Literature DB >> 2502833

Another Swedish family with complete properdin deficiency: association with fulminant meningococcal disease in one male family member.

C Söderström1, A G Sjöholm, R Svensson, S Ostenson.   

Abstract

Inherited deficiency of the complement component properdin is described in a Swedish family without any previous history of meningococcal infections. The properdin-deficient index patient died from a fulminant infection caused by Neisseria meningitidis serogroup Y. Family investigation revealed properdin deficiency in the patient's half-brother and in the maternal grandfather. The half-brother had a history of pneumococcal pneumonia and meningitis probably caused by Borrelia burgdorferi. Opsonic and bactericidal functions of serum were examined in the half-brother after immunization with tetravalent meningococcal vaccine. Vaccination promoted opsonization of N. meningitidis serogroups C and Y but not of serogroups A and W-135. The serum bactericidal activity increased against serogroup C and to some extent against serogroup W-135. This report emphasizes the importance of investigating the complement system even in families with single cases of fulminant meningococcal disease. Individuals with properdin deficiency might be protected from infection by immunization.

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Year:  1989        PMID: 2502833     DOI: 10.3109/00365548909035695

Source DB:  PubMed          Journal:  Scand J Infect Dis        ISSN: 0036-5548


  5 in total

1.  Properdin deficiency in a large Swiss family: identification of a stop codon in the properdin gene, and association of meningococcal disease with lack of the IgG2 allotype marker G2m(n).

Authors:  P J Späth; A G Sjöholm; G N Fredrikson; G Misiano; R Scherz; U B Schaad; B Uhring-Lambert; G Hauptmann; J Westberg; M Uhlén; C Wadelius; L Truedsson
Journal:  Clin Exp Immunol       Date:  1999-11       Impact factor: 4.330

2.  Meningococcal group W-135 and Y capsular polysaccharides paradoxically enhance activation of the alternative pathway of complement.

Authors:  Sanjay Ram; Lisa A Lewis; Sarika Agarwal
Journal:  J Biol Chem       Date:  2011-01-18       Impact factor: 5.157

Review 3.  Infectious diseases associated with complement deficiencies.

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

Review 4.  Factor H and neisserial pathogenesis.

Authors:  Jo Anne Welsch; Sanjay Ram
Journal:  Vaccine       Date:  2008-12-30       Impact factor: 3.641

5.  Loss of properdin exacerbates C3 glomerulopathy resulting from factor H deficiency.

Authors:  Marieta M Ruseva; Katherine A Vernon; Allison M Lesher; Wilhelm J Schwaeble; Youssif M Ali; Marina Botto; Terence Cook; Wenchao Song; Cordula M Stover; Matthew Caleb Pickering
Journal:  J Am Soc Nephrol       Date:  2012-11-26       Impact factor: 14.978

  5 in total

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