Journal Article

Actinomyces in Chronic Granulomatous Disease: An Emerging and Unanticipated Pathogen

Janine Reichenbach, Uri Lopatin, Nizar Mahlaoui, Bojana Beovic, Ulrich Siler, Reinhard Zbinden, Reinhard A. Seger, Louise Galmiche, Nicole Brousse, Samer Kayal, Tayfun Güngör, Stéphane Blanche and Steven M. Holland

in Clinical Infectious Diseases

Published on behalf of Infectious Diseases Society of America

Volume 49, issue 11, pages 1703-1710
Published in print December 2009 | ISSN: 1058-4838
Published online December 2009 | e-ISSN: 1537-6591 | DOI: http://dx.doi.org/10.1086/647945
Actinomyces in Chronic Granulomatous Disease: An Emerging and Unanticipated Pathogen

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Background.Chronic granulomatous disease (CGD) is a rare inherited disease of the phagocyte NADPH oxidase system that causes defective production of toxic oxygen metabolites, impaired bacterial and fungal killing, and recurrent life-threatening infections, mostly by catalase-producing organisms. We report for the first time, to our knowledge, chronic infections with Actinomyces species in 10 patients with CGD. Actinomycosis is a chronic granulomatous condition that commonly manifests as cervicofacial, pulmonary, or abdominal disease, caused by slowly progressive infection with oral and gastrointestinal commensal Actinomyces species. Treatment of actinomycosis is usually simple in immunocompetent individuals, requiring long-term, high-dose intravenous penicillin, but is more complicated in those with CGD because of delayed diagnosis and an increased risk of chronic invasive or debilitating disease.

Methods.Actinomyces was identified by culture, staining, 16S ribosomal DNA polymerase chain reaction, and/or a complement fixation test in 10 patients with CGD.

Results.All 10 patients presented with a history of fever and elevated inflammatory signs without evident focus. Diagnosis was delayed and clinical course severe and protracted despite high-dose intravenous antibiotic therapy and/or surgery. These results suggest an unrecognized and unanticipated susceptibility to weakly pathogenic Actinomyces species in patients with CGD because these are catalase-negative organisms previously thought to be nonpathogenic in CGD.

Conclusions.Actinomycosis should be vigorously sought and promptly treated in patients with CGD presenting with uncommon and prolonged clinical signs of infection. Actinomycosis is a catalase-negative infection important to consider in CGD.

Journal Article.  4412 words.  Illustrated.

Subjects: Infectious Diseases ; Immunology ; Public Health and Epidemiology ; Microbiology

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