Document Detail


Carrier detection of pyruvate carboxylase deficiency in fibroblasts and lymphocytes.
MedLine Citation:
PMID:  116187     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Pyruvate carboxylase (E.C. 6.4.1.1) activity was determined in the circulating peripheral lymphocytes and cultured skin fibroblasts from the family of a patient with hepatic, cerebral, renal cortical, leukocyte, and fibroblast pyruvate carboxylase deficiency (PC Portland deficiency). Lymphocyte activities were: mother, 33--39%; father, 11--29%; brother, 82--103%; and sister, 38--48% of the lowest normal. Fibroblasts from the patient's mother and father had 42 and 34%, respectively, of the activity of the lowest normal. These data demonstrate that the disease is inherited in an autosomal recessive manner and that lymphocytes and fibroblasts can be used to detect carriers. Neither pyruvate carboxylase nor mitochondrial PEPCK activity in lymphocytes was increased by a 21-hr fast.
Authors:
B M Atkin
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Publication Detail:
Type:  Journal Article    
Journal Detail:
Title:  Pediatric research     Volume:  13     ISSN:  0031-3998     ISO Abbreviation:  Pediatr. Res.     Publication Date:  1979 Oct 
Date Detail:
Created Date:  1980-01-28     Completed Date:  1980-01-28     Revised Date:  2007-11-15    
Medline Journal Info:
Nlm Unique ID:  0100714     Medline TA:  Pediatr Res     Country:  UNITED STATES    
Other Details:
Languages:  eng     Pagination:  1101-4     Citation Subset:  IM    
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MeSH Terms
Descriptor/Qualifier:
Adolescent
Adult
Cells, Cultured
Child
Child, Preschool
Citrate (si)-Synthase / metabolism
Clinical Enzyme Tests*
Fasting
Female
Fibroblasts / enzymology*
Heterozygote Detection / methods*
Humans
Infant
Infant, Newborn
Lymphocytes / enzymology*,  ultrastructure
Male
Middle Aged
Mitochondria / enzymology
Phosphoenolpyruvate Carboxykinase (GTP) / metabolism
Pyruvate Carboxylase / metabolism
Pyruvate Carboxylase Deficiency Disease*
Chemical
Reg. No./Substance:
EC 2.3.3.1/Citrate (si)-Synthase; EC 4.1.1.32/Phosphoenolpyruvate Carboxykinase (GTP); EC 6.4.1.1/Pyruvate Carboxylase

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


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