نتایج جستجو برای: glutaric aciduria

تعداد نتایج: 1568  

Journal: :The Journal of clinical investigation 2007
William J Zinnanti Jelena Lazovic Cathy Housman Kathryn LaNoue James P O'Callaghan Ian Simpson Michael Woontner Stephen I Goodman James R Connor Russell E Jacobs Keith C Cheng

Glutaric acidemia type I (GA-I) is an inherited disorder of lysine and tryptophan metabolism presenting with striatal lesions anatomically and symptomatically similar to Huntington disease. Affected children commonly suffer acute brain injury in the context of a catabolic state associated with nonspecific illness. The mechanisms underlying injury and age-dependent susceptibility have been unkno...

Journal: :Folia Medica 2023

Introduction : Acute hemiparesis is an emergency of various etiologies and possible fatal outcome. Aim The aim this study was to determine the etiology, clinical manifestations, prognosis acute in childhood. Materials methods This a retrospective 52 children (28 boys 24 girls) aged 4 months 16 yrs 11 with hemiparesis, admitted Clinic Pediatrics at St George University Hospital between 2013 2020...

Journal: :Brain : a journal of neurology 2007
Klaus Gempel Haluk Topaloglu Beril Talim Peter Schneiderat Benedikt G H Schoser Volkmar H Hans Beatrix Pálmafy Gulsev Kale Aysegul Tokatli Catarina Quinzii Michio Hirano Ali Naini Salvatore DiMauro Holger Prokisch Hanns Lochmüller Rita Horvath

Coenzyme Q10 (CoQ10) deficiency is an autosomal recessive disorder with heterogenous phenotypic manifestations and genetic background. We describe seven patients from five independent families with an isolated myopathic phenotype of CoQ10 deficiency. The clinical, histological and biochemical presentation of our patients was very homogenous. All patients presented with exercise intolerance, fat...

2018
Peter Augustin Marina Toplak Katharina Fuchs Eva Christine Gerstmann Ruth Prassl Andreas Winkler Peter Macheroux

The heterodimeric human (h) electron-transferring flavoprotein (ETF) transfers electrons from at least 13 different flavin dehydrogenases to the mitochondrial respiratory chain through a non-covalently bound FAD cofactor. Here, we describe the discovery of an irreversible and pH-dependent oxidation of the 8α-methyl group to 8-formyl-FAD (8f-FAD), which represents a unique chemical modification ...

Journal: :Brazilian journal of medical and biological research = Revista brasileira de pesquisas medicas e biologicas 2001
A R Silva C Ruschel C Helegda A T Wyse C M Wannmacher M Wajner C S Dutra-Filho

2-Hydroxybutyric acid appears at high concentrations in situations related to deficient energy metabolism (e.g., birth asphyxia) and also in inherited metabolic diseases affecting the central nervous system during neonatal development, such as "cerebral" lactic acidosis, glutaric aciduria type II, dihydrolipoyl dehydrogenase (E3) deficiency, and propionic acidemia. The present study was carried...

Journal: :Human molecular genetics 2015
Silvia Olivera-Bravo César A J Ribeiro Eugenia Isasi Emiliano Trías Guilhian Leipnitz Pablo Díaz-Amarilla Michael Woontner Cheryl Beck Stephen I Goodman Diogo Souza Moacir Wajner Luis Barbeito

Glutaric acidemia type I (GA-I) is an inherited neurometabolic childhood disorder caused by defective activity of glutaryl CoA dehydrogenase (GCDH) which disturb lysine (Lys) and tryptophan catabolism leading to neurotoxic accumulation of glutaric acid (GA) and related metabolites. However, it remains unknown whether GA toxicity is due to direct effects on vulnerable neurons or mediated by GA-i...

2009
J. Lazovic W. J. Zinnanti R. E. Jacobs

INTRODUCTION: Glutaric Acidemia type I (GA-1) is an inborn error of lysine, hydroxylysine and tryptophan catabolism. Due to deficiency in glutarylCoA dehydrogenase (Gcdh), these amino acids can not be completely catabolized and lead to accumulation of glutaric acid in the brain, blood and urine. The world wide prevalence is approximately 1:30,000. The common findings at autopsy are severe neuro...

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