IntroductionIn this report, we describe an unusual case of post-operative Acremonium falciforme endophthalmitis with orbital and extra-orbital involvement following combined cataract and glaucoma surgery.Case presentationA 68-year-old Caucasian man with glaucoma presented with endophthalmitis characterized by pain, redness and impaired vision in the left eye fifteen days after combined cataract and filtering surgery. He subsequently underwent a pars plana vitrectomy, with vitreous sampling, silicone oil placement and intra-vitreal injection of antibiotics, but only after a second vitrectomy we identified Acremonium falciforme as the causative agent for the endophthalmitis. An antifungal systemic and topical therapy was started, but meanwhile the infection extended to orbital and peri-orbital tissues. Following these procedures, even if the eye went slowly in phthisis, we were able to limit the further extension and circumscribe the orbital and extra-orbital involvement.ConclusionTo our knowledge, this report is the first describe Acremonium falciforme endophthalmitis with orbital and extra-orbital involvement, following anterior segment combined surgery. Ophthalmologists and physicians should be aware of the extension risk of a fungal panophthalmitis, but also to potentially serious side effects related to systemic therapy.