Kamatani Takaaki, Katada Ryogo, Watanabe Masataka, Shirota Tatsuo
Department of Oral and Maxillofacial Surgery, Showa University School of Dentistry, Tokyo 145-8515, Japan.
Heliyon. 2024 Nov 21;10(23):e40535. doi: 10.1016/j.heliyon.2024.e40535. eCollection 2024 Dec 15.
Pulmonary metastasis of ameloblastoma is a rare associated with the histopathologically plexiform types of ameloblastoma. In this report, we present an exceptionally rare case of pulmonary metastatic ameloblastoma without local recurrence, emerging 12 years post-initial resection. A female patient, initially diagnosed with mandibular desmoplastic ameloblastoma, revealed masses in both lung fields of the lung on chest radiography, while chest computed tomography revealed more than 10 nodules in both lungs. Histopathological analysis of a lung biopsy specimen revealed a nest-like structure of tumor cells surrounded by highly columnar cells, exhibiting a palisade-like arrangement, and squamous metaplasia. Subsequently, the diagnosis of pulmonary metastatic ameloblastoma was confirmed through immunohistochemical examination. Despite clinical trial treatment with molecular targeted therapy for three years, the lung disease remained clinically stable. This case highlights the need for oral surgeons to recognize that pulmonary metastasis of desmoplastic ameloblastoma can manifest as a late recurrence, extending beyond a decade after initial curative surgery.
成釉细胞瘤的肺转移罕见，与组织病理学上的丛状型成釉细胞瘤相关。在本报告中，我们呈现了一例极为罕见的肺转移性成釉细胞瘤病例，无局部复发，在初次切除术后12年出现。一名女性患者最初被诊断为下颌骨促结缔组织增生性成釉细胞瘤，胸部X线检查显示双肺野有肿块，而胸部计算机断层扫描显示双肺有10多个结节。肺活检标本的组织病理学分析显示肿瘤细胞呈巢状结构，周围为高柱状细胞，呈栅栏状排列，并有鳞状化生。随后，通过免疫组织化学检查确诊为肺转移性成釉细胞瘤。尽管进行了三年的分子靶向治疗临床试验，肺部疾病在临床上仍保持稳定。该病例强调口腔外科医生需要认识到促结缔组织增生性成釉细胞瘤的肺转移可表现为晚期复发，在初次根治性手术后十多年出现。
