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Pyoderma gangrenosum (PG) is a noninfectious, inflammatory, ulcerative neutrophilic dermatosis that mainly involves the extensor surfaces of the legs. PG characteristically presents as a painful wound with suppurative as well as attenuated borders. PG-like wounds have been rarely reported in patients with APS and systemic lupus erythematous (SLE) patients. Rituximab can be effective in the treatment of PG. We report the case of a woman with SLE and APS who developed extensive and progressive lesions consistent with PG that showed rituximab can be a potential treatment for it.