Infection and Drug Resistance (Sep 2020)

Chronic Invasive Fungal Rhinosinusitis with Atypical Clinical Presentation in an Immunocompromised Patient

  • Kawaji-Kanayama Y,
  • Nishimura A,
  • Yasuda M,
  • Sakiyama E,
  • Shimura Y,
  • Tsukamoto T,
  • Kobayashi T,
  • Mizutani S,
  • Okamoto S,
  • Ohmura G,
  • Hirano S,
  • Konishi E,
  • Shibuya K,
  • Kuroda J

Journal volume & issue
Vol. Volume 13
pp. 3225 – 3232

Abstract

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Yuka Kawaji-Kanayama,1 Ayako Nishimura,2 Makoto Yasuda,3 Emiko Sakiyama,1,4 Yuji Shimura,1 Taku Tsukamoto,1 Tsutomu Kobayashi,1 Shinsuke Mizutani,1 Shota Okamoto,3 Gaku Ohmura,3 Shigeru Hirano,3 Eiichi Konishi,2 Kazutoshi Shibuya,5 Junya Kuroda1 1Division of Hematology and Oncology, Department of Medicine, Kyoto Prefectural University of Medicine, Kyoto, Japan; 2Division of Surgical Pathology, Department of Pathology, Kyoto Prefectural University of Medicine, Kyoto, Japan; 3Department of Otolaryngology-Head and Neck Surgery, Kyoto Prefectural University of Medicine, Kyoto, Japan; 4Department of Hematology, Matsushita Memorial Hospital, Moriguchi, Japan; 5Department of Surgical Pathology, Toho University Graduate School of Medicine, Tokyo, JapanCorrespondence: Junya KurodaDivision of Hematology and Oncology, Department of Medicine, Kyoto Prefectural University of Medicine, 465 Kajii-Cho, Kamigyo-Ku, Kyoto 602-8566, JapanTel +81 75 251 5740Fax +81 75 251 5743Email [email protected]: Invasive fungal rhinosinusitis (FRS) is a rare but intractable infectious disease of the sinonasal region with destructive direct infiltration into surrounding tissues, such as the bone, orbit and brain, and potential dissemination to systemic organs. Symptomatic assessments and imaging are frequently not sufficiently diagnostic, and histopathological examination is essential for definite diagnosis of FRS. We herein report a case of chronic invasive FRS (CIFRS) in a 58-year-old Japanese male with end-stage diabetic nephropathy that required maintenance dialysis after graft rejection of living kidney transplantation. His initial main clinical presentation was sinus gangrene, which gradually progressed from the paranasal sinus to the nasal septum and oral palate, but not towards the intracranial or orbital region, for two months. The patient was first strongly suspected to have extranodal natural killer/T cell lymphoma (ENKTL), nasal type, a subtype of malignant lymphoma, based on the macroscopic appearance of the gangrene, expansion pattern and high serum soluble interleukin-2 level; however, repeated biopsies and eventual resection led to diagnosis of CIFRS due to Aspergillus niger and Mucor. The disease was improved by surgical resection in combination with antifungal pharmacologic treatment with liposomal amphotericin B and voriconazole. CIFRS typically occurs in immunocompetent patients and shows intracranial progression, but this case shows that atypical CIFRS with an uncommon expansion pattern can occur in an immunodeficient patient.Keywords: chronic invasive fungal rhinosinusitis, diabetes mellitus, maintenance dialysis, extranodal natural killer/T cell lymphoma, nasal type

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