AIM: To evaluate the efficacy of intravenous immunoglobulin (IVIg) in the treatment of Birdshot retinochoroiditis. PATIENTS AND METHODS: Eighteen patients were followed-up prospectively in a total number of 37 patients recorded. Inclusion criteria were birdshot retinochoroiditis (BRC) according to the criteria defined by Ryan et al., and a decrease in visual acuity (VA). Efficacy was assessed by measurements of visual acuity and a decrease in inflammation and macular edema on fluorescein angiograms. RESULTS: Sex ratio=1, mean age was 51 (range 29 to 72 years), HLA A29 was positive in 100% of the patients. VA at baseline was 0.6+/-2.4 (range 0.25 to 0.9). Angiography showed retinal vasculitis in 32 patients (86.48%) and cystoid macular edema in 16 patients (43.24%). IVIg was administered at a dosage of 0.4g/kg/d for 4 days then 0.6g/kg/d for 2 days every 4 weeks. Follow-up lasted a mean of 2.7+/-2.0 years (range, 4 months to 5.6 years). Visual acuity of 35 out of 66 eyes (53%) increased by 2.6+/-1.5 (range 1 to 5). In 19 eyes out of 66 (29%), VA remained stable and in 12 eyes out of 66 (18%), VA decreased by -1.8+/-0.8 (ranges-4 to -1). Inflammation on fluorescein angiography improved in 17 patients (81%) and cystoid macular edema decreased in 65% of the cases. Side effects were rare. Treatment was discontinued for side effects in only 3 patients. DISCUSSION: BRC is a chronique disease threatening vision. Treatments including systemic corticosteroids and cyclosporin A may result in severe long-term side effects. IVIg is well tolerated and may be therapeutic alternative for the treatment of BRC.
AIM: To evaluate the efficacy of intravenous immunoglobulin (IVIg) in the treatment of Birdshot retinochoroiditis. PATIENTS AND METHODS: Eighteen patients were followed-up prospectively in a total number of 37 patients recorded. Inclusion criteria were birdshot retinochoroiditis (BRC) according to the criteria defined by Ryan et al., and a decrease in visual acuity (VA). Efficacy was assessed by measurements of visual acuity and a decrease in inflammation and macular edema on fluorescein angiograms. RESULTS: Sex ratio=1, mean age was 51 (range 29 to 72 years), HLA A29 was positive in 100% of the patients. VA at baseline was 0.6+/-2.4 (range 0.25 to 0.9). Angiography showed retinal vasculitis in 32 patients (86.48%) and cystoid macular edema in 16 patients (43.24%). IVIg was administered at a dosage of 0.4g/kg/d for 4 days then 0.6g/kg/d for 2 days every 4 weeks. Follow-up lasted a mean of 2.7+/-2.0 years (range, 4 months to 5.6 years). Visual acuity of 35 out of 66 eyes (53%) increased by 2.6+/-1.5 (range 1 to 5). In 19 eyes out of 66 (29%), VA remained stable and in 12 eyes out of 66 (18%), VA decreased by -1.8+/-0.8 (ranges-4 to -1). Inflammation on fluorescein angiography improved in 17 patients (81%) and cystoid macular edema decreased in 65% of the cases. Side effects were rare. Treatment was discontinued for side effects in only 3 patients. DISCUSSION: BRC is a chronique disease threatening vision. Treatments including systemic corticosteroids and cyclosporin A may result in severe long-term side effects. IVIg is well tolerated and may be therapeutic alternative for the treatment of BRC.
Authors: Evangelos Minos; Robert J Barry; Sue Southworth; Annie Folkard; Philip I Murray; Jay S Duker; Pearse A Keane; Alastair K Denniston Journal: Orphanet J Rare Dis Date: 2016-05-12 Impact factor: 4.123