Abstract

BackgroundThe rise in incidence and mortality of gastrointestinal mixed adenoneuroendocrine carcinoma (MANEC) has not been well focused. The aim of our study was to examine epidemiological trends in incidence and incidence-based (IB) mortality of gastrointestinal MANEC at a population level.MethodsThe incidence and IB mortality of gastrointestinal MANEC as well as data on affected patients from 2000 to 2016 were obtained from the Surveillance, Epidemiology, and End Results database. Trends in incidence and IB mortality were assessed using Joinpoint regression. The Kaplan–Meier method and log-rank test were used for survival analysis. Cox proportional hazards regression was used to identify independent predictors of mortality.Results581 patients diagnosed with gastrointestinal MANEC were enrolled. Gastrointestinal MANEC incidence was 0.23 cases per 1,000,000 individuals in 2000 and 1.16 cases per 1,000,000 individuals in 2016, with an annual percent change (APC) of 8.0% (95% CI 5.7–10.3%, P < 0.05). IB mortality also showed a sustained increase (APC 12.9%, 95% CI 9.0–16.8%, P < 0.05). In Cox regression analysis, age at diagnosis, tumor grade and stage, lymph node metastasis, surgery, and tumor size were independently associated with mortality. Median survival was 75 months (95% CI 60–128 months). Median survival of appendiceal MANEC was significantly longer than that of cecal MANEC (115 vs. 31 months; P < 0.001).ConclusionsWe found a sustained and rapid increase both in incidence and IB mortality of gastrointestinal MANEC, manifesting that there has been no significant improvement in patient outcomes, nor progress in prevention and treatment. Additional resources should be devoted to gastrointestinal MANEC research.

Highlights

  • The rise in incidence and mortality of gastrointestinal mixed adenoneuroendocrine carcinoma (MANEC) has not been well focused

  • The annual percent change (APC) was 2.9%

  • Gastrointestinal MANEC showed a sustained increase in IB mortality over the study period (Fig. 2d), with an APC of 12.9%

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Summary

Introduction

The rise in incidence and mortality of gastrointestinal mixed adenoneuroendocrine carcinoma (MANEC) has not been well focused. Mixed adenoneuroendocrine carcinoma (MANEC) is a rare subtype of neuroendocrine neoplasm consisting of both adenocarcinomatous and neuroendocrine cells. Cordier first described gastrointestinal tumors containing epithelial and neuroendocrine components in 1924, and in the years since, many different terms have been used to describe this hybrid tumor [2]. These include composite glandular-neuroendocrine mixed tumor, mucin-producing carcinoid, composite carcinoidadenocarcinoma, collision tumor, adenocarcinoma exgoblet cell carcinoid and other names [3,4,5,6,7]. This phenomenon led to a lack of awareness of this disease, which eventually related to little attention

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