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通过荧光原位杂交(FISH)意外检测到两个22号染色体长臂端粒缺失。

Two 22q telomere deletions serendipitously detected by FISH.

作者信息

Precht K S, Lese C M, Spiro R P, Huttenlocher P R, Johnston K M, Baker J C, Christian S L, Kittikamron K, Ledbetter D H

机构信息

Department of Human Genetics, The University of Chicago, IL 60637, USA.

出版信息

J Med Genet. 1998 Nov;35(11):939-42. doi: 10.1136/jmg.35.11.939.

Abstract

Cryptic telomere deletions have been proposed to be a significant cause of idiopathic mental retardation. We present two unrelated subjects, with normal G banding analysis, in whom 22q telomere deletions were serendipitously detected at two different institutions using fluorescence in situ hybridisation (FISH). Both probands presented with several of the previously described features associated with 22q deletions, including hypotonia, developmental delay, and absence of speech. Our two cases increase the total number of reported 22q telomere deletions to 19, the majority of which were identified by cytogenetic banding analysis. With the limited sensitivity of routine cytogenetic studies (approximately 2-5 Mb), these two new cases suggest that the actual prevalence of 22q telomere deletions may be higher than currently documented. Of additional interest is the phenotypic overlap with Angelman syndrome (AS) as it raises the possibility of a 22q deletion in patients in whom AS has been ruled out. The use of telomeric probes as diagnostic reagents would be useful in determining an accurate prevalence of chromosome 22q deletions and could result in a significantly higher detection rate of subtelomeric rearrangements.

摘要

隐匿性端粒缺失被认为是特发性智力迟钝的一个重要原因。我们报告了两名无关个体,其G带分析正常,在两个不同机构通过荧光原位杂交(FISH)意外检测到22号染色体端粒缺失。两名先证者均表现出一些先前描述的与22q缺失相关的特征,包括肌张力减退、发育迟缓以及无言语能力。我们的这两个病例使已报道的22q端粒缺失总数增加到19例,其中大多数是通过细胞遗传学带型分析确定的。鉴于常规细胞遗传学研究的敏感性有限(约2 - 5 Mb),这两个新病例表明22q端粒缺失的实际患病率可能高于目前记录的水平。另外值得关注的是与天使综合征(AS)的表型重叠,因为这增加了在已排除AS的患者中存在22q缺失的可能性。使用端粒探针作为诊断试剂将有助于确定22号染色体缺失的准确患病率,并可能显著提高亚端粒重排的检测率。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/80ee/1051488/1c402cbe6fb5/jmedgene00240-0060-a.jpg

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