TY - JOUR
T1 - Bilateral uveitis in a patient with autoimmune lymphoproliferative syndrome
AU - Lim, Wee Kiak
AU - Ursea, Roxana
AU - Rao, Koneti
AU - Buggage, Ronald R.
AU - Suhler, Eric B.
AU - Dugan, Faith
AU - Chan, Chi Chao
AU - Straus, Stephen E.
AU - Nussenblatt, Robert B.
PY - 2005/3
Y1 - 2005/3
N2 - 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.
AB - 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.
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U2 - 10.1016/j.ajo.2004.09.017
DO - 10.1016/j.ajo.2004.09.017
M3 - Article
C2 - 15767081
AN - SCOPUS:15044365268
SN - 0002-9394
VL - 139
SP - 562
EP - 563
JO - American journal of ophthalmology
JF - American journal of ophthalmology
IS - 3
ER -