Objective: To report the first case of inadvertent cyclodialysis cleft and hypotony requiring surgical repair following ab-interno trabeculotomy (AIT) using the Trabectome device, and the postoperative clinical results following direct suture cyclopexy.

Methods: A 55-year-old man with hypotonous maculopathy secondary to cyclodialysis cleft inadvertently created 3 years earlier during AIT using the Trabectome device was referred for repair. Direct suture cyclopexy was performed and topical homatropine and dexamethasone drops were prescribed postoperatively.

Results: Gonioscopic examination revealed complete cleft closure on postoperative day 1 confirmed by anterior segment optical coherence tomography. At 11 weeks postoperatively, visual acuity had improved from 20/400 to 20/40, with resolution of preoperative macular folds on fundoscopic examination. At 9 months postoperatively, visual acuity had further improved to 20/20 with intraocular pressure stable at 9 mm Hg maintained on travoprost and brimonidine.

Conclusions: Inadvertent cyclodialysis cleft from a malpositioned AIT and resultant hypotony is rare and in this case was successfully treated by direct suture cyclopexy.