Prognostic factors and survival in MEN1 patients with gastrinomas: Results from the DutchMEN study group (DMSG).


Journal

Journal of surgical oncology
ISSN: 1096-9098
Titre abrégé: J Surg Oncol
Pays: United States
ID NLM: 0222643

Informations de publication

Date de publication:
Nov 2019
Historique:
received: 23 07 2019
accepted: 28 07 2019
pubmed: 12 8 2019
medline: 24 10 2019
entrez: 12 8 2019
Statut: ppublish

Résumé

Gastrinomas are the most prevalent functioning neuroendocrine tumors (NET) in multiple endocrine neoplasia type 1 (MEN1). Guidelines suggest medical therapy in most patients, but surgery may be considered in a subgroup. Currently, factors to guide management are necessary. This population-based cohort study assessed prognostic factors of survival in patients with MEN1-related gastrinomas. Patients with MEN1 having gastrinomas were identified in the Dutch MEN1 database from 1990 to 2014 based on fasting serum gastrin (FSG) levels and/or pathology. Predictors of overall survival were assessed using Cox regression. Sixty-three patients with gastrinoma (16% of the MEN1 population) were identified. Five- and 10-year overall survival rates were 83% and 65%, respectively. Prognostic factors associated with overall survival were initial FSG levels ≥20x upper limit of normal (ULN) (hazard ratio [HR], 6.2 [95% confidence interval, 1.7-23.0]), pancreatic NET ≥2 cm (HR 4.5; [1.5-13.1]), synchronous liver metastases (HR 8.9; [2.1-36.7]), gastroduodenoscopy suspicious for gastric NETs (HR 12.7; [1.4-115.6]), and multiple concurrent NETs (HR 5.9; [1.2-27.7]). Life expectancy of patients with MEN1 gastrinoma is reduced. FSG levels and pancreatic NETs ≥2 cm are prognostic factors. FSG levels might guide surveillance intensity, step-up to additional diagnostics, or provide arguments in selecting patients who might benefit from surgery.

Sections du résumé

BACKGROUND AND OBJECTIVES OBJECTIVE
Gastrinomas are the most prevalent functioning neuroendocrine tumors (NET) in multiple endocrine neoplasia type 1 (MEN1). Guidelines suggest medical therapy in most patients, but surgery may be considered in a subgroup. Currently, factors to guide management are necessary. This population-based cohort study assessed prognostic factors of survival in patients with MEN1-related gastrinomas.
METHODS METHODS
Patients with MEN1 having gastrinomas were identified in the Dutch MEN1 database from 1990 to 2014 based on fasting serum gastrin (FSG) levels and/or pathology. Predictors of overall survival were assessed using Cox regression.
RESULTS RESULTS
Sixty-three patients with gastrinoma (16% of the MEN1 population) were identified. Five- and 10-year overall survival rates were 83% and 65%, respectively. Prognostic factors associated with overall survival were initial FSG levels ≥20x upper limit of normal (ULN) (hazard ratio [HR], 6.2 [95% confidence interval, 1.7-23.0]), pancreatic NET ≥2 cm (HR 4.5; [1.5-13.1]), synchronous liver metastases (HR 8.9; [2.1-36.7]), gastroduodenoscopy suspicious for gastric NETs (HR 12.7; [1.4-115.6]), and multiple concurrent NETs (HR 5.9; [1.2-27.7]).
CONCLUSION CONCLUSIONS
Life expectancy of patients with MEN1 gastrinoma is reduced. FSG levels and pancreatic NETs ≥2 cm are prognostic factors. FSG levels might guide surveillance intensity, step-up to additional diagnostics, or provide arguments in selecting patients who might benefit from surgery.

Identifiants

pubmed: 31401809
doi: 10.1002/jso.25667
pmc: PMC6852496
doi:

Substances chimiques

MEN1 protein, human 0
Proto-Oncogene Proteins 0

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

966-975

Subventions

Organisme : Ipsen Pharmaceutical

Informations de copyright

© 2019 The Authors. Journal of Surgical Oncology published by Wiley Periodicals, Inc.

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Auteurs

Dirk-Jan van Beek (DJ)

Department of Endocrine Surgical Oncology, University Medical Center Utrecht, Utrecht, The Netherlands.

Sjoerd Nell (S)

Department of Endocrine Surgical Oncology, University Medical Center Utrecht, Utrecht, The Netherlands.

Carolina R C Pieterman (CRC)

Department of Endocrine Oncology, University Medical Center Utrecht, Utrecht, The Netherlands.

Wouter W de Herder (WW)

Department of Internal Medicine, Erasmus Medical Center, Rotterdam, The Netherlands.

Annenienke C van de Ven (AC)

Department of Endocrinology, Radboud University Medical Center, Nijmegen, The Netherlands.

Olaf M Dekkers (OM)

Departments of Endocrinology and Metabolism and Clinical Epidemiology, Leiden University Medical Center, Leiden, The Netherlands.

Anouk N van der Horst-Schrivers (AN)

Department of Endocrinology, University Medical Center Groningen, University of Groningen, Groningen, The Netherlands.

Madeleine L Drent (ML)

Department of Internal Medicine, Section of Endocrinology, Amsterdam UMC location, VU University Medical Center, Amsterdam, The Netherlands.

Peter H Bisschop (PH)

Department of Endocrinology and Metabolism, Amsterdam UMC location Academic Medical Center, Amsterdam, The Netherlands.

Bas Havekes (B)

Department of Internal Medicine, Division of Endocrinology, Maastricht University Medical Center, Maastricht, The Netherlands.

Inne H M Borel Rinkes (IHM)

Department of Endocrine Surgical Oncology, University Medical Center Utrecht, Utrecht, The Netherlands.

Menno R Vriens (MR)

Department of Endocrine Surgical Oncology, University Medical Center Utrecht, Utrecht, The Netherlands.

Gerlof D Valk (GD)

Department of Endocrine Oncology, University Medical Center Utrecht, Utrecht, The Netherlands.

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Classifications MeSH