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一名患有新发16p13.3 - 13.2间质性缺失男孩的复杂表型

Complex Phenotype of a Boy With De Novo 16p13.3-13.2 Interstitial Deletion.

作者信息

Milone Roberta, Ferrari Anna Rita, Pasquariello Rosa, Bargagna Stefania

机构信息

Department of Developmental Neuroscience, IRCCS Fondazione Stella Maris, Pisa, Italy.

Department of Clinical and Experimental Medicine, University of Pisa, Via Roma 67, Pisa, Italy.

出版信息

Child Neurol Open. 2016 Dec 16;3:2329048X16676153. doi: 10.1177/2329048X16676153. eCollection 2016 Jan-Dec.

Abstract

Interstitial deletions encompassing chromosome 16p13.3-13.2 are rarely described in the literature, whereas terminal deletions or duplications involving this region are slightly more frequently described. The authors describe a boy harboring a de novo 16p13.3-13.2 interstitial deletion, with intellectual disability, verbal dyspraxia, epilepsy, and a distinctive brain magnetic resonance finding, namely a nodular heterotopia. The authors found partial genotype-phenotype correspondences regarding epilepsy and intellectual disability, which have been associated with 16p1 region. Conversely, nodular heterotopia and verbal dyspraxia have not been clearly related to this region. These data are in agreement with the emerging concept that similar copy number variants may be the general risk factors for distinct disorders. Verbal dyspraxia, which has not responded to speech therapy, is the child's most disabling trait. In view of the above, genetic studies should be appraised in cases of serious speech difficulties, especially if they are associated with intellectual disability and epilepsy.

摘要

文献中很少描述包含染色体16p13.3 - 13.2的间质性缺失,而涉及该区域的末端缺失或重复的描述则略为常见。作者描述了一名患有新发16p13.3 - 13.2间质性缺失的男孩,伴有智力障碍、言语失用症、癫痫以及独特的脑磁共振成像表现,即结节性异位。作者发现了与癫痫和智力障碍相关的部分基因型 - 表型对应关系,这些与16p1区域有关。相反,结节性异位和言语失用症与该区域的关系尚不明确。这些数据与新出现的概念一致,即相似的拷贝数变异可能是不同疾病的常见风险因素。言语失用症对言语治疗无反应,是该患儿最致残的特征。鉴于上述情况,对于严重言语困难的病例,尤其是伴有智力障碍和癫痫的病例,应进行基因研究评估。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b83d/5417293/22cbbcf34fcc/10.1177_2329048X16676153-fig1.jpg

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