Wynne Sarah M, Kwon-Chung Kyung J, Shea Yvonne R, Filie Armando C, Varma Ashok, Lupo Petrizia, Holland Steven M
NIAID, NIH, Bethesda, MD 20892, USA.
J Allergy Clin Immunol. 2004 Dec;114(6):1418-24. doi: 10.1016/j.jaci.2004.07.066.
A 9-year-old girl with autosomal recessive chronic granulomatous disease (CGD) presented with asymptomatic bilateral pulmonary infiltrates on routine computed tomography. Fine-needle aspirate of the infiltrates was obtained and showed fungal cells resembling Trichosporon inkin . The specimen grew in culture, and testing by means of both API 20C and PCR amplification confirmed the diagnosis of T inkin . The infiltrates increased in size, despite sequential therapy with voriconazole, liposomal amphotericin B, caspofungin, and posaconazole. The patient required resection of the infected lung tissue, after which she recovered completely. While she was undergoing therapy, her 13-year-old brother, also with CGD, was given a diagnosis of bilateral T inkin -induced pulmonary infection. He also required bilateral pulmonary resection for cure. These cases demonstrate the predisposition of patients with CGD to have invasive infections with unusual fungal organisms, such as T inkin . They also illustrate the difficulty of treating invasive T inkin infections with antifungal agents alone. There are 9 previously reported cases of invasive infections caused by T inkin , 3 of which are in patients with CGD. All patients required removal of infected prosthetic devices or surgical resection of infected tissue for cure.
一名患有常染色体隐性慢性肉芽肿病（CGD）的9岁女孩在常规计算机断层扫描中显示双侧肺部无症状浸润。对浸润灶进行细针抽吸，发现真菌细胞类似于皮状丝孢酵母。标本在培养中生长，通过API 20C和PCR扩增检测确诊为皮状丝孢酵母。尽管先后使用伏立康唑、脂质体两性霉素B、卡泊芬净和泊沙康唑进行治疗，浸润灶仍增大。患者需要切除受感染的肺组织，术后完全康复。在她接受治疗期间，她13岁的弟弟，同样患有CGD，被诊断为双侧皮状丝孢酵母引起的肺部感染。他也需要双侧肺切除以治愈。这些病例表明，CGD患者易发生由不常见真菌病原体（如皮状丝孢酵母）引起的侵袭性感染。它们还说明了仅用抗真菌药物治疗侵袭性皮状丝孢酵母感染的困难。先前有9例关于皮状丝孢酵母引起侵袭性感染的报道，其中3例发生在CGD患者中。所有患者都需要移除受感染的假体装置或手术切除受感染组织以治愈。
