OBJECTIVE: To determine the efficacy of sustained topical therapy with dorzolamide hydrochloride, 2%, on visual acuity and cystic macular lesions in patients with juvenile X-linked retinoschisis (XLRS). DESIGN: Retrospective analysis. SETTING: University hospital, tertiary care referral center. PATIENTS: Twenty-nine eyes of 15 patients with XLRS receiving treatment with the topical dorzolamide formulation for 4 to 41 months were enrolled. MAIN OUTCOME MEASURES: Changes in visual acuity, cystic macular lesions, and central foveal zone thickness on optical coherence tomography during follow-up for the duration of treatment. RESULTS: Among the 15 patients with XLRS, 20 eyes (69%) of 11 patients showed a positive response to treatment. Five of the 20 eyes (25%) in 3 of the 11 patients showed an initial response and a subsequent rebound of macular cysts. In 4 eyes (14%) of 3 patients, there was no response to treatment, but the macular cysts did not worsen compared with the baseline level. In 5 additional eyes (17%) of 4 patients, there was no response to treatment, and the macular cysts worsened when compared with the baseline level. Sixteen eyes (55%) of 12 patients had improvement in best-corrected visual acuity by at least 7 letters in at least 1 eye at the most recent follow-up visit. Seventeen eyes (59%) of 10 patients showed a reduction in the central foveal zone thickness in at least 1 eye when compared with the pretreatment level. CONCLUSION: Patients with XLRS have the potential to experience a beneficial effect from sustained treatment with dorzolamide, 2%.
OBJECTIVE: To determine the efficacy of sustained topical therapy with dorzolamide hydrochloride, 2%, on visual acuity and cystic macular lesions in patients with juvenile X-linked retinoschisis (XLRS). DESIGN: Retrospective analysis. SETTING: University hospital, tertiary care referral center. PATIENTS: Twenty-nine eyes of 15 patients with XLRS receiving treatment with the topical dorzolamide formulation for 4 to 41 months were enrolled. MAIN OUTCOME MEASURES: Changes in visual acuity, cystic macular lesions, and central foveal zone thickness on optical coherence tomography during follow-up for the duration of treatment. RESULTS: Among the 15 patients with XLRS, 20 eyes (69%) of 11 patients showed a positive response to treatment. Five of the 20 eyes (25%) in 3 of the 11 patients showed an initial response and a subsequent rebound of macular cysts. In 4 eyes (14%) of 3 patients, there was no response to treatment, but the macular cysts did not worsen compared with the baseline level. In 5 additional eyes (17%) of 4 patients, there was no response to treatment, and the macular cysts worsened when compared with the baseline level. Sixteen eyes (55%) of 12 patients had improvement in best-corrected visual acuity by at least 7 letters in at least 1 eye at the most recent follow-up visit. Seventeen eyes (59%) of 10 patients showed a reduction in the central foveal zone thickness in at least 1 eye when compared with the pretreatment level. CONCLUSION:Patients with XLRS have the potential to experience a beneficial effect from sustained treatment with dorzolamide, 2%.
Authors: Guo-Jie Ye; Ewa Budzynski; Peter Sonnentag; Paul E Miller; Alok K Sharma; James N Ver Hoeve; Kellie Howard; David R Knop; Martha Neuringer; Trevor McGill; Jonathan Stoddard; Jeffrey D Chulay Journal: Hum Gene Ther Clin Dev Date: 2015-09 Impact factor: 5.032
Authors: Jacque L Duncan; Kavitha Ratnam; David G Birch; Sanna M Sundquist; Anna S Lucero; Yuhua Zhang; Meira Meltzer; Nizar Smaoui; Austin Roorda Journal: Invest Ophthalmol Vis Sci Date: 2011-12-20 Impact factor: 4.799