Frontiers in Neurology · 2021 · 21 citations · 22 references
Life SciencesGeneticsMolecular GeneticsHuman GeneticsMitochondrial BiologyMitochondrial GeneticsGenetic DisordersHereditary NeuropathiesMitochondrial DiseaseMitochondrial DisordersMitochondrial InheritanceNeurogenetic DisordersInherited Optic NeuropathiesLeber Hereditary Optic NeuropathyMitochondrial Dna MutationsMitochondrial Dna Point MutationsMitochondrial Gene VariantsPoint MutationsRare Genetic VariantsTissue Specificity Determinants
Leber's hereditary optic neuropathy (LHON) is due to missense point mutations affecting mitochondrial DNA (mtDNA); 90% of cases harbor the m.3460G>A, m.11778G>A, and m.14484T>C primary mutations. Here, we report and discuss five families with patients affected by symptomatic LHON, in which we found five novel mtDNA variants. Remarkably, these mtDNA variants are located in complex I genes, though without strong deleterious effect on respiration in cellular models: this finding is likely linked to the tissue specificity of LHON. This study observes that in the case of a strong clinical suspicion of LHON, it is recommended to analyze the whole mtDNA sequence, since new rare mtDNA pathogenic variants causing LHON are increasingly identified.
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Gene–environment interactions in Leber hereditary optic neuropathy
Matthew A. Kirkman, Patrick Yu‐Wai‐Man, Alex Korsten et al. · Brain · 2009 · 355 citations · Full text