Literature DB >> 16771861

Molecular defects of the C7 gene in two patients with complement C7 deficiency.

Sonia Barroso1, Claudine Rieubland, Antonia José álvarez, Margarita López-Trascasa, Pierre-Alexandre Bart, Antonio Núñez-Roldán, Berta Sánchez.   

Abstract

Different genetic mutations have been described in complement components resulting in total or subtotal deficiency states. In this work we report the genetic basis of C7 deficiency in a previously reported Spanish patient exhibiting a combined total deficiency of C7 and C4B associated with systemic lupus erythematosus. Exon-specific polymerase chain reaction and sequencing revealed a not previously described single base mutation in exon 10 (T1458A) leading to a stop codon that causes the premature truncation of the C7 protein (C464X). Additionally, a C to A transversion at position 1561 (exon 11) was found in the patient resulting in an amino acid change (R499S). This latter mutation has been previously reported in individuals with subtotal C7 deficiency or with combined subtotal C6/C7 deficiency from widely spaced geographical areas. Another novel mutation was found in a second patient with meningococcal meningitis of Bolivian and Czech origin; a 11-base pair deletion of nucleotides 631-641 in exon 6 leading to the generation of a downstream stop codon causing the premature truncation of the C7 protein product (T189 x 193). This patient was found to be a heterozygous compound for another mutation in C7; a two-base pair deletion of nucleotides 1922 and 1923, 1923 and 1924 or 1924 and 1925 in exon 14 (1922delAG/1923delGA/1924delAG), leading again to the generation of a downstream stop codon that provokes the truncation of the C7 protein (S620x630). This latter mutation has been recently reported by our group in another Spanish family. Our results provide more evidences for the heterogeneous molecular basis of C7 deficiency.

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Year:  2006        PMID: 16771861      PMCID: PMC1782290          DOI: 10.1111/j.1365-2567.2006.02364.x

Source DB:  PubMed          Journal:  Immunology        ISSN: 0019-2805            Impact factor:   7.397


  20 in total

Review 1.  Reference typing report for complement components C6, C7 and C9 including mutations leading to deficiencies.

Authors:  R Würzner; K Witzel-Schlömp; K Tokunaga; B A Fernie; M J Hobart; A Orren
Journal:  Exp Clin Immunogenet       Date:  1998

Review 2.  Deficiencies of the complement MAC II gene cluster (C6, C7, C9): is subtotal C6 deficiency of particular evolutionary benefit?

Authors:  R Würzner
Journal:  Clin Exp Immunol       Date:  2003-08       Impact factor: 4.330

3.  Importance of the third thrombospondin repeat of C6 for terminal complement complex assembly.

Authors:  R Würzner; D Mewar; B A Fernie; M J Hobart; P J Lachmann
Journal:  Immunology       Date:  1995-06       Impact factor: 7.397

4.  Molecular bases of combined subtotal deficiencies of C6 and C7: their effects in combination with other C6 and C7 deficiencies.

Authors:  B A Fernie; R Würzner; A Orren; B P Morgan; P C Potter; A E Platonov; I V Vershinina; G A Shipulin; P J Lachmann; M J Hobart
Journal:  J Immunol       Date:  1996-10-15       Impact factor: 5.422

5.  Molecular bases of C7 deficiency: three different defects.

Authors:  B A Fernie; A Orren; G Sheehan; M Schlesinger; M J Hobart
Journal:  J Immunol       Date:  1997-07-15       Impact factor: 5.422

6.  Deficiency of C7 with systemic lupus erythematosus: solubilization of immune complexes in complement-deficient sera.

Authors:  H J Zeitz; G W Miller; T F Lint; M A Ali; H Gewurz
Journal:  Arthritis Rheum       Date:  1981-01

7.  Complement component C7 deficiency in two Spanish families.

Authors:  Sonia Barroso; Berta Sánchez; Antonia José Alvarez; Margarita López-Trascasa; Amparo Lanuza; Rafael Luque; Ingeborg Wichmann; Antonio Núñez-Roldán
Journal:  Immunology       Date:  2004-12       Impact factor: 7.397

Review 8.  Complement deficiency states and infection: epidemiology, pathogenesis and consequences of neisserial and other infections in an immune deficiency.

Authors:  S C Ross; P Densen
Journal:  Medicine (Baltimore)       Date:  1984-09       Impact factor: 1.889

9.  Complement component C7 deficiency in a Spanish family.

Authors:  M F Vázquez-Bermúdez; S Barroso; K Walter; A J Alvarez; A Alarcón; M López-Trascasa; I Wichmann; F Aguilar; A Núñez-Roldán; B Sánchez
Journal:  Clin Exp Immunol       Date:  2003-08       Impact factor: 4.330

10.  Two novel mutations in the C7 gene in a Korean patient with complement C7 deficiency.

Authors:  Chang-Seok Ki; Jong-Won Kim; Hee-Jin Kim; Sung-Min Choi; Gyoung-Yim Ha; Hee Jung Kang; Won-Duck Kim
Journal:  J Korean Med Sci       Date:  2005-04       Impact factor: 2.153

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  1 in total

Review 1.  Human genetics of meningococcal infections.

Authors:  Stephanie Hodeib; Jethro A Herberg; Michael Levin; Vanessa Sancho-Shimizu
Journal:  Hum Genet       Date:  2020-02-17       Impact factor: 4.132

  1 in total

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