Fever, Rhinosinusitis and Glomerulonephritis with Systemic Inflammation and Antimyeloperoxidase Antibody

© Author(s) (or their employer(s)) 2022. No commercial reuse. See rights and permissions. Published by BMJ. A woman in her 80s presented with a monthlong fever and nasal discharge, which did not subside by clarithromycin and levofloxacin. She did not have a history of allergic diseases including bronchial asthma. Physical examination showed no significant findings. However, laboratory tests revealed a high level of Creactive protein (129 mg/L, reference range <1.4), positive antimyeloperoxidase antibody and active urine sediments including haematuria, proteinuria and cellular casts. Wholebody CT demonstrated no remarkable findings except abnormal soft tissue filling left maxillary sinus (figure 1A). The patient was referred to our department under the suspected diagnosis of antineutrophil cytoplasmic antibody (ANCA)associated vasculitis with nasal manifestation and glomerulonephritis. However, CT reassessment revealed slight speckled calcification without bone destruction in the left maxillary sinus (figure 1B). MR fatsaturatedT2weighted imaging also demonstrated lowintensity areas in the left maxillary sinus without dural thickening (figure 1C). In the nasopharyngoscopy procedure, the septum was found to be normal, but the lateral wall of nasal cavity was retracted with purulent discharges (figure 1D). (1–3)-β-Dglucan, serum fungal antigen and blood culture test results were negative. We suspected left fungal sinusitis and performed surgical treatment. Evaluation of the maxillary sinus indicated acuteangle branching, septate, filamentous fungi, suggesting Aspergillus spp (figure 1E). The mucosal biopsy of maxillary sinus exhibited inflammatory cells around vessels without fungal invasion (figure 1F). The fever immediately resolved after surgery. We eventually diagnosed the patient with noninvasive chronic fungal sinusitis, which immunologically triggered ANCAassociated vasculitis with glomerulonephritis. Previous reports demonstrated that cases of ANCAassociated vasculitis are frequently subjected to invasive mycoses in which Aspergillus spp are the leading strain. 2 Sporadic cases of systemic vasculitis due to invasive aspergillosis were noticed even in immunocompetent individuals. Physicians should remember that noninvasive aspergillosis would induce autoimmunity of ANCAassociated vasculitis as in our case. Three months after the surgical removal of maxillary fungal balls, systemic inflammation, ANCA and active urine sediments had completely diminished without immunosuppressive therapy (see online supplemental table 1 for the followup results of laboratory findings).