Clinical findings in Patient 1 with extensive conjunctival melanoma before and after treatment with immune checkpoint blockade. (A,B)Before treatment, primary acquired melanosis and wide areas of pigmentation were present involving the bulbar, forniceal, and tarsal conjunctiva as well as the upper punctum, semilunar fold, and caruncle. (C,D)There was modest response after 4 cycles of pembrolizumab. (E,F) After 8 months (10 cycles) of pembrolizumab, there was marked improvement of all sites of pigmentation. After 12 months of therapy with pembrolizumab, there was near-total resolution of all pigmented lesions (G,H). Multiple biopsies of all residual conjunctival pigmentation showed pigmented macrophages and no melanoma cells.