BACKGROUND Spinal bronchogenic cysts are rare nonneoplastic congenital variants of neurenteric cysts. The natural history and surgical management of these lesions are poorly understood. OBSERVATIONS A 25-year-old male presented with progressive back pain and bilateral lower limb sciatica of 6 months’ duration. He had undergone subtotal resection of an intramedullary bronchogenic conus medullaris cyst 5 years prior. Magnetic resonance imaging revealed a recurrent bilobed intramedullary and extramedullary conus medullaris cystic lesion. The authors resected the lesion via a posterior approach with the aid of intraoperative neuromonitoring. Gross total resection was precluded by the tightly adherent nature of the cyst and the fact that stimulation of a residual intramedullary portion of the lesion evoked external anal sphincter responses. LESSONS This is the first reported case of a bilobed intramedullary and extramedullary bronchogenic cyst of the conus medullaris. This unique case lends insight into the poorly defined embryogenesis of bronchogenic cysts by favoring the split notochord syndrome theory rather than the ectopic ectoderm proposal. The importance of neuromonitoring when resecting these tightly adherent lesions is demonstrated. Finally, although the secretory nature of these lesions portends a tendency for cyst reaccumulation, it is imperative to recognize that this is usually a slow process.