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伴有8号染色体短臂缺失的遗传性球形细胞增多症。

Hereditary spherocytic anemia with deletion of the short arm of chromosome 8.

作者信息

Okamoto N, Wada Y, Nakamura Y, Nakayama M, Chiyo H, Murayama K, Inoue T, Kanzaki A, Yawata Y, Hirono A

机构信息

Department of Planning and Research, Osaka Medical Center and Research Institute for Maternal and Child Health, Japan.

出版信息

Am J Med Genet. 1995 Sep 11;58(3):225-9. doi: 10.1002/ajmg.1320580306.

Abstract

We describe a 30-month-old boy with multiple anomalies and mental retardation with hereditary spherocytic anemia. His karyotype was 46,XY,del(8)(p11.23p21.1). Genes for ankyrin and glutathione reductase (GSR) were localized to chromosome areas 8p11.2 and 8p21.1, respectively. Six patients with spherocytic anemia and interstitial deletion of 8p- have been reported. In these patients, severe mental retardation and multiple anomalies are common findings. This is a new contiguous gene syndrome. Lux et al. [1990: Nature 345:736-739] established that ankyrin deficiency and associated deficiencies of spectrin and protein 4.2 were responsible for spherocytosis in this syndrome. We reviewed the manifestations of this syndrome. Patients with spherocytic anemia and multiple congenital anomalies should be investigated by high-resolution chromosomal means to differentiate this syndrome.

摘要

我们描述了一名患有多种异常和智力发育迟缓并伴有遗传性球形红细胞增多症的30个月大男孩。他的核型为46,XY,del(8)(p11.23p21.1)。锚蛋白和谷胱甘肽还原酶(GSR)基因分别定位于染色体区域8p11.2和8p21.1。已有6例球形红细胞增多症和8p-间质缺失的患者被报道。在这些患者中,严重智力发育迟缓和多种异常是常见表现。这是一种新的相邻基因综合征。勒克斯等人[1990年:《自然》345:736 - 739]证实,锚蛋白缺乏以及相关的血影蛋白和蛋白4.2缺乏是该综合征中球形红细胞增多症的病因。我们回顾了该综合征的表现。对于患有球形红细胞增多症和多种先天性异常的患者,应通过高分辨率染色体检查手段进行调查,以鉴别该综合征。

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