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进一步描述 White-Sutton 综合征的临床谱:12 例新病例及文献复习。

Further delineation of the clinical spectrum of White-Sutton syndrome: 12 new individuals and a review of the literature.

机构信息

Institute of Medical Genetics, University Hospital of Wales, Cardiff, UK.

Department of Medical Genetics, Haukeland University Hospital, Bergen, Norway.

出版信息

Eur J Hum Genet. 2022 Jan;30(1):95-100. doi: 10.1038/s41431-021-00961-3. Epub 2021 Oct 14.

Abstract

White-Sutton syndrome (WHSUS) is a neurodevelopmental disorder caused by heterozygous loss-of-function variants in POGZ. Through the Deciphering Developmental Disorders study and clinical testing, we identified 12 individuals from 10 families with pathogenic or likely pathogenic variants in POGZ (eight de novo and two inherited). Most individuals had delayed development and/or intellectual disability. We analyzed the clinical findings in our series and combined it with data from 89 previously reported individuals. The results demonstrate WHSUS is associated with variable developmental delay or intellectual disability, increased risk of obesity, visual defects, craniofacial dysmorphism, sensorineural hearing loss, feeding problems, seizures, and structural brain malformations. Our series includes further individuals with rod-cone dystrophy, cleft lip and palate, congenital diaphragmatic hernia, and duplicated renal drainage system, suggesting these are rare complications of WHSUS. In addition, we describe an individual with a novel, de novo missense variant in POGZ and features of WHSUS. Our work further delineates the phenotypic spectrum of WHSUS highlighting the variable severity of this disorder and the observation of familial pathogenic POGZ variants.

摘要

White-Sutton 综合征(WHSUS)是一种神经发育障碍,由 POGZ 杂合功能丧失变异引起。通过解析发育障碍研究和临床测试,我们在 10 个家族中发现了 12 名个体,他们携带 POGZ 的致病性或可能致病性变异(8 个新发和 2 个遗传)。大多数个体存在发育迟缓或智力残疾。我们分析了我们系列中的临床发现,并将其与之前报道的 89 名个体的数据相结合。结果表明,WHSUS 与可变的发育迟缓或智力残疾、肥胖风险增加、视觉缺陷、颅面畸形、感觉神经性听力损失、喂养问题、癫痫发作和结构性脑畸形有关。我们的系列还包括更多患有杆状 - 圆锥营养不良、唇腭裂、先天性膈疝和重复的肾脏引流系统的个体,表明这些是 WHSUS 的罕见并发症。此外,我们描述了一名患有 POGZ 新型、新发错义变异的个体,其具有 WHSUS 的特征。我们的工作进一步描绘了 WHSUS 的表型谱,突出了这种疾病的可变严重程度以及家族性致病性 POGZ 变异的观察。

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