生物
表型
基因
遗传学
线粒体DNA
氧化磷酸化
线粒体
核基因
疾病
粒线体疾病
生物信息学
病理
医学
生物化学
作者
Eduardo Perrone,Thiago R. Cavole,Manuella G. Oliveira,Luiza do Amaral Virmond,Marina de França B. Silva,Maria de Fátima Soares,Simone Iglesias,Ariane Falconi,Juliana Saragiotto Silva,Viviane Nakano,Maria Fernanda Grillo Milanezi,Carmen Mendes,Marco A. Curiati,Cecília Micheletti
标识
DOI:10.1590/1678-4685-gmb-2018-0271
摘要
Leigh syndrome is an early onset progressive disorder caused by defects in mitochondrial oxidative phosphorylation. Pathogenic variants in nuclear and mitochondrial genes are associated with the syndrome. Homozygous pathogenic variants in the C12orf65 gene impair the mitochondrial oxidative phosphorylation system. We describe a new case of Leigh syndrome caused by a novel pathogenic variant of the C12orf65 gene resulting in the lack of the Gly-Gly-Gln (GGQ) domain in the predicted protein, and review clinical and molecular data from previously reported patients. Our study supports that the phenotype caused by C12orf65 gene variants is heterogeneous and varies from spastic paraparesis to Leigh syndrome. Loss-of-function variants are more likely to cause the disease, and variants affecting the GGQ domain tend to be associated with more severe phenotypes, reinforcing a possible genotype-phenotype correlation.
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