We report a case of CMV-related Transverse Myelitis in a patient with AIDS. A 44-year-old HIV-positive man presented to the Unit of Infectious Diseases of our hospital, complaining of fever, paresthesia and hypotonia in his legs, and acute urinary retention. He had been diagnosed with AIDS one week before admission and had been started on tenofovir/emtricitabine and darunavir/ritonavir. At admission, neurological exam showed moderate neck rigidity, paraparesis and dysesthesia. The MRI revealed the presence of inflammatory signs extending from T1 to T12. CMV DNA in cerebrospinal fluid was positive. The patient was treated with steroids and ganciclovir but did not significantly recovered. He was finally transferred to the Rehabilitation Spinal Unit. Ganciclovir was interrupted six months after discharge. At the 18-month follow up, the patient had only partially recovered sensibility, with no significant improvement in ambulation.