Purpose: To report an unusual presentation of pyoderma gangrenosum with ocular and extracutaneous involvement. Design: A single, interventional case report. Methods: A 28-year-old female presented with nodular scleritis in the right eye and left orbital inflammation lasting 1 year. The patient initially responded to systemic steroids, but then ocular inflammation became more severe, associated with onset of fever, myalgias, and elevation of the erythrocyte sedimentation rate, accompanied by formation of multiple cutaneous abscesses. Main Outcome Measures: Ocular and systemic disease control. Results: The diagnosis of pyoderma gangrenosum was made 1 year after the occurrence of ocular symptomatology, when the patient presented with ocular and skin lesions and abscesses in the spleen and liver. Conclusions: Pyoderma gangrenosum is a rare, necrotizing, noninfectious ulcerating skin disease. Ocular and extracutaneous involvement rarely occur, but the disease should be included in the differential diagnosis of scleritis and orbital inflammation.
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