We report a rare case of cervical dermal sinus associated with a dermoid cy
st in a 10-month-old infant, who presented with a 1-month history of motor
weakness of the right upper extremity. Magnetic resonance imaging showed an
intradural extramedullary dermoid cyst extending from C3 to C4, which was
connected with the skin dimple along the sinus tract. Total excision of the
dermoid cyst and the sinus tract with C3-5 laminectomy resulted in good fu
nctional recovery.