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塞克尔综合征的中枢神经系统异常:一个新家族的报告及文献综述

Central nervous system anomalies in Seckel syndrome: report of a new family and review of the literature.

作者信息

Shanske A, Caride D G, Menasse-Palmer L, Bogdanow A, Marion R W

机构信息

Department of Pediatrics, Montefiore Medical Center/Albert Einstein College of Medicine, Bronx, New York 10467, USA.

出版信息

Am J Med Genet. 1997 May 16;70(2):155-8. doi: 10.1002/(sici)1096-8628(19970516)70:2<155::aid-ajmg10>3.0.co;2-i.

Abstract

Seckel syndrome (SS) is a rare, heterogeneous form of primordial dwarfism. The clinical delineation of this disorder has been inconsistent, using even Seckel's original criteria. As a result, probably fewer than one-third of reported cases are truly affected with SS. Among these, there have been only six familial cases, all of whom were born to normal parents, and in only one case has a detailed description of the central nervous system (CNS) anomalies been given. We describe a family in which three of eight children were affected with SS. CNS anomalies seen in our patients included agenesis of the corpus callosum, a dysgenetic cerebral cortex, a large dorsal cerebral cyst, and pachygyria, suggesting an underlying neuronal migration disorder. The parents are first cousins, representing only the second instance of consanguinity, supporting an autosomal recessive mode of inheritance.

摘要

塞克尔综合征(SS)是一种罕见的、异质性的原始侏儒症。即使采用塞克尔最初的标准,这种疾病的临床描述也不一致。因此,报告的病例中可能只有不到三分之一是真正患有SS的。其中,只有6例家族性病例,所有患儿的父母均正常,并且仅有1例对中枢神经系统(CNS)异常进行了详细描述。我们描述了一个家庭,8个孩子中有3个患有SS。我们的患者中观察到的CNS异常包括胼胝体发育不全、发育异常的大脑皮质、一个大的脑背侧囊肿和巨脑回,提示存在潜在的神经元迁移障碍。父母是近亲结婚,这是第二例近亲结婚的情况,支持常染色体隐性遗传模式。

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