Bilateral uveitis in a patient with autoimmune lymphoproliferative syndrome

Am J Ophthalmol. 2005 Mar;139(3):562-3. doi: 10.1016/j.ajo.2004.09.017.

Abstract

Purpose: We report a case of autoimmune lymphoproliferative syndrome (ALPS) presenting with bilateral uveitis.

Design: Observational case report.

Methods: Review of case record, serum and aqueous IL-10 and IL-6 cytokine results, and immunosuppressive treatment of a patient with a mutation in the gene encoding Fas.

Results: Control of the intermediate uveitis required sustained doses of topical and periocular corticosteroids as well as systemic cyclosporine. The serum IL-10 level was elevated, as commonly seen in ALPS, but the aqueous IL-10 was not.

Conclusions: Despite a Th2 immune predominance in ALPS, uveitis, a Th1-mediated disease, may still manifest in these patients. The pathogenesis of uveitis in ALPS may differ from that of the systemic disease overall. Long-term follow-up is required for patients with uveitis associated with ALPS.

Publication types

  • Case Reports

MeSH terms

  • Aqueous Humor / metabolism
  • Autoimmune Diseases / complications*
  • Autoimmune Diseases / drug therapy
  • Autoimmune Diseases / immunology
  • Child
  • Drug Therapy, Combination
  • Female
  • Glucocorticoids / therapeutic use
  • Humans
  • Immunosuppressive Agents / therapeutic use
  • Interleukin-10 / blood
  • Interleukin-6 / blood
  • Lymphoproliferative Disorders / complications*
  • Lymphoproliferative Disorders / drug therapy
  • Lymphoproliferative Disorders / immunology
  • Mutation
  • Th1 Cells / immunology
  • Uveitis, Intermediate / complications*
  • Uveitis, Intermediate / drug therapy
  • Uveitis, Intermediate / immunology
  • fas Receptor / genetics

Substances

  • Glucocorticoids
  • Immunosuppressive Agents
  • Interleukin-6
  • fas Receptor
  • Interleukin-10