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四个家族中杜氏肌营养不良症通过父系家族性发生。

Familial occurrence of Duchenne dystrophy through paternal lines in four families.

作者信息

Zatz M, Passos-Bueno M R, Rapaport D, Vainzof M

机构信息

Departamento de Biologia, Universidade de São Paulo, Brazil.

出版信息

Am J Med Genet. 1991 Jan;38(1):80-4. doi: 10.1002/ajmg.1320380118.

Abstract

In a survey of 454 families with patients affected with Duchenne muscular dystrophy (DMD) we have found 4 genealogies with 2 or more affected patients who were related through paternal lines. In 1 of these families, 2 affected cousins showed different DNA deletions suggesting 2 independent mutations; in the other 2, in which only the propositus could be studied DNA deletions were also detected in the dystrophin gene. In the last one, with 3 affected patients, no DNA deletions were detected but immunohistochemical study of muscle biopsies showed a negative dystrophin pattern typical of DMD. Although one of these families might have occurred by chance, the probability of finding the other 3 in our sample of families with DMD patients constitutes a rare event. It is suggested that other mechanisms, such as the presence of transposable elements in other sites of the genome, could be responsible in some families, for a greater predisposition for the occurrence of pathogenic deletions, duplications or mutations.

摘要

在一项对454个患有杜氏肌营养不良症(DMD)患者家庭的调查中,我们发现了4个家系,其中有2名或更多受影响的患者通过父系相关。在其中1个家庭中,2名受影响的表亲显示出不同的DNA缺失,提示有2个独立的突变;在另外2个家庭中,仅对先证者进行了研究,在肌营养不良蛋白基因中也检测到了DNA缺失。在最后一个家庭中,有3名受影响的患者,未检测到DNA缺失,但肌肉活检的免疫组织化学研究显示出典型的DMD阴性肌营养不良蛋白模式。虽然这些家庭中的一个可能是偶然发生的,但在我们的DMD患者家庭样本中发现另外3个的概率是一个罕见事件。有人提出,其他机制,如基因组其他位点存在转座元件,在一些家庭中可能是导致致病性缺失、重复或突变发生倾向增加的原因。

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