The authors present a case of sacral meningeal cyst (s.m.c.) being the cause of chronic pain related to perineal, sacral and pelvic regions. The aim of the report is to emphasize the role played by s.m.c. in the aetiology of pain arising in these regions. As it can result from a wide variety of pathologies, like gynaecological, urological and anorectal the differential diagnosis is very difficult. Until recent years s.m.c. was rarely encountered in clinical practice and identified as a cause of chronic pain in perineal, sacral or pelvic areas. Now in the era of MRI and with increasing access to sophisticated imaging diagnostic methods s.m.c. will probably be recognized in more cases of perineal, sacral or pelvic pain.