Literature DB >> 23545042

Novel therapeutic approaches for Leber's hereditary optic neuropathy.

Shilpa Iyer1.   

Abstract

Many human childhood mitochondrial disorders result from abnormal mitochondrial DNA (mtDNA) and altered bioenergetics. These abnormalities span most of the mtDNA, demonstrating that there are no "unique" positions on the mitochondrial genome that when deleted or mutated produce a disease phenotype. This diversity implies that the relationship between mitochondrial genotype and clinical phenotype is very complex. The origins of clinical phenotypes are thus unclear, fundamentally difficult-to-treat, and are usually clinically devastating. Current treatment is largely supportive and the disorders progress relentlessly causing significant morbidity and mortality. Vitamin supplements and pharmacological agents have been used in isolated cases and clinical trials, but the efficacy of these interventions is unclear. In spite of recent advances in the understanding of the pathogenesis of mitochondrial diseases, a cure remains elusive. An optimal cure would be gene therapy, which involves introducing the missing gene(s) into the mitochondria to complement the defect. Our recent research results indicate the feasibility of an innovative protein-transduction ("protofection") technology, consisting of a recombinant mitochondrial transcription factor A (TFAM) that avidly binds mtDNA and permits efficient targeting into mitochondria in situ and in vivo. Thus, the development of gene therapy for treating mitochondrial disease offers promise, because it may circumvent the clinical abnormalities and the current inability to treat individual disorders in affected individuals. This review aims to focus on current treatment options and future therapeutics in mitochondrial disease treatment with a special emphasis on Leber's hereditary optic neuropathy.

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Year:  2013        PMID: 23545042      PMCID: PMC5652312     

Source DB:  PubMed          Journal:  Discov Med        ISSN: 1539-6509            Impact factor:   2.970


  60 in total

1.  Functional analysis of lymphoblast and cybrid mitochondria containing the 3460, 11778, or 14484 Leber's hereditary optic neuropathy mitochondrial DNA mutation.

Authors:  M D Brown; I A Trounce; A S Jun; J C Allen; D C Wallace
Journal:  J Biol Chem       Date:  2000-12-22       Impact factor: 5.157

2.  Targeting proteins to mitochondria using TAT.

Authors:  Victoria Del Gaizo; James A MacKenzie; R Mark Payne
Journal:  Mol Genet Metab       Date:  2003 Sep-Oct       Impact factor: 4.797

3.  Mitochondrial DNA C4171A/ND1 is a novel primary causative mutation of Leber's hereditary optic neuropathy with a good prognosis.

Authors:  Ji Yeon Kim; Jeong-Min Hwang; Sung Sup Park
Journal:  Ann Neurol       Date:  2002-05       Impact factor: 10.422

4.  DQAsome-mediated delivery of plasmid DNA toward mitochondria in living cells.

Authors:  Gerard G M D'Souza; Ram Rammohan; Shing-Ming Cheng; Vladimir P Torchilin; Volkmar Weissig
Journal:  J Control Release       Date:  2003-09-19       Impact factor: 9.776

5.  Stable transformation of CHO Cells and human NARP cybrids confers oligomycin resistance (oli(r)) following transfer of a mitochondrial DNA-encoded oli(r) ATPase6 gene to the nuclear genome: a model system for mtDNA gene therapy.

Authors:  S J Zullo; W T Parks; M Chloupkova; B Wei; H Weiner; W A Fenton; J M Eisenstadt; C R Merril
Journal:  Rejuvenation Res       Date:  2005       Impact factor: 4.663

Review 6.  Mitochondrial DNA transmission, replication and inheritance: a journey from the gamete through the embryo and into offspring and embryonic stem cells.

Authors:  Justin C St John; Joao Facucho-Oliveira; Yan Jiang; Richard Kelly; Rana Salah
Journal:  Hum Reprod Update       Date:  2010-03-15       Impact factor: 15.610

Review 7.  Leber hereditary optic neuropathy.

Authors:  P Yu-Wai-Man; D M Turnbull; P F Chinnery
Journal:  J Med Genet       Date:  2002-03       Impact factor: 6.318

8.  Recombinant mitochondrial transcription factor A with N-terminal mitochondrial transduction domain increases respiration and mitochondrial gene expression.

