Submandibular Gland Hemangioma

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.

Hemangioma in the submandibular gland or submandibular space is rare.¹ In this study, Hemangioma in the submandibular gland in 31-year-old female patient was reported. Moreover, literature review was performed in the submandibular gland hemangioma.

CLINICAL REPORT

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. She had no medical history and no medication. Magnetic resonance imaging (MRI) findings revealed that the mass showed an equal signal in T1-weighted sequence and a high-signal intensity in T2-weighted sequence. MRI STIR sequence showed a 38 × 29 mm well-circumscribed, lobulated mass with high-signal intensity attached with the submandibular gland (Fig. 1). Computed tomography showed phlebolith formation. A clinical diagnosis of suspected hemangioma in submandibular gland was made and scheduled for surgical excision under general anesthesia. Submandibular gland excision including mass was performed with submandibular approach without complications. No unusual bleeding during the surgery was observed, and the total blood loss during the surgery equaled 133 mL. The resected specimen showed well-circumscribed and connected to the submandibular gland (Fig. 2A). X-ray image of the resected specimen showed phlebolith formation (Fig. 2B). 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 (Fig. 3). The final diagnosis was venous hemangioma in submandibular gland. No postoperative recurrence was recognized in 1-year follow-up.

Fig. 1.

MRI finding. MRI STIR sequence coronal image showed a 38 × 29 mm well-circumscribed, lobulated mass with high signal intensity attached with the submandibular gland.

Fig. 2.

Photograph of the resected specimen and the X-ray image. A, The resected specimen. The mass (H) was well circumscribed and connected to the SG. B, x-ray image of the resected specimen. It showed phlebolith formation. The mass (left side) was well circumscribed and connected to the SG (right side). H, hemangioma; SG, submandibular gland.

Fig. 3.

HE-stained specimen. HE finding indicated that proliferation of dilated vessels was observed. Although the boundary with the SG was mainly clear, some parts were irregularly occupied with a component of hemangioma (arrow shows). H, hemangioma; HE, hematoxylin and eosin; SG, submandibular gland.

DISCUSSION

Hemangioma in the submandibular gland is rare. There is no hemangioma in submandibular gland in 779 cases of salivary gland tumors during 25 years in a single hospital, although there are 3 cases of hemangioma in parotid gland.¹ A search of PubMed revealed only 18 cases associated with hemangioma in the submandibular gland in 12 English articles.^2–12 Of these 18 cases, 11 cases (61%) occurred in female and 7 cases (39%) occurred in male. The mean age of the patients was 37.6 ± 4.0 years old (range, 7–70 years). Pathological type of hemangioma in submandibular gland was cavernous (17 cases) and venous (1 case).^2–12 Venous hemangioma is rare in submandinbular gland hemangioma,³ although our case showed venous hemangioma. Although in this case the patient showed painless swelling of submandibular gland, 67% patients showed painful swelling of submandibular gland such as sialadenitis.^2,3,6–12 There is no report of massive bleeding in surgical excision.^2–12 Cho et al.⁶ reported that the amount of bleeding was not severe, except in the case in which the feeding vessel was not ligated in their 4 cases. Aynali et al.⁴ reported that there was an unusual bleeding during dissection from hemangiomatous areas. Kumar et al.⁷ reported that preoperative embolization conferred no benefit in terms of reducing intraoperative blood loss (mean blood loss: 310 mL, with embolization, n = 2, versus 340 mL, without embolization, n = 2). No recurrence has been reported.^2–12 In conclusion, hemangioma in submandibular gland is rare, literature review indicated that (1) approximately 70% patients showed painful swelling^2,3,6–12 and (2) x-ray or computed tomography may indicate the phlebolith calcification.^{4,6,7,10–12} It may make an erroneous diagnosis of sialolithiasis.^4,10,12 MRI would be required for the diagnosis.