Acute invasive fungal sinusitis is a fungal infectious disease in which fungal hyphae invade the nasal mucosa, blood vessels and nerves, causing tissue necrosis. Acute invasive fungal sinusitis is often complicated by intracranial infection, which is a critical disease with a high mortality rate. Meanwhile, ocular manifestations such as orbital cusp syndrome, cavernous sinus syndrome, and a sudden drop in visual acuity may suggest that the disease has been complicated by intracranial or intraorbital infection. Therefore, early diagnosis and early antifungal and anti-infectious treatment are essential to improve patient survival, and recognition of specific ocular signs is crucial for early diagnosis and treatment of acute invasive fungal sinusitis. In this article, we report a typical and rare case of acute invasive fungal sinusitis complicated by central retinal artery occlusion caused by Trichoderma infection. Central retinal artery occlusion is a kind of emergency and serious disease in ophthalmology, which has a rapid onset and rapid development. Failure to relieve the occlusion in time will result in permanent blindness of the patient, which will seriously affect the quality of life of the patient.Retinal artery obstruction relies heavily on imaging:Fundus Fluorescein Angiography: Demonstrates arterial and choroidal blood flow patterns to assess retinal blood flow and vascular obstruction. Patients present with very sluggish retinal arterial filling, and large areas of no perfusion;Optical Coherence Tomography : provides high-resolution images of retinal anatomy, thickness, and vitreoretinal interface in patients presenting with thickening of the inner retinal layer with edema and a highly reflective signal. Although the patient was treated by multidisciplinary diagnosis and treatment and transferred to the intensive care unit in a timely manner, he eventually died of systemic multi-organ failure due to complicated intracranial infection and sepsis. This case was discussed and analyzed from the ophthalmology point of view, and the relevant literature was reviewed in order to provide a reference for clinicians to recognize the initial symptoms of acute invasive fungal sinusitis, to provide timely antifungal and anti-infective treatment, to reduce missed diagnosis and misdiagnosis, and to avoid further deterioration of the patient's condition and death.In particular, we ophthalmologists are cautioned that retinal artery occlusion is not a purely ophthalmic disease, but is closely related to systemic diseases.