Document Detail


Absence of the G1528C (E474Q) mutation in the alpha-subunit of the mitochondrial trifunctional protein in women with acute fatty liver of pregnancy.
MedLine Citation:
PMID:  11978893     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Acute fatty liver of pregnancy (AFLP) is a rare and dreaded complication of pregnancy, almost exclusively seen in the third trimester. The histopathologic features of AFLP closely resemble those seen in metabolic disorders characterized by deficiency of fatty acid oxidative enzymes. Several reports have established a strong association between AFLP in the mother and fetal deficiency of the enzyme long-chain L-3-hydroxyacyl-CoA dehydrogenase (LCHAD). However, these studies have an inevitable selection bias resulting from ascertainment through an affected infant, rather than an unselected population of patients with AFLP. We retrospectively examined a series of 10 women with pregnancies complicated by AFLP to determine the prevalence of the common LCHAD mutation (G1528C) in this population. The existing LCHAD primers, which produce a 640-bp amplicon (IJlst L, Ruiter JP, Hoovers JM, Jakobs ME, Wanders RJ: J Clin Invest 98:1028-1033, 1996), were modified to make them amenable to analysis of fragmented DNA obtained from microdissected formalin-fixed material. None of the patients were found to harbor the common G1528C mutation. It is likely that AFLP arising in the context of fetal LCHAD deficiency represents only one of the possible etiologies for this uncommon disorder, and the metabolic basis of AFLP is more heterogeneous than previously believed.
Authors:
Anibran Maitra; Rana Domiati-Saad; Nicole Yost; Gary Cunningham; Beverly Barton Rogers; Michael J Bennett
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Publication Detail:
Type:  Journal Article; Review    
Journal Detail:
Title:  Pediatric research     Volume:  51     ISSN:  0031-3998     ISO Abbreviation:  Pediatr. Res.     Publication Date:  2002 May 
Date Detail:
Created Date:  2002-04-29     Completed Date:  2002-10-25     Revised Date:  2005-11-16    
Medline Journal Info:
Nlm Unique ID:  0100714     Medline TA:  Pediatr Res     Country:  United States    
Other Details:
Languages:  eng     Pagination:  658-61     Citation Subset:  IM    
Affiliation:
Department of Pathology, University of Texas Southwestern Medical Center, Dallas, Texas 75390, USA.
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MeSH Terms
Descriptor/Qualifier:
3-Hydroxyacyl CoA Dehydrogenases / deficiency,  genetics*
Acute Disease
Adult
Amino Acid Substitution*
DNA Mutational Analysis
Fatty Liver / enzymology*,  epidemiology,  genetics
Female
Humans
Mitochondria / enzymology
Multienzyme Complexes / genetics*
Mutation, Missense*
Point Mutation*
Pregnancy
Pregnancy Complications / enzymology*,  epidemiology
Protein Subunits
Retrospective Studies
Texas / epidemiology
Chemical
Reg. No./Substance:
0/Multienzyme Complexes; 0/Protein Subunits; 0/fatty acid beta-oxidation multienzyme complex; EC 1.1.1.211/long-chain 3-hydroxyacyl CoA dehydrogenase; EC 1.1.1.35/3-Hydroxyacyl CoA Dehydrogenases

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


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