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

Abstract Background 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. Ca...

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Main Authors: Yuko Komatsu, Ikuya Miyamoto, Yu Ohashi, Katsunori Katagiri, Daisuke Saito, Mizuki Obara, Yasunori Takeda, Kiyoto Shiga, Hiroyuki Yamada
Format: Article
Language:English
Published: BMC 2020-10-01
Series:World Journal of Surgical Oncology
Subjects:
Online Access:http://link.springer.com/article/10.1186/s12957-020-01999-1
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author Yuko Komatsu
Ikuya Miyamoto
Yu Ohashi
Katsunori Katagiri
Daisuke Saito
Mizuki Obara
Yasunori Takeda
Kiyoto Shiga
Hiroyuki Yamada
author_facet Yuko Komatsu
Ikuya Miyamoto
Yu Ohashi
Katsunori Katagiri
Daisuke Saito
Mizuki Obara
Yasunori Takeda
Kiyoto Shiga
Hiroyuki Yamada
author_sort Yuko Komatsu
collection DOAJ
description Abstract Background 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. Case presentation 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. Conclusions 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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spelling doaj.art-c39f531d11e04e62b2b77ef83faade392022-12-21T23:58:15ZengBMCWorld Journal of Surgical Oncology1477-78192020-10-011811610.1186/s12957-020-01999-1Primary epithelioid angiosarcoma originating from the mandibular gingiva: a case report of an extremely rare oral lesionYuko Komatsu0Ikuya Miyamoto1Yu Ohashi2Katsunori Katagiri3Daisuke Saito4Mizuki Obara5Yasunori Takeda6Kiyoto Shiga7Hiroyuki Yamada8Division of Oral and Maxillofacial Surgery, Department of Oral and Maxillofacial Reconstructive Surgery, School of Dentistry, Iwate Medical UniversityDivision of Oral and Maxillofacial Surgery, Department of Oral and Maxillofacial Reconstructive Surgery, School of Dentistry, Iwate Medical UniversityDivision of Oral and Maxillofacial Surgery, Department of Oral and Maxillofacial Reconstructive Surgery, School of Dentistry, Iwate Medical UniversityHead and Neck Cancer Center, Iwate Medical UniversityHead and Neck Cancer Center, Iwate Medical UniversityDivision of Oral and Maxillofacial Surgery, Department of Oral and Maxillofacial Reconstructive Surgery, School of Dentistry, Iwate Medical UniversityDivision of Clinical Pathology, Department of Oral and Maxillofacial Reconstructive Surgery, School of Dentistry, Iwate Medical UniversityHead and Neck Cancer Center, Iwate Medical UniversityDivision of Oral and Maxillofacial Surgery, Department of Oral and Maxillofacial Reconstructive Surgery, School of Dentistry, Iwate Medical UniversityAbstract Background 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. Case presentation 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. Conclusions 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.http://link.springer.com/article/10.1186/s12957-020-01999-1Epithelioid angiosarcomaMandibular gingivaVascular tumor
spellingShingle Yuko Komatsu
Ikuya Miyamoto
Yu Ohashi
Katsunori Katagiri
Daisuke Saito
Mizuki Obara
Yasunori Takeda
Kiyoto Shiga
Hiroyuki Yamada
Primary epithelioid angiosarcoma originating from the mandibular gingiva: a case report of an extremely rare oral lesion
World Journal of Surgical Oncology
Epithelioid angiosarcoma
Mandibular gingiva
Vascular tumor
title Primary epithelioid angiosarcoma originating from the mandibular gingiva: a case report of an extremely rare oral lesion
title_full Primary epithelioid angiosarcoma originating from the mandibular gingiva: a case report of an extremely rare oral lesion
title_fullStr Primary epithelioid angiosarcoma originating from the mandibular gingiva: a case report of an extremely rare oral lesion
title_full_unstemmed Primary epithelioid angiosarcoma originating from the mandibular gingiva: a case report of an extremely rare oral lesion
title_short Primary epithelioid angiosarcoma originating from the mandibular gingiva: a case report of an extremely rare oral lesion
title_sort primary epithelioid angiosarcoma originating from the mandibular gingiva a case report of an extremely rare oral lesion
topic Epithelioid angiosarcoma
Mandibular gingiva
Vascular tumor
url http://link.springer.com/article/10.1186/s12957-020-01999-1
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