Literature DB >> 2532396

Hereditary, complete deficiency of complement factor H associated with recurrent meningococcal disease.

H E Nielsen1, K C Christensen, C Koch, B S Thomsen, N H Heegaard, J Tranum-Jensen.   

Abstract

Complement factor H (beta-1H globulin) is an important regulatory protein which inhibits the spontaneous complement activation via the alternative pathway. We describe a 15-year-old girl without any detectable factor H in plasma. She has had two episodes of meningococcal disease, but is otherwise completely healthy. Secondary to the factor-H deficiency, the levels of factor B, properdin, C3, and C5-C9 were strongly reduced due to spontaneous in vivo activation of the alternative complement pathway. Plasma C3dg was strongly elevated in spite of the factor-H deficiency; apparently erythrocyte CR1 substitutes for factor H in C3 degradation. Neither C3 nor complement lesions were demonstrable on her erythrocytes which did, however, show increased, spontaneous haemolysis in vitro in citrate plasma, but not in serum. The patient is a single child and her parents, who are unrelated and healthy, had half-normal levels of factor H. This reduction of factor H is sufficient to cause increased, spontaneous activation of the alternative pathway.

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Year:  1989        PMID: 2532396     DOI: 10.1111/j.1365-3083.1989.tb02480.x

Source DB:  PubMed          Journal:  Scand J Immunol        ISSN: 0300-9475            Impact factor:   3.487


  17 in total

1.  The molecular basis for hereditary porcine membranoproliferative glomerulonephritis type II: point mutations in the factor H coding sequence block protein secretion.

Authors:  Guido A Hegasy; Tamara Manuelian; Kolbjorn Hogasen; Johan H Jansen; Peter F Zipfel
Journal:  Am J Pathol       Date:  2002-12       Impact factor: 4.307

2.  Vaccines, reverse vaccinology, and bacterial pathogenesis.

Authors:  Isabel Delany; Rino Rappuoli; Kate L Seib
Journal:  Cold Spring Harb Perspect Med       Date:  2013-05-01       Impact factor: 6.915

3.  Characterization of diverse subvariants of the meningococcal factor H (fH) binding protein for their ability to bind fH, to mediate serum resistance, and to induce bactericidal antibodies.

Authors:  Kate L Seib; Brunella Brunelli; Barbara Brogioni; Emmanuelle Palumbo; Stefania Bambini; Alessandro Muzzi; Federica DiMarcello; Sara Marchi; Arie van der Ende; Beatrice Aricó; Silvana Savino; Maria Scarselli; Maurizio Comanducci; Rino Rappuoli; Marzia M Giuliani; Mariagrazia Pizza
Journal:  Infect Immun       Date:  2010-12-13       Impact factor: 3.441

Review 4.  Infections of people with complement deficiencies and patients who have undergone splenectomy.

Authors:  Sanjay Ram; Lisa A Lewis; Peter A Rice
Journal:  Clin Microbiol Rev       Date:  2010-10       Impact factor: 26.132

5.  Factor H mutations in hemolytic uremic syndrome cluster in exons 18-20, a domain important for host cell recognition.

Authors:  A Richards; M R Buddles; R L Donne; B S Kaplan; E Kirk; M C Venning; C L Tielemans; J A Goodship; T H Goodship
Journal:  Am J Hum Genet       Date:  2001-01-17       Impact factor: 11.025

6.  Factor H-binding protein is important for meningococcal survival in human whole blood and serum and in the presence of the antimicrobial peptide LL-37.

Authors:  K L Seib; D Serruto; F Oriente; I Delany; J Adu-Bobie; D Veggi; B Aricò; R Rappuoli; M Pizza
Journal:  Infect Immun       Date:  2008-10-13       Impact factor: 3.441

Review 7.  aHUS caused by complement dysregulation: new therapies on the horizon.

Authors:  Aoife M Waters; Christoph Licht
Journal:  Pediatr Nephrol       Date:  2010-06-18       Impact factor: 3.714

Review 8.  Infectious diseases associated with complement deficiencies.

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

Review 9.  Translational mini-review series on complement factor H: renal diseases associated with complement factor H: novel insights from humans and animals.

Authors:  M C Pickering; H T Cook
Journal:  Clin Exp Immunol       Date:  2008-02       Impact factor: 4.330

10.  Hereditary porcine membranoproliferative glomerulonephritis type II is caused by factor H deficiency.

Authors:  K Høgåsen; J H Jansen; T E Mollnes; J Hovdenes; M Harboe
Journal:  J Clin Invest       Date:  1995-03       Impact factor: 14.808

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