Enhanced generation of reactive oxygen species by interferon-γ may have contributed to successful treatment of invasive pulmonary aspergillosis in a patient with chronic granulomatous disease

被引:0
作者
Kouhei Yamashita
Takashi Miyoshi
Yasuyuki Arai
Kiyomi Mizugishi
Akifumi Takaori-Kondo
Takehiko Ueyama
机构
[1] Kyoto University,Department of Hematology and Oncology, Graduate School of Medicine
[2] Biosignal Research Center,Laboratory of Molecular Pharmacology
[3] Kobe University,undefined
来源
International Journal of Hematology | 2013年 / 97卷
关键词
Chronic granulomatous disease; Invasive pulmonary aspergillosis; Interferon-γ; Reactive oxygen species; Voriconazole;
D O I
暂无
中图分类号
学科分类号
摘要
Invasive pulmonary aspergillosis (IPA) is a life-threatening complication of chronic granulomatous disease (CGD), a rare inherited disorder of phagocytes that is characterized by a defect in the production of reactive oxygen species (ROS) caused by mutations in NADPH oxidase 2. Here, we report a case of successful treatment of IPA complicated with CGD by the administration of interferon-γ (IFN-γ) in combination with voriconazole. The patient carried a splice site mutation in the CYBB gene, and the neutrophils could produce a certain amount of ROS. In this case, augmentation of ROS generation in the patient’s neutrophils was observed after in vivo IFN-γ treatment, which may be attributable to the induction of a normal CYBB gene in the myeloid progenitor cells. This treatment, in combination with voriconazole, may have contributed to the reversal of IPA in this patient. These results suggest that the in vivo use of IFN-γ may augment ROS generation in CGD neutrophils, thus leading to the successful treatment of severe IPA.
引用
收藏
页码:505 / 510
页数:5
相关论文
共 84 条
[1]  
Segal BH(2000)Genetic, biomedical, and clinical features of chronic granulomatous disease Medicine (Baltimore) 79 170-200
[2]  
Leto TL(2000)Statistical and mutational analysis of chronic granulomatous disease in Japan with special reference to gp91- Hum Genet 106 473-481
[3]  
Gallin JI(2001) and p22- Blood 98 436-441
[4]  
Malech HL(2009)Improved superoxide-generating ability by interferon-γ due to splicing pattern change of transcripts in neutrophils from patients with a splice site mutation in Med Mycol 47 217-220
[5]  
Holland SM(1987) gene J Clin Invest 80 1009-1016
[6]  
Ishibashi F(1991)Multiple-azole-resistant J Infect Dis 163 849-852
[7]  
Nunoi H(2005) osteomyelitis in a patient with chronic granulomatous disease successfully treated with long-term oral posaconazole and surgery Int J Hematol 81 220-227
[8]  
Endo F(2011)Recombinant interferon gamma augments phagocyte superoxide production and X-chronic granulomatous disease gene expression in X-linked variant chronic granulomatous disease J Biol Chem 286 40693-40705
[9]  
Matsuda I(2004)In vivo interferon- Jpn J Infect Dis 57 S25-S26
[10]  
Kanegasaki S(1982) therapy augments the in vitro ability of chronic granulomatous disease neutrophils to damage Nucleic Acids Res 10 459-472