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Children who have lymphatic malformations frequently experience functional limitations and aesthetic abnormalities that have a significant impact on their quality of life and may pose a threat to their lives. Conventional treatments such as surgery or sclerotherapy are rarely curative, demonstrating the great need for new treatment modalities. Recently, oral or topical administration of sirolimus has successfully treated lymphatic malformations. We report the 6-month treatment outcome of a 7-year-old boy with lesions consistent with lymphangioma circumscriptum on the left side of the neck since birth who was successfully treated with topical sirolimus.