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22q11.2 重复:临床表现的扩展

22q11.2 duplications: Expanding the clinical presentation.

作者信息

Bartik Lauren E, Hughes Susan S, Tracy Meghan, Feldt M Max, Zhang Lei, Arganbright Jill, Kaye Alison

机构信息

Division of Clinical Genetics, Children's Mercy Hospital, Kansas City, Missouri, USA.

University of Missouri-Kansas City School of Medicine, Kansas City, Missouri, USA.

出版信息

Am J Med Genet A. 2022 Mar;188(3):779-787. doi: 10.1002/ajmg.a.62577. Epub 2021 Nov 29.

DOI:10.1002/ajmg.a.62577
PMID:34845825
Abstract

22q11.2 duplication syndrome has a frequency of ~1/700 in the intellectual disability population. Despite this frequency, there is limited information on the variable clinical presentation. Although the phenotype and incidence of congenital anomalies are well described for 22q11.2 deletion syndrome, they are not as well understood for individuals with 22q11.2 duplication syndrome. This study is a single-center, retrospective review of patients diagnosed with 22q11.2 duplication syndrome designed to categorize the variable phenotype seen in these individuals. The data suggest that the incidence of congenital anomalies may be higher than previously reported for this syndrome. Affected individuals are at increased risk for a variety of problems including gastrointestinal complications, endocrine dysfunction, ophthalmologic abnormalities, palatal anomalies, congenital heart disease, musculoskeletal differences, and neurologic abnormalities. Individuals with 22q11.2 duplication syndrome would benefit from care coordinated by a multidisciplinary team and managed according to the 22q11.2 deletion syndrome guidelines.

摘要

22q11.2重复综合征在智力残疾人群中的发生率约为1/700。尽管有这样的发生率,但关于其临床表现多样性的信息有限。虽然22q11.2缺失综合征的表型和先天性异常的发生率已有充分描述,但对于22q11.2重复综合征患者,人们对其了解并不多。本研究是一项针对诊断为22q11.2重复综合征患者的单中心回顾性研究,旨在对这些个体中观察到的可变表型进行分类。数据表明,先天性异常的发生率可能高于该综合征之前报告的发生率。受影响个体面临多种问题的风险增加,包括胃肠道并发症、内分泌功能障碍、眼科异常、腭裂、先天性心脏病、肌肉骨骼差异和神经异常。22q11.2重复综合征患者将受益于多学科团队协调的护理,并根据22q11.2缺失综合征指南进行管理。

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