Fujioka Keisuke, Nagai Takashi, Kinoshita Yukiko, Urushihara Maki, Hamasaki Yuko, Shishido Seiichiro, Kagami Shoji
Department of Pediatrics, Institute of Biochemical Sciences, Tokushima University Graduate School, Kuramoto-cho 3-18-15, Tokushima, Tokushima, 770-8503, Japan.
Department of Pediatric Nephrology, Faculty of Medicine, Toho University, Tokyo, Japan.
CEN Case Rep. 2019 Nov;8(4):261-265. doi: 10.1007/s13730-019-00403-6. Epub 2019 Jun 3.
Cryptococcosis is an invasive fungal infection that is common among organ transplant recipients, and it is challenging to treat among these patients because of their immunocompromised status. Fluconazole (FLCZ) is recommended as a first-line treatment modality for pulmonary cryptococcosis in organ transplant recipients. However, cases of FLCZ resistance among Cryptococcus neoformans isolates have been reported from the Asia Pacific region. Previous studies have reported the efficacy of voriconazole (VRCZ) in patients with FLCZ-resistant fungal infections. Herein, we report a case of FLCZ-resistant pulmonary cryptococcosis after renal transplantation that was successfully treated with VRCZ combined with amphotericin B-liposome (L-AMB). The patient was a-23-year-old woman who underwent living-donor kidney transplantation at age 20 years. She has attended our hospital since before for mental retardation, epilepsy, and dilated cardiomyopathy. At age 23 years, she presented to our hospital with fever and cough. She was diagnosed with pulmonary cryptococcosis based on positive-serum cryptococcal antigen. Chest radiography showed bilateral consolidations. Fosfluconazole (F-FLCZ) was administered, and her condition improved. However, she developed cough and fever again on day 60 of hospitalization. Cryptococcosis recurrence was suspected due to the high degree of cryptococcal antigen titers showed (1:2048) taken on the same day. Therefore, L-AMB was added, and F-FLCZ was substituted with VRCZ. Her condition improved, but L-AMB was discontinued due to hyponatremia, hypokalemia, and elevated serum creatinine. This indicates that VRCZ caused the remission. She was discharged after 6 months of admission. In conclusion, this case shows the efficacy of VRCZ combined with L-AMB for refractory pulmonary cryptococcosis.
隐球菌病是一种侵袭性真菌感染,在器官移植受者中很常见,由于这些患者免疫功能低下,治疗具有挑战性。氟康唑(FLCZ)被推荐作为器官移植受者肺隐球菌病的一线治疗方式。然而,亚太地区已报告了新型隐球菌分离株对FLCZ耐药的病例。先前的研究报道了伏立康唑(VRCZ)在FLCZ耐药真菌感染患者中的疗效。在此,我们报告1例肾移植后FLCZ耐药肺隐球菌病患者,经VRCZ联合两性霉素B脂质体(L-AMB)成功治疗。该患者为一名23岁女性,20岁时接受活体供肾移植。她此前因智力发育迟缓、癫痫和扩张型心肌病一直在我院就诊。23岁时,她因发热和咳嗽就诊于我院。基于血清隐球菌抗原阳性,她被诊断为肺隐球菌病。胸部X线片显示双侧实变。给予氟伏沙明(F-FLCZ)治疗后,她的病情有所改善。然而,住院第60天时她再次出现咳嗽和发热。由于当天测得的隐球菌抗原滴度很高(1:2048),怀疑隐球菌病复发。因此,加用L-AMB,并将F-FLCZ换为VRCZ。她的病情有所改善,但由于低钠血症、低钾血症和血清肌酐升高,停用了L-AMB。这表明VRCZ导致了病情缓解。入院6个月后她出院。总之,本病例显示了VRCZ联合L-AMB治疗难治性肺隐球菌病的疗效。