Literature DB >> 1710153

Point mutations in the beta-subunit of cytochrome b558 leading to X-linked chronic granulomatous disease.

B G Bolscher1, M de Boer, A de Klein, R S Weening, D Roos.   

Abstract

The NADPH:O2 oxidoreductase of phagocytic leukocytes is an important enzyme for the bactericidal activity of these cells. Cytochrome b558 is a membrane component of this enzyme. In X-linked chronic granulomatous disease (Xb- CGD) the phagocytes are defective in the beta-subunit (gp91-phox) of this cytochrome. We have studied the genetic defect in a group of six X-linked CGD patients characterized by complete or partial loss of cytochrome b558 with the use of the polymerase chain reaction. All patients had a different single point mutation in the gp91-phox gene, indicating that the genetic defect in Xb- CGD is very heterogeneous. In one patient the mutation leads to a premature termination codon. In the other five cases these mutations predict incorporation of a different amino acid. The mutations were with one exception found in the N-terminal half of the protein, suggesting that this part of cytochrome b558 is important for the binding of the heme or for formation of a stable complex with p22-phox. Two histidyl residues were found that might be ligands of the heme iron.

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Year:  1991        PMID: 1710153

Source DB:  PubMed          Journal:  Blood        ISSN: 0006-4971            Impact factor:   22.113


  28 in total

Review 1.  Hematologically important mutations: X-linked chronic granulomatous disease (third update).

Authors:  Dirk Roos; Douglas B Kuhns; Anne Maddalena; Joachim Roesler; Juan Alvaro Lopez; Tadashi Ariga; Tadej Avcin; Martin de Boer; Jacinta Bustamante; Antonio Condino-Neto; Gigliola Di Matteo; Jianxin He; Harry R Hill; Steven M Holland; Caroline Kannengiesser; M Yavuz Köker; Irina Kondratenko; Karin van Leeuwen; Harry L Malech; László Marodi; Hiroyuki Nunoi; Marie-José Stasia; Anna Maria Ventura; Carl T Witwer; Baruch Wolach; John I Gallin
Journal:  Blood Cells Mol Dis       Date:  2010-08-21       Impact factor: 3.039

2.  X-Linked chronic granulomatous disease: mutations in the CYBB gene encoding the gp91-phox component of respiratory-burst oxidase.

Authors:  J Rae; P E Newburger; M C Dinauer; D Noack; P J Hopkins; R Kuruto; J T Curnutte
Journal:  Am J Hum Genet       Date:  1998-06       Impact factor: 11.025

3.  Analysis of glycosylation sites on gp91phox, the flavocytochrome of the NADPH oxidase, by site-directed mutagenesis and translation in vitro.

Authors:  T M Wallach; A W Segal
Journal:  Biochem J       Date:  1997-02-01       Impact factor: 3.857

4.  A family with complement factor D deficiency.

Authors:  D H Biesma; A J Hannema; H van Velzen-Blad; L Mulder; R van Zwieten; I Kluijt; D Roos
Journal:  J Clin Invest       Date:  2001-07       Impact factor: 14.808

5.  A point mutation in gp91-phox of cytochrome b558 of the human NADPH oxidase leading to defective translocation of the cytosolic proteins p47-phox and p67-phox.

Authors:  J H Leusen; M de Boer; B G Bolscher; P M Hilarius; R S Weening; H D Ochs; D Roos; A J Verhoeven
Journal:  J Clin Invest       Date:  1994-05       Impact factor: 14.808

6.  Hereditary eosinophil peroxidase deficiency: immunochemical and spectroscopic studies and evidence for a compound heterozygosity of the defect.

Authors:  M Romano; P Patriarca; C Melo; F E Baralle; P Dri
Journal:  Proc Natl Acad Sci U S A       Date:  1994-12-20       Impact factor: 11.205

7.  A newly recognized point mutation in the cytochrome b558 heavy chain gene replacing alanine57 by glutamic acid, in a patient with cytochrome b positive X-linked chronic granulomatous disease.

Authors:  T Ariga; Y Sakiyama; K Tomizawa; S Imajoh-Ohmi; S Kanegasaki; S Matsumoto
Journal:  Eur J Pediatr       Date:  1993-06       Impact factor: 3.183

8.  Identification of allele-specific p22-phox mutations in a compound heterozygous patient with chronic granulomatous disease by mismatch PCR and restriction enzyme analysis.

Authors:  J P Hossle; M de Boer; R A Seger; D Roos
Journal:  Hum Genet       Date:  1994-04       Impact factor: 4.132

9.  Mutations in the promoter region of the gene for gp91-phox in X-linked chronic granulomatous disease with decreased expression of cytochrome b558.

Authors:  P E Newburger; D G Skalnik; P J Hopkins; E A Eklund; J T Curnutte
Journal:  J Clin Invest       Date:  1994-09       Impact factor: 14.808

10.  Point mutation in the cytoplasmic domain of the neutrophil p22-phox cytochrome b subunit is associated with a nonfunctional NADPH oxidase and chronic granulomatous disease.

Authors:  M C Dinauer; E A Pierce; R W Erickson; T J Muhlebach; H Messner; S H Orkin; R A Seger; J T Curnutte
Journal:  Proc Natl Acad Sci U S A       Date:  1991-12-15       Impact factor: 11.205

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