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Prepublished online as a Blood First Edition Paper on August 28, 2003; DOI 10.1182/blood-2003-04-1065. This Article Full Text (PDF) All Versions of this Article: 2003-04-1065v1 103/2/673    most recent Alert me when this article is cited Alert me if a correction is posted Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Download to citation manager Rights and Permissions Citing Articles Citing Articles via HighWire Citing Articles via CrossRef Citing Articles via Google Scholar Google Scholar Articles by Hoeflich, C. Articles by Suttorp, N. Search for Related Content PubMed PubMed Citation Articles by Hoeflich, C. Articles by Suttorp, N. Social Bookmarking                   What's this? Submitted April 7, 2003 Accepted August 17, 2003 Naturally occurring anti-IFN auto-antibody and severe infections with Mycobacterium cheloneae and Burkholderia cocovenenans Conny Hoeflich*, Robert Sabat, Simone Rosseau, Bettina Temmesfeld, Hortense Slevogt, Wolf-Dietrich Docke, Gerald Gruetz, Christian Meisel, Elke Halle, Ulf B Goebel, Hans-Dieter Volk, and Norbert Suttorp Institute for Medical Immunology, University Hospital Charite, Humboldt University, Berlin, Germany Department of Internal Medicine, University Hospital Charite, Humboldt University, Berlin, Germany Institute for Microbiology, University Hospital Charite, Humboldt University, Berlin, Germany * Corresponding author; email: conny.hoeflich{at}charite.de. Recently various genetic defects in IFN mediated immunity have been described including mutations in the interferon- receptor 1 (IFNgR1) and receptor 2 (IFNgR2), Stat1 and IL-12 receptor beta 1 (IL-12Rb1) and IL-12 p40 genes. These mutations are associated with the occurrence of severe infections with intracellular pathogens especially nontuberculous mycobacteria and vaccine-associated BCG. Here we report on a previously healthy adult patient primarilary presenting with severe infections with Burkholderia cocovenenans and subsequently Mycobacterium cheloneae. We found a strong inhibitory anti-IFN activity in the patient's plasma and identified a high-affinity neutralizing anti-IFN autoantibody. Unfortunately, the patient died due to severe sepsis before knowing the nature of the inhibitory activity. The application of alternative therapeutic approaches such as IVIG or immunoadsorption may have been beneficial in this case. Screening for neutralizing anti-IFN autoantibodies should supplement testing for IFN- and IL-12 pathway defects in patients with recurrent infections with intracellular pathogens, especially with NTM. CiteULike    Connotea    Del.icio.us    Digg    Reddit    Technorati    What's this? This article has been cited by other articles: M. Buckland, R. Ali, G. Bothamley, D. Lammas, R. Doffinger, and H. Longhurst Recurrent mycobacterial infections in a patient with IL-12 deficiency BMJ Case Reports, December 1, 2009; 2009(dec01_1): bcr1120081278 - bcr1120081278. [Abstract] [Full Text] S Y Patel, R Doffinger, G Barcenas-Morales, and D S Kumararatne Genetically determined susceptibility to mycobacterial infection J. Clin. Pathol., September 1, 2008; 61(9): 1006 - 1012. [Abstract] [Full Text] [PDF] P. Sexton and A. C. Harrison Susceptibility to nontuberculous mycobacterial lung disease Eur. Respir. J., June 1, 2008; 31(6): 1322 - 1333. [Abstract] [Full Text] [PDF] A. Puel, C. Picard, M. Lorrot, C. Pons, M. Chrabieh, L. Lorenzo, M. Mamani-Matsuda, E. Jouanguy, D. Gendrel, and J.-L. Casanova Recurrent Staphylococcal Cellulitis and Subcutaneous Abscesses in a Child with Autoantibodies against IL-6 J. Immunol., January 1, 2008; 180(1): 647 - 654. [Abstract] [Full Text] [PDF] G. Plitas, U. I. Chaudhry, T. P. Kingham, J. R. Raab, and R. P. DeMatteo NK Dendritic Cells Are Innate Immune Responders to Listeria monocytogenes Infection J. Immunol., April 1, 2007; 178(7): 4411 - 4416. [Abstract] [Full Text] [PDF] P. Kelleher, A. Goodsall, A. Mulgirigama, H. Kunst, D. C. Henderson, R. Wilson, A. Newman-Taylor, and M. Levin Interferon-{gamma} therapy in two patients with progressive chronic pulmonary aspergillosis. Eur. Respir. J., June 1, 2006; 27(6): 1307 - 1310. [Abstract] [Full Text] [PDF] A. E. Medvedev, I. Sabroe, J. D. Hasday, and S. N. Vogel Invited review: Tolerance to microbial TLR ligands: molecular mechanisms and relevance to disease Innate Immunity, June 1, 2006; 12(3): 133 - 150. [Abstract] [PDF] S. Y. Patel, L. Ding, M. R. Brown, L. Lantz, T. Gay, S. Cohen, L. A. Martyak, B. Kubak, and S. M. Holland Anti-IFN-{gamma} Autoantibodies in Disseminated Nontuberculous Mycobacterial Infections J. Immunol., October 1, 2005; 175(7): 4769 - 4776. [Abstract] [Full Text] [PDF]

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