Komatsu Yuko, Miyamoto Ikuya, Ohashi Yu, Katagiri Katsunori, Saito Daisuke, Obara Mizuki, Takeda Yasunori, Shiga Kiyoto, Yamada Hiroyuki
Division of Oral and Maxillofacial Surgery, Department of Oral and Maxillofacial Reconstructive Surgery, School of Dentistry, Iwate Medical University, 19-1 Uchimaru, Morioka, Iwate, 020-8505, Japan.
Head and Neck Cancer Center, Iwate Medical University, 2-1-1 Idaidori, Yahaba-cho, Shiwa-gun, Iwate, 028-3694, Japan.
World J Surg Oncol. 2020 Oct 3;18(1):260. doi: 10.1186/s12957-020-01999-1.
Angiosarcoma occurs very rarely in the oral cavity, and the epithelioid type is even rarer. Here, we report a rare case involving an elderly man with a primary epithelioid angiosarcoma that originated from the mandibular gingiva and resembled a dentigerous cyst on radiographs.
A 66-year-old Japanese man visited our hospital with a chief complaint of gingival swelling in right mandibular third molar region. A panoramic radiograph showed bone resorption around the crown of right mandibular third molar, which was impacted. Incisional biopsy confirmed a diagnosis of epithelioid angiosarcoma. The lesion exhibited aggressive proliferation after biopsy resulting in uncontrolled bleeding and difficulty in closing the mouth. Mandibular segmental resection including the tumor was performed without reconstruction. Because of the aggressive preoperative course of the tumor, the patient received adjuvant chemotherapy. There were no signs of recurrence during a 2-year follow-up period.
A review of the literature yielded only four reported cases of epithelioid angiosarcoma in the jaw region, with the lesions occurring in the maxilla in three cases. To our knowledge, this is the second case of primary epithelioid angiosarcoma in the mandible.
血管肉瘤在口腔中极为罕见，上皮样型则更为罕见。在此，我们报告一例罕见病例，患者为一名老年男性，患有源自下颌牙龈的原发性上皮样血管肉瘤，X线片上类似含牙囊肿。
一名66岁的日本男性因右下颌第三磨牙区域牙龈肿胀为主诉前来我院就诊。全景X线片显示右下颌第三磨牙牙冠周围骨质吸收，该牙为阻生牙。切开活检确诊为上皮样血管肉瘤。活检后病变呈侵袭性增殖，导致出血不止且张口困难。行包括肿瘤在内的下颌骨节段性切除，未进行重建。由于肿瘤术前进展迅速，患者接受了辅助化疗。在2年的随访期内无复发迹象。
文献回顾仅报道了4例颌骨区域的上皮样血管肉瘤病例，其中3例病变发生在上颌骨。据我们所知，这是下颌骨原发性上皮样血管肉瘤的第二例病例。
