Literature DB >> 28766179

Glutaric Aciduria Type 3: Three Unrelated Canadian Cases, with Different Routes of Ascertainment.

Paula J Waters1, Thomas M Kitzler2, Annette Feigenbaum3,4, Michael T Geraghty5, Osama Al-Dirbashi5,6, Patrick Bherer7, Christiane Auray-Blais7, Serge Gravel7, Nathan McIntosh5, Komudi Siriwardena3,8, Yannis Trakadis2, Catherine Brunel-Guitton9, Walla Al-Hertani2,10.   

Abstract

Glutaric aciduria type 3 (GA3) is associated with decreased conversion of free glutaric acid to glutaryl-coA, reflecting deficiency of succinate-hydroxymethylglutarate coA-transferase, caused by variants in the SUGCT (C7orf10) gene. GA3 remains less well known, characterised and understood than glutaric aciduria types 1 and 2. It is generally considered a likely "non-disease," but this is based on limited supporting information, with only nine individuals with GA3 described in the literature. Clinicians encountering a patient with GA3 therefore still face a dilemma of whether or not this should be dismissed as irrelevant.We have identified three unrelated Canadian patients with GA3. Two came to clinical attention because of symptoms, while the third was identified by a population urine-based newborn screening programme and has so far remained asymptomatic. We describe the clinical histories, biochemical characterisation and genotypes of these individuals. Examination of allele frequencies underlines the fact that GA3 is underdiagnosed. While one probable factor is that some GA3 patients remain asymptomatic, we highlight other plausible reasons whereby this diagnosis might be overlooked.Gastrointestinal disturbances were previously reported in some GA3 patients. In one of our patients, severe episodes of cyclic vomiting were the major problem. A trial of antibiotic treatment, to minimise bacterial GA production, was followed by significant clinical improvement.At present, there is insufficient evidence to define any specific clinical phenotype as attributable to GA3. However, we consider that it would be premature to assume that this condition is completely benign in all individuals at all times.

Entities:  

Keywords:  ACY1; Aminoacylase 1 deficiency; Benign condition; C7orf10; Glutaric acid; Glutaric aciduria type 3; Glutaric aciduria type III; Non-disease; SUGCT gene; Succinate-hydroxymethylglutarate CoA-transferase

Year:  2017        PMID: 28766179      PMCID: PMC5953897          DOI: 10.1007/8904_2017_49

Source DB:  PubMed          Journal:  JIMD Rep        ISSN: 2192-8304


  19 in total

1.  Diagnosis of glutaric aciduria type 1 by measuring 3-hydroxyglutaric acid in dried urine spots by liquid chromatography tandem mass spectrometry.

Authors:  Osama Y Al-Dirbashi; Stefan Kölker; Dione Ng; Lawrence Fisher; Tony Rupar; Nathalie Lepage; Mohamed S Rashed; Tomofumi Santa; Stephen I Goodman; Michael T Geraghty; Johannes Zschocke; Ernst Christensen; Georg F Hoffmann; Pranesh Chakraborty
Journal:  J Inherit Metab Dis       Date:  2010-10-27       Impact factor: 4.982

2.  Neurological findings in aminoacylase 1 deficiency.

Authors:  J O Sass; H Olbrich; V Mohr; C Hart; B Woldseth; S Krywawych; B Bjurulf; P K Lakhani; R M Buchdahl; H Omran
Journal:  Neurology       Date:  2007-06-12       Impact factor: 9.910

3.  Aminoacylase 1 deficiency associated with autistic behavior.

Authors:  Anna Tylki-Szymanska; Wanda Gradowska; Anke Sommer; Angelina Heer; Melanie Walter; Christina Reinhard; Heymut Omran; Jörn Oliver Sass; Agnieszka Jurecka
Journal:  J Inherit Metab Dis       Date:  2010-05-18       Impact factor: 4.982

Review 4.  Proposed recommendations for diagnosing and managing individuals with glutaric aciduria type I: second revision.

Authors:  Nikolas Boy; Chris Mühlhausen; Esther M Maier; Jana Heringer; Birgit Assmann; Peter Burgard; Marjorie Dixon; Sandra Fleissner; Cheryl R Greenberg; Inga Harting; Georg F Hoffmann; Daniela Karall; David M Koeller; Michael B Krawinkel; Jürgen G Okun; Thomas Opladen; Roland Posset; Katja Sahm; Johannes Zschocke; Stefan Kölker
Journal:  J Inherit Metab Dis       Date:  2016-11-16       Impact factor: 4.982

Review 5.  HSD10 disease: clinical consequences of mutations in the HSD17B10 gene.

Authors:  Johannes Zschocke
Journal:  J Inherit Metab Dis       Date:  2011-11-30       Impact factor: 4.982

6.  Metabolic, nutritional, iatrogenic, and artifactual sources of urinary organic acids: a comprehensive table.

Authors:  Alain Kumps; Pierre Duez; Yves Mardens
Journal:  Clin Chem       Date:  2002-05       Impact factor: 8.327

7.  Glutaric aciduria type III: a distinctive non-disease?

Authors:  I Knerr; J Zschocke; U Trautmann; L Dorland; T J de Koning; P Müller; E Christensen; F K Trefz; G F Wündisch; W Rascher; G F Hoffmann
Journal:  J Inherit Metab Dis       Date:  2002-10       Impact factor: 4.982

8.  Gas chromatographic-mass spectrometric analysis of N-acetylated amino acids: the first case of aminoacylase I deficiency.

Authors:  Erik Gerlo; Rudy Van Coster; Willy Lissens; Gerda Winckelmans; Linda De Meirleir; Ron Wevers
Journal:  Anal Chim Acta       Date:  2006-05-05       Impact factor: 6.558

9.  C7orf10 encodes succinate-hydroxymethylglutarate CoA-transferase, the enzyme that converts glutarate to glutaryl-CoA.

Authors:  Simon Marlaire; Emile Van Schaftingen; Maria Veiga-da-Cunha
Journal:  J Inherit Metab Dis       Date:  2013-07-27       Impact factor: 4.982

10.  Molecular insights into HSD10 disease: impact of SDR5C1 mutations on the human mitochondrial RNase P complex.

Authors:  Elisa Vilardo; Walter Rossmanith
Journal:  Nucleic Acids Res       Date:  2015-04-29       Impact factor: 16.971

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