Tsumura N, Akasu Y, Yamane H, Ikezawa S, Hirata T, Oda K, Sakata Y, Shirahama M, Inoue A, Kato H
Department of Pediatrics, Kurume University School of Medicine, Japan.
Kurume Med J. 1999;46(1):87-90. doi: 10.2739/kurumemedj.46.87.
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.
在此，我们描述了一名患有常染色体隐性慢性肉芽肿病（CGD）的9岁男孩的胫骨曲霉菌骨髓炎。该患者表现出p67-吞噬细胞氧化酶（phox）缺乏，这在日本是一种罕见的CGD类型。初始治疗包括手术清创和使用两性霉素B（AMPH）进行抗生素治疗，但未能控制感染。从引流液和坏死骨组织中纯分离出的烟曲霉菌（A. fumigatus）对包括AMPH、氟康唑和氟胞嘧啶在内的抗真菌药物敏感性较低。随后给予重组干扰素γ，其在控制严重侵袭性曲霉菌病病程方面有效。本报告表明，对于一名患有CGD且表现出p67-phox缺乏、对AMPH常规治疗无效的儿童胫骨曲霉菌骨髓炎，使用干扰素γ可能有助于控制病情。
