To report the treatment results of a frontotemporal dermoid cyst with a cutaneous fistula and sinus tract that caused recurrent periorbital cellulitis in a child.

A 4-year-old girl who presented with left orbital swelling and tenderness visited our hospital. She had a cutaneous fistula with a small amount of purulent discharge at the left frontotemporal area. Orbital computed tomography scans showed a well-defined low density lesion in the fronto-zygomatic suture, and there was a bony defect in the left greater wing of the sphenoid bone of the orbit. Orbital magnetic resonance imaging showed a cutaneous fistula and sinus tract that extended into the middle cranial fossa. The patient was treated with intravenous antibiotics until the inflammation was resolved. Surgery was performed to remove the dermoid cyst with sinus tract. After surgery, there was no evidence of recurrence, and complications included neurologic and ophthalmic symptoms.

Orbitofacial lesions, particularly frontotemporal cutaneous fistulas that present with recurrent discharge, should be regarded with suspicion in cases of deep extended dermoid cysts with sinus tract. Additionally, imaging tests should be carefully conducted before surgery.