A 63-year-old man had a history of diabetes mellitus for more than 10 years and took oral hypoglycemic agents regularly. He visited Taipei Veterans General Hospital with the complaint of progressive weakness in all 4 limbs and neck pain for 6 months. Computed tomography of the cervical spine revealed increased soft tissue density in the epidural space from C2 to C5 with cord compression. Surgical decompression was done and Aspergillus flavus was isolated from the inflammatory tissue. He was initially treated with oral itraconazole 200 mg 3 times per day for 4 days and then twice daily. Later, the treatment regimen was shifted to intravenous amphotericin B 25 mg/d. He died of intraventricular hemorrhage and complicated fungal meningoencephalitis 2 weeks postlaminectomy. This case reminds us that a prolonged history of back pain accompanied with peripheral neuropathy in diabetic patients should raise the suspicion of Aspergillus epidural abscess. Prompt aggressive diagnostic testing and management is needed to improve the likelihood of a good outcome of these patients.