Aspergillus osteomyelitis in a child who has p67-phox-deficient chronic granulomatous disease.
Here we describe Aspergillus osteomyelitis of the tibia in a 9-year-old boy who has an autosomal recessive form of chronic granulomatous disease (CGD). The patient showed a p67-phagocyte oxidase (phox) deficiency, which is rare type of CGD in Japan. The initial treatment which consisted of surgical debridement and antibiotic therapy with amphotericin B (AMPH), did not control the infection. Aspergillus fumigatus (A. fumigatus) pure isolated from drainage fluid and necrotic bone tissue demonstrated less susceptible to antifungal agents, including AMPH, fluconazole and flucytosine. Recombinant interferon gamma was then administrated, and it was effective in controlling the course of severe invasive aspergillosis. This report indicates the use of interferon gamma might be helpful in control for Aspergillus osteomyelitis of the tibia in a child with CGD demonstrated p67-phox deficiency refractory to conventional therapy with AMPH.
D000666:Amphotericin B / Q000627:therapeutic use; D001228:Aspergillosis / Q000628:therapy*; D002648:Child; D006105:Granulomatous Disease, Chronic / Q000628:therapy*; D006801:Humans; D007371:Interferon-gamma / Q000627:therapeutic use*; D008297:Male; D010019:Osteomyelitis / Q000628:therapy*; D010750:Phosphoproteins / Q000172:deficiency*; D011994:Recombinant Proteins; D013977:Tibia