INTRODUCTION:Posterior epidural sequestrated disc is a fairly uncommon condition. We report a case of an unusual presentation of posterior epidural migration with contralateral symptoms. We emphasize a high index of suspicion for early recognition and treatment to promote a good neurological recovery. CASE PRESENTATION: A 58-year-old man with a history of severe back pain for 1 week developed sudden right leg weakness while walking. Neurological examination revealed motor and sensory impairments of the right lower extremities (positive right straight leg raising test, right tibialis anterior grade 2/5, right extensor hallucis longus grade 1/5, decreased pinprick sensation of L4-5 dermatome right side) according to the Standards for Neurological Classification of Spinal Cord Injury as Asia Impairment Scale D. Magnetic resonance images showed an acute disc herniation at L4-5 on the left side, cranially migrated and sequestrated to the posterior epidural area, causing severe compression to the cauda equina in the thecal sac. The patient was successfully treated with surgery (transforaminal lumbar interbody fusion) on an urgent basis. We saw significant neurological recovery on the first day after surgery. Motor power recovery was achieved with a minor deficit at 4 weeks. At the follow-up examination at 3 months, the patient had no residual neurological deficits as Asia Impairment Scale E. DISCUSSION: Posterior epidural sequestrated disc with contralateral radiculopathy is very rare. This case brings a new presentation of posterior epidural sequestrated disc. To our knowledge, this is the first report of a case with an unusual presentation of contralateral radiculopathy.