The authors treated a 38-year-old man with nanophthalmic uveal effusion with scleral windows and local application of mitomycin-C. The patient underwent two separate surgical procedures, involving placement of scleral windows in all four quadrants and topical application of mitomycin-C in one quadrant. Episcleral scarring led to closure of scleral windows in all quadrants except one quadrant where mitomycin-C was applied during the first surgery. The uveal effusion resolved following the second surgery. No complications related to mitomycin-C were observed. Topical mitomycin-C may be a useful adjunct in the surgical management of uveal effusion syndrome.