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

J AAPOS. 2011 Dec;15(6):598-600. doi: 10.1016/j.jaapos.2011.07.011.

Abstract

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.

Publication types

  • Case Reports

MeSH terms

  • Adjuvants, Pharmaceutic / therapeutic use
  • Adolescent
  • Cataract / drug therapy
  • Cataract / pathology
  • Drug Therapy, Combination
  • Enzyme Inhibitors / therapeutic use*
  • Eye Diseases / drug therapy*
  • Eye Diseases / pathology
  • Glucocorticoids / therapeutic use*
  • Humans
  • Male
  • Methotrexate / therapeutic use*
  • Treatment Outcome
  • Xanthogranuloma, Juvenile / drug therapy*
  • Xanthogranuloma, Juvenile / pathology

Substances

  • Adjuvants, Pharmaceutic
  • Enzyme Inhibitors
  • Glucocorticoids
  • Methotrexate