| Summary: | Summary:. Hemangioma in the submandibular gland or submandibular space is rare. A 31-year-old woman was referred to our department. She became aware of painless swelling in the right submandibular gland at 9 months ago. The right submandibular gland showed swelling with no tenderness, and the palpation was softer than the normal submandibular gland. Magnetic resonance imaging short T1 inversion recovery (STIR) sequence showed a 38 × 29 mm well-circumscribed, lobulated mass with high signal intensity attached with the submandibular gland. Computed tomography showed phlebolith formation. A clinical diagnosis of suspected hemangioma in submandibular gland was made, and submandibular gland excision including mass was performed with submandibular approach without complications. No unusual bleeding during the surgery was observed. Pathological finding indicated that thick walled vessels containing smooth muscle were proliferated with dilatation. Some vascular lumen included phlebolith calcification. Although the boundary with the submandibular gland was mainly clear, some parts were irregularly occupied with a component of hemangioma. The final diagnosis was venous hemangioma in submandibular gland. No postoperative recurrence was recognized in 1-year follow-up. A search of PubMed revealed that only 18 cases associated with hemangioma in the submandibular gland in 12 English articles. Literature review indicated that (1) approximately 70% patients showed painful swelling and (2) x-ray or computed tomography may indicate the phlebolith calcification. It may make an erroneous diagnosis of sialolithiasis. Magnetic resonance imaging finding would be needed for the diagnosis.
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