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西米德兰兹郡严重智力迟钝的社区研究以及脆性X染色体在其病因学中的重要性。

A community study of severe mental retardation in the West Midlands and the importance of the fragile X chromosome in its aetiology.

作者信息

Bundey S, Webb T P, Thake A, Todd J

出版信息

J Med Genet. 1985 Aug;22(4):258-66. doi: 10.1136/jmg.22.4.258.

Abstract

This paper describes a community based study of 156 boys with idiopathic, severe mental retardation. The boys were examined and a pedigree taken before the cytogenetic results were known. The prevalence of the fragile X chromosome among this group of boys was high: 9% in the whole group and 11% after 39 boys with specific features had been excluded. The fragile X syndrome is therefore an important cause of idiopathic, severe retardation. Its clinical features of large head, large testes, and IQ in the 35 to 70 range were often but not always present in the 14 boys identified in this study. In the whole group, the recurrence of severe mental subnormality was high: 1 in 8 for brothers and 1 in 25 for sisters. This high recurrence was partly due to the fragile X syndrome, partly to X linked mental retardation not accompanied by cytogenetic abnormalities, and partly due to autosomal recessive disease. Autosomal recessive disease was perhaps higher in the West Midlands than elsewhere (such as British Columbia, for example 1) because of the disproportionate contribution by Asian immigrants.

摘要

本文描述了一项针对156名患有特发性重度智力障碍男孩的社区研究。在细胞遗传学结果出来之前,对这些男孩进行了检查并绘制了家谱。在这群男孩中,脆性X染色体的患病率很高:整个群体中为9%,在排除39名具有特定特征的男孩后为11%。因此,脆性X综合征是特发性重度智力障碍的一个重要原因。在本研究中确定的14名男孩中,其大头、大睾丸以及智商在35至70范围内的临床特征经常出现,但并非总是出现。在整个群体中,严重智力低下的复发率很高:兄弟为八分之一,姐妹为二十五分之一。这种高复发率部分归因于脆性X综合征,部分归因于伴有细胞遗传学异常的X连锁智力障碍,部分归因于常染色体隐性疾病。由于亚洲移民的比例过高,西米德兰兹地区的常染色体隐性疾病患病率可能比其他地方(如不列颠哥伦比亚)更高(例如1)。

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