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Abstract Background Tarlov cysts are a commonly misdiagnosed condition, which can present with many rare symptoms. We report a case of a Tarlov cyst with spermatorrhea and review the pertinent literature. Case presentation A 42-year-old male patient had a history of spermatorrhea for > 10 years, but was incorrectly diagnosed as the patient and the doctors consistently mistook the symptoms for a genitourinary disease. Magnetic resonance imaging showed that two cysts in the sacral canal. The diagnosis was Tarlov cyst. We performed surgery to remove the cyst and the symptoms of spermatorrhea disappeared after the operation. Conclusions This case demonstrates that orthopedics and urologists should improve their understanding of Tarlov cysts to avoid misdiagnosis and mistreatment.