Literature DB >> 33185731

Leber hereditary optic neuropathy-new insights and old challenges.

Srilekha Sundaramurthy1, Ambika SelvaKumar2, Jared Ching3,4, Vidhya Dharani2, Sripriya Sarangapani5, Patrick Yu-Wai-Man3,4,6.   

Abstract

Leber hereditary optic neuropathy (LHON) is the most common primary mitochondrial DNA (mtDNA) disorder with the majority of patients harboring one of three primary mtDNA point mutations, namely, m.3460G>A (MTND1), m.11778G>A (MTND4), and m.14484T>C (MTND6). LHON is characterized by bilateral subacute loss of vision due to the preferential loss of retinal ganglion cells (RGCs) within the inner retina, resulting in optic nerve degeneration. This review describes the clinical features associated with mtDNA LHON mutations and recent insights gained into the disease mechanisms contributing to RGC loss in this mitochondrial disorder. Although treatment options remain limited, LHON research has now entered an active translational phase with ongoing clinical trials, including gene therapy to correct the underlying pathogenic mtDNA mutation.

Entities:  

Keywords:  Gene therapy; Haplogroup; Heteroplasmy; LHON; Mitochondrial disease; Optic neuropathy

Year:  2020        PMID: 33185731     DOI: 10.1007/s00417-020-04993-1

Source DB:  PubMed          Journal:  Graefes Arch Clin Exp Ophthalmol        ISSN: 0721-832X            Impact factor:   3.117


  8 in total

1.  Varying Clinical Phenotypes of Mitochondrial DNA T12811C Mutation: A Case Series Report.

Authors:  Qingdan Xu; Ping Sun; Chaoyi Feng; Qian Chen; Xinghuai Sun; Yuhong Chen; Guohong Tian
Journal:  Front Med (Lausanne)       Date:  2022-07-04

2.  Brain Gray Matter Atrophy and Functional Connectivity Remodeling in Patients With Chronic LHON.

Authors:  Qin Tian; Ling Wang; Yu Zhang; Ke Fan; Meng Liang; Dapeng Shi; Wen Qin; Hao Ding
Journal:  Front Neurosci       Date:  2022-05-12       Impact factor: 5.152

3.  Prevalence of primary mutations in Leber hereditary optic neuropathy: A five-year report from a tertiary eye care center in India.

Authors:  Srilekha Sundaramurthy; Ambika Selvakumar; Vidhya Dharani; Nagasamy Soumittra; Jayaprakash Mani; Karthiyayini Thirumalai; Porkodi Periyasamy; Sinnakaruppan Mathavan; Sarangapani Sripriya
Journal:  Mol Vis       Date:  2021-12-11       Impact factor: 2.367

4.  A broad comparative genomics approach to understanding the pathogenicity of Complex I mutations.

Authors:  Galya V Klink; Hannah O'Keefe; Amrita Gogna; Georgii A Bazykin; Joanna L Elson
Journal:  Sci Rep       Date:  2021-10-01       Impact factor: 4.379

Review 5.  Clinical Overview of Leber Hereditary Optic Neuropathy.

Authors:  Almina Stramkauskaitė; Ieva Povilaitytė; Brigita Glebauskienė; Rasa Liutkevičienė
Journal:  Acta Med Litu       Date:  2022-06-29

Review 6.  Mitochondrial Impairment: A Common Motif in Neuropsychiatric Presentation? The Link to the Tryptophan-Kynurenine Metabolic System.

Authors:  Masaru Tanaka; Ágnes Szabó; Eleonóra Spekker; Helga Polyák; Fanni Tóth; László Vécsei
Journal:  Cells       Date:  2022-08-21       Impact factor: 7.666

7.  Toxic and nutritional factors trigger Leber hereditary optic neuropathy due to a mitochondrial tRNA mutation.

Authors:  Ana Vela-Sebastián; Ester López-Gallardo; Sonia Emperador; Carmen Hernández-Ainsa; David Pacheu-Grau; Ignacio Blanco; Andrea Ros; Ester Pascual-Benito; Neus Rabaneda-Lombarte; Silvia Presas-Rodríguez; Pilar García-Robles; Julio Montoya; Eduardo Ruiz-Pesini
Journal:  Clin Genet       Date:  2022-07-18       Impact factor: 4.296

8.  DNAJC30 biallelic mutations extend mitochondrial complex I-deficient phenotypes to include recessive Leber's hereditary optic neuropathy.

Authors:  Janey L Wiggs
Journal:  J Clin Invest       Date:  2021-03-15       Impact factor: 14.808

  8 in total

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