Background: Scedosporium Apiospermum is a fungal species that can cause invasive fungal infection. We report a rare case of invasive fungal sinusitis and dacryocystitis due to Scedosporium apiospermum leading to orbital exenteration in an immunosuppressed patient post bone marrow transplant. Case Report: A 56-year-old male with multiple myeloma developed left sinusitis and dacryocystitis seven days after bone marrow transplant. Despite medical interventions including antimicrobials, intravenous steroids and blood transfusions, the infection progressed with complete vision loss from optic neuropathy. Imaging demonstrated maxillary sinusitis, a lacrimal sac abscess, and orbital cellulitis. Surgical interventions included lateral canthotomy and cantholysis, functional endoscopic sinus surgery with medial orbital decompression and ultimately orbital exenteration. Histology revealed Scedosporium apiospermum species. The infection remained cleared at the 3-month follow-up. Discussion: Invasive fungal sinusitis often has a poor prognosis especially in immunosuppressed patients. Treatment often needs a combination of medical treatment and extensive surgical debridement. Conclusion: This is the first reported case of Scedosporium apiospermum dacryocystitis with associated orbital cellulitis requiring exenteration. It highlights the need for early multidisciplinary input, including surgical debridement and targeted antifungal therapy, in immunocompromised patients with suspected invasive fungal infections. Reference: 1. O’Rourke M, Tang YF, Pick Z, Tan JS, Tan PE, Athavale DD, O’Donnell B, Selva D, Gajdatsy A, Hardy TG, McNab A. Orbital Cellulitis Secondary to Dacryocystitis: A Case Series and Literature Review. Ophthalmic Plastic & Reconstructive Surgery. 2025 May 1;41(3):306-14. 2. Farooq AV, Patel RM, Lin AY, Setabutr P, Sartori J, Aakalu VK. Fungal orbital cellulitis: presenting features, management and outcomes at a referral center. Orbit. 2015 May 4;34(3):152-9.