Pyoderma gangrenosum (PG) is a vasculitic process, often associated with systemic diseases, including Takayasu's arteritis (TA), rheumatoid arthritis, and inflammatory bowel disease. Most cases of PG associated with TA have been observed in Japan. However, this association is not commonly observed in North American and European patients. PG is often refractory to therapy, requiring high dosages of glucocorticoids. Here, we describe a case of recalcitrant PG that was associated with TA and successfully treated with methotrexate.