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The authors present a case of posterior fossa arachnoid cyst associated with syringomyelia. A 58-year-old woman was admitted to our hospital due to progressive headache, gait disturbance, and pain at the left upper extremity. Magnetic resonance(MR) image showed a huge retrocerebellar arachnoid cyst associated with syringomyelia from the second to the sixth cervical vertebral level. After decompression of the posterior fossa including removal of posterior lip of foramen magnum and lamina of the atlas and fenestration between arachnoid cyst and subarachnoid space, the patient's symptoms were improved and the arachnoid cyst and syrinx were shrunken on follow-up MR imaging. The authors discuss the pathogenesis of the syringomyelia associated with a huge posterior fossa arachnoid cyst and the management option. Key words:Posterior fossa;Arachnoid cyst;Syringomyelia.