Masaki Machida1,2, Itaru Nakamura3, Akihiro Sato4, Shigeki Nakamura5, Yoshitsugu Miyazaki6, Hidehiro Watanabe1. 1. Tokyo Medical University Hospital, Department of Infection Prevention and Control, 6-7-1 Nishishinjuku, Shinjuku-ku, Tokyo, 160-0023, Japan. 2. Tokyo Medical University, Department of Preventive Medicine and Public Health, 6-1-1 Shinjuku, Shinjuku-ku, Tokyo, 160-8402, Japan. 3. Tokyo Medical University Hospital, Department of Infection Prevention and Control, 6-7-1 Nishishinjuku, Shinjuku-ku, Tokyo, 160-0023, Japan. task300@tokyo-med.ac.jp. 4. KARADA Internal Medicine Clinic, 1-2-8-10 Nishigotanda, Shinagawa-ku, Tokyo, 141-0031, Japan. 5. Tokyo Medical University, Department of Microbiology, 6-1-1 Shinjuku, Shinjuku-ku, Tokyo, 160-8402, Japan. 6. National Institute of Infectious Diseases, Department of Chemotherapy and Mycoses, 1-23-1Shinjuku-ku, Toyama, 162-8640, Japan.
Abstract
BACKGROUND: The pathogenic fungus Schizophyllum sp. can cause allergic fungal rhinosinusitis and allergic bronchopulmonary mycosis in humans. Sinus and lung infections due to Schizophyllum sp. have been reported globally; however, no case of hypertrophic pachymeningitis due to this pathogen has been reported yet. Herein, we describe for the first time, a case of hypertrophic pachymeningitis due to Schizophyllum sp. CASE PRESENTATION: A 69-year-old woman visited the hospital with chief complaints of headache, right trigeminal neuralgia (third branch), ataxic gait, and deafness in the right ear. Magnetic resonance imaging revealed a tumor in the right sphenoidal sinus and thickening of the dura mater surrounding the right porus acusticus internus. Endoscopic sinus surgery and neuroendoscopic biopsy were performed to remove sinus lesions and intracranial lesions, respectively. Both pathological specimens showed findings indicative of filamentous fungi on Grocott's staining. DNA sequencing with the sinus specimen revealed Schizophyllum sp. as the causative pathogen, consistent with the diagnosis of fungal sinusitis and hypertrophic pachymeningitis. Intravenous liposomal amphotericin B was started, but owing to lack of improvement, the treatment was switched to intravenous voriconazole. We observed improvements in both radiological findings and symptoms. However, the symptoms exacerbated again when the trough level of voriconazole decreased. Upon increasing the voriconazole dose, a higher trough level was obtained and the symptoms improved. CONCLUSION: Our study suggests that when symptoms of central nervous system infection due to Schizophyllum sp. do not improve with liposomal amphotericin B, voriconazole can be administered at high trough levels to improve the symptoms.
BACKGROUND: The pathogenic fungus Schizophyllum sp. can cause allergic fungal rhinosinusitis and allergic bronchopulmonary mycosis in humans. Sinus and lung infections due to Schizophyllum sp. have been reported globally; however, no case of hypertrophic pachymeningitis due to this pathogen has been reported yet. Herein, we describe for the first time, a case of hypertrophic pachymeningitis due to Schizophyllum sp. CASE PRESENTATION: A 69-year-old woman visited the hospital with chief complaints of headache, right trigeminal neuralgia (third branch), ataxic gait, and deafness in the right ear. Magnetic resonance imaging revealed a tumor in the right sphenoidal sinus and thickening of the dura mater surrounding the right porus acusticus internus. Endoscopic sinus surgery and neuroendoscopic biopsy were performed to remove sinus lesions and intracranial lesions, respectively. Both pathological specimens showed findings indicative of filamentous fungi on Grocott's staining. DNA sequencing with the sinus specimen revealed Schizophyllum sp. as the causative pathogen, consistent with the diagnosis of fungal sinusitis and hypertrophic pachymeningitis. Intravenous liposomal amphotericin B was started, but owing to lack of improvement, the treatment was switched to intravenous voriconazole. We observed improvements in both radiological findings and symptoms. However, the symptoms exacerbated again when the trough level of voriconazole decreased. Upon increasing the voriconazole dose, a higher trough level was obtained and the symptoms improved. CONCLUSION: Our study suggests that when symptoms of central nervous system infection due to Schizophyllum sp. do not improve with liposomal amphotericin B, voriconazole can be administered at high trough levels to improve the symptoms.
Authors: C Lorentz; A Rivier; A Debourgogne; J Sokolowska-Gillois; J-M Vignaud; R Jankowski; M Machouart Journal: Mycoses Date: 2011-07-08 Impact factor: 4.377
Authors: J P Z Siqueira; D Sutton; J Gené; D García; M Guevara-Suarez; C Decock; N Wiederhold; J Guarro Journal: J Clin Microbiol Date: 2016-07-20 Impact factor: 5.948