Hypertrophic pachymeningitis caused by Schizophyllum sp.: a novel case report
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CASE REPORT
Hypertrophic pachymeningitis caused by Schizophyllum sp.: a novel case report Masaki Machida1,2 · Itaru Nakamura1 · Akihiro Sato3 · Shigeki Nakamura4 · Yoshitsugu Miyazaki5 · Hidehiro Watanabe1 Received: 10 August 2020 / Accepted: 23 October 2020 © Springer-Verlag GmbH Germany, part of Springer Nature 2020
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. Keywords Hypertrophic pachymeningitis · Filamentous basidiomycete · Schizophyllum species · Sinusitis · Case report
Background * Itaru Nakamura task300@tokyo‑med.ac.jp 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
KARADA Internal Medicine Clinic, 1‑2‑8‑10 Nishigotanda, Shinagawa‑ku, Tokyo 141‑0031, Japan
4
Tokyo Medical University, Department of Microbiology, 6‑1‑1 Shinjuku, Shinjuku‑ku, Tokyo 160‑8402, Japan
5
National Institute of Infectious Diseases, Department of Chemotherapy and Mycoses, 1‑23‑1Shinjuku‑ku, Toyama 162‑8640, Japan
Schizophyllum sp. is a widely distributed basidiomycete that inhabits rotting wood [1]. The Schizophyllum genus has approximately 20 species [2], in
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