Literature DB >> 24037495

L-2-hydroxyglutaric aciduria: report of two Indian families.

Mahesh Kamate1, Gowda Parameshwar Prashanth, Virupaxi Hattiholi.   

Abstract

L-2-Hydroxyglutaric aciduria (L-2-HGA) is a rare type of organic acidemia that has characteristic neurological manifestations including macrocephaly, developmental delay, epilepsy and cerebellar ataxia. Worldwide, few hundred cases of L-2-HGA are reported till date. The authors report the first three cases of L-2-HGA from two Indian families. Pertinent clinical aspects of this rare neurometabolic disorder namely, lack of acute exacerbations, and predisposition to brain tumors, are highlighted. In the present series, all cases had infantile onset of symptoms in the form of global developmental delay, seizures and cerebellar ataxia without extra-pyramidal signs or macrocephaly. One child presented as acute febrile encephalopathy which has not been described as a presenting feature.

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Year:  2013        PMID: 24037495     DOI: 10.1007/s12098-013-1194-5

Source DB:  PubMed          Journal:  Indian J Pediatr        ISSN: 0019-5456            Impact factor:   1.967


  9 in total

1.  A successfully treated adult patient with L-2-hydroxyglutaric aciduria.

Authors:  M Samuraki; K Komai; Y Hasegawa; M Kimura; S Yamaguchi; N Terada; M Yamada
Journal:  Neurology       Date:  2008-03-25       Impact factor: 9.910

2.  L-2-Hydroxyglutaric aciduria: clinical, biochemical and magnetic resonance imaging in six Portuguese pediatric patients.

Authors:  C Barbot; I Fineza; L Diogo; M Maia; J Melo; A Guimarães; M M Pires; M L Cardoso; L Vilarinho
Journal:  Brain Dev       Date:  1997-06       Impact factor: 1.961

Review 3.  Cerebral neoplasms in L-2 hydroxyglutaric aciduria: 3 new cases and meta-analysis of literature data.

Authors:  Z Patay; J C Mills; U Löbel; A Lambert; A Sablauer; D W Ellison
Journal:  AJNR Am J Neuroradiol       Date:  2012-01-12       Impact factor: 3.825

4.  In vivo proton magnetic resonance spectroscopy of the brain in a patient with L-2-hydroxyglutaric acidemia.

Authors:  F Hanefeld; B Kruse; H Bruhn; J Frahm
Journal:  Pediatr Res       Date:  1994-05       Impact factor: 3.756

5.  L-2-Hydroxyglutaric aciduria: identification of a mutant gene C14orf160, localized on chromosome 14q22.1.

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Journal:  Hum Mol Genet       Date:  2004-09-22       Impact factor: 6.150

Review 6.  An overview of L-2-hydroxyglutarate dehydrogenase gene (L2HGDH) variants: a genotype-phenotype study.

Authors:  Marjan E Steenweg; Cornelis Jakobs; Abdellatif Errami; Silvy J M van Dooren; Maria T Adeva Bartolomé; Peter Aerssens; Persephone Augoustides-Savvapoulou; Ivo Baric; Matthias Baumann; Luisa Bonafé; Brigitte Chabrol; Joe T R Clarke; Peter Clayton; Mahmut Coker; Sarah Cooper; Tzipora Falik-Zaccai; Mark Gorman; Andreas Hahn; Alev Hasanoglu; Mary D King; Hans B C de Klerk; Stanley H Korman; Céline Lee; Allan Meldgaard Lund; Vlatka Mejaski-Bosnjak; Ignacio Pascual-Castroviejo; Aparna Raadhyaksha; Terje Rootwelt; Agathe Roubertie; Maria L Ruiz-Falco; Emmanuel Scalais; Ulf Schimmel; Manuel Seijo-Martinez; Mohnish Suri; Jolanta Sykut-Cegielska; Friedrich K Trefz; Graziella Uziel; Vassili Valayannopoulos; Christine Vianey-Saban; Stefan Vlaho; Julia Vodopiutz; Moacir Wajner; John Walter; Claudia Walter-Derbort; Zuhal Yapici; Dimitrios I Zafeiriou; Marieke D Spreeuwenberg; Jacopo Celli; Johan T den Dunnen; Marjo S van der Knaap; Gajja S Salomons
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7.  Riboflavin treatment in a case with l-2-hydroxyglutaric aciduria.

Authors:  Kutluhan Yilmaz
Journal:  Eur J Paediatr Neurol       Date:  2008-03-17       Impact factor: 3.140

8.  L-2-Hydroxyglutaric aciduria: pattern of MR imaging abnormalities in 56 patients.

Authors:  Marjan E Steenweg; Gajja S Salomons; Zuhal Yapici; Graziella Uziel; Emmanuel Scalais; Dimitrios I Zafeiriou; Maria L Ruiz-Falco; Vlatka Mejaski-Bosnjak; Persephone Augoustides-Savvopoulou; Moacir Wajner; John Walter; Nanda M Verhoeven-Duif; Eduard A Struys; Cornelis Jakobs; Marjo S van der Knaap
Journal:  Radiology       Date:  2009-06       Impact factor: 11.105

Review 9.  Progress in understanding 2-hydroxyglutaric acidurias.

Authors:  Martijn Kranendijk; Eduard A Struys; Gajja S Salomons; Marjo S Van der Knaap; Cornelis Jakobs
Journal:  J Inherit Metab Dis       Date:  2012-03-06       Impact factor: 4.982

  9 in total
  2 in total

1.  Postural tremor in L-2-hydroxyglutaric aciduria is associated with cerebellar atrophy.

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Journal:  Neurol Sci       Date:  2021-08-24       Impact factor: 3.307

Review 2.  Treatment, Therapy and Management of Metabolic Epilepsy: A Systematic Review.

Authors:  Vanessa Lin Lin Lee; Brandon Kar Meng Choo; Yin-Sir Chung; Uday P Kundap; Yatinesh Kumari; Mohd Farooq Shaikh
Journal:  Int J Mol Sci       Date:  2018-03-15       Impact factor: 5.923

  2 in total

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