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一个家族的个体表现出巨大的表型变异,该家族与一种新型与小头畸形、癫痫以及行为和认知缺陷相关的突变有关。

Large Phenotypic Variation of Individuals from a Family with a Novel Mutation Associated with Microcephaly, Epilepsy, and Behavioral and Cognitive Deficits.

机构信息

Department of Epileptology, University Hospital Bonn, 53127 Bonn, Germany.

German Center for Neurodegenerative Diseases (DZNE), 53127 Bonn, Germany.

出版信息

Genes (Basel). 2022 Feb 25;13(3):429. doi: 10.3390/genes13030429.

Abstract

Here, we report a consanguineous family harboring a novel homozygous frame-shift mutation in leading to a truncation of the ASPM protein after amino acid position 1830. The phenotype of the patients was associated with microcephaly, epilepsy, and behavioral and cognitive deficits. Despite the obvious genetic similarity, the affected patients show a considerable phenotypic heterogeneity regarding the degree of mental retardation, presence of epilepsy and MRI findings. Interestingly, the degree of mental retardation and the presence of epilepsy correlates well with the severity of abnormalities detected in brain MRI. On the other hand, we detected no evidence for substantial nonsense-mediated transcript decay in blood samples. This indicates that other factors than ASPM expression levels are relevant for the variability of structural changes in brain morphology seen in patients with primary hereditary microcephaly caused by mutations.

摘要

在这里,我们报道了一个近亲家族,该家族携带有一个新的纯合框移突变,导致 ASPM 蛋白在第 1830 位氨基酸后截短。患者的表型与小头畸形、癫痫以及行为和认知缺陷有关。尽管存在明显的遗传相似性,但受影响的患者在智力障碍程度、癫痫发作和 MRI 发现方面表现出相当大的表型异质性。有趣的是,智力障碍的程度和癫痫发作与脑 MRI 检测到的异常严重程度密切相关。另一方面,我们在血液样本中未检测到大量无意义介导的转录物衰减的证据。这表明,除了 ASPM 表达水平之外,其他因素对于由 突变引起的原发性遗传性小头畸形患者的脑形态结构变化的可变性也很重要。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8b11/8956106/ead30aab6f6e/genes-13-00429-g001.jpg

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