Pyoderma gangrenosum is a known, though infrequent, ulcerative skin lesion that is often associated with systemic inflammatory and immunologic diseases. Objective: To present the efficacy of Adalimumab, a fully human Tumour Necrosis Factor-α antagonist, in the treatment of pyoderma gangrenosum associated with Crohn's disease. Methods: A young male patient with pyoderma gangrenosum of the shin as the first sign of clinically active Crohn's disease, who was treated with Adalimumab, is herein presented. Results: Almost complete healing of the lesion was achieved after 12 weeks of treatment. Conclusions: Successful treatment outcome suggests that Adalimumab is not only a therapeutic option for Crohn's disease but may also be a safe and effective therapy for Pyoderma gangrenosum associated with the disease. Pyoderma gangrenosum should be suspected in any cutaneous ulcerative lesion occurring in patients with Crohn's disease.