Literature DB >> 9578969

Mild or absent clinical signs in twin sisters with short-chain acyl-CoA dehydrogenase deficiency.

A Ribes1, E Riudor, B Garavaglia, G Martinez, A Arranz, F Invernizzi, P Briones, E Lamantea, M Sentís, A Barceló, M Roig.   

Abstract

UNLABELLED: Two HLA-identical twin sisters are reported, of whom one has remained essentially asymptomatic, and an episode of hypotonia and decreased level of conciousness being the only relevant clinical finding in the other. Organic acid-analysis revealed that ethylmalonate was constantly, although sometimes only slightly, increased. No abnormal acylglycines or acylcarnitines could be detected. Enzyme assay in cultured skin fibroblasts confirmed short-chain acyl-CoA dehydrogenase deficiency.
CONCLUSION: The lack of appropriate biochemical markers for this deficiency makes the diagnosis difficult and consequently, the low number of patients described may be the result of underdiagnosis.

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Year:  1998        PMID: 9578969     DOI: 10.1007/s004310050819

Source DB:  PubMed          Journal:  Eur J Pediatr        ISSN: 0340-6199            Impact factor:   3.183


  9 in total

Review 1.  Short-chain acyl-coenzyme A dehydrogenase deficiency.

Authors:  Reena Jethva; Michael J Bennett; Jerry Vockley
Journal:  Mol Genet Metab       Date:  2008-11-05       Impact factor: 4.797

Review 2.  Disorders of mitochondrial fatty acyl-CoA beta-oxidation.

Authors:  R J Wanders; P Vreken; M E den Boer; F A Wijburg; A H van Gennip; L IJlst
Journal:  J Inherit Metab Dis       Date:  1999-06       Impact factor: 4.982

3.  A Rare Case of Short-Chain Acyl-COA Dehydrogenase Deficiency: The Apparent Rarity of the Disorder Results in Under Diagnosis.

Authors:  G Shilpa Reddy; M Sujatha
Journal:  Indian J Clin Biochem       Date:  2011-06-08

Review 4.  Short-chain acyl-CoA dehydrogenase deficiency: from gene to cell pathology and possible disease mechanisms.

Authors:  Zahra Nochi; Rikke Katrine Jentoft Olsen; Niels Gregersen
Journal:  J Inherit Metab Dis       Date:  2017-05-17       Impact factor: 4.982

5.  Ethylmalonic acid induces permeability transition in isolated brain mitochondria.

Authors:  Cristiane Cecatto; Alexandre Umpierrez Amaral; Guilhian Leipnitz; Roger Frigério Castilho; Moacir Wajner
Journal:  Neurotox Res       Date:  2014-02-21       Impact factor: 3.911

6.  Recurrent vomiting and ethylmalonic aciduria associated with rare mutations of the short-chain acyl-CoA dehydrogenase gene.

Authors:  J Seidel; S Streck; K Bellstedt; C Vianey-Saban; C B Pedersen; J Vockley; H Korall; M Roskos; T Deufel; K F Trefz; A C Sewell; E Kauf; F Zintl; W Lehnert; N Gregersen
Journal:  J Inherit Metab Dis       Date:  2003       Impact factor: 4.982

7.  The ACADS gene variation spectrum in 114 patients with short-chain acyl-CoA dehydrogenase (SCAD) deficiency is dominated by missense variations leading to protein misfolding at the cellular level.

Authors:  Christina B Pedersen; Steen Kølvraa; Agnete Kølvraa; Vibeke Stenbroen; Margrethe Kjeldsen; Regina Ensenauer; Ingrid Tein; Dietrich Matern; Piero Rinaldo; Christine Vianey-Saban; Antonia Ribes; Willy Lehnert; Ernst Christensen; Thomas J Corydon; Brage S Andresen; Søren Vang; Lars Bolund; Jerry Vockley; Peter Bross; Niels Gregersen
Journal:  Hum Genet       Date:  2008-06-04       Impact factor: 4.132

8.  A new case of short-chain acyl-CoA dehydrogenase deficiency: clinical, biochemical, genetic and (1)H-NMR spectroscopic studies.

Authors:  C Battisti; F Forte; M Molinelli; S Funghini; E Pasquini; M Tassini; M T Dotti; A Federico
Journal:  Neurol Sci       Date:  2008-01-04       Impact factor: 3.307

9.  Biochemical correction of short-chain acyl-coenzyme A dehydrogenase deficiency after portal vein injection of rAAV8-SCAD.

Authors:  Stuart G Beattie; Eric Goetzman; Thomas Conlon; Sean Germain; Glenn Walter; Martha Campbell-Thompson; Dietrich Matern; Jerry Vockley; Terence R Flotte
Journal:  Hum Gene Ther       Date:  2008-06       Impact factor: 5.695

  9 in total

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