2008European Journal of Gastroenterology & HepatologyRequires access

Efficiency of hydroxychloroquine in the treatment of granulomatous complications in chronic granulomatous disease

Jean‐Benoît Arlet, Achile Aouba, Felipe Suárez, Stéphane Blanche, Dominique Valeyre, Alain Fischer, Diane Damotte, Christophe Cellier, Olivier Hermine

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Abstract

Chronic granulomatous disease (CGD) represents a group of genetic disorders in which impaired intracellular microbial killing by phagocytes leads to recurrent bacterial and fungal infections and granuloma formation. Granulomatous lesions involved gastrointestinal tract in more than one-third of all CGD patients, with various clinical presentations, and may be life threatening. Corticosteroid therapy may relieve symptoms but increase the risk of infections in such immunocompromised patients. We report here the first successful management, with hydroxychloroquine therapy alone, of a 29-year-old CGD man with severe gastric granulomatous involvement. Hydroxychloroquine appears to be a safe and effective alternative treatment of granulomas in CGD patients.

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What this paper is about

Chronic granulomatous disease (CGD) represents a group of genetic disorders in which impaired intracellular microbial killing by phagocytes leads to recurrent bacterial and fungal infections and granuloma formation. Granulomatous lesions involved gastrointestinal tract in more than one-third of all CGD patients, with various clinical presentations, and may be life threatening. Corticosteroid therapy may relieve symptoms but increase the risk of infections in such immunocompromised patients. We report here the first successful management, with hydroxychloroquine therapy alone, of a 29-year-old CGD man with severe gastric granulomatous involvement. Hydroxychloroquine appears to be a safe and effective alternative treatment of granulomas in CGD patients.

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Available abstract

Chronic granulomatous disease (CGD) represents a group of genetic disorders in which impaired intracellular microbial killing by phagocytes leads to recurrent bacterial and fungal infections and granuloma formation. Granulomatous lesions involved gastrointestinal tract in more than one-third of all CGD patients, with various clinical presentations, and may be life threatening. Corticosteroid therapy may relieve symptoms but increase the risk of infections in such immunocompromised patients. We report here the first successful management, with hydroxychloroquine therapy alone, of a 29-year-old CGD man with severe gastric granulomatous involvement. Hydroxychloroquine appears to be a safe and effective alternative treatment of granulomas in CGD patients.

Key concepts: Chronic granulomatous disease, Hydroxychloroquine, Medicine, Granulomatous disease, Granuloma, Granulomatous inflammation, Immunology, Disease

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