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儿童型脊髓性肌萎缩症的分子遗传学研究

Molecular genetic study of a childhood form of spinal muscular atrophy.

作者信息

Wong V, Chan V

机构信息

Department of Paediatrics, Queen Mary Hospital, Faculty of Medicine, The University of Hong Kong.

出版信息

J Child Neurol. 2001 Apr;16(4):291-4. doi: 10.1177/088307380101600412.

Abstract

Molecular genetic studies were performed in 28 cases of childhood-onset spinal muscular atrophy (24 unrelated families). This consisted of type 1 (severe) (n = 5), type 2 (intermediate form) (n = 8), and type 3 (mild) (n = 15). Deletion of exons 7 and 8 of the SMNt gene was found in 100%, 100%, and 93%, respectively, in type 1, 2, and 3 spinal muscular atrophy. Deletion of exons 5 and 6 of the NAIP gene was found in 3 of 5 (60%) of type 1 and none of the type 2 and 3 cases. None of the 32 asymptomatic relatives had homozygous deletions in the SMNt and NAIP genes. Thus, the role of the NAIP gene needs to be specifically defined in spinal muscular atrophy. In a suspected case of spinal muscular atrophy, deletion of the SMNt gene is a useful laboratory marker for confirmation of the diagnosis.

摘要

对28例儿童期发病的脊髓性肌萎缩症患者(来自24个无亲缘关系的家庭)进行了分子遗传学研究。其中包括1型(严重型)(n = 5)、2型(中间型)(n = 8)和3型(轻型)(n = 15)。在1型、2型和3型脊髓性肌萎缩症患者中,分别有100%、100%和93%的患者检测到SMNt基因外显子7和8缺失。在5例1型患者中有3例(60%)检测到NAIP基因外显子5和6缺失,2型和3型患者均未检测到。32名无症状亲属中,无一例在SMNt和NAIP基因中存在纯合缺失。因此,NAIP基因在脊髓性肌萎缩症中的作用需要进一步明确。在疑似脊髓性肌萎缩症病例中,SMNt基因缺失是确诊的一项有用的实验室指标。

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