Primary intraosseous adenoid cystic carcinoma of the mandible: a comprehensive review and analysis of four new cases with emphasis on morphologic, immunophenotypic, and molecular characteristics

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• 作者：Jing Han ; DDS^a ; Ting Gu ; DDS^b ; Xi Yang ; DDS^a ; Longwei Hu ; DDS^a ; Ronghui Xia ; DDS ; PhD^b ; Zhen Tian ; DDS ; MDS ; PhD^b ; Jiang Li ; DDS ; MDS ; PhD^b ; Ling Zhu ; DDS ; PhD^c ; Liqun Xu ; DDS ; MDS ; PhD^a ; Chunye Zhang ; DDS ; MDS ; PhD^b ; ^{yezi1806@126.com}
• 刊名：Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology
• 出版年：2017
• 出版时间：March 2017
• 年：2017
• 卷：123
• 期：3
• 页码：365-373
• 全文大小：4400 K
• 卷排序：123

文摘

To comprehensively review the clinical manifestations, imaging, diagnosis, treatment, and pathologic features of primary intraosseous adenoid cystic carcinoma (PIACC) of the mandible and analyze PIACC histopathology and molecular features in four cases.Study DesignWe reviewed the literature and retrospectively studied four cases of PIACC.ResultsThe clinical and imaging findings of PIACC are similar to other malignant or borderline-malignant mandible tumors. The four cases of PIACC included three males and one female (aged between 41 and 58 years). The histopathologic features of the tumors resembled those of ACC. We observed abundant osteoclasts resorbing bone at the leading edge of the tumors characterized by solid structure histology but not by the cribriform subtype. Additionally, all four cases showed abnormalities in the MYB gene and high expression of MYB protein. All patients survived for the duration of follow-up, and two patients had distant metastases (followed up for 3 to 36 months).ConclusionsPIACC is extremely rare and is often clinically misdiagnosed. Different histologic subtypes could show different mechanisms of invasion of the mandible. MYB gene and protein expression abnormalities can be used as indicators for the precise diagnosis of PIACC.