Background: Two patients, a 13-year-old girl and a 31-year-old man, ha
d an orbital dermoid tumor located in the lateral rectus muscle. This
unusual location of dermoid tumors has not been reported previously. M
ethods: Computed tomography and magnetic resonance imaging showed a cy
stic mass within the belly of the lateral rectus in each patient. Surg
ical excision through a lateral orbitotomy showed a well-circumscribed
mass surrounded by lateral rectus fibers. No periorbital attachment w
as noted. Results: Results of histopathologic evaluations showed a der
moid cyst. Postoperatively, the diplopia and proptosis resolved. In on
e patient, an unusual subconjunctival deposition of fat droplets was s
een. Conclusion: The findings in patients demonstrate an unusual prese
ntation of dermoid cysts and make an addition to the differential diag
nosis of enlarged extraocular muscles.