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Background Lumbar disc herniation into the dural space is a very rare phenomenon of degenerative lumbar lesions in the elderly population, and its potential pathogenesis and natural course remain unclear. Case description We describe a rare case of intradural lumbar disc herniation. A 68-year-old man presented with progressive lower back pain and radiating pain and numbness in both legs for 3 years. Magnetic resonance imaging revealed a large herniated disc at L4-L5. Posterior discectomy and fusion of the L4-L5 was performed after conservative treatment failed. Intraoperatively, only minimal disc fragments in the epidural space were found after meticulous probing following laminectomy of the L4-L5 vertebrae. The dorsal dura mater was saturated, tense, and bulged at the L4-L5 levels; additionally, an intradural mass was palpable and confirmed by intraoperative ultrasonography. Subsequently, dorsal middle durotomy was performed. Upon opening the dural sac, a large cauliflower-like mass similar to nucleus pulposus tissue was found near the arachnoid membrane. The mass was dissociative and could be completely resected. The dorsal dural incisions were closed after careful exploration, followed by fixation and fusion of the L4-L5 levels. Pathological examination revealed disc tissue with central balloon-type cystic degenerative changes. The patient’s lower back pain and radiating pain and numbness of both legs improved remarkably postoperatively, and he became asymptomatic at 3 months postoperatively. Conclusion Intradural lumbar disc herniation should be highly suspected when intraoperative findings are incompatible with findings from the preoperative imaging examination, and it could be further confirmed via intraoperative ultrasonography and pathological examination of the resected tissue from the dural space. Prompt surgery is recommended, and surgical results are usually favorable. We also reviewed the literature and discussed the potential pathogenesis, natural course, diagnosis, and treatment of intradural lumbar disc herniation.