A sialoblastoma is a rare congenital epithelial tumor that arises in a major salivary gland. To our knowledge, only 24 cases of sialoblastoma have been previously reported in the English-language literature. We report a new case, that of a 15-month-old boy who presented with a submandibular mass. Surgical excision of the mass was undertaken. Intraoperatively, the mass appeared to be adjacent to the submandibular gland, but it had not invaded it. The mass was excised, and the submandibular gland was left in place. Pathology identified the tumor as a sialoblastoma. However, pathology also revealed that residual tumor was present at the surgical margin. The patient was returned to the operating room for excision of the left submandibular gland and the level I lymph nodes. Following revision surgery, the surgical margins were negative. The patient remained disease-free at the 1-year follow-up. Despite the need for revision surgery, this case provides support for the idea that surgery alone is sufficient for curative treatment.
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