Unlabelled: Fungal sinusitis are very often caused by Aspergillus spp. Dematiaceae spp. and mucomycoses. Sinusitis with Scedosporium spp is very rare, especially in immunocompetent humans. A 42-year-old female presented with long history of facial pain and headache. Isolated opacification of maxillary sinus with double density sign was noted on left side in the CT scan. Diagnosed as fungal rhino sinusitis and endoscopic sinus surgery was carried out to clear the disease of the sinus and further mycological evaluation proved the organism to be Scedosporium apiospermum. We report the rare case of a non-invasive isolated fungal sinusitis caused by fungi Scedosporium apiospermum.

Supplementary Information: The online version contains supplementary material available at 10.1007/s12070-024-05167-9.

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