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强直性肌营养不良症异质性的临床证据。

Clinical evidence for heterogeneity in myotonic dystrophy.

作者信息

Bundey S

出版信息

J Med Genet. 1982 Oct;19(5):341-8. doi: 10.1136/jmg.19.5.341.

Abstract

In a study of 35 index patients who developed myotonic dystrophy between birth and 30 years (neonatal cases aware excluded), 30 could be categorised into two clinical types. The 13 type 1 patients had a more severe limb weakness, of patchy distribution, associated with proportional facial weakness. The 17 type 2 patients had a milder and more diffuse limb weakness; their facial weakness, however, was very pronounced and preceded the limb weakness by several years. All but one of the 25 affected relatives who were examined belonged to the same category as their index relative, providing evidence that the cause of the clinical heterogeneity was genetic. Subsequent observations showed that mental retardation, male infertility, and neonatally affected offspring were commoner in type 2 patients. Congenital myotonic dystrophy could occur among the offspring of either affected males or affected females, but neonatal symptoms were confined to the offspring of affected women. The overall risk for having neonatally affected offspring for this prospective study of young adult patients was 7 in 38, and for the offspring of affected females 7 in 27. The risk for having a surviving child whose mental or physical handicap or both required special schooling was 1 in 12 for males and 4 in 27 for females.

摘要

在一项针对35例出生至30岁之间患强直性肌营养不良的索引患者(不包括已确诊的新生儿病例)的研究中,30例可分为两种临床类型。13例1型患者肢体无力更严重,呈斑片状分布,伴有相应程度的面部无力。17例2型患者肢体无力较轻且更广泛;然而,他们的面部无力非常明显,且比肢体无力早数年出现。在接受检查的25名受影响亲属中,除一人外,其他所有人都与其索引亲属属于同一类型,这证明临床异质性的原因是遗传因素。后续观察表明,2型患者中智力迟钝、男性不育和新生儿受影响的后代更为常见。先天性强直性肌营养不良可发生在受影响男性或受影响女性的后代中,但新生儿症状仅限于受影响女性的后代。在这项针对年轻成年患者的前瞻性研究中,新生儿受影响后代的总体风险为38例中有7例,受影响女性的后代中为27例中有7例。男性存活的孩子出现精神或身体残疾或两者都需要特殊教育的风险为12例中有1例,女性为27例中有4例。

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