| Summary: | Abstract Background Among benign salivary gland tumors, pleomorphic adenoma is one of the most common tumor involving submandibular gland, but it is rare to encounter recurrent pleomorphic adenoma in submandibular gland after submandibular gland excision. Case presentation We present a case of 22-year-old female patient who presented to us with recurrent pleomorphic adenoma of submandibular gland region after 10 years of removal of submandibular gland, diagnosis was established with the help of ultrasound and fine-needle aspiration cytology, and thereafter, patient was taken up for surgical excision under general anesthesia. Histopathology confirmed the diagnosis. Conclusions This case report highlights the importance of special caution in removal of submandibular gland along with its capsule especially in pediatric population where due to small facial structures, chances of capsular breech are high, which can result in tumor seedling and future recurrence of tumor.
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