affected patients.

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