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Ismaël Chehaibou, Raphaelle Ores, Claire Marie Dhaenens, Camille Andrieux, Michel Paques, Saddek Mohand-Said, José Alain sahel, Isabelle S Audo; Carbonic anhydrase inhibitors for the mangement of cystic macular lesions in X-linked retinoschisis. Invest. Ophthalmol. Vis. Sci. 2017;58(8):614.
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To evaluate the efficacy of carbonic anhydrase inhibitors (CAI) in the treatment of cystic macular lesions in patients with X-linked retinoschisis (XLRS).
Retrospective interventional study of XLRS patients treated either with oral or topical CAI. Data were collected from patient charts including demographic features, clinical and spectral-domain optical coherence tomography (SD-OCT) findings, initial and final best-corrected visual acuity (BCVA). All included patients presented molecularly confirmed XLRS. We evaluated the effect of the first line treatment on both foveal zone thickness (FZT) measured on the same SD-OCT and VA. An intermediate analyzis of FZT was performed between 3 to 6 months after initiation of treatment. A FZT change was considered statistically significant if more than 17.1% of baseline.
Thirty-three eyes of 17 patients (mean age 21.90 years, range 4 - 64) were included in this study. The mean duration of the first line treatment was 19.3 months (range 3 - 60 months). Twelve eyes were treated orally (acetazolamide) and 21 eyes topically (dorzolamide or brinzolamide). The mean BCVA remained unchanged from baseline to the end of first line therapy (from 0.43 to 0.43 LogMAR, p=0.96). Mean FZT decreased of 3.7% from 407.09 µm +/- 108.83 to 392.00 µm +/- 118.04 (p=0.43) after the first line therapy. Six of the 33 eyes (18.20%) had a statistically significant reduction in the FZT. In the systemic treatment subgroup, 4 of 12 eyes (30%) had a significant improvement of the FZT and 1 of 21 eyes (4.71%) in the topical therapy subgroup. Intermediate analyzis was available for 26 eyes and revealed mean reduction of 11.71% of the initial FZT, from 407.09 +/- 108.83 µm to 365.65 µm +/- 120.14 (p=0.004). Five eyes (19.20%) had a statistically significant improvement in FZT. We did not observed significant increase of FZT in any eye.
This study outlines the fluctuation of macular cysts during the CAI therapy in patients with XLRS. Visual acuity remained stable during the follow-up period and we could not demonstrate significant long term reduction in FZT. A subgroup analysis with assessment of the hyper reflective structure of external retina is needed with a longer-term follow-up to evaluate whether selected patients may benefit of CAI treatment.
This is an abstract that was submitted for the 2017 ARVO Annual Meeting, held in Baltimore, MD, May 7-11, 2017.
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