Homologous dinucleotide (GT or TG) deletion in Japanese patients with chronic granulomatous disease with p47-phox deficiency

Biochem Biophys Res Commun. 1994 Mar 30;199(3):1372-7. doi: 10.1006/bbrc.1994.1382.

Abstract

The cytosolic 47-kDa protein designated as p47-phox (phagocyte oxidase) is one of the essential components of the superoxide-generating system in phagocytes, and its defect is known to cause chronic granulomatous disease (CGD). Five unrelated CGD patients with p47-phox deficiency were found among 82 CGD patients in Japan. We sequenced the cDNAs and the genomic DNAs corresponding to p47-phox derived from these patients. In all cases examined, the defect was identified to be a GT (or TG) dinucleotide deletion at bases 75/76 (or 74/75, respectively) in the coding sequence for the protein. The same mutation was reported previously for a total of 9 alleles from 5 CGD patients in England and in the United States. It seems, therefore, that the dinucleotide GT deletion is the common mutation in 47-phox deficient CGD due to certain structural issues.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • B-Lymphocytes / enzymology
  • Base Sequence
  • Cell Line, Transformed
  • DNA Primers
  • Female
  • Granulomatous Disease, Chronic / enzymology*
  • Granulomatous Disease, Chronic / genetics*
  • Herpesvirus 4, Human / genetics
  • Humans
  • Japan
  • Male
  • Molecular Sequence Data
  • NADPH Dehydrogenase / deficiency*
  • NADPH Dehydrogenase / genetics*
  • NADPH Oxidases
  • Oligonucleotides*
  • Phosphoproteins / deficiency*
  • Phosphoproteins / genetics*
  • Polymerase Chain Reaction
  • Sequence Deletion*

Substances

  • DNA Primers
  • Oligonucleotides
  • Phosphoproteins
  • NADPH Oxidases
  • neutrophil cytosolic factor 1
  • NADPH Dehydrogenase