Primary epithelioid angiosarcoma originating from the mandibular gingiva: a case report of an extremely rare oral lesion.

Primary epithelioid angiosarcoma originating from the mandibular gingiva: a case report of an extremely rare oral lesion.
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DOI:
10.1186/s12957-020-01999-1
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发表时间:
2020-10-03
影响因子:
3.2
通讯作者:
Yamada H
Yamada H
中科院分区:
医学3区
文献类型:
--
作者:
Komatsu Y;Miyamoto I;Ohashi Y;Katagiri K;Saito D;Obara M;Takeda Y;Shiga K;Yamada H

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血管肉瘤很少发生在口腔,上皮样型更是罕见。在这里,我们报告一个罕见的情况下,涉及一个老年男子与原发性上皮样血管肉瘤,起源于下颌牙龈和类似的含牙囊肿的放射线照片。一位66岁的日本男性来我院就诊,主诉为右下颌第三磨牙区牙龈肿胀。曲面断层片显示右侧下颌第三磨牙牙冠周围骨吸收,该磨牙被阻生。切开活检证实了上皮样血管肉瘤的诊断。病变表现出积极的增殖活检后,导致不受控制的出血和难以关闭的嘴。包括肿瘤在内的下颌骨节段切除术未进行重建。由于肿瘤的侵袭性术前病程,患者接受了辅助化疗。在2年的随访期间没有复发的迹象。文献回顾发现只有4例报告的颌骨上皮样血管肉瘤,其中3例发生在上颌骨。据我们所知,这是第二例原发于下颌骨的上皮样血管肉瘤。
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.
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