Department of Pathology, Memorial Sloan Kettering Cancer Center, New York, NY, USA.
Department of Pathology, Hackensack University Medical Center, Hackensack, NJ, USA.
Mod Pathol. 2021 Aug;34(8):1570-1587. doi: 10.1038/s41379-021-00799-6. Epub 2021 Mar 26.
Mesonephric carcinoma of the cervix is a rare tumor derived from Wolffian remnants. Mesonephric-like carcinomas of the ovary and endometrium, while morphologically similar, do not have obvious Wolffian derivation. Here, we sought to characterize the repertoire of genetic alterations in primary mesonephric and mesonephric-like carcinomas, in the distinct histologic components of mixed cases, as well as in matched primary tumors and metastases. DNA from microdissected tumor and normal tissue from mesonephric carcinomas (cervix, n = 8) and mesonephric-like carcinomas (ovarian n = 15, endometrial n = 13) were subjected to sequencing targeting 468 cancer-related genes. The histologically distinct components of four cases with mixed histology and four primary tumors and their matched metastases were microdissected and analyzed separately. Mesonephric-like carcinomas were underpinned by somatic KRAS mutations (25/28, 89%) akin to mesonephric carcinomas (8/8, 100%), but also harbored genetic alterations more frequently reported in Müllerian tumors. Mesonephric-like carcinomas that lacked KRAS mutations harbored NRAS (n = 2, ovary) or BRAF (n = 1, endometrium) hotspot mutations. PIK3CA mutations were identified in both mesonephric-like (8/28, 28%) and mesonephric carcinomas (2/8, 25%). Only mesonephric-like tumors harbored CTNNB1 hotspot (4/28, 14%) and PTEN (3/13, 23%) mutations. Copy number analysis revealed frequent gains of chromosomes 1q and 10 in both mesonephric (87% 1q; 50% chromosome 10) and mesonephric-like tumors (89% 1q; 43% chromosome 10). Chromosome 12 gains were more frequent in ovarian mesonephric-like carcinomas, and losses of chromosome 9 were more frequent in mesonephric than in mesonephric-like carcinomas (both p = 0.01, Fisher's exact test). The histologically distinct components of four mixed cases were molecularly related and shared similar patterns of genetic alterations. The progression from primary to metastatic lesions involved the acquisition of additional mutations, and/or shifts from subclonal to clonal mutations. Our findings suggest that mesonephric-like carcinomas are derived from a Müllerian substrate with differentiation along Wolffian/mesonephric lines.
宫颈苗勒管癌是一种罕见的来源于沃夫管残余物的肿瘤。卵巢和子宫内膜的苗勒管样癌虽然形态上相似,但没有明显的沃夫管来源。在这里,我们试图描述原发性苗勒管和苗勒管样癌、混合病例中不同组织学成分、以及匹配的原发性肿瘤和转移瘤中遗传改变的全貌。对 8 例宫颈苗勒管癌和 28 例卵巢苗勒管样癌、13 例子宫内膜苗勒管样癌的肿瘤和正常组织的微切割 DNA 进行了靶向 468 个癌症相关基因的测序。对 4 例混合组织学和 4 例原发性肿瘤及其匹配转移瘤的不同组织学成分进行了微切割和单独分析。苗勒管样癌存在体细胞 KRAS 突变(25/28,89%),类似于苗勒管癌(8/8,100%),但也存在更常报道的米勒管肿瘤的遗传改变。缺乏 KRAS 突变的苗勒管样癌存在 NRAS(n=2,卵巢)或 BRAF(n=1,子宫内膜)热点突变。PIK3CA 突变在苗勒管样癌(8/28,28%)和苗勒管癌(2/8,25%)中均有发现。只有苗勒管样肿瘤存在 CTNNB1 热点(4/28,14%)和 PTEN(3/13,23%)突变。拷贝数分析显示,苗勒管(87% 1q;50% 染色体 10)和苗勒管样癌(89% 1q;43% 染色体 10)均存在 1q 和 10 染色体频繁获得。卵巢苗勒管样癌中 12 号染色体获得更为频繁,而苗勒管癌中 9 号染色体丢失更为频繁(均为 p=0.01,Fisher 确切检验)。4 例混合病例的不同组织学成分在分子上是相关的,具有相似的遗传改变模式。从原发性病变到转移性病变的进展涉及到获得额外的突变,以及/或从亚克隆突变到克隆突变的转变。我们的研究结果表明,苗勒管样癌来源于米勒管基质,沿着沃夫管/苗勒管分化。