This study reports a case of Blastomyces dermatitidis soft tissue infection resulting in a disfiguring lower eyelid ectropion from cicatricial and postinflammatory cutaneous changes. Primary treatment included intravenous amphotericin B followed by long-term oral itraconazole, which resulted in complete remission of the disease without debridement, after which cicatricial ectropion was repaired surgically with scar release, full-thickness skin graft, and temporary Frost tarsorraphy. Cutaneous blastomycosis may cause severe oculofacial sequelae, ranging from eyelid ectropion to widespread facial cicatrix, and may mimic other more common infectious processes, in addition to malignancy. Recommended antifungal therapy includes induction with intravenous amphotericin B and a long course of oral antifungals, preferably coordinated in conjunction with an infectious disease specialist. Ectropion repair should be delayed until the inflammatory response has completely healed. If the ocular surface is compromised or nearby ocular structures are threatened, primary debridement and repair may be indicated.