Literature DB >> 2212005

Molecular basis of hereditary C3 deficiency.

M Botto1, K Y Fong, A K So, A Rudge, M J Walport.   

Abstract

Hereditary deficiency of complement component C3 in a 10-yr-old boy was studied. C3 could not be detected by RIA of serum from the patient. Segregation of C3 S and C3 F allotypes within the family confirmed the presence of a null gene for C3, for which the patient was homozygous. 30 exons have been characterized, spanning the entire beta chain of C3 and the alpha chain as far as the C3d region. Sequence analysis of the exons derived from the C3 null gene showed no abnormalities in the coding sequences. A GT-AT mutation at the 5' donor splice site of the intervening sequence 18 was found in the C3 null gene. Exons 17-21 were amplified by the polymerase chain reaction (PCR) from first-strand cDNA synthesized from mRNA obtained from peripheral blood monocytes stimulated with LPS. This revealed a 61-bp deletion in exon 18, resulting from splicing of a cryptic 5' donor splice site in exon 18 with the normal 3' splice site in exon 19. This deletion leads to a disturbance of the reading frame of the mRNA with a stop codon 17 bp downstream from the abnormal splice in exon 18. His parents had both the normal and abnormal C3 mRNA and were shown to be heterozygous for this mutation by sequence analysis of genomic DNA amplified by PCR. Similar splice mutants have previously been reported in the beta-globin, phenylalanine hydroxylase, and porphobilinogen deaminase genes. This mutation is sufficient to cause the deficiency of C3 in the patient.

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Year:  1990        PMID: 2212005      PMCID: PMC296845          DOI: 10.1172/JCI114821

Source DB:  PubMed          Journal:  J Clin Invest        ISSN: 0021-9738            Impact factor:   14.808


  35 in total

1.  Genomic organization of human complement component C3.

Authors:  K Y Fong; M Botto; M J Walport; A K So
Journal:  Genomics       Date:  1990-08       Impact factor: 5.736

2.  Linkage of beta-thalassaemia mutations and beta-globin gene polymorphisms with DNA polymorphisms in human beta-globin gene cluster.

Authors:  S H Orkin; H H Kazazian; S E Antonarakis; S C Goff; C D Boehm; J P Sexton; P G Waber; P J Giardina
Journal:  Nature       Date:  1982-04-15       Impact factor: 49.962

3.  Assignment of the structural gene for the third component of human complement to chromosome 19.

Authors:  A S Whitehead; E Solomon; S Chambers; W F Bodmer; S Povey; G Fey
Journal:  Proc Natl Acad Sci U S A       Date:  1982-08       Impact factor: 11.205

4.  A catalogue of splice junction sequences.

Authors:  S M Mount
Journal:  Nucleic Acids Res       Date:  1982-01-22       Impact factor: 16.971

5.  Complete absence of the third component of complement in a patient with repeated infections.

Authors:  K H Hsieh; C Y Lin; T C Lee
Journal:  Clin Immunol Immunopathol       Date:  1981-09

6.  A single-base change at a splice site in a beta 0-thalassemic gene causes abnormal RNA splicing.

Authors:  R Treisman; N J Proudfoot; M Shander; T Maniatis
Journal:  Cell       Date:  1982-07       Impact factor: 41.582

7.  Ovalbumin gene: evidence for a leader sequence in mRNA and DNA sequences at the exon-intron boundaries.

Authors:  R Breathnach; C Benoist; K O'Hare; F Gannon; P Chambon
Journal:  Proc Natl Acad Sci U S A       Date:  1978-10       Impact factor: 11.205

8.  Inherited deficiency of the third component of complement associated with recurrent pyogenic infections, circulating immune complexes, and vasculitis in a Dutch family.

Authors:  J J Roord; M Daha; W Kuis; H A Verbrugh; J Verhoef; B J Zegers; J W Stoop
Journal:  Pediatrics       Date:  1983-01       Impact factor: 7.124

9.  A nucleotide change at a splice junction in the human beta-globin gene is associated with beta 0-thalassemia.

Authors:  M Baird; C Driscoll; H Schreiner; G V Sciarratta; G Sansone; G Niazi; F Ramirez; A Bank
Journal:  Proc Natl Acad Sci U S A       Date:  1981-07       Impact factor: 11.205

10.  Hereditary deficiency of the third component of complement in two sisters with systemic lupus erythematosus-like symptoms.

Authors:  Y Sano; H Nishimukai; H Kitamura; K Nagaki; S Inai; Y Hamasaki; I Maruyama; A Igata
Journal:  Arthritis Rheum       Date:  1981-10
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  18 in total

1.  A new assay to monitor the degranulation process in phagocytizing human neutrophils.

Authors:  Violetta Borelli; Maria Giovanna Perrotta; Francesca Vita; Maria Rosa Soranzo; Giuliano Zabucchi
Journal:  Inflammation       Date:  2002-02       Impact factor: 4.092

2.  Homozygous hereditary C3 deficiency due to a premature stop codon.

Authors:  Edimara Da Silva Reis; Gisele Vanessa Baracho; Adriana Sousa Lima; Chuck S Farah; Lourdes Isaac
Journal:  J Clin Immunol       Date:  2002-11       Impact factor: 8.317

Review 3.  Immunological loss-of-function due to genetic gain-of-function in humans: autosomal dominance of the third kind.

Authors:  Bertrand Boisson; Pierre Quartier; Jean-Laurent Casanova
Journal:  Curr Opin Immunol       Date:  2015-01-31       Impact factor: 7.486

4.  Carrier detection in families with properdin deficiency by microsatellite haplotyping.

Authors:  K Kölble; A J Cant; A C Fay; K Whaley; M Schlesinger; K B Reid
Journal:  J Clin Invest       Date:  1993-01       Impact factor: 14.808

5.  An association between homozygous C3 deficiency and low levels of anti-pneumococcal capsular polysaccharide antibodies.

Authors:  M A Hazlewood; D S Kumararatne; A D Webster; M Goodall; P Bird; M Daha
Journal:  Clin Exp Immunol       Date:  1992-03       Impact factor: 4.330

6.  Homozygous hereditary C3 deficiency due to a partial gene deletion.

Authors:  M Botto; K Y Fong; A K So; R Barlow; R Routier; B J Morley; M J Walport
Journal:  Proc Natl Acad Sci U S A       Date:  1992-06-01       Impact factor: 11.205

Review 7.  Infectious diseases associated with complement deficiencies.

Authors:  J E Figueroa; P Densen
Journal:  Clin Microbiol Rev       Date:  1991-07       Impact factor: 26.132

8.  Compound heterozygous complement C3 deficiency.

Authors:  Y Katz; R A Wetsel; M Schlesinger; Z Fishelson
Journal:  Immunology       Date:  1995-01       Impact factor: 7.397

9.  Intrinsic differences between authentic and cryptic 5' splice sites.

Authors:  Xavier Roca; Ravi Sachidanandam; Adrian R Krainer
Journal:  Nucleic Acids Res       Date:  2003-11-01       Impact factor: 16.971

10.  Nonsense-codon-mediated decay in human hereditary complement C3 deficiency.

Authors:  Edimara S Reis; Victor Nudelman; Lourdes Isaac
Journal:  Immunogenetics       Date:  2003-11-25       Impact factor: 2.846

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