Literature DB >> 18593870

Identification of new mutations in the ETHE1 gene in a cohort of 14 patients presenting with ethylmalonic encephalopathy.

R Mineri1, M Rimoldi, A B Burlina, S Koskull, C Perletti, B Heese, U von Döbeln, P Mereghetti, I Di Meo, F Invernizzi, M Zeviani, G Uziel, V Tiranti.   

Abstract

BACKGROUND: Ethylmalonic encephalopathy (EE) is a rare autosomal recessive metabolic disorder characterised by progressive encephalopathy, recurrent petechiae, acrocyanosis and chronic diarrhoea, with a fatal outcome in early in life.
METHODS: 14 patients with EE were investigated for mutations in the ETHE1 gene.
RESULTS: Of the 14 patients, 5 were found to carry novel mutations.
CONCLUSIONS: This work expands our knowledge of the causative mutations of EE.

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Year:  2008        PMID: 18593870     DOI: 10.1136/jmg.2008.058271

Source DB:  PubMed          Journal:  J Med Genet        ISSN: 0022-2593            Impact factor:   6.318


  30 in total

1.  Multiple sources of metabolic disturbance in ETHE1-related ethylmalonic encephalopathy.

Authors:  Magalie Barth; Chris Ottolenghi; Laurence Hubert; Dominique Chrétien; Valérie Serre; Stéphanie Gobin; Stéphane Romano; Anne Vassault; Aziz Sefiani; Daniel Ricquier; Nathalie Boddaert; Michèle Brivet; Yves de Keyzer; Arnold Munnich; Marinus Duran; Daniel Rabier; Vassili Valayannopoulos; Pascale de Lonlay
Journal:  J Inherit Metab Dis       Date:  2010-10-27       Impact factor: 4.982

2.  Turkish case of ethylmalonic encephalopathy misdiagnosed as short chain acyl-CoA dehydrogenase deficiency.

Authors:  Fatma Derya Bulut; Deniz Kör; Berna Şeker-Yılmaz; Gülen Gül-Mert; Sebile Kılavuz; Neslihan Önenli-Mungan
Journal:  Metab Brain Dis       Date:  2017-11-20       Impact factor: 3.584

3.  Characterization of patient mutations in human persulfide dioxygenase (ETHE1) involved in H2S catabolism.

Authors:  Omer Kabil; Ruma Banerjee
Journal:  J Biol Chem       Date:  2012-11-09       Impact factor: 5.157

4.  Distribution, diversity, and activities of sulfur dioxygenases in heterotrophic bacteria.

Authors:  Honglei Liu; Yufeng Xin; Luying Xun
Journal:  Appl Environ Microbiol       Date:  2014-01-03       Impact factor: 4.792

5.  Evidence that Thiosulfate Inhibits Creatine Kinase Activity in Rat Striatum via Thiol Group Oxidation.

Authors:  Mateus Grings; Belisa Parmeggiani; Alana Pimentel Moura; Leonardo de Moura Alvorcem; Angela T S Wyse; Moacir Wajner; Guilhian Leipnitz
Journal:  Neurotox Res       Date:  2018-07-28       Impact factor: 3.911

6.  Combined treatment with oral metronidazole and N-acetylcysteine is effective in ethylmalonic encephalopathy.

Authors:  Carlo Viscomi; Alberto B Burlina; Imad Dweikat; Mario Savoiardo; Costanza Lamperti; Tatjana Hildebrandt; Valeria Tiranti; Massimo Zeviani
Journal:  Nat Med       Date:  2010-07-25       Impact factor: 53.440

Review 7.  Chemical Biology of H2S Signaling through Persulfidation.

Authors:  Milos R Filipovic; Jasmina Zivanovic; Beatriz Alvarez; Ruma Banerjee
Journal:  Chem Rev       Date:  2017-11-07       Impact factor: 60.622

Review 8.  Altered sulfide (H(2)S) metabolism in ethylmalonic encephalopathy.

Authors:  Valeria Tiranti; Massimo Zeviani
Journal:  Cold Spring Harb Perspect Biol       Date:  2013-01-01       Impact factor: 10.005

Review 9.  Sulfur as a signaling nutrient through hydrogen sulfide.

Authors:  Omer Kabil; Victor Vitvitsky; Ruma Banerjee
Journal:  Annu Rev Nutr       Date:  2014       Impact factor: 11.848

Review 10.  Enzymology of H2S biogenesis, decay and signaling.

Authors:  Omer Kabil; Ruma Banerjee
Journal:  Antioxid Redox Signal       Date:  2013-06-07       Impact factor: 8.401

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