Pennsylvania State University College of Medicine, Hershey, Pennsylvania, USA.
Penn State Health Milton S. Hershey Medical Center, Hershey, Pennsylvania, USA.
Am J Med Genet A. 2022 Mar;188(3):896-899. doi: 10.1002/ajmg.a.62568. Epub 2021 Nov 19.
NDUFAF5 encodes a Complex I assembly factor which is critical to the modification of a core subunit, NDUFS7, in early Complex I factor assembly. Mutations in NDUFAF5 have been previously shown to cause Complex I deficiency leading to mitochondrial respiratory chain impairment. More than 15 individuals affected by variants in NDUFAF5 have been described; however, there is phenotypic heterogeneity within this cohort. Some individuals display features of classical Leigh syndrome with early onset neurodegeneration whereas others live into early adulthood with progressive neurological deficits. Here, we present a clinical report of a 17-year-old African American individual with compound heterozygous mutations in NDUFAF5. The individual presented with childhood onset bilateral optic atrophy and developed progressive neuromuscular decline with relatively preserved cognition over time.
NDUFAF5 编码一个复合体 I 组装因子,对于早期复合体 I 因子组装中核心亚基 NDUFS7 的修饰至关重要。先前的研究表明,NDUFAF5 中的突变会导致复合体 I 缺陷,从而导致线粒体呼吸链损伤。已有 15 多名受 NDUFAF5 变异影响的个体被描述;然而,在这一组中存在表型异质性。一些个体表现出经典 Leigh 综合征的特征,伴有早期神经退行性变,而另一些个体则在成年早期出现进行性神经功能缺损。在这里,我们报告了一个 17 岁的非洲裔美国人个体的临床病例,该个体存在 NDUFAF5 的复合杂合突变。该个体在儿童时期出现双侧视神经萎缩,并随着时间的推移逐渐出现进行性神经肌肉衰退,但认知相对保留。