Recurrent clear cell odontogenic carcinoma initially diagnosed as ameloblastoma: A case report and review of literature

Q3 Dentistry
Thijs Bauwens , Frederik Piccart , Matthias Ureel , David Creytens , Wouter Bauters , Fréderic Duprez , Renaat Coopman
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引用次数: 0

Abstract

Clear cell odontogenic carcinoma (CCOC) is a rare, malignant tumor primarily affecting the mandible in middle-aged women, known to be highly recurrent post-surgery. A 56-year-old female, with an initial diagnosis of ameloblastoma based on radiological findings, had her diagnosis revised to CCOC at Ghent University Hospital after histopathological analysis and EWSR1::CREB1 gene fusion confirmation. Despite undergoing a Brown-Shah IID maxillectomy, radical neck dissection, and fibula flap reconstruction, she faced a recurrence within a year, complicated by an Aspergillus fumigatus infection requiring antifungal treatments and debridements. Diagnosis relies on immunohistochemical markers p40, p63, CK5, SMA, Calponin, and S-100. Additionally, over 80 % of CCOC cases show EWSR1 gene rearrangements. Treatment includes surgical resection and fibula free flap reconstruction. Adjuvant radiotherapy may be beneficial in cases with compromised margins or positive lymph nodes. Prophylactic antifungal treatment is recommended for at-risk patients. The complex nature of CCOC requires thorough follow-up and potentially adjuvant therapies.
最初诊断为成釉细胞瘤的复发性透明细胞性牙源性癌1例报告及文献复习
透明细胞牙源性癌(CCOC)是一种罕见的恶性肿瘤,主要影响中年妇女的下颌骨,已知是术后高度复发。一名56岁女性,最初诊断为成膜细胞瘤,基于影像学表现,在根特大学医院进行组织病理学分析和EWSR1::CREB1基因融合确认后,诊断修改为CCOC。尽管接受了Brown-Shah ii型上颌切除术、根治性颈部清扫术和腓骨瓣重建,她在一年内面临复发,并伴有烟曲霉感染,需要抗真菌治疗和清创。诊断依赖免疫组织化学标志物p40、p63、CK5、SMA、钙钙蛋白和S-100。此外,超过80%的CCOC病例显示EWSR1基因重排。治疗包括手术切除和腓骨游离皮瓣重建。对于边缘受损或淋巴结阳性的病例,辅助放疗可能是有益的。建议高危患者进行预防性抗真菌治疗。CCOC的复杂性需要彻底的随访和潜在的辅助治疗。
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来源期刊
Oral and Maxillofacial Surgery Cases
Oral and Maxillofacial Surgery Cases Medicine-Otorhinolaryngology
CiteScore
0.60
自引率
0.00%
发文量
43
审稿时长
69 days
期刊介绍: Oral and Maxillofacial Surgery Cases is a surgical journal dedicated to publishing case reports and case series only which must be original, educational, rare conditions or findings, or clinically interesting to an international audience of surgeons and clinicians. Case series can be prospective or retrospective and examine the outcomes of management or mechanisms in more than one patient. Case reports may include new or modified methodology and treatment, uncommon findings, and mechanisms. All case reports and case series will be peer reviewed for acceptance for publication in the Journal.
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