Literature DB >> 8188292

Correction of the metabolic defect in propionic acidemia fibroblasts by microinjection of a full-length cDNA or RNA transcript encoding the propionyl-CoA carboxylase beta subunit.

A M Lamhonwah1, D Leclerc, M Loyer, R Clarizio, R A Gravel.   

Abstract

Propionyl-CoA carboxylase (PCC) is a mitochondrial, biotin-dependent enzyme, composed of an equal number of alpha and beta subunits, that functions in the catabolism of branched-chain amino acids and other metabolites. Mutations of the PCCA (alpha subunit) or PCCB (beta subunit) gene cause the inherited metabolic disease, propionic acidemia. We report the cloning of a full-length cDNA encoding the beta subunit of human PCC. The open reading frame encodes a pre-beta polypeptide of 539 amino acids (58,205 Da). The cDNA was introduced into the expression vector, pRc/CMV, and microinjected into the nucleus or, as ribotranscripts, into the cytoplasm of fibroblast lines from patients with defects of the beta subunit. The restoration of function was monitored by autoradiography of PCC-dependent [14C]-propionate incorporation into cellular protein. These results confirm the completeness of the clone and demonstrate the capacity for beta subunits derived from the microinjected cDNA or RNA to be transported into mitochondria and assembled with endogenously derived alpha subunits to form functional PCC.

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Year:  1994        PMID: 8188292     DOI: 10.1006/geno.1994.1099

Source DB:  PubMed          Journal:  Genomics        ISSN: 0888-7543            Impact factor:   5.736


  8 in total

1.  Mapping of the ATP2B2 and PCCB genes on porcine chromosome 13.

Authors:  M Van Poucke; A Sjoberg; M Mattheeuws; A Van Zeveren; Y Bouquet; B P Chowdhary; L J Peelman
Journal:  Mamm Genome       Date:  1997       Impact factor: 2.957

2.  Propionyl coenzyme A carboxylase is required for development of Myxococcus xanthus.

Authors:  Y Kimura; R Sato; K Mimura; M Sato
Journal:  J Bacteriol       Date:  1997-11       Impact factor: 3.490

Review 3.  The management and outcome of propionic and methylmalonic acidaemia.

Authors:  J V Leonard
Journal:  J Inherit Metab Dis       Date:  1995       Impact factor: 4.982

4.  Molecular cloning and characterization of two genes for the biotin carboxylase and carboxyltransferase subunits of acetyl coenzyme A carboxylase in Myxococcus xanthus.

Authors:  Y Kimura; R Miyake; Y Tokumasu; M Sato
Journal:  J Bacteriol       Date:  2000-10       Impact factor: 3.490

5.  Human propionyl-CoA carboxylase beta subunit gene: exon-intron definition and mutation spectrum in Spanish and Latin American propionic acidemia patients.

Authors:  P Rodríguez-Pombo; J Hoenicka; S Muro; B Pérez; C Pérez-Cerdá; E Richard; L R Desviat; M Ugarte
Journal:  Am J Hum Genet       Date:  1998-08       Impact factor: 11.025

6.  Transcarboxylase 12S crystal structure: hexamer assembly and substrate binding to a multienzyme core.

Authors:  Pamela R Hall; Yan-Fei Wang; Rosa E Rivera-Hainaj; Xiaojing Zheng; Marianne Pustai-Carey; Paul R Carey; Vivien C Yee
Journal:  EMBO J       Date:  2003-05-15       Impact factor: 11.598

7.  Mutations participating in interallelic complementation in propionic acidemia.

Authors:  R A Gravel; B R Akerman; A M Lamhonwah; M Loyer; A Léon-del-Rio; I Italiano
Journal:  Am J Hum Genet       Date:  1994-07       Impact factor: 11.025

8.  Absence of acidosis in the initial presentation of propionic acidaemia.

Authors:  J H Walter; J E Wraith; M A Cleary
Journal:  Arch Dis Child Fetal Neonatal Ed       Date:  1995-05       Impact factor: 5.747

  8 in total

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