巴基斯坦新纯合TTI2变异导致常染色体隐性综合征智力残疾和原发性小头畸形:1例报告(外显子组报告)。

Case Reports in Genetics Pub Date : 2022-08-12 eCollection Date: 2022-01-01 DOI:10.1155/2022/2766957
Zul Qarnain, Fatima Khan, Fizza Akbar, Salman Kirmani
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引用次数: 0

摘要

我们描述了一个男性患者与新的TTI2变异,这是以前没有与人类表型相关。其特征包括智力残疾、原发性小头畸形、精神运动发育迟缓、言语发育迟缓、身材矮小、面部畸形、内斜视、脊柱后凸和行为异常(图)。下一代测序显示常染色体隐性TTI2变异,意义不确定,记为c.21_22insAAGCGCTCTG (p.Glu8Lysfs × 12)。TTI2编码DNA损伤反应的调节因子,并帮助维持PIKK蛋白激酶家族的稳定水平。没有发现其他可能与他的临床表现有关的致病基因变异。我们报告了一种新的TTI2功能丧失纯合变异,可导致综合征性智力残疾和原发性小头畸形。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

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Novel Homozygous TTI2 Variant Causing Autosomal Recessive Syndromic Intellectual Disability and Primary Microcephaly from Pakistan: A Case Report (Exome Report).

We describe a male patient with a novel TTI2 variant, which has not been previously associated with a human phenotype. His features include intellectual disability, primary microcephaly, delayed psychomotor development, speech delay, short stature, dysmorphic facial features, esotropia, kyphoscoliosis, and behavior abnormalities (Figure). Next generation sequencing revealed autosomal recessive TTI2 variant with uncertain significance, denoted as c.21_22insAAGCGCTCTG (p.Glu8Lysfs × 12). TTI2 encodes a regulator of DNA damage response and helps maintain steady levels of the PIKK family of protein kinases. No disease-causing variants in other genes potentially linked to his clinical presentation were identified. We report a novel loss-of-function homozygous variant in TTI2 that leads to syndromic intellectual disability and primary microcephaly.

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