نتایج جستجو برای: leigh syndrome

تعداد نتایج: 623686  

Journal: :emergency journal 0
farzad ashrafi brain mapping research center, shahid beheshti university of medical sciences, tehran, iran. hossein pakdaman brain mapping research center, shahid beheshti university of medical sciences, tehran, iran. mehran arabahmadi brain mapping research center, shahid beheshti university of medical sciences, tehran, iran. behdad behnam brain mapping research center, shahid beheshti university of medical sciences, tehran, iran.

leigh syndrome is a severe progressive neurodegenerative disorder with different clinical presentationsthat usually becomes apparent in the first year of life and rarely in late childhood and elderly years. it is causedby failure of mitochondrial respiratory chain and often results in regression of both mental and motor skills and might even lead to death. in some of the inherited neurodegenera...

Journal: :Journal of Neuropathology & Experimental Neurology 2015

2010
AL Kartikasalwah Ngu LH

Leigh syndrome is a progressive neurodegenerative disorder of childhood. The symmetrical necrotic lesions in the basal ganglia and/or brainstem which appear as hyperintense lesions on T2-weighted MRI is characteristic and one of the essential diagnostic criteria. Recognising this MR imaging pattern in a child with neurological problems should prompt the clinician to investigate for Leigh syndro...

Journal: :Molecular genetics and metabolism 2012
Diego Martinelli Michela Catteruccia Fiorella Piemonte Anna Pastore Giulia Tozzi Carlo Dionisi-Vici Giuseppe Pontrelli Tiziana Corsetti Susanna Livadiotti Viktoria Kheifets Andrew Hinman William D Shrader Martin Thoolen Matthew B Klein Enrico Bertini Guy Miller

BACKGROUND Genetically defined Leigh syndrome is a rare, fatal inherited neurodegenerative disorder that predominantly affects children. No treatment is available. EPI-743 is a novel small molecule developed for the treatment of Leigh syndrome and other inherited mitochondrial diseases. In compassionate use cases and in an FDA Expanded Access protocol, children with Leigh syndrome treated with ...

Journal: :Brain : a journal of neurology 2013
Mike Gerards Rick Kamps Jo van Oevelen Iris Boesten Eveline Jongen Bart de Koning Hans R Scholte Isabel de Angst Kees Schoonderwoerd Abdelaziz Sefiani Ilham Ratbi Wouter Coppieters Latifa Karim René de Coo Bianca van den Bosch Hubert Smeets

Leigh syndrome is an early onset, often fatal progressive neurodegenerative disorder caused by mutations in the mitochondrial or nuclear DNA. Until now, mutations in more than 35 genes have been reported to cause Leigh syndrome, indicating an extreme genetic heterogeneity for this disorder, but still only explaining part of the cases. The possibility of whole exome sequencing enables not only m...

2015
Mariella Simon Elodie M. Richard Xinjian Wang Mohsin Shahzad Vincent H. Huang Tanveer A. Qaiser Prasanth Potluri Sarah E. Mahl Antonio Davila Sabiha Nazli Saege Hancock Margret Yu Jay Gargus Richard Chang Nada Al-sheqaih William G. Newman Jose Abdenur Arnold Starr Rashmi Hegde Thomas Dorn Anke Busch Eddie Park Jie Wu Hagen Schwenzer Adrian Flierl Catherine Florentz Marie Sissler Shaheen N. Khan Ronghua Li Min-Xin Guan Thomas B. Friedman Doris K. Wu Vincent Procaccio Sheikh Riazuddin Douglas C. Wallace Zubair M. Ahmed Taosheng Huang Saima Riazuddin

Here we demonstrate association of variants in the mitochondrial asparaginyl-tRNA synthetase NARS2 with human hearing loss and Leigh syndrome. A homozygous missense mutation ([c.637G>T; p.Val213Phe]) is the underlying cause of nonsyndromic hearing loss (DFNB94) and compound heterozygous mutations ([c.969T>A; p.Tyr323*] + [c.1142A>G; p.Asn381Ser]) result in mitochondrial respiratory chain defici...

2014
Kalliopi Sofou Irenaeus F M De Coo Pirjo Isohanni Elsebet Ostergaard Karin Naess Linda De Meirleir Charalampos Tzoulis Johanna Uusimaa Isabell B De Angst Tuula Lönnqvist Helena Pihko Katariina Mankinen Laurence A Bindoff Már Tulinius Niklas Darin

BACKGROUND Leigh syndrome is a progressive neurodegenerative disorder, associated with primary or secondary dysfunction of the mitochondrial oxidative phosphorylation. Despite the fact that Leigh syndrome is the most common phenotype of mitochondrial disorders in children, longitudinal natural history data is missing. This study was undertaken to assess the phenotypic and genotypic spectrum of ...

2010
Helen A. L. Tuppen Vanessa E. Hogan Langping He Emma L. Blakely Lisa Worgan Mazhor Al-Dosary Gabriele Saretzki Charlotte L. Alston Andrew A. Morris Michael Clarke Simon Jones Anita M. Devlin Sahar Mansour Zofia M. A. Chrzanowska-Lightowlers David R. Thorburn Robert McFarland Robert W. Taylor

Isolated complex I deficiency is the most frequently observed oxidative phosphorylation defect in children with mitochondrial disease, leading to a diverse range of clinical presentations, including Leigh syndrome. For most patients the genetic cause of the biochemical defect remains unknown due to incomplete understanding of the complex I assembly process. Nonetheless, a plethora of pathogenic...

Journal: :Annals of neurology 1999
R H Triepels L P van den Heuvel J L Loeffen C A Buskens R J Smeets M E Rubio Gozalbo S M Budde E C Mariman F A Wijburg P G Barth J M Trijbels J A Smeitink

Leigh syndrome is the phenotypical expression of a genetically heterogeneous cluster of disorders, with pyruvate dehydrogenase complex deficiency and respiratory chain disorders as the main biochemical causes. We report the first missense mutation within the nuclear encoded complex I subunit, NDUFS7, in 2 siblings with neuropathologically proven complex I-deficient Leigh syndrome.

2017
Mei Wang Ya-Ping Huang Han Wu Ke Song Cong Wan A-Ni Chi Ya-Mei Xiao Xiao-Yang Zhao

BACKGROUND The NDUFS4 gene encodes an 18-kD subunit of mitochondria complex I, and mutations in this gene lead to the development of a severe neurodegenerative disease called Leigh syndrome (LS) in humans. To investigate the disease phenotypes and molecular mechanisms of Leigh syndrome, the Ndufs4 knockout (KO) mouse has been widely used as a novel animal model. Because the homozygotes cannot s...

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