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Abstract：
BACKGROUND: Odontogenic carcinoma with dentinoid (OCD) is a rare and controversial entity, which has not yet been included in the current World Health Organization classification of odontogenic lesions. Owing to the small number of reported cases, the clinicopathological characteristics, biological behavior, prognosis, and appropriate treatment strategies for OCD remain to be defined. Herein, we present an additional case of OCD with a focus on the differential diagnosis and review of the pertinent literature, in order to enable better recognition by oral clinicians and pathologists and further characterization of this entity.
METHODS: This paper reports a case of OCD in the posterior mandible of a 22-year-old female. Radiography showed a well-defined unilocular radiolucency with radiopaque materials. The intraoperative frozen section pathology gave a non-committed diagnosis of odontogenic neoplasm with uncertain malignant potential. Then a partial mandibulectomy with free iliac crest bone graft and titanium implants was performed. Microscopically, the tumor consisted of sheets, islands, and cords of round to polygonal epithelial cells associated with an abundant dentinoid matrix. Immunohistochemically, the tumor cells were diffusely positive for CK19, p63, and β-catenin (cytoplasmic and nuclear). No rearrangement of the EWSR1 gene was detected. The final diagnosis was OCD. There has been no evidence of recurrence or metastasis for 58 months after surgery. We also provide a literature review of OCD cases, including one case previously reported as ghost cell odontogenic carcinoma from our hospital.
CONCLUSIONS: OCD is a locally aggressive low grade malignancy without apparent metastatic potential. Wide surgical excision with clear margins and long-term period follow-up to identify any possible recurrence or metastases are recommended. Histopathological examination is essential to conclude the diagnosis. Special care must be taken to distinguish OCD from ghost cell odontogenic carcinoma and clear cell odontogenic carcinoma, as misdiagnosis might lead to unnecessary overtreatment. Study of additional cases is required to further characterize the clinicopathological features and clarify the nosologic status and biological behavior of this tumor.
METHODS: This paper reports a case of OCD in the posterior mandible of a 22-year-old female. Radiography showed a well-defined unilocular radiolucency with radiopaque materials. The intraoperative frozen section pathology gave a non-committed diagnosis of odontogenic neoplasm with uncertain malignant potential. Then a partial mandibulectomy with free iliac crest bone graft and titanium implants was performed. Microscopically, the tumor consisted of sheets, islands, and cords of round to polygonal epithelial cells associated with an abundant dentinoid matrix. Immunohistochemically, the tumor cells were diffusely positive for CK19, p63, and β-catenin (cytoplasmic and nuclear). No rearrangement of the EWSR1 gene was detected. The final diagnosis was OCD. There has been no evidence of recurrence or metastasis for 58 months after surgery. We also provide a literature review of OCD cases, including one case previously reported as ghost cell odontogenic carcinoma from our hospital.
CONCLUSIONS: OCD is a locally aggressive low grade malignancy without apparent metastatic potential. Wide surgical excision with clear margins and long-term period follow-up to identify any possible recurrence or metastases are recommended. Histopathological examination is essential to conclude the diagnosis. Special care must be taken to distinguish OCD from ghost cell odontogenic carcinoma and clear cell odontogenic carcinoma, as misdiagnosis might lead to unnecessary overtreatment. Study of additional cases is required to further characterize the clinicopathological features and clarify the nosologic status and biological behavior of this tumor.
摘要：
背景：牙源性牙样癌（OCD）是一种罕见且有争议的实体，目前尚未纳入世界卫生组织牙源性病变分类。由于报告的案件数量很少，临床病理特征，生物学行为,预后,强迫症的适当治疗策略仍有待确定。在这里，我们介绍了另一例强迫症病例，重点是鉴别诊断和相关文献的回顾，以便使口腔临床医生和病理学家更好地识别并进一步表征该实体。
方法：本文报告1例22岁女性下颌骨后部强迫症。射线照相术显示出不透射线的材料具有明确的单眼射线可透性。术中冰冻切片病理诊断为牙源性肿瘤，恶性潜能不确定。然后进行部分下颌骨切除术，并进行游离骨移植和钛植入物。微观上,肿瘤由床单组成，岛屿,以及与丰富的牙质基质相关的圆形至多边形上皮细胞的索。免疫组织化学，肿瘤细胞对CK19，p63和β-catenin（细胞质和细胞核）呈弥漫性阳性。未检测到EWSR1基因的重排。最终诊断为强迫症。术后58个月没有复发或转移的证据。我们还提供了强迫症病例的文献综述，包括1例以前从我们医院报告的鬼细胞牙源性癌。
结论：强迫症是一种局部侵袭性低级别恶性肿瘤，无明显转移潜力。建议广泛的手术切除，边缘清晰，长期随访以确定任何可能的复发或转移。组织病理学检查对于确定诊断至关重要。必须特别注意将OCD与鬼细胞牙源性癌和透明细胞牙源性癌区分开来，因为误诊可能导致不必要的过度治疗。需要对其他病例进行研究，以进一步表征临床病理特征，并阐明该肿瘤的疾病状态和生物学行为。
方法：本文报告1例22岁女性下颌骨后部强迫症。射线照相术显示出不透射线的材料具有明确的单眼射线可透性。术中冰冻切片病理诊断为牙源性肿瘤，恶性潜能不确定。然后进行部分下颌骨切除术，并进行游离骨移植和钛植入物。微观上,肿瘤由床单组成，岛屿,以及与丰富的牙质基质相关的圆形至多边形上皮细胞的索。免疫组织化学，肿瘤细胞对CK19，p63和β-catenin（细胞质和细胞核）呈弥漫性阳性。未检测到EWSR1基因的重排。最终诊断为强迫症。术后58个月没有复发或转移的证据。我们还提供了强迫症病例的文献综述，包括1例以前从我们医院报告的鬼细胞牙源性癌。
结论：强迫症是一种局部侵袭性低级别恶性肿瘤，无明显转移潜力。建议广泛的手术切除，边缘清晰，长期随访以确定任何可能的复发或转移。组织病理学检查对于确定诊断至关重要。必须特别注意将OCD与鬼细胞牙源性癌和透明细胞牙源性癌区分开来，因为误诊可能导致不必要的过度治疗。需要对其他病例进行研究，以进一步表征临床病理特征，并阐明该肿瘤的疾病状态和生物学行为。
