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. 2010 Sep;128(9):1146-50.
doi: 10.1001/archophthalmol.2010.172.

Efficacy of sustained topical dorzolamide therapy for cystic macular lesions in patients with retinitis pigmentosa and usher syndrome

Mohamed A Genead  1 , Gerald A Fishman
Affiliations

Efficacy of sustained topical dorzolamide therapy for cystic macular lesions in patients with retinitis pigmentosa and usher syndrome

Mohamed A Genead et al. Arch Ophthalmol. 2010 Sep.

Abstract

Objective: To determine the efficacy of sustained topical therapy with dorzolamide hydrochloride, 2%, on visual acuity and cystic macular lesions in patients with retinitis pigmentosa and Usher syndrome.

Methods: In a retrospective case series at a university hospital, 64 eyes of 32 patients with retinitis pigmentosa or Usher syndrome receiving treatment with the topical dorzolamide formulation for 6 to 58 months were enrolled. Changes in visual acuity on the Early Treatment Diabetic Retinopathy Study chart and central foveal zone thickness on optical coherence tomography were measured during follow-up for the duration of treatment.

Results: Among the study cohort, 20 of 32 patients (63%) showed a positive response to treatment in at least 1 eye and 13 patients (41%) showed a positive response in both eyes. Four patients (20%) showed an initial response and a subsequent rebound of macular cysts. In 8 patients (25%), there was no response to treatment and the macular cysts worsened when compared with the pretreatment level. Ten patients (31%) had improvement in visual acuity by 7 or more letters in at least 1 eye at the most recent follow-up visit. Sixteen patients (67%) showed a reduction of more than 11% in the central foveal zone thickness in at least 1 eye when compared with the pretreatment level.

Conclusions: Patients with either retinitis pigmentosa or Usher syndrome who received treatment of cystoid macular edema with topical dorzolamide followed by an optical coherence tomography-guided strategy showed a decrease in central foveal zone thickness in most cases. Visual acuity improved in almost one-third of the cases, suggesting a potential corresponding visual benefit.

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Conflict of interest statement

None of the authors have any proprietary interest in this work.

Figures

Figure 1
Figure 1
Horizontal OCT scans in 2 patients [1 patient with USH syndrome type-I (left column) and 1 with RP (right column)] on treatment with topical dorzolamide hydrochloride 2%. Both patients demonstrated marked improvement of their cystic macular lesions on spectral-domain OCT (left column) and time-domain OCT (right column).
Figure 2
Figure 2
Horizontal time-domain OCT scans of a patient with RP. The sequence of scans demonstrate an example of an initial improvement of macular cysts and subsequent rebound after decreasing the dose of topical dorzolamide hydrochloride 2% from three time a day to twice a day followed subsequently by an improvement of the macular cysts after an increase in the dose of topical dorzolamide back to three times a day.
Figure 3
Figure 3
Horizontal time-domain OCT scans of a patient with RP. The scans demonstrate an example of mild worsening of macular cysts while on treatment with topical dorzolamide hydrochloride 2%.
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