INTRODUCTION: Currarino syndrome (Currarino triad) was described in 1981 as a 
triad syndrome with a common embryogenesis in infants and with three 
characteristics: anorectal stenosis, a defect in the sacral bone, and a 
presacral mass. We describe here an unusual case of Currarino syndrome in an 
adult presenting with a presacral abscess but no meningitis.
CASE PRESENTATION: A 32-year-old Japanese man presented with fever, arthralgia 
and buttock pain. A digital rectal examination showed mild rectal stenosis with 
local warmth and tenderness in the posterior wall of his rectum. Computed 
tomography showed a scimitar-shaped deformity of his sacrum and an 8cm presacral 
mass, which continued to a pedicle of his deformed sacrum. This was diagnosed as 
Currarino syndrome with a presacral abscess. The abscess was drained by a 
perianal approach with our patient treated with antibiotics. His symptoms soon 
disappeared. After three months, an excision was performed through a posterior 
sagittal approach. His postoperative course was uneventful and he was discharged 
10 days after surgery. A histopathological examination revealed an infected 
epidermoid cyst. He has been free from recurrence as of four years and six 
months after surgery.
CONCLUSIONS: We report a case of Currarino syndrome in an adult who presented 
with a presacral abscess but no meningitis. Abscess drainage followed by radical 
surgery resulted in a successful outcome.