Juvenile xanthogranulomatosis with bilateral and multifocal ocular lesions of the iris, cornealscleral limbus, and choroid.

A 14-month-old boy with juvenile xanthogranuloma skin lesions presented with increased intraocular pressure, hyphema, anterior uveitis, iris mass, and a subconjunctival limbal mass of the right eye. He subsequently developed a subretinal mass in the left eye. The anterior uveitis resolved after 2 periocular injections of triamcinolone in addition to the administration of topical prednisolone and oral prednisone and methotrexate. The subretinal mass in the left eye also resolved during the course of 1 year. He developed a cataract in the right eye and underwent lensectomy with anterior vitrectomy. This is the first published case in which methotrexate was used as an adjunctive treatment of juvenile xanthogranuloma in a child.