Kilic Mehmet Akif, Kipoglu Osman, Coskun Orhan, Karacabey Burcin Nazli, Yesilyurt Ahmet, Yildiz Edibe Pembegul, Aydinli Nur, Caliskan Meliha Mine
Department of Pediatric Neurology, Istanbul Medical Faculty, Turkey.
Department of Pediatric Neurology, Istanbul Medical Faculty, Turkey.
Brain Dev. 2021 Nov;43(10):1039-1043. doi: 10.1016/j.braindev.2021.06.001. Epub 2021 Jun 16.
In recent years, with advances in molecular genetics, many new mutations with various ataxic syndromes have been identified. Recently, homozygous sequestosome 1 (SQSTM1) gene variant with a progressive childhood-onset cerebellar ataxia, dystonia and gaze palsy was described. Here we describe a patient with progressive cerebellar ataxia and gaze palsy, as well as myoclonus, cognitive impairment and growth retardation with a homozygous SQSTM1 variant NM_003900.5:c.55G > T (p.Glu19*). Our case had brainstem lesions on brain magnetic resonance imaging that have not been previously reported. This novel finding expands the SQSTM1 gene-associated neuroradiologic spectrum. Homozygous SQSTM1 variant should be considered in the differential diagnosis in patients presenting with cerebellar findings, gaze palsy, and cognitive impairment to facilitate early diagnosis and genetic counseling.
近年来,随着分子遗传学的进展,许多与各种共济失调综合征相关的新突变已被鉴定出来。最近,有人描述了一种纯合的聚集体小体1(SQSTM1)基因变异,其与儿童期起病的进行性小脑共济失调、肌张力障碍和凝视麻痹有关。在此,我们描述了一名患有进行性小脑共济失调和凝视麻痹,以及肌阵挛、认知障碍和生长发育迟缓的患者,其携带纯合的SQSTM1变异NM_003900.5:c.55G>T(p.Glu19*)。我们的病例在脑磁共振成像上有脑干病变,这在以前尚未见报道。这一新发现扩展了与SQSTM1基因相关的神经放射学谱。对于出现小脑病变、凝视麻痹和认知障碍的患者,在鉴别诊断中应考虑纯合的SQSTM1变异,以促进早期诊断和遗传咨询。