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五位新的 WAC 相关智力障碍个体的临床和分子特征:一种新型剪接变异体的致病性证据。

Clinical and molecular characterization of five new individuals with WAC-related intellectual disability: Evidence of pathogenicity for a novel splicing variant.

机构信息

Department of Pediatrics, Medical Genetics Division, Stanford University, Stanford, California, USA.

Department of Clinical and Molecular Genetics, University Hospital Vall d'Hebron, Barcelona, Spain.

出版信息

Am J Med Genet A. 2022 May;188(5):1396-1406. doi: 10.1002/ajmg.a.62648. Epub 2022 Jan 12.

Abstract

WAC-related intellectual disability (ID) is a rare genetic condition characterized by a spectrum of neurodevelopmental disorders of varying severity, including global developmental delay (GDD), ID, and autism spectrum disorder. Here, we describe five affected individuals, age range 9-20 years, and provide proof of pathogenicity of a novel splicing variant. All individuals presented with GDD, some degree of ID, and variable dysmorphism. Except for feeding difficulties, all patients were healthy without major congenital malformations or medical comorbidities. All individuals were heterozygous for de novo, previously unreported, loss of function variants in WAC. Three unrelated patients from different ethnic backgrounds shared the intronic variant c.381+4_381+7delAGTA, which was predicted to alter splicing and was initially classified as a variant of uncertain significance. Reverse transcription-polymerase chain reaction analysis from one patient's cells confirmed aberrant splicing of the WAC transcript resulting in premature termination and a truncated protein p.(Gly92Alafs*2). These functional studies and the identification of several nonrelated individuals provide sufficient evidence to classify this variant as pathogenic. The clinical description of these five individuals and the three novel variants expand the genotypic and phenotypic spectrum of this ultrarare disease.

摘要

WAC 相关智力障碍(ID)是一种罕见的遗传性疾病,其特征是一系列严重程度不同的神经发育障碍,包括全面发育迟缓(GDD)、智力障碍和自闭症谱系障碍。在这里,我们描述了五名受影响的个体,年龄在 9-20 岁之间,并提供了一种新型剪接变异的致病性证据。所有个体均表现为 GDD、不同程度的智力障碍和不同程度的畸形。除了喂养困难外,所有患者均健康,无重大先天性畸形或合并症。所有个体均为杂合子,存在新发现的、以前未报道的 WAC 功能丧失变异。来自不同种族背景的三位无关患者共享内含子变异 c.381+4_381+7delAGTA,该变异预计会改变剪接,并最初被归类为意义不明的变异。对一名患者细胞的逆转录-聚合酶链反应分析证实了 WAC 转录本的异常剪接,导致提前终止和截短的蛋白 p.(Gly92Alafs*2)。这些功能研究和多个非相关个体的鉴定提供了足够的证据将该变异归类为致病性变异。这五名个体的临床描述和三个新变异扩展了这种超罕见疾病的基因型和表型谱。

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