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另一位患者存在 NFIB 致病性无义变异,表现为畸形、自闭症谱系障碍、胼胝体发育不全和智力障碍。

Pathogenic nonsense variant in NFIB in another patient with dysmorphism, Autism Spectrum Disorder, agenesis of the corpus callosum, and intellectual disability.

机构信息

Hunter New England Local Health District, Hunter Genetics, Cnr. Turton & Tinonee Rds, Waratah, NSW, AUS 2298, Australia; University of Sydney, Faculty of Medicine and Health, Edward Ford Building (A27), Fisher Road, Camperdown, NSW, AUS 2006, Australia.

Hunter New England Local Health District, Hunter Genetics, Cnr. Turton & Tinonee Rds, Waratah, NSW, AUS 2298, Australia; University of Newcastle, Callaghan, NSW, AUS 2308, Australia.

出版信息

Eur J Med Genet. 2020 Dec;63(12):104092. doi: 10.1016/j.ejmg.2020.104092. Epub 2020 Oct 28.

Abstract

The Nuclear Factor I (NFI) transcription family (NFIA, NFIB and NFIX) have been implicated in a range of developmental pathologies, including corpus callosum, craniofacial, urinary tract abnormalities, as well in the development of a number of neurodevelopmental developmental phenotypes including muscular hypotonia, motor and speech delay, attention deficit disorder, autism spectrum disorder, and behavioural abnormalities. NFIB haploinsufficiency has only recently been presented as a cause for macrocephaly-intellectual disability syndrome, with comparable phenotypes to NFIA related disorder. We add another patient with a previously reported nonsense variant in the NFIB who has Autism Spectrum Disorder level 2, agenesis of the corpus callosum, ADHD, obsessive compulsive Disorder and an intellectual disability. A clinical exome analysis identified a nonsense variant, c.265C > T, p.(Arg89*) involving exon 2 of NFIB (ClinVar variation ID: 424,344). A brain MRI demonstrated agenesis of the corpus callosum.

摘要

核因子 I(NFI)转录家族(NFIA、NFIB 和 NFIX)与多种发育病理学有关,包括胼胝体、颅面、泌尿道异常,以及许多神经发育发育表型的发展,包括肌肉张力减退、运动和言语延迟、注意力缺陷障碍、自闭症谱系障碍和行为异常。NFIB 单倍不足最近才被提出作为巨脑-智力障碍综合征的原因,与 NFIA 相关疾病具有可比的表型。我们添加了另一位患者,该患者在 NFIB 中存在先前报道的无意义变异,患有自闭症谱系障碍 2 级、胼胝体发育不全、ADHD、强迫症和智力障碍。临床外显子组分析确定了一个无意义变异 c.265C>T,p.(Arg89*),涉及 NFIB 的外显子 2(ClinVar 变异 ID:424,344)。脑部 MRI 显示胼胝体发育不全。

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