描述与16p13.11重复相关的终生多发病:文献回顾、荟萃分析和案例研究

IF 3.9 2区 医学 Q1 CLINICAL NEUROLOGY
Rose Mary Xavier, Wenxin Bian, Fadhah Alshammari, Matthew K Harner, Tyler E Dietterich, Maya Lichtenstein, Robert Stowe, Martilias Farrell, Jin P Szatkiewicz, Rita A Shaughnessy, Jonathan S Berg, Patrick F Sullivan, Richard C Josiassen
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引用次数: 0

摘要

16p13.11重复是一种罕见的拷贝数变异(CNV),在整个生命周期中似乎会增加多病的风险。最初的报告暗示了自闭症谱系障碍、智力残疾和各种先天性异常的重复;然而,在“表型正常”的个体中也观察到不完全外显或无致病性。最近的研究表明,复制携带者经常出现多重发病,但需要更多的数据来阐明相关表型的全部范围。终生多发病尚不清楚,目前也没有文献综述。我们对已发表的表型特征进行了系统的文献回顾和荟萃分析。语言迟缓、发育迟缓、智力残疾、学习障碍和自闭症症状在bbbb30 %的病例中报告。肌肉骨骼异常和心血管疾病是常见的报道。其中包括一名71岁女性的终身病例报告,她有行为障碍和治疗难治性精神分裂症的病史,被确定为16p13.11复制携带者。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Delineating lifetime multimorbidity associated with 16p13.11 duplication: A literature review, meta-analysis, and case study.

The 16p13.11 duplication is a rare copy number variation (CNV) that appears to increase risk for multimorbidity across the life span. Initial reports implicated the duplication in autism spectrum disorder, intellectual disability, and various congenital anomalies; however, it was also observed in "phenotypically normal" individuals suggesting incomplete penetrance or non-pathogenicity. Recent studies suggest that duplication carriers often present with multimorbidity, but more data are needed to elucidate the full range of associated phenotypes. Lifetime multimorbidity remains unclear, and no reviews summarizing this literature currently exist. We report a systematic literature review and meta-analysis of published phenotypic characteristics. Speech delays, developmental delays, intellectual disability, learning disability, and autistic symptoms were reported in >30 % of cases. Musculoskeletal abnormalities and cardiovascular disorders were commonly reported. Included is a lifespan case report of a 71-year-old female with a history of behavioral disturbance and treatment-resistant schizophrenia, identified as being a 16p13.11 duplication carrier.

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来源期刊
CiteScore
12.00
自引率
1.80%
发文量
153
审稿时长
56 days
期刊介绍: Progress in Neuro-Psychopharmacology & Biological Psychiatry is an international and multidisciplinary journal which aims to ensure the rapid publication of authoritative reviews and research papers dealing with experimental and clinical aspects of neuro-psychopharmacology and biological psychiatry. Issues of the journal are regularly devoted wholly in or in part to a topical subject. Progress in Neuro-Psychopharmacology & Biological Psychiatry does not publish work on the actions of biological extracts unless the pharmacological active molecular substrate and/or specific receptor binding properties of the extract compounds are elucidated.
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