Rationale:Trichilemmal carcinoma (TLC) is a rare malignant cutaneous adnexal tumor usually accept surgery. This report describes an elderly patient with recurrence TLC of the periorbital region after surgery who was subsequently treated with IMRT radiotherapy. After 2-years follow-up visit, there was no progress or metastasis.Introduction:TLC is a rare malignant cutaneous adnexal tumor. It usually occurs on sun-exposed areas in elderly people but rarely occurs in the periorbital region. Most cases accept surgery or micrographic Mohs surgery. Recurrence or metastasis of this neoplasm was seldom reported in the medical literature after enough tumor-free margin surgery. And radiotherapy was seldom reported in the treatment for patients of TLC.Patient concerns:Here we report an elderly patient with recurrence TLC of the periorbital region after surgery who was subsequently treated with radiotherapy with a total dose of 66 Gy. Two years later, the patient was admitted head, neck, chest, abdomen CT scan, and no progress or metastasis was detected after 2-years follow-up.Diagnosis:Trichilemmal carcinoma of the periorbital region.Interventions:We describe the clinical characteristics, pathological features, and choice of examination methods of a patient with TLC in the periorbital region. And we use the radical radiotherapy to treat this case.Outcomes:There are no progress or metastasis after 2-years follow-up.Conclusion:Radiotherapy is a good option for patients with TLC if the patient refuses surgery or fails to achieve a satisfactory tumor-free margin or relapses after surgery.