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与18号环状染色体相关的自身免疫性多内分泌病

Autoimmune polyendocrinopathy associated with ring chromosome 18.

作者信息

Jain Nina, Reitnauer Pamela J, Rao Kathleen W, Aylsworth Arthur S, Calikoglu Ali S

机构信息

Department of Pediatrics, Division of Pediatric Endocrinology, CB#7039, 3341 MBRB, University of North Carolina, Chapel Hill, NC 27599-703, USA.

出版信息

J Pediatr Endocrinol Metab. 2011;24(9-10):847-50. doi: 10.1515/jpem.2011.320.

Abstract

Phenotypic and clinical features of individuals with ring chromosome 18 [r(18)] vary with the extent of deletion of the short (18p-) or long arm (18q-). Most patients with r(18), therefore, demonstrate a clinical spectrum of both 18p- and 18q- deletions. Short stature, microcephaly, mental and motor retardation, craniofacial dysmorphism and extremity abnormalities are the most commonly reported features in patients with r(18). Abnormalities of chromosome 18, especially 18p- syndrome, are often reported with autoimmune thyroid disease and growth hormone deficiency, but reports of endocrine abnormalities associated with r(18) are rare. Here, we report a case of an African-American female with hyperthyroidism, type 1 diabetes mellitus, vitiligo and IgA deficiency associated with a r(18) chromosome complement. This patient additionally had mild intellectual disability and dysmorphic features. Karyotype analysis showed a de novo ring chromosome 18 (deletion 18q23-18qter and deletion 18p11.3-18pter). Although this unique association of autoimmune polyglandular endocrinopathy with ring chromosome 18 could be coincidental, we speculate that a gene or genes on chromosome 18 might play a role in the autoimmune process.

摘要

18号环状染色体[r(18)]个体的表型和临床特征因短臂(18p-)或长臂(18q-)缺失程度而异。因此,大多数r(18)患者表现出18p-和18q-缺失的临床谱。身材矮小、小头畸形、智力和运动发育迟缓、颅面畸形和肢体异常是r(18)患者最常报告的特征。18号染色体异常,尤其是18p-综合征,常与自身免疫性甲状腺疾病和生长激素缺乏有关,但与r(18)相关的内分泌异常报告很少。在此,我们报告一例非裔美国女性,患有甲状腺功能亢进、1型糖尿病、白癜风和IgA缺乏症,其染色体组成为r(18)。该患者还伴有轻度智力残疾和畸形特征。核型分析显示为新发的18号环状染色体(18q23-18q末端缺失和18p11.3-18p末端缺失)。虽然自身免疫性多腺体内分泌病与18号环状染色体的这种独特关联可能是巧合,但我们推测18号染色体上的一个或多个基因可能在自身免疫过程中起作用。

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