Department of Pathology, Cumming School of Medicine and Alberta Precision Laboratories, University of Calgary, Calgary, Alberta, T2N 2T9, Canada.
Department of Pathology, Cumming School of Medicine and Alberta Precision Laboratories, University of Calgary, Calgary, Alberta, T2N 2T9, Canada.
Hum Pathol. 2022 Apr;122:40-49. doi: 10.1016/j.humpath.2022.01.008. Epub 2022 Feb 1.
Low-grade appendiceal mucinous neoplasms (LAMNs) can occur concurrently with appendiceal sessile serrated lesions (SSLs). To interrogate relatedness, we performed multigene and immunohistochemical characterizations of paired and unpaired SSLs and LAMNs. We evaluated 62 serrated lesions from 50 appendectomy specimens for hotspot mutations in BRAF, KRAS, and GNAS genes. Cases were subdivided into 3 groups: 20 unpaired SSLs, 18 unpaired LAMNs, and 12 with an SSL and concurrent LAMN. β-catenin and Annexin A10 immunostaining were performed on the SSL and LAMN components in the 12 paired cases, and 14 colonic SSLs served as controls. There was no significant difference in KRAS hotspot mutation rates in appendiceal SSLs (17/26; 65.4%) and LAMNs (16/30; 53.3%) (p = 0.42). BRAF V600E was identified in a single case (1/50; 2.0%) of SSL and concurrent LAMN (p = 1.0). Mutations in GNAS were more common in LAMNs (6/30; 20.0%) than in SSLs (1/31; 3.2%) (p = 0.05). The molecular genotypes between paired SSLs and LAMNs were concordant in most cases (10/12; 83.3%). Annexin A10 immunostaining was significantly greater in colonic SSLs (14/14; 100%) than in appendiceal SSLs (1/12; 8.3%) (p < 0.0001). β-catenin immunostaining was significantly increased in LAMNs (10/12; 83.3%) compared with their paired appendiceal SSLs (2/12; 16.7%) (p = 0.003). Overall, appendiceal SSLs are predominantly driven by KRAS mutations and are not characterized by Annexin A10 immunostaining. Our data suggest that at least a subset of LAMNs may arise from a precursor SSL in which GNAS mutations and/or upregulation of the WNT signaling pathway are likely key events modulating this progression.
低级别阑尾黏液性肿瘤 (LAMN) 可与阑尾无蒂锯齿状病变 (SSL) 同时发生。为了探究两者之间的关系,我们对配对和非配对 SSL 和 LAMN 进行了多基因和免疫组织化学特征分析。我们评估了 50 个阑尾切除术标本中的 62 个锯齿状病变,以检测 BRAF、KRAS 和 GNAS 基因中的热点突变。将病例分为 3 组:20 个非配对 SSL、18 个非配对 LAMN 和 12 个有 SSL 和同时存在的 LAMN。对 12 对病例中的 SSL 和 LAMN 成分进行了 β-连环蛋白和膜联蛋白 A10 免疫染色,14 个结肠 SSL 作为对照。阑尾 SSL 的 KRAS 热点突变率(17/26;65.4%)和 LAMN(16/30;53.3%)(p=0.42)无显著差异。SSL 和同时存在的 LAMN 中仅发现 1 例 BRAF V600E(1/50;2.0%)(p=1.0)。GNAS 突变在 LAMN 中更为常见(6/30;20.0%),而在 SSL 中则较为少见(1/31;3.2%)(p=0.05)。大多数情况下,配对的 SSL 和 LAMN 之间的分子基因型是一致的(10/12;83.3%)。膜联蛋白 A10 免疫染色在结肠 SSL 中(14/14;100%)明显高于阑尾 SSL(1/12;8.3%)(p<0.0001)。与配对的阑尾 SSL 相比,LAMN 的 β-连环蛋白免疫染色明显增加(10/12;83.3%)(p=0.003)。总体而言,阑尾 SSL 主要由 KRAS 突变驱动,不具有膜联蛋白 A10 免疫染色特征。我们的数据表明,至少一部分 LAMN 可能起源于前体 SSL,其中 GNAS 突变和/或 WNT 信号通路的上调可能是调节这种进展的关键事件。