Anatomic Pathology Unit, Department of Molecular Medicine, University of Pavia, Via Carlo Forlanini 16 - 27100, Pavia, Italy.
Anatomic Pathology, Fondazione IRCCS San Matteo Hospital, Pavia, Italy.
Endocr Pathol. 2022 Jun;33(2):274-288. doi: 10.1007/s12022-022-09720-6. Epub 2022 May 13.
Neuroendocrine neoplasms (NENs) of the major and minor ampulla are rare diseases with clinico-pathologic features distinct from non-ampullary-duodenal NENs. However, they have been often combined and the knowledge on prognostic factors specific to ampullary NENs (Amp-NENs) is limited. The aim of this study was to identify factors associated with metastatic potential and patient prognosis in Amp-NENs. We clinically and histologically investigated an international series of 119 Amp-NENs, comprising 93 ampullary neuroendocrine tumors (Amp-NETs) and 26 neuroendocrine carcinomas (Amp-NECs). Somatostatin-producing tubulo-acinar NET represented the predominant Amp-NET histologic subtype (58 cases, 62%, 12 associated with type 1 neurofibromatosis). Compared to Amp-NETs, Amp-NECs arose in significantly older patients and showed a larger tumor size, a more frequent small vessel invasion, a deeper level of invasion and a higher rate of distant metastasis, and, importantly, a tremendously worse disease-specific patient survival. In Amp-NETs, the WHO grade proved to be a strong predictor of disease-specific survival (hazard ratio: 12.61, p < 0.001 for G2 vs G1), as well as patient age at diagnosis > 60 years, small vessel invasion, pancreatic invasion, and distant metastasis at diagnosis. Although nodal metastatic disease was not associated with survival by itself, patients with > 3 metastatic lymph nodes showed a worse outcome in comparison with the remaining Amp-NET cases with lymphadenectomy. Tumor epicenter in the major ampulla, small vessel invasion, and tumor size > 16 mm were independent predictors of nodal metastases in Amp-NETs. In conclusion, we identified prognostic factors, which may eventually help guide treatment decisions in Amp-NENs.
神经内分泌肿瘤(NENs)发生于主、副乳头者较为罕见,其临床病理特征与非十二指肠乳头来源的 NENs 不同。然而,这些肿瘤常被合并研究,且目前有关主、副乳头来源的神经内分泌肿瘤(Amp-NENs)特有预后因素的知识有限。本研究旨在明确与 Amp-NENs 转移潜能和患者预后相关的因素。我们对包含 93 例主乳头神经内分泌肿瘤(Amp-NET)和 26 例神经内分泌癌(Amp-NEC)在内的国际系列 119 例 Amp-NEN 进行了临床和组织学研究。由管状-腺泡结构组成的生长抑素阳性 NET 是主要的 Amp-NET 组织学亚型(58 例,62%,其中 12 例与 1 型神经纤维瘤病相关)。与 Amp-NETs 相比,Amp-NECs 发生于年龄更大的患者,且肿瘤体积更大、更常发生小血管浸润、浸润更深、远处转移率更高,重要的是,其疾病特异性患者生存率显著更差。在 Amp-NETs 中,WHO 分级被证实是疾病特异性生存的强有力预测因子(G2 与 G1 相比,危险比:12.61,p<0.001),此外,诊断时患者年龄>60 岁、小血管浸润、胰腺浸润和远处转移也是重要的预后因素。虽然淋巴结转移本身与生存无关,但与淋巴结清扫的其余 Amp-NET 病例相比,存在>3 个转移淋巴结的患者预后更差。主乳头肿瘤中心、小血管浸润和肿瘤直径>16mm 是 Amp-NETs 淋巴结转移的独立预测因子。总之,我们确定了一些预后因素,这些因素可能最终有助于指导 Amp-NENs 的治疗决策。