Mixed adenoneuroendocrine carcinoma (MANEC) of the lower gastrointestinal tract: A systematic review with Bayesian hierarchical survival analysis

Published on May 14, 2021in Ejso4.424
· DOI :10.1016/J.EJSO.2021.05.021
Emma V. Carrington12
Estimated H-index: 12
(ICL: Imperial College London),
M. P. Romano10
Estimated H-index: 10
(UNIPD: University of Padua)
Abstract Background Mixed adeno-neuroendocrine carcinomas (MANEC) are a subgroup of mixed neuroendocrine non-neuroendocrine neoplasms (MiNEN) described as mixed neoplasms containing dual neuroendocrine and non-neuroendocrine components. The aim of this study was to appraise the prevalence of MANEC in the lower gastrointestinal (GI) tract and provide reliable estimates of survival. Method A systematic review was undertaken in accordance with PRISMA guidelines using PubMed, Embase, Cochrane Library of Systematic Review, Web of Science, and Scopus databases, and a Bayesian hierarchical survival pooled analysis was performed. Results Of 182 unique records identified, 71 studies reporting on 752 patients met the inclusion criteria. Mean age was 64.2 ± 13.6, with a male-to-female ratio of 1.25. Overall, 60.3% of MANEC were located in the appendix, 29.3% in the colon, and 10.4% in the anorectum. More than a quarter (29%) of patients had stage IV disease at diagnosis, with higher prevalence in appendiceal than colonic and anorectal primaries. More than 80% had a high-grade (G3) endocrine component. Of the 152 patients followed up for a median of 20 months (interquartile range limits, 16.5–32), median overall survival was 12.3 months (95% credible interval [95%CrI], 11.3–13.7), with a 1.12 [95%CrI, 0.67–1.83] age-adjusted hazard ratio between metastatic and non-metastatic MANEC. Stage IV disease at diagnosis was more prognostically unfavorable in cases of colonic compared to anorectal origin. Conclusion MANEC is a clinically aggressive pathological entity. The results of this study provide new insights for the understanding of tumor location within the lower GI tract and its prognosis in terms of overall survival.
#1Hideaki Kinugasa (Okayama University)H-Index: 14
#2Sakiko Hiraoka (Okayama University)H-Index: 19
Last. Hiroyuki Okada (Okayama University)H-Index: 63
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#1Jennifer E. Hrabe (UIHC: University of Iowa Hospitals and Clinics)H-Index: 8
Neuroendocrine neoplasms of the colon and rectum are rare, although surgeons are likely to encounter appendiceal neuroendocrine tumors while caring for patients with appendicitis. Surgery remains the primary treatment, provided disease is resectable, although for small rectal lesions endoscopic resection is often sufficient. Metastastic disease has a variety of treatment options. Poorly differentiated neuroendocrine carcinomas continue to have a poor prognosis.
: Twenty-six Krukenberg tumors (16 lower gastrointestinal, 4 upper gastrointestinal, and 6 of unknown origin) and their primaries when known were stained with CDX2, SATB2, GATA3, TTF1, and PAX8 using a tissue microarray containing predominantly or exclusively signet ring cells. The most common primary was appendiceal mixed adenoneuroendocrine carcinoma. CDX2 and SATB2 were positive in all known lower gastrointestinal primary tumors and negative in nearly all known upper gastrointestinal primary ...
#1Samuel L. Brilleman (Monash University)H-Index: 9
#2Eren Metin Elçi (Bayer)H-Index: 5
Last. Rory Wolfe (Monash University)H-Index: 82
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Survival data is encountered in a range of disciplines, most notably health and medical research. Although Bayesian approaches to the analysis of survival data can provide a number of benefits, they are less widely used than classical (e.g. likelihood-based) approaches. This may be in part due to a relative absence of user-friendly implementations of Bayesian survival models. In this article we describe how the rstanarm R package can be used to fit a wide range of Bayesian survival models. The r...
#1Yuji Kanazawa (Kyoto University)H-Index: 8
#2Masahiro Kikuchi (Kyoto University)H-Index: 8
Last. Shogo ShinoharaH-Index: 16
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Patients with mixed neuroendocrine-nonneuroendocrine neoplasms (MiNENs) of the colon have poor prognosis. Herein, we report a patient with MiNEN of the colon with metastases to the liver and the thyroid gland, with long-term survival. A 45-year-old man presented with anterior neck swelling. Histopathological examination of the thyroid tumor revealed neuroendocrine carcinoma (NEC), suggesting that a primary NEC in another organ had metastasized to the thyroid gland. Computed tomography to identif...
#1Federica GrilloH-Index: 24
#2Luca ValleH-Index: 5
Last. Luca MastracciH-Index: 32
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#1Handan TokmakH-Index: 2
#2Oktar AsogluH-Index: 16
Last. Burak KozaH-Index: 1
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#1Melissa FrizzieroH-Index: 11
Last. Mairéad G McNamara (University of Manchester)H-Index: 27
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Mixed neuroendocrine non-neuroendocrine neoplasms (MiNENs) represent a rare diagnosis of the gastro-entero-pancreatic tract. Evidence from the current literature regarding their epidemiology, biology, and management is of variable quality and conflicting. Based on available data, the MiNEN has an aggressive biological behaviour, mostly driven by its (often high-grade) neuroendocrine component, and a dismal prognosis. In most cases, the non-neuroendocrine component is of adenocarcinoma histology....
#1Li Xian Lim (Wollongong Hospital)H-Index: 1
#2Marie Shella De Robles (Wollongong Hospital)H-Index: 1
Last. Kimberly Anne Hart (Wollongong Hospital)H-Index: 1
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Abstract Introduction Mixed adeno-neuroendocrine carcinoma (MANEC) is a rare disease, and much of the available literature to date has consisted of case reports. A recent systematic review revealed heterogeneity in the data as not all reports documented treatment regimens and course of disease. The recent 2019 WHO update on neuroendocrine carcinoma nomenclature adds to the pre-existing classification system based on biologic activity, to better represent the spectrum of neuroendocrine non-neuroe...
#1Audrius Dulskas (Vilnius University)H-Index: 13
#2Algirdas PilvelisH-Index: 1
Abstract Introduction Mixed adeno-neuroendocrine carcinomas (MANEC) are rare tumors of gastrointestinal tract mostly found in stomach. There are very few case reports and case series with location of colon and rectum. We aimed to report our results in the treatment of colorectal MANEC. Methods We performed a prospective analysis of patients with MANEC diagnosis from December 2011 to March 2019 at National Cancer Institute, Lithuania. The demographic information, tumor details, immunohistochemica...
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