Literature DB >> 2037281

Genetic heterogeneity of propionic acidemia: analysis of 15 Japanese patients.

T Ohura1, S Miyabayashi, K Narisawa, K Tada.   

Abstract

Propionic acidemia is an autosomal recessive metabolic disease resulting from a deficiency of propionyl CoA carboxylase (PCC) activity. We have analyzed the molecular heterogeneity of Japanese propionic acidemia patients using anti-human PCC antiserum and cDNA clones coding for the two protein subunits (alpha and beta) of the enzyme. The steady state levels of both alpha and beta subunits of PCC from 15 Japanese patients were determined by Western blot. Three patients had neither alpha nor beta subunits, and the amounts of both alpha and beta subunits were low in 3 other patients. According to our previous data, we classified these 6 patients as having alpha subunit deficiency. In the remaining 8 patients, alpha subunits were normal, but the beta subunits were aberrant. Two patients had low levels of normal-sized beta subunits and 6 had beta subunits smaller than normal in size and greatly reduced in quantity. These 8 patients were assigned to the beta subunit deficiency category. One patient had apparently normal alpha and beta subunits. We could not determine this patient's primary defect. These data reveal the genetic heterogeneity of molecular defects causing propionic acidemia in the Japanese. Southern blot analysis did not reveal any gross alteration in gene structure when DNA was digested with HindIII, EcoRI and TaqI. However, DNA from 3 beta-subunit-deficient patients, when digested with MspI and probed with beta PCC cDNA, revealed a unique 2.7-kb band not observed in blots of DNA from any other patient or 15 normal controls. We conclude that this altered MspI restriction map is the result of a mutation in the beta subunit gene of these patients.

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Year:  1991        PMID: 2037281     DOI: 10.1007/bf01213089

Source DB:  PubMed          Journal:  Hum Genet        ISSN: 0340-6717            Impact factor:   4.132


  13 in total

1.  Genetic complementation of propionyl-CoA carboxylase deficiency in cultured human fibroblasts.

Authors:  R A Gravel; K F Lam; K J Scully; Y Hsia
Journal:  Am J Hum Genet       Date:  1977-07       Impact factor: 11.025

2.  Primer-directed enzymatic amplification of DNA with a thermostable DNA polymerase.

Authors:  R K Saiki; D H Gelfand; S Stoffel; S J Scharf; R Higuchi; G T Horn; K B Mullis; H A Erlich
Journal:  Science       Date:  1988-01-29       Impact factor: 47.728

3.  Isolation of cDNA clones coding for the alpha and beta chains of human propionyl-CoA carboxylase: chromosomal assignments and DNA polymorphisms associated with PCCA and PCCB genes.

Authors:  A M Lamhonwah; T J Barankiewicz; H F Willard; D J Mahuran; F Quan; R A Gravel
Journal:  Proc Natl Acad Sci U S A       Date:  1986-07       Impact factor: 11.205

4.  Electrophoretic transfer of proteins from polyacrylamide gels to nitrocellulose sheets: procedure and some applications.

Authors:  H Towbin; T Staehelin; J Gordon
Journal:  Proc Natl Acad Sci U S A       Date:  1979-09       Impact factor: 11.205

5.  Propionicacidemia (ketotic hyperglycinemia): dietary treatment resulting in normal growth and development.

Authors:  I K Brandt; Y E Hsia; D H Clement; S A Provence
Journal:  Pediatrics       Date:  1974-03       Impact factor: 7.124

6.  Direct identification of sickle cell anemia by blot hybridization.

Authors:  R F Geever; L B Wilson; F S Nallaseth; P F Milner; M Bittner; J T Wilson
Journal:  Proc Natl Acad Sci U S A       Date:  1981-08       Impact factor: 11.205

7.  Isolation and characterization of propionyl-CoA carboxylase from normal human liver. Evidence for a protomeric tetramer of nonidentical subunits.

Authors:  F Kalousek; M D Darigo; L E Rosenberg
Journal:  J Biol Chem       Date:  1980-01-10       Impact factor: 5.157

8.  Asymptomatic propionyl CoA carboxylase deficiency in a 13-year-old girl.

Authors:  B Wolf; E P Paulsen; Y E Hsia
Journal:  J Pediatr       Date:  1979-10       Impact factor: 4.406

9.  Biogenesis of the mitochondrial enzyme methylmalonyl-CoA mutase. Synthesis and processing of a precursor in a cell-free system and in cultured cells.

Authors:  W A Fenton; A M Hack; D Helfgott; L E Rosenberg
Journal:  J Biol Chem       Date:  1984-05-25       Impact factor: 5.157

10.  Unequal synthesis and differential degradation of propionyl CoA carboxylase subunits in cells from normal and propionic acidemia patients.

Authors:  T Ohura; J P Kraus; L E Rosenberg
Journal:  Am J Hum Genet       Date:  1989-07       Impact factor: 11.025

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  8 in total

1.  Mutation analysis of the propionyl-CoA carboxylase alpha-subunit gene in four Japanese patients with propionic acidaemia.

Authors:  T Ohura; K Narisawa; K Iinuma
Journal:  J Inherit Metab Dis       Date:  1999-10       Impact factor: 4.982

2.  An 84 bp insertion found in a propionic acidaemia patient is not a disease-causing mutation but a product of cryptic mRNA.

Authors:  T Ohura; K Narisawa; K Tada; K Iinuma
Journal:  J Inherit Metab Dis       Date:  1999-06       Impact factor: 4.982

3.  High incidence of propionic acidemia in greenland is due to a prevalent mutation, 1540insCCC, in the gene for the beta-subunit of propionyl CoA carboxylase.

Authors:  K Ravn; M Chloupkova; E Christensen; N J Brandt; H Simonsen; J P Kraus; I M Nielsen; F Skovby; M Schwartz
Journal:  Am J Hum Genet       Date:  2000-05-16       Impact factor: 11.025

4.  A novel splicing mutation in propionic acidemia associated with a tetranucleotide direct repeat in the PCCB gene.

Authors:  T Ohura; K Narisawa; K Tada; K Iinuma
Journal:  Hum Genet       Date:  1995-06       Impact factor: 4.132

5.  Propionic acidaemia: sequence analysis of mutant mRNAs from Japanese beta subunit-deficient patients.

Authors:  T Ohura; K Narisawa; K Tada
Journal:  J Inherit Metab Dis       Date:  1993       Impact factor: 4.982

6.  The molecular defect in propionic acidemia: exon skipping caused by an 8-bp deletion from an intron in the PCCB allele.

Authors:  T Ohura; M Ogasawara; H Ikeda; K Narisawa; K Tada
Journal:  Hum Genet       Date:  1993-10       Impact factor: 4.132

Review 7.  Methylmalonic and propionic acidemias: clinical management update.

Authors:  Jamie L Fraser; Charles P Venditti
Journal:  Curr Opin Pediatr       Date:  2016-12       Impact factor: 2.856

8.  Three independent mutations in the same exon of the PCCB gene: differences between Caucasian and Japanese propionic acidaemia.

Authors:  T Tahara; J P Kraus; T Ohura; L E Rosenberg; W A Fenton
Journal:  J Inherit Metab Dis       Date:  1993       Impact factor: 4.982

  8 in total

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