Document Detail


Epilepsy in a young adult caused by L-2-hydroxyglutaric aciduria: a case report.
MedLine Citation:
PMID:  18408399     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
OBJECTIVE: To report the importance of a rare organic acid metabolic disorder, L-2-hydroxyglutaric aciduria, and its characteristic neuroimaging cerebral white matter abnormalities in a case of epilepsy. SUBJECT AND METHODS: A 19-year-old male presented with an 11-year history consisting of school failures, intellectual deterioration and generalized tonic-clonic convulsions. RESULTS: Neurological examination showed mental subnormality, mild dysarthria and bilateral pyramidal signs. Computed tomography and magnetic resonance imaging (MRI) of the brain showed characteristic white matter lesions, suggestive of L-2-hydroxyglutaric aciduria. The diagnosis of this disease was confirmed by elevated urinary concentrations of L-2-hydroxyglutaric acid. The epilepsy was partially controlled with antiepileptic drugs. CONCLUSION: This report indicates the importance of routine examination of urinary organic acids in children and young adults presenting with chronic encephalopathy and epilepsy with characteristic MRI white matter lesions. L-2-hydroxyglutaric aciduria should be considered as one of the differential diagnoses of epilepsy.
Authors:
Vembu Periasamy; Mohd Rudwan; Girish Yadav; Riyadh A Khan
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Publication Detail:
Type:  Case Reports; Journal Article     Date:  2008-04-10
Journal Detail:
Title:  Medical principles and practice : international journal of the Kuwait University, Health Science Centre     Volume:  17     ISSN:  1423-0151     ISO Abbreviation:  Med Princ Pract     Publication Date:  2008  
Date Detail:
Created Date:  2008-04-14     Completed Date:  2008-06-02     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  8901334     Medline TA:  Med Princ Pract     Country:  Switzerland    
Other Details:
Languages:  eng     Pagination:  258-61     Citation Subset:  IM    
Copyright Information:
(c) 2008 S. Karger AG, Basel
Affiliation:
Department of Neurology, Ibn Sina Hospital, Kuwait. drperiasamy06@yahoo.com
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MeSH Terms
Descriptor/Qualifier:
Adult
Alcohol Oxidoreductases / urine*
Anticonvulsants / therapeutic use
Biological Markers / urine
Brain Diseases, Metabolic / complications*,  diagnosis*,  physiopathology,  urine
Cerebrospinal Fluid
Diagnosis, Differential
Electroencephalography
Epilepsy, Tonic-Clonic / drug therapy,  etiology*,  physiopathology,  urine
Gas Chromatography-Mass Spectrometry
Humans
Lysine / metabolism*
Magnetic Resonance Imaging
Male
Psychological Tests
Psychometrics
Risk Factors
Chemical
Reg. No./Substance:
0/Anticonvulsants; 0/Biological Markers; 56-87-1/Lysine; EC 1.1.-/Alcohol Oxidoreductases; EC 1.1.99.2/L2HGDH protein, human

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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