Division of Pathology, IEO, European Institute of Oncology IRCCS, Milan, Italy.
Department of Oncology and Hemato-Oncology, University of Milan, Milan, Italy.
Cancer Med. 2023 May;12(9):10725-10737. doi: 10.1002/cam4.5754. Epub 2023 Mar 14.
Mucoepidermoid carcinoma (MEC) of the breast is an extremely rare salivary gland-type tumor characterized by epidermoid, basaloid, intermediate, and/or mucinous cells arranged in solid and cystic patterns. Despite their triple-negative phenotype, breast MECs are generally considered low-risk malignancies but their biology is largely unexplored; therefore, guidelines for clinical management are lacking. Here, we sought to characterize the molecular landscape of breast MECs. Thirteen cases were histologically reviewed, characterized for tumor-infiltrating lymphocytes (TILs), and were subjected to immunohistochemistry for programmed death-ligand 1 (PD-L1, clone 22C3), EGFR, and amphiregulin (AREG). Rearrangements in MAML2 and EWSR1 were investigated by fluorescent in situ hybridization. Targeted next-generation sequencing of 161 genes was performed on eight cases. Most MECs had low histological grade (n = 10, 77%), with the presence of TILs (n = 9/12; 75%) and PD-L1 combined positive score ranging from 10 to 20 (n = 4/6; 67%). All cases showed EGFR and AREG overexpression and were fusion negative. Enrichment of genetic alterations was observed in PI3K/AKT/mTOR and cell cycle regulation pathways, while only one case harbored TP53 mutations. This is the first study providing extensive molecular data on breast MECs and the largest collection of cases available to date in the literature. Breast MECs lack TP53 mutations found in high-grade forms of triple-negative breast cancers and MAML2 or EWSR1 rearrangements pathognomonic of salivary MECs. Triple-negativity and PD-L1 positivity suggest a window of opportunity for immunotherapy in these patients. The EGFR/AREG axis activation, coupled with the mutational patterns in PI3K/AKT/mTOR and cell cycle pathways warrants caution in considering MECs as low-risk neoplasms.
乳腺黏液表皮样癌(MEC)是一种极为罕见的唾液腺型肿瘤,其特征为表皮样、基底样、中间型和/或黏液细胞呈实性和囊性排列。尽管其为三阴性表型,但乳腺 MEC 通常被认为是低风险的恶性肿瘤,但它们的生物学特性在很大程度上仍未被探索;因此,缺乏临床管理指南。在这里,我们试图描绘乳腺 MEC 的分子特征。对 13 例病例进行了组织学回顾、肿瘤浸润淋巴细胞(TILs)分析,并进行了程序性死亡配体 1(PD-L1,克隆 22C3)、表皮生长因子受体(EGFR)和 Amphiregulin(AREG)的免疫组织化学染色。采用荧光原位杂交检测 MAML2 和 EWSR1 的重排。对 8 例病例进行了 161 个基因的靶向下一代测序。大多数 MEC 组织学分级较低(n=10,77%),其中 9/12 例(75%)存在 TILs,PD-L1 联合阳性评分范围为 10-20(n=4/6;67%)。所有病例均显示 EGFR 和 AREG 过表达,且融合阴性。PI3K/AKT/mTOR 和细胞周期调控通路中观察到基因改变的富集,而仅有 1 例存在 TP53 突变。这是首次提供乳腺 MEC 广泛分子数据的研究,也是迄今为止文献中最大的病例集合。乳腺 MEC 缺乏在高级别三阴性乳腺癌中发现的 TP53 突变,也缺乏唾液腺 MEC 特有的 MAML2 或 EWSR1 重排。三阴性和 PD-L1 阳性提示这些患者有免疫治疗的机会。EGFR/AREG 轴的激活,加上 PI3K/AKT/mTOR 和细胞周期通路的突变模式,需要谨慎对待将 MEC 视为低风险肿瘤。