The aim of this study was to present a unique case of intramedullary brucellar granuloma (IBG) and to discuss the diagnosis and management. To our knowledge, only one case of thoracic IBG has been reported previously, and our case is the first in cervical spine. A 35-year-old female patient was admitted with headache, pain and weakness in her four extremities. She had no gastrointestinal symptoms and fever. She had been diagnosed with Brucella meningitis 3 months ago and a triple therapy of doxycyclin, rifampicin and trimetoprim/sulfametoxazol (TMP/SMZ) had been started. Medical history revealed that she had ingested raw cheese and taken her medication improperly. Loss of strength was detected in her four extremities, which led us to assume the formation of a mass lesion at cervical level. Therefore, we performed a magnetic resonance imaging scan and found enhancement of an intramedullary mass lesion at cervical 1-2 level. Diagnosis of neurobrucellosis was confirmed by titer of >1/160 Brucella antibodies both in blood and cerebrospinal fluid. Based on these findings, brucellar granuloma of cervical spine was diagnosed and a combination therapy of doxycyclin, TMP/SMZ and rifampicin was administered for additional 6 months. At the ninth month of treatment, the patient recovered both radiologically and clinically. Our case is unique, in terms of cervical IBG formation. The excellent response to antimicrobial therapy in our patient suggests that, a trial of medical treatment for 6 months may be effective in such cases.