Mahpara Hasan, Gayatra Mainali, Ermal Aliu, Sita Paudel
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引用次数: 0
摘要
常染色体隐性智力发育障碍5型(MRT5, OMIM # 611091)是由双等位基因致病变异引起的,导致NSUN2基因功能丧失,该基因编码一种参与从应激反应到神经发育等多种生物过程的甲基转移酶(Hussain 2021)。目前的文献显示,MRT5通常表现为智力残疾、面部畸形、青少年白内障、慢性肾炎、听力障碍、癫痫发作、小脑萎缩和小头畸形(Pingree et al. 2021)。我们描述了一个MRT5患者在青少年时期发展为癫痫的病例,这是一种罕见的临床表现,尚未在文献中进行详细讨论。我们的患者是一名15岁的男性,有自闭症、发育迟缓和局灶性癫痫的病史,他进行了基因检测,发现NSUN2有一个纯合子移码突变,预计会导致功能丧失。本病例强调癫痫可能是MRT5患者的一种表型表现。
A Case of Autosomal Recessive Intellectual Developmental Disorder Type 5 Presenting with Epilepsy.
Autosomal recessive intellectual developmental disorder type 5 (MRT5, OMIM # 611091) is caused by biallelic pathogenic variants, leading to loss of function of the NSUN2 gene which encodes a methyltransferase involved in several biological processes, ranging from stress response to neurodevelopment (Hussain 2021). The current literature shows that MRT5 typically manifests with intellectual disability, facial dysmorphism, juvenile cataracts, chronic nephritis, hearing impairment, seizures, cerebellar atrophy, and microcephaly (Pingree et al. 2021). We describe a case of a patient with MRT5 who developed epilepsy in his teens, a rare clinical presentation that has not yet been discussed at length in the literature. Our patient is a 15-year-old male with a history of autism, developmental delay, and focal epilepsy who underwent genetic testing and was found to have a homozygous frameshift mutation in NSUN2 predicted to cause loss of function. This case emphasizes that epilepsy can be a phenotypic manifestation in patients with MRT5.