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韦斯特综合征的产前病因。

Prenatal etiologies of West syndrome.

作者信息

Ohtahara S, Ohtsuka Y, Yamatogi Y, Oka E, Yoshinaga H, Sato M

机构信息

Department of Child Neurology, Okayama University Medical School, Japan.

出版信息

Epilepsia. 1993 Jul-Aug;34(4):716-22. doi: 10.1111/j.1528-1157.1993.tb00451.x.

Abstract

We investigated the etiology of West syndrome (WS) with special reference to prenatal factors in 180 cases. Prenatal cause was the most frequent diagnosis (77 cases, 42.8%), followed by perinatal (25 cases, 13.9%) and postnatal factors (12 cases, 6.7%); 48 cases (26.7%) were of uncertain etiology; eighteen cases (10.0%) were idiopathic. Of the three forms of age-dependent epileptic encephalopathy, prenatal cause was present in 12 of 15 cases (80.0%) of early-infantile epileptic encephalopathy with suppression-burst, 77 of 180 cases (42.8%) of WS, and 31 of 123 cases (25.2%) of Lennox-Gastaut syndrome (LGS). Prenatal factors of WS included tuberous sclerosis (23), chromosome abnormalities (10), cerebral dysgenesis (10), porencephaly (7), hydrocephalus (5), Aicardi syndrome (3), Aicardi syndrome associated with chromosome abnormality (1), and other causes (18). Chromosome abnormalities with WS consisted of 6 cases with 21 trisomy and one case each with 18q duplication, t(1;y) translocation, 7q duplication, and partial 2p trisomy. One patient with Aicardi syndrome also had a t(12;21) translocation. No significant difference was observed in the age of onset of WS among the five etiologic groups. The evolution from WS to LGS was not influenced by etiology, except for the idiopathic group. In patients followed for over 3 years, seizure remission occurred in 46.8% (22 of 47 cases) of the prenatal group. This was lower than the other four groups. Intellectual prognosis was also relatively poor in those with prenatal onset. Pyridoxal phosphate (PAL-P) treatment was effective in 9 of 70 (12.9%) prenatal cases and 5 of 18 (27.8%) idiopathic cases.(ABSTRACT TRUNCATED AT 250 WORDS)

摘要

我们对180例韦斯特综合征(WS)的病因进行了调查,特别关注产前因素。产前病因是最常见的诊断(77例,42.8%),其次是围产期病因(25例,13.9%)和产后因素(12例,6.7%);48例(26.7%)病因不明;18例(10.0%)为特发性。在三种年龄依赖性癫痫性脑病中,产前病因在15例早发性婴儿癫痫性脑病伴抑制爆发中的12例(80.0%)、180例WS中的77例(42.8%)以及123例伦诺克斯-加斯东综合征(LGS)中的31例(25.2%)中存在。WS的产前因素包括结节性硬化症(23例)、染色体异常(10例);脑发育不全(10例)、脑穿通畸形(7例)、脑积水(5例)、艾卡迪综合征(3例)、与染色体异常相关的艾卡迪综合征(1例)以及其他病因(18例)。与WS相关的染色体异常包括6例21三体综合征,以及各1例18q重复、t(1;y)易位、7q重复和部分2p三体。1例艾卡迪综合征患者还存在t(¨2;21)易位。五个病因组中WS的发病年龄未观察到显著差异。除特发性组外,WS向LGS的演变不受病因影响。在随访超过3年的患者中,产前组46.8%(47例中的22例)癫痫发作缓解。这低于其他四组。产前发病患者的智力预后也相对较差。磷酸吡哆醛(PAL-P)治疗在70例产前病例中的9例(12.9%)和18例特发性病例中的5例(27.8%)有效。 (摘要截选至250字)

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