Literature DB >> 7510656

3-Methylglutaconic aciduria in the Iraqi-Jewish 'optic atrophy plus' (Costeff) syndrome.

O N Elpeleg1, H Costeff, A Joseph, Y Shental, R Weitz, K M Gibson.   

Abstract

Eleven new patients of Iraqi-Jewish origin with bilateral optic atrophy, neurological abnormalities ('optic atrophy plus' syndrome) and 3-methylglutaconic aciduria (type III) are described. Clinical abnormalities in decreasing order of frequency were bilateral optic atrophy, extrapyramidal signs, spasticity, ataxia, dysarthria and cognitive deficit. An association with age was found only for spasticity. Spasticity, extrapyramidal signs and optic atrophy frequently led to major disability, in contrast to ataxia, dysarthria and cognitive deficit. The combined excretion of 3-methylglutaconic and 3-methylglutaric acid ranged between 9 and 187 mmol/mol creatinine. The primary enzymatic defect possibly may reside in the mitochondrial respiratory chain.

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Year:  1994        PMID: 7510656     DOI: 10.1111/j.1469-8749.1994.tb11825.x

Source DB:  PubMed          Journal:  Dev Med Child Neurol        ISSN: 0012-1622            Impact factor:   5.449


  8 in total

1.  Type III 3-methylglutaconic aciduria (optic atrophy plus syndrome, or Costeff optic atrophy syndrome): identification of the OPA3 gene and its founder mutation in Iraqi Jews.

Authors:  Y Anikster; R Kleta; A Shaag; W A Gahl; O Elpeleg
Journal:  Am J Hum Genet       Date:  2001-10-19       Impact factor: 11.025

2.  OPA3, mutated in 3-methylglutaconic aciduria type III, encodes two transcripts targeted primarily to mitochondria.

Authors:  Marjan Huizing; Heidi Dorward; Lien Ly; Enriko Klootwijk; Robert Kleta; Flemming Skovby; Wuhong Pei; Benjamin Feldman; William A Gahl; Yair Anikster
Journal:  Mol Genet Metab       Date:  2010-03-16       Impact factor: 4.797

3.  A model of Costeff Syndrome reveals metabolic and protective functions of mitochondrial OPA3.

Authors:  Wuhong Pei; Lisa E Kratz; Isa Bernardini; Raman Sood; Tohei Yokogawa; Heidi Dorward; Carla Ciccone; Richard I Kelley; Yair Anikster; Harold A Burgess; Marjan Huizing; Benjamin Feldman
Journal:  Development       Date:  2010-08-01       Impact factor: 6.868

4.  Mitochondrial complex deficiencies in a male with cardiomyopathy and 3-methylglutaconic aciduria.

Authors:  G T Besley; M Lendon; D M Broadhead; J Till; L E Heptinstall; B Phillips
Journal:  J Inherit Metab Dis       Date:  1995       Impact factor: 4.982

5.  Costeff syndrome: clinical features and natural history.

Authors:  Gilad Yahalom; Yair Anikster; Ruth Huna-Baron; Chen Hoffmann; Lubov Blumkin; Dorit Lev; Rakefet Tsabari; Zeev Nitsan; Sheera F Lerman; Bruria Ben-Zeev; Ben Pode-Shakked; Shira Sofer; Avraham Schweiger; Tally Lerman-Sagie; Sharon Hassin-Baer
Journal:  J Neurol       Date:  2014-09-09       Impact factor: 4.849

Review 6.  The 3-methylglutaconic acidurias: what's new?

Authors:  Saskia B Wortmann; Leo A Kluijtmans; Udo F H Engelke; Ron A Wevers; Eva Morava
Journal:  J Inherit Metab Dis       Date:  2010-09-30       Impact factor: 4.982

7.  Autosomal dominant optic atrophy and cataract "plus" phenotype including axonal neuropathy.

Authors:  Alejandro Horga; Enrico Bugiardini; Andreea Manole; Fion Bremner; Zane Jaunmuktane; Lois Dankwa; Adriana P Rebelo; Catherine E Woodward; Iain P Hargreaves; Andrea Cortese; Alan M Pittman; Sebastian Brandner; James M Polke; Robert D S Pitceathly; Stephan Züchner; Michael G Hanna; Steven S Scherer; Henry Houlden; Mary M Reilly
Journal:  Neurol Genet       Date:  2019-04-01

8.  Neonatal presentation of ventricular tachycardia and a Reye-like syndrome episode associated with disturbed mitochondrial energy metabolism.

Authors:  Fernando Scaglia; Angela E Scheuerle; Jeffrey A Towbin; Dawna L Armstrong; Lawrence Sweetman; Lee-Jun C Wong
Journal:  BMC Pediatr       Date:  2002-12-30       Impact factor: 2.125

  8 in total

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