نتایج جستجو برای: mitochondrial encephalomyopathy

تعداد نتایج: 132426  

Journal: :American journal of human genetics 2013
Xiaowu Gai Daniele Ghezzi Mark A Johnson Caroline A Biagosch Hanan E Shamseldin Tobias B Haack Aurelio Reyes Mai Tsukikawa Claire A Sheldon Satish Srinivasan Matteo Gorza Laura S Kremer Thomas Wieland Tim M Strom Erzsebet Polyak Emily Place Mark Consugar Julian Ostrovsky Sara Vidoni Alan J Robinson Lee-Jun Wong Neal Sondheimer Mustafa A Salih Emtethal Al-Jishi Christopher P Raab Charles Bean Francesca Furlan Rossella Parini Costanza Lamperti Johannes A Mayr Vassiliki Konstantopoulou Martina Huemer Eric A Pierce Thomas Meitinger Peter Freisinger Wolfgang Sperl Holger Prokisch Fowzan S Alkuraya Marni J Falk Massimo Zeviani

Whole-exome sequencing and autozygosity mapping studies, independently performed in subjects with defective combined mitochondrial OXPHOS-enzyme deficiencies, identified a total of nine disease-segregating FBXL4 mutations in seven unrelated mitochondrial disease families, composed of six singletons and three siblings. All subjects manifested early-onset lactic acidemia, hypotonia, and developme...

2016
Mona Faramawy Najla Al Kuwaiti Abdulla Al-Amri Amar Al Shibli

Mitochondrial neuro-gastrointestinal encephalomyopathy (MNGIE), is an autosomal recessive disease, is one of the mitochondrial disorders, and is a multisystem disease clinically defined by progressive ophthalmoplegia, peripheral neuropathy, leukoencephalopathy, mitochondrial abnormalities and severe gastrointestinal involvement. Mitochondrial disorders have clinical manifestations reflecting th...

Journal: :Proceedings of the National Academy of Sciences 1989

2006
Alicia M. Celotto Adam C. Frank Steven W. McGrath Tim Fergestad Wayne A. Van Voorhies Karolyn F. Buttle Carmen A. Mannella Michael J. Palladino

Alicia M. Celotto,1,2 Adam C. Frank,1,2 Steven W. McGrath,1,2 Tim Fergestad,3 Wayne A. Van Voorhies,5 Karolyn F. Buttle,4 Carmen A. Mannella,4 and Michael J. Palladino1,2 1Department of Pharmacology and 2Pittsburgh Institute for Neurodegenerative Diseases, University of Pittsburgh School of Medicine, Pittsburgh, Pennsylvania 15261, 3Laboratory of Genetics, University of Wisconsin–Madison, Madis...

Journal: :Annals of neurology 1993
B Koo L E Becker S Chuang F Merante B H Robinson D MacGregor I Tein V B Ho D A McGreal J R Wherrett

We reviewed 10 patients (5 males, 5 females) with mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes. The age of symptom onset ranged from 3 months to 12 years. All had lactic acidosis, multiple stroke-like events with secondary neurological deficits, radiological changes of progressive brain infarction, and muscle biopsy showing ragged-red fibers. In patients with earli...

2013
Xiaoshan Zhou Kristina Kannisto Sophie Curbo Ulrika von Döbeln Kjell Hultenby Sindra Isetun Mats Gåfvels Anna Karlsson

Thymidine kinase 2 (TK2) deficiency in humans causes mitochondrial DNA (mtDNA) depletion syndrome. To study the molecular mechanisms underlying the disease and search for treatment options, we previously generated and described a TK2 deficient mouse strain (TK2(-/-)) that progressively loses its mtDNA. The TK2(-/-) mouse model displays symptoms similar to humans harboring TK2 deficient infantil...

2016
Thanes Termglinchan Seito Hisamatsu Junko Ohmori Hiroshi Suzumura Noriko Sumitomo George Imataka Osamu Arisaka Nobuyuki Murakami Narihiro Minami Ishiyama Akihiko Masayuki Sasaki Yuichi Goto Satoru Noguchi Ikuya Nonaka Satomi Mitsuhashi Ichizo Nishino

Recessive mutations in TK2 cause a severe mitochondrial DNA depletion syndrome (MDS),(1) characterized by severe myopathy from early infancy. Recent reports have suggested a wider clinical spectrum including encephalomyopathic form.(1,2) We report a patient with infantile-onset fatal encephalomyopathy presenting with extreme muscle fiber immaturity.

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