Abstract
Background: Midline developmental neck lesions primarily consist of thyroglossal duct remnants. Their recurrence is uncommon following thorough resection, which includes hyoid removal (the Sistrunk procedure). Case report: A 3-year-old girl presented with mucoid secretion drainage and swelling in the anterior mid-neck region, clinically resembling a thyroglossal duct remnant. Following an initial Sistrunk procedure, the lesion recurred, prompting a subsequent resection. Histological analysis revealed a mucocele alongside acinar and mucous ectopic salivary glands. Conclusions: The ectopic salivary gland can manifest along the midline of the neck and may clinically resemble the signs and symptoms of a thyroglossal duct cyst. Importantly, it can exhibit recurrence post-surgery, even following hyoid resection