A 34-year-old woman with right-sided headache for 8 days, blindness in the right eye with swelling for 5 days, and blindness in the left eye for 1 day was admitted to our hospital on July 13, 2021. Her medical history included left nephrectomy and macrosomia for three times. MRI showed the patient had sphenoid sinus, sinuses, and frontal lobe lesions (,). Meanwhile, we observed thickening of the right eye ring, overstriking of the extraocular muscles and optic nerve, and proptosis of the right eye (). No abnormalities were observed via magnetic resonance angiography (). In terms of facial features, the patient presented with proptosis and fixation of the right eye, conjunctival hyperemia and edema, drooping eyelids, and askew mouth (,). Additionally, blood and cerebrospinal fluid metagenomic sequencing (mNGS) revealed Rhizopus oryzae infection. In pertinent laboratory examinations, her glycated hemoglobin (HbA1c) was 17.8%, and fungus β-D-glucan was 240.64 pg/ml. Finally, she was diagnosed with ROCM and diabetes.