Moltyaner Y, Geerts W H, Chamberlain D W, Heyworth P G, Noack D, Rae J, Doyle J J, Downey G P
Division of Respirology, University of Toronto, Toronto, Ontario, Canada.
Thorax. 2003 Dec;58(12):1096-8. doi: 10.1136/thorax.58.12.1096.
We present a case of bronchocentric granulomatosis in a woman with no history of asthma who was colonised with Aspergillusfumigatus. A family history of chronic granulomatous disease prompted further testing that demonstrated severely depressed neutrophil oxidant production and gp91(phox) deficiency compatible with the X linked carrier state of chronic granulomatous disease. Only one report of the association of these two rare diseases has previously appeared in the literature. We postulate that an ineffective immune response led to the prolonged colonisation of Afumigatus resulting in a hypersensitivity reaction that was manifest clinically as bronchocentric granulomatosis.
我们报告一例支气管中心性肉芽肿病病例，患者为一名无哮喘病史的女性，其感染了烟曲霉。慢性肉芽肿病的家族史促使进一步检查，结果显示中性粒细胞氧化剂生成严重降低，且gp91(phox)缺乏，这与慢性肉芽肿病的X连锁携带者状态相符。此前文献中仅出现过一篇关于这两种罕见疾病关联的报告。我们推测，无效的免疫反应导致烟曲霉长期定植，进而引发超敏反应，临床上表现为支气管中心性肉芽肿病。
