Literature DB >> 28139165

Leber hereditary optic neuropathy due to a new ND1 mutation.

Patrick Soldath1,2, Marianne Wegener1,3, Birgit Sander3, Thomas Rosenberg1,3, Morten Duno4, Flemming Wibrand4, John Vissing1,2.   

Abstract

We report a proband with Leber hereditary optic neuropathy (LHON), in whom we have identified a novel homoplasmic m.3,395A>G mutation in the ND1 gene. The mutation alters a highly conserved amino acid in codon 30 which previously has been associated with LHON and leads to a severe selective complex I deficiency. By providing further evidence for pathogenicity we conclude that m.3,395A>G is pathogenic. High definition optical coherence tomography of the retina and peripapillary retinal nerve fiber layer (pRNFL) confirms recent reports that retinal ganglion cell loss precedes axonal loss in LHON and is present in the early stage of the disease. Furthermore, evaluation of two unaffected mutation carriers disclosed asymptomatic borderline ganglion cell loss and thin pRNFL in one.

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Keywords:  Complex I deficiency; LHON; ND1 gene; leber hereditary optic neuropathy; mitochondrial DNA; optical coherence tomography; retinal ganglion cell; retinal nerve fiber layer

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Year:  2017        PMID: 28139165     DOI: 10.1080/13816810.2016.1253108

Source DB:  PubMed          Journal:  Ophthalmic Genet        ISSN: 1381-6810            Impact factor:   1.803


  1 in total

1.  Foveal pit morphological changes in asymptomatic carriers of the G11778A mutation with Leber's hereditary optic neuropathy.

Authors:  Xin-Ting Liu; Mei-Xiao Shen; Chong Chen; Sheng-Hai Huang; Xi-Ran Zhuang; Qing-Kai Ma; Qi Chen; Fan Lu; Yi-Min Yuan
Journal:  Int J Ophthalmol       Date:  2020-05-18       Impact factor: 1.779

  1 in total

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