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TASP1 在一名发育迟缓、小头畸形、特征性面部特征和多种先天性异常的婴儿中缺失。

TASP1 is deleted in an infant with developmental delay, microcephaly, distinctive facial features, and multiple congenital anomalies.

机构信息

Division of Neurology, Department of Pediatrics, Tawam Hospital, and Department of Pediatrics, College of Medicine and Health Sciences, United Arab Emirates University, Al Ain, United Arab Emirates.

PreventionGenetics, LLC, Marshfield, Wisconsin.

出版信息

Clin Genet. 2018 Jul;94(1):170-173. doi: 10.1111/cge.13258. Epub 2018 May 10.

DOI:10.1111/cge.13258
PMID:29633245
Abstract

We report a 20p12.1 homozygous deletion including exons 5-10 of the TASP1 gene in an infant with developmental delay, acquired microcephaly, distinctive facial features, and multiple congenital anomalies involving skeletal, cardiac, and renal systems. TASP1 encodes taspase 1 which is responsible for cleaving, thus activating, a number of transcription factors including the mixed lineage leukemia 1 (MLL1). Taspase 1-deficient mice showed early lethality, skeletal abnormalities, and growth failure, which support a potentially causal role of TASP1 deletion in this infant. Furthermore, the infant reported here had many of the features seen in Wiedemann-Steiner syndrome which is caused by MLL1 defects. Such observation further supports that TASP1 is a novel disease-related gene that is associated with a disease phenotype overlapping with Wiedemann-Steiner syndrome as both are caused by defects in the same pathway.

摘要

我们报道了一名患有发育迟缓、获得性小头畸形、特征性面部特征和多种先天性畸形(涉及骨骼、心脏和肾脏系统)的婴儿,其 TASP1 基因 20p12.1 纯合缺失,包括外显子 5-10。TASP1 编码 taspase 1,负责切割,从而激活许多转录因子,包括混合谱系白血病 1(MLL1)。Taspase 1 缺陷小鼠表现出早期致死性、骨骼异常和生长衰竭,这支持 TASP1 缺失在该婴儿中的潜在因果作用。此外,这里报道的婴儿具有 Wiedemann-Steiner 综合征的许多特征,该综合征是由 MLL1 缺陷引起的。这种观察结果进一步支持 TASP1 是一种新的疾病相关基因,与 Wiedemann-Steiner 综合征的疾病表型重叠,因为两者都是由同一途径的缺陷引起的。

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