Document Detail


An adult onset case of alpha-methyl-acyl-CoA racemase deficiency.
MedLine Citation:
PMID:  20821052     Owner:  NLM     Status:  Publisher    
Abstract/OtherAbstract:
alpha-Methyl-acyl-CoA-racemase (AMACR) deficiency (OMIM 604489) is a rare peroxisomal disorder with a variable age of onset from infancy to late adulthood. We describe a 45-year-old male with a history of seizures who presented with relapsing encephalopathy. Laboratory studies revealed an elevated serum pristanic acid concentration, an elevated pristanic/phytanic acid ratio, as well as the previously described homozygous mutation in the AMACR gene, c.154T>C, consistent with AMACR deficiency. This homozygous mutation is associated with a variable phenotype ranging from neonatal cholestasis to late-onset sensorimotor neuropathy. Dietary pristanic acid restriction was attempted to improve clinical status and the patient has remained in remission for more than 16 months.
Authors:
Emily Helen Smith; Dimitar K Gavrilov; Devin Oglesbee; William D Freeman; Michael W Vavra; Dietrich Matern; Silvia Tortorelli
Publication Detail:
Type:  JOURNAL ARTICLE     Date:  2010-9-4
Journal Detail:
Title:  Journal of inherited metabolic disease     Volume:  -     ISSN:  1573-2665     ISO Abbreviation:  -     Publication Date:  2010 Sep 
Date Detail:
Created Date:  2010-9-7     Completed Date:  -     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  7910918     Medline TA:  J Inherit Metab Dis     Country:  -    
Other Details:
Languages:  ENG     Pagination:  -     Citation Subset:  -    
Affiliation:
Biochemical Genetics Laboratory - Hilton 3-10-02, Department of Laboratory Medicine and Pathology, Mayo Clinic, 200 First Street SW, Rochester, MN, 55905, USA, smith.emily1@mayo.edu.
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