Authors:  Shilpa Iyer; Ravindar R Thomas; Francisco R Portell; Lisa D Dunham; Caitlin K Quigley; James P Bennett
Journal:  Mitochondrion       Date:  2009-02-04       Impact factor: 4.160

Review 9.  Mitochondrial dysfunction in optic neuropathies: animal models and therapeutic options.

Authors:  Valerio Carelli; Chiara La Morgia; Alfredo A Sadun
Journal:  Curr Opin Neurol       Date:  2013-02       Impact factor: 5.710

10.  Persistence of the treatment effect of idebenone in Leber's hereditary optic neuropathy.

Authors:  T Klopstock; G Metz; P Yu-Wai-Man; B Büchner; C Gallenmüller; M Bailie; N Nwali; P G Griffiths; B von Livonius; L Reznicek; J Rouleau; N Coppard; T Meier; P F Chinnery
Journal:  Brain       Date:  2013-02-06       Impact factor: 13.501

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  7 in total

1.  mRNA Reprogramming of T8993G Leigh's Syndrome Fibroblast Cells to Create Induced Pluripotent Stem Cell Models for Mitochondrial Disorders.

Authors:  Harrison E Grace; Patrick Galdun; Edward J Lesnefsky; Franklin D West; Shilpa Iyer
Journal:  Stem Cells Dev       Date:  2019-05-20       Impact factor: 3.272

2.  Arts, Science, Engineering and Medicine Collaborate to Educate Public on Bioenergetics.

Authors:  Emily Tompkins; Sarah Faris; Laura Hughes; Eugene Maurakis; Edward Joseph Lesnefsky; Raj Raghavendra Rao; Shilpa Iyer
Journal:  Res Rep (Montgomery, Ala.)       Date:  2017-08-16

3.  Developments in Ocular Genetics: 2013 Annual Review.

Authors:  Inas F Aboobakar; R Rand Allingham
Journal:  Asia Pac J Ophthalmol (Phila)       Date:  2014 May-Jun

4.  Evaluation of Leber's hereditary optic neuropathy patients prior to a gene therapy clinical trial.

Authors:  Shuo Yang; Hong Yang; Si-Qi Ma; Shuai-Shuai Wang; Heng He; Min-Jian Zhao; Bin Li
Journal:  Medicine (Baltimore)       Date:  2016-10       Impact factor: 1.889

Review 5.  Current and Emerging Treatment Modalities for Leber's Hereditary Optic Neuropathy: A Review of the Literature.

Authors:  Anna Theodorou-Kanakari; Spyridon Karampitianis; Vasiliki Karageorgou; Eleni Kampourelli; Efstathios Kapasakis; Panagiotis Theodossiadis; Irini Chatziralli
Journal:  Adv Ther       Date:  2018-09-01       Impact factor: 3.845

6.  A Typical Case Presentation with Spontaneous Visual Recovery in Patient Diagnosed with Leber Hereditary Optic Neuropathy due to Rare Point Mutation in MT-ND4 Gene (m.11253T>C) and Literature Review.

Authors:  Rasa Liutkeviciene; Agne Sidaraite; Lina Kuliaviene; Brigita Glebauskiene; Neringa Jurkute; Lina Aluzaite-Baranauskiene; Arvydas Gelzinis; Reda Zemaitiene
Journal:  Medicina (Kaunas)       Date:  2021-02-26       Impact factor: 2.430

7.  Predictive modeling targets thymidylate synthase ThyX in Mycobacterium tuberculosis.

Authors:  Kamel Djaout; Vinayak Singh; Yap Boum; Victoria Katawera; Hubert F Becker; Natassja G Bush; Stephen J Hearnshaw; Jennifer E Pritchard; Pauline Bourbon; Peter B Madrid; Anthony Maxwell; Valerie Mizrahi; Hannu Myllykallio; Sean Ekins
Journal:  Sci Rep       Date:  2016-06-10       Impact factor: 4.379

  7 in total

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