Pyoderma gangrenosum(PG)is a rare autoinflammatory disease,characterized mainly by painful and necrotic skin ulcers.The etiology of PG is unknown,and its treatment is quite challenging.This article reports a case of recurrent pyoderma gangrenosum successfully treated with adalimumab combined with tofacitinib,along with a review of relevant literature.The patient,a 54-year-old male,presented with scrotal ulcers accompanied by pain for 20 days,worsening and involving the groin area for 5 days,and fever for 1 day.Dermatological examination revealed two painful ulcers on the scrotum with raised edges,clear boundaries,and a small amount of purulent discharge on the surface;the right groin area and the medial left thigh showed irregular infiltrative erythematous plaques,with scattered clustered pustules on them,central necrosis appearing purplish-brown,surrounded by a red halo with clear boundaries.Based on the patient's past medical history,clinical manifestations,and auxiliary examinations,the diagnosis was consistent with pyoderma gangrenosum.After treatment with adalimumab combined with tofacitinib,the patient's symptoms were significantly improved,and the ulcers healed.At the 6-month follow-up,the skin lesions had not recurred.For refractory PG that is ineffective to conventional treatments,adalimumab combined with tofacitinib is an effective and safe treatment option,providing a new combination therapy regimen for PG.