Leigh syndrome in a patient with a novel C12orf65 pathogenic variant: case report and literature review
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 phosphor...
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Veröffentlicht in: | Genetics and molecular biology 2020-01, Vol.43 (2), p.e20180271-e20180271 |
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Sprache: | eng |
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Zusammenfassung: | 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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ISSN: | 1415-4757 1678-4685 1678-4685 |
DOI: | 10.1590/1678-4685-gmb-2018-0271 |