Purpose: The axillary web syndrome (AWS) is a selflimiting cause of morbidity in the early postoperative period after axillary surgery. This article presents a rare complication developed after surgical treatment for axillary osmidrosis. Methods: A 55-year-old male patient underwent surgical excision of skin and glandular tissue for axillary osmidrosis. Three weeks after the surgery, he visited our department due to a visible web of left axillary skin overlying palpable cord extends into the medial ipsilateral arm. There was a taut and tender cord of tissue under the skin and shoulder abduction was limited to less than 90 degrees. He was diagnosed with AWS and treated with conservative management. Results: Four months later, AWS resolved completely without any treatment and patient was free of pain or motion restriction. Conculsion: Axillary web syndrome has been described as frequent complication after axillary lymph node dissection or sentinel node biopsy, but not after surgical treatment of axillary osmidrosis. Surgeons must be aware of the risk of axillary web syndrome after treatment of axillary osmidrosis.