Oculodermal melanocytosis (ODM) is a rare condition with glaucoma being the most common complication in these eyes. Open angle glaucoma is the commonest variant and presence of angle closure in these eyes should raise a suspicion of ciliary body or uveal tumor, which needs to be evaluated before any surgical intervention is planned. We report a case of unilateral ODM with secondary angle closure glaucoma. Angle closure in an eye with ODM is extremely rare, ultrasound B-scan showed thickened choroid, and ultrasound biomicroscopy showed 360° ciliary body hypertrophy. Incisional biopsy & fine needle aspiration cytology of the ciliary body tissue was performed, and histopathological features were suggestive of a benign hypertrophy ruling out ciliary body melanoma. Subsequently, a glaucoma drainage device surgery was performed in the right eye. To the best of our knowledge, benign ciliary body hypertrophy causing intractable angle closure glaucoma in a case of ODM has not been reported.

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