Intravesical administration of bacille Calmette-Guérin (BCG) is an important component of the gold standard in treating non-muscle invasive bladder cancer (NMIBC). However, complications of this treatment include infections caused by the dissemination of Mycobacterium bovis. We present a case of a 62-year-old man who had been treated with intravesical BCG for bladder cancer and developed an M. bovis infection of his vertebral column. About four months after completing the BCG treatment, he developed an acute onset of severe upper thoracic radicular back pain, with radiation anteriorly to his sternum. Examination revealed the presence of early myelopathy. After other causes were ruled out, he was diagnosed with the infection four months later. He was investigated for the pain, with resulting imaging identifying an erosive ventral epidural mass at the T4-T5 levels causing cord compression. The patient underwent a transthoracic procedure to evacuate the paraspinal mass lesion and obtain a diagnostic biopsy, followed by a posterolateral decompression of the lesion and posterior instrumented stabilization. Pathology resulted in the identification of a granuloma with a single acid-fast bacillus (AFB) from the paraspinal abscess, thus being diagnostic of a mycobacterial granuloma with paraspinal involvement.We subsequently performed an extensive review of current literature, looking at articles on spinal osteomyelitis following intravesical BCG treatment of bladder cancer. We identified 26 documented cases in English literature. We present our case report with a good outcome at 24 months, resolving with appropriate chemotherapy. Additionally, we completed a systematic review of the literature and discuss this infrequent iatrogenic pathology. Our report reveals the good response to targeted therapy in the case of osteomyelitis at other skeletal sites and that practitioners caring for these patients maintain a high degree of suspicion in the workup of these patients. Early identification and treatment can appropriately treat osteomyelitis with good long-term outcomes.