Moon Ja Un, Yum Sook Kyung
Department of Pediatrics, Yeouido St. Mary's Hospital, College of Medicine, The Catholic University of Korea, Seoul 07345, Republic of Korea.
Department of Pediatrics, Seoul St. Mary's Hospital, College of Medicine, The Catholic University of Korea, Seoul 06591, Republic of Korea.
Children (Basel). 2023 Aug 27;10(9):1461. doi: 10.3390/children10091461.
Partial deletion of the long arm (q) in chromosome 21 is an extremely rare condition with various phenotypes, including microcephaly, neurodevelopmental delay, dysmorphic features, and epileptic seizures. Neonatal hypoxic-ischemic encephalopathy (HIE) is an encephalopathy associated with a hypoxic-ischemic event in the brain where seizures usually occur in the earliest days of life. Neonatal encephalopathy is a distinct entity resulting from metabolic disorders, congenital infections or genetic abnormalities that could often mimic HIE features, leading to a misdiagnosis of HIE. Here, we present a case of a newborn who was initially misdiagnosed with HIE due to HIE-like features, and eventually was diagnosed to have a de novo ring chromosome 21 with 21q microdeletion. Clinical findings, including severe hypotonia with respiratory/feeding difficulties and intractable seizures, and radiologic findings of ischemic encephalopathy were discovered. Subsequent atypical findings of the clinical presentation ultimately led to her undergoing genetic testing confirming that she had a neonatal encephalopathy with a genetic abnormality. Our case highlights the importance of identifying non-HI neonatal encephalopathy by careful and structured evaluation for current history with a clinical course and a multidisciplinary approach including genetic testing, to provide an accurate diagnosis, treat curable inherited disorders, and develop future genetic counseling.
21号染色体长臂(q)部分缺失是一种极为罕见的病症,具有多种表型,包括小头畸形、神经发育迟缓、畸形特征和癫痫发作。新生儿缺氧缺血性脑病(HIE)是一种与脑部缺氧缺血事件相关的脑病,癫痫发作通常发生在生命的最初几天。新生儿脑病是一种由代谢紊乱、先天性感染或遗传异常引起的独特病症,常常会模仿HIE的特征,导致HIE的误诊。在此,我们报告一例新生儿病例,该患儿最初因类似HIE的特征被误诊为HIE,最终被诊断为新发的21号环状染色体伴21q微缺失。发现了包括严重肌张力低下伴呼吸/喂养困难和顽固性癫痫发作等临床症状,以及缺血性脑病的影像学表现。随后临床表现出现的非典型特征最终促使她接受基因检测,证实她患有伴有遗传异常的新生儿脑病。我们的病例强调了通过对当前病史、临床病程进行仔细且结构化的评估,并采用包括基因检测在内的多学科方法来识别非HI新生儿脑病的重要性,以便提供准确的诊断、治疗可治愈的遗传性疾病并开展未来的遗传咨询。