• Xue Wu, Lili Qiao, Tiantian Tian, Yang Shu, and Yingying Zhang.
• Clinical Medical College of Jining Medical University, Jining, Shandong Province, China.
• Medicine (Baltimore). 2023 Sep 1; 102 (35): e34544e34544.

RationaleAdenoid cystic carcinoma (ACC) of orbit is a very rare epithelial tumor, often originating from the lacrimal glands. At the same time, treatment options are currently limited, such as radiation, chemotherapy. We report a case of a patient treated with antirotinib combined with radiotherapy.Patient ConcernsA 13-year-old girl was initially admitted with "left eye swelling for over half a year, 12 days after surgery for left orbital adenoid cystic carcinoma". Initial swelling of the lateral upper eyelid of the left eye, with gradual enlargement and occasional pain.DiagnosesLeft orbital adenoid cystic carcinoma.InterventionsAfter diagnosis of orbital ACC, she underwent resection of the left orbital mass, and received 33 times of adjuvant radiotherapy, but brain metastases appeared later. She refused further treatment, and received 25 times of radiotherapy and anlotinib therapy after the disease progressed again.OutcomesNow the patient has been followed up for 8 months, but no progress was found.LessonsBased on this, we hypothesized that radiation therapy in combination with anlotinib is effective for ACC or ACC metastases.Copyright © 2023 the Author(s). Published by Wolters Kluwer Health, Inc.

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