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PMID: 16361598 Published · ppublish English Case Reports Journal Article Research Support, Non-U.S. Gov't

Leigh syndrome caused by mutations in the flavoprotein (Fp) subunit of succinate dehydrogenase (SDHA).

Journal of neurology, neurosurgery, and psychiatry ·Vol. 77 ·No. 1 ·2006-01-00 ·Pages 74-6

Horváth R, Abicht A, Holinski-Feder E, Laner A, Gempel K, Prokisch H, Lochmüller H, Klopstock T, Jaksch M

Abstract

Detailed clinical, neuroradiological, histological, biochemical, and genetic investigations were undertaken in a child suffering from Leigh syndrome. The clinical symptoms started at age five months and led to a severe progressive neurodegenerative disorder causing epilepsy, psychomotor retardation, and tetraspasticity. Biochemical measurement of skeletal muscle showed a severe decrease in mitochondrial complex II. Sequencing of SDHA revealed compound heterozygosity for a nonsense mutation in exon 4 (W119X) and a missense mutation in exon 3 (A83V), both absent in normal controls. In six additional patients--five with Leigh or Leigh-like syndrome and one with neuropathy and ataxia associated with isolated deficiency of complex II--mutations in SDHA were not detected, indicating genetic heterogeneity.

MeSH Terms
Atrophy/pathology Biopsy Brain/pathology Cerebral Cortex/pathology Child Child, Preschool DNA/analysis DNA, Complementary/analysis Disease Progression Exons/genetics Female Flavoproteins/genetics Functional Laterality Humans Immunoblotting Infant Leigh Disease/diagnosis,genetics Magnetic Resonance Imaging Male Muscle, Skeletal/pathology Point Mutation/genetics Polymerase Chain Reaction Protein Subunits/genetics RNA/analysis Succinate Dehydrogenase/genetics
Chemicals
DNA, Complementary Flavoproteins Protein Subunits RNA DNA Succinate Dehydrogenase
Authors & Affiliations
9 authors, click to expand affiliations / ORCID
Horváth R
Metabolic Disease Centre Munich-Schwabing, Institute of Clinical Chemistry, Academic Hospital Schwabing, Kölner Platz 1, 80804 Munich, Germany. Rita.Horvath@lrz.uni-muenchen.de
Abicht A
Holinski-Feder E
Laner A
Gempel K
Prokisch H
Lochmüller H
Klopstock T
Jaksch M
References (10)
10 references, click to expand
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Article Info
Journal
Journal of neurology, neurosurgery, and psychiatry
Abbr.
J Neurol Neurosurg Psychiatry
ISSN
0022-3050
Published
2006-01-00
Pages
74-6
Language
English
Region
England
NLM ID
2985191R
PMCID
PMC2117401
Subset
IM
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