Purpose To determine the incidence rate, principal causes, and clinical course of eyes developing no light perception (NLP) visual acuity (VA) following Boston Keratoprosthesis (B-KPro) type 1 surgery. Secondary objectives include determining the incidence rate, relative risk (RR), and survival probability with respect to NLP outcomes among eyes with congenital aniridia. Design Retrospective, interventional case series. Subjects All patients undergoing B-KPro type 1 surgery between October 2008 and June 2016 by a single surgeon at CHUM – Hopital Notre-Dame. Methods Records of patients having undergone B-KPro implantation were reviewed. Eyes with a final outcome of NLP were further reviewed to determine best recorded postoperative VA, time to NLP onset, clinical course, and principal cause. Descriptive statistics, incidence rates, Kaplan-Meier survival curves, and the RR of NLP outcomes among eyes with aniridia were determined. Statistical significance was defined as P Results Records of 119 patients were included, with an average follow-up of 49.1 ± 26.8 months postoperatively. Nineteen eyes had a final outcome of NLP, representing 16.0%. The incidence rate of NLP was 0.04 cases per eye-year of follow-up. The most common principal causes were inoperable retinal detachment (n = 7, 36.8%), terminal glaucoma (n = 6, 31.6%), and carrier graft melt-related complications (n = 5, 26.3%). The RR of developing NLP among eyes with aniridia was 3.04 ( P = .01). Conclusions No light perception is a devastating but uncommon outcome of B-KPro surgery. Patients with aniridia seem to be at increased risk. In spite of all available medical and surgical interventions, some eyes may still suffer this outcome.