Literature DB >> 26959136

Leber Hereditary Optic Neuropathy: Bringing the Lab to the Clinic.

Nailyn Rasool1, Simmons Lessell1, Dean M Cestari1.   

Abstract

Leber hereditary optic neuropathy (LHON) was the first clinically characterized mitochondrial disorder. Since its first description in 1871, much has been discovered regarding the genetics and pathophysiology of the disease. This has enabled the development of in vitro cell and animal models that can be used to try to determine not only the effects of the genetic mutation upon the clinical phenotype but to also test potential novel therapies. Treatments for LHON have ranged from vitamins and minerals to immunosuppressants and, more recently, targeted gene therapy. This article reviews the pathophysiology and clinical features of LHON with a focus on translational research.

Entities:  

Keywords:  Hereditary optic neuropathy; Leber hereditary optic neuropathy; mitochondrial optic neuropathy; optic neuropathy

Mesh:

Substances:

Year:  2016        PMID: 26959136     DOI: 10.3109/08820538.2015.1115251

Source DB:  PubMed          Journal:  Semin Ophthalmol        ISSN: 0882-0538            Impact factor:   1.975


  6 in total

1.  MECR Mutations Cause Childhood-Onset Dystonia and Optic Atrophy, a Mitochondrial Fatty Acid Synthesis Disorder.

Authors:  Gali Heimer; Juha M Kerätär; Lisa G Riley; Shanti Balasubramaniam; Eran Eyal; Laura P Pietikäinen; J Kalervo Hiltunen; Dina Marek-Yagel; Jeffrey Hamada; Allison Gregory; Caleb Rogers; Penelope Hogarth; Martha A Nance; Nechama Shalva; Alvit Veber; Michal Tzadok; Andreea Nissenkorn; Davide Tonduti; Florence Renaldo; Ichraf Kraoua; Celeste Panteghini; Lorella Valletta; Barbara Garavaglia; Mark J Cowley; Velimir Gayevskiy; Tony Roscioli; Jonathon M Silberstein; Chen Hoffmann; Annick Raas-Rothschild; Valeria Tiranti; Yair Anikster; John Christodoulou; Alexander J Kastaniotis; Bruria Ben-Zeev; Susan J Hayflick
Journal:  Am J Hum Genet       Date:  2016-11-03       Impact factor: 11.025

2.  Mutational analysis of mitochondrial tRNA genes in 138 patients with Leber's hereditary optic neuropathy.

Authors:  Jie Shuai; Jian Shi; Ya Liang; Fangfang Ji; Luo Gu; Zhilan Yuan
Journal:  Ir J Med Sci       Date:  2021-05-29       Impact factor: 1.568

3.  Gene therapy restores mitochondrial function and protects retinal ganglion cells in optic neuropathy induced by a mito-targeted mutant ND1 gene.

Authors:  Yuan Liu; Jeremy D Eastwood; Diego E Alba; Sindhu Velmurugan; Ning Sun; Vittorio Porciatti; Richard K Lee; William W Hauswirth; John Guy; Hong Yu
Journal:  Gene Ther       Date:  2022-04-06       Impact factor: 4.184

4.  Severe inflammatory disease activity 14 months after cessation of Natalizumab in a patient with Leber's optic neuropathy and multiple sclerosis - a case report.

Authors:  Trygve Holmøy; Antonie G Beiske; Svetozar Zarnovicky; Aija Zuleron Myro; Egil Røsjø; Emilia Kerty
Journal:  BMC Neurol       Date:  2016-10-18       Impact factor: 2.474

5.  Optimization of a genotyping screening based on hydrolysis probes to detect the main mutations related to Leber hereditary optic neuropathy (LHON).

Authors:  Fábio Tadeu Arrojo Martins; Paulo Maurício do Amor Divino Miranda; Marcela Scabello Amaral Fernandes; Andréa Trevas Maciel-Guerra; Edi Lúcia Sartorato
Journal:  Mol Vis       Date:  2017-07-21       Impact factor: 2.367

Review 6.  Biomarkers for Detecting Mitochondrial Disorders.

Authors:  Josef Finsterer; Sinda Zarrouk-Mahjoub
Journal:  J Clin Med       Date:  2018-01-30       Impact factor: 4.241

  6 in total

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