Division of Human Genetics, Children's Hospital of Philadelphia, Pennsylvania, Pennsylvania, USA.
Department of Radiology, Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, USA.
Am J Med Genet A. 2023 Oct;191(10):2602-2609. doi: 10.1002/ajmg.a.63230. Epub 2023 May 9.
Eukaryotic translation elongation factor 2 (eEF2), encoded by the gene EEF2, is an essential factor involved in the elongation phase of protein translation. A specific heterozygous missense variant (p.P596H) in EEF2 was originally identified in association with autosomal dominant adult-onset spinocerebellar ataxia-26 (SCA26). More recently, additional heterozygous missense variants in this gene have been described to cause a novel, childhood-onset neurodevelopmental disorder with benign external hydrocephalus. Herein, we report two unrelated individuals with a similar gene-disease correlation to support this latter observation. Patient 1 is a 7-year-old male with a previously reported, de novo missense variant (p.V28M) who has motor and speech delay, autism spectrum disorder, failure to thrive with relative macrocephaly, unilateral microphthalmia with coloboma and eczema. Patient 2 is a 4-year-old female with a novel de novo nonsense variant (p.Q145X) with motor and speech delay, hypotonia, macrocephaly with benign ventricular enlargement, and keratosis pilaris. These additional cases help to further expand the genotypic and phenotypic spectrum of this newly described EEF2-related neurodevelopmental syndrome.
真核翻译延伸因子 2(eEF2),由 EEF2 基因编码,是参与蛋白质翻译延伸阶段的必需因子。EEF2 中的一个特定杂合错义变异(p.P596H)最初与常染色体显性成人发病的脊髓小脑共济失调 26 型(SCA26)相关联。最近,该基因中的其他杂合错义变异也被描述为导致一种新的、儿童发病的神经发育障碍,并伴有良性外部脑积水。在此,我们报告了两个无关联的个体,他们具有相似的基因-疾病相关性,以支持这一观察结果。患者 1 是一名 7 岁男性,携带先前报道的、新生的错义变异(p.V28M),具有运动和言语延迟、自闭症谱系障碍、生长不良伴相对大头畸形、单侧小眼球伴视网膜色素上皮脱离和湿疹。患者 2 是一名 4 岁女性,携带新的、新生的无义变异(p.Q145X),具有运动和言语延迟、肌张力低下、大头畸形伴良性脑室扩大和毛发角化病。这些额外的病例有助于进一步扩大该新描述的 EEF2 相关神经发育综合征的基因型和表型谱。