Determinants of disease-specific survival in patients with and without metastatic pheochromocytoma and paraganglioma.

Adrenal Gland Neoplasms Humans Metanephrine Neoplasms, Second Primary Paraganglioma / pathology Pheochromocytoma / pathology Retrospective Studies
Metastases Methoxytyramine Paraganglioma Pheochromocytoma Predictors Survival

Journal

European journal of cancer (Oxford, England : 1990)
ISSN: 1879-0852
Titre abrégé: Eur J Cancer
Pays: England
ID NLM: 9005373

Informations de publication

Date de publication:
07 2022
Historique:
received: 30 01 2022
revised: 23 03 2022
accepted: 23 03 2022
pubmed: 3 5 2022
medline: 14 6 2022
entrez: 2 5 2022
Statut: ppublish

Résumé

Pheochromocytomas and paragangliomas (PPGLs) have a heterogeneous prognosis, the basis of which remains unclear. We, therefore, assessed disease-specific survival (DSS) and potential predictors of progressive disease in patients with PPGLs and head/neck paragangliomas (HNPGLs) according to the presence or absence of metastases. This retrospective study included 582 patients with PPGLs and 57 with HNPGLs. DSS was assessed according to age, location and size of tumours, recurrent/metastatic disease, genetics, plasma metanephrines and methoxytyramine. Among all patients with PPGLs, multivariable analysis indicated that apart from older age (HR = 5.4, CI = 2.93-10.29, P < 0.0001) and presence of metastases (HR = 4.8, CI = 2.41-9.94, P < 0.0001), shorter DSS was also associated with extra-adrenal tumour location (HR = 2.6, CI = 1.32-5.23, P = 0.0007) and higher plasma methoxytyramine (HR = 1.8, CI = 1.11-2.85, P = 0.0170) and normetanephrine (HR = 1.8, CI = 1.12-2.91, P = 0.0160). Among patients with HNPGLs, those with metastases presented with longer DSS compared to patients with metastatic PPGLs (33.4 versus 20.2 years, P < 0.0001) and only plasma methoxytyramine (HR = 13, CI = 1.35-148, P = 0.0380) was an independent predictor of DSS. For patients with metastatic PPGLs, multivariable analysis revealed that apart from older age (HR = 6.2, CI = 3.20-12.20, P < 0.0001), shorter DSS was associated with the presence of synchronous metastases (HR = 4.9, CI = 2.78-8.80, P < 0.0001), higher plasma methoxytyramine (HR = 2.4, CI = 1.44-4.14, P = 0.0010) and extensive metastatic burden (HR = 2.1, CI = 1.07-3.79, P = 0.0290). DSS among patients with PPGLs/HNPGLs relates to several presentations of the disease that may provide prognostic markers. In particular, the independent associations of higher methoxytyramine with shorter DSS in patients with HNPGLs and metastatic PPGLs suggest the utility of this biomarker to guide individualized management and follow-up strategies in affected patients.

Sections du résumé

BACKGROUND
Pheochromocytomas and paragangliomas (PPGLs) have a heterogeneous prognosis, the basis of which remains unclear. We, therefore, assessed disease-specific survival (DSS) and potential predictors of progressive disease in patients with PPGLs and head/neck paragangliomas (HNPGLs) according to the presence or absence of metastases.
METHODS
This retrospective study included 582 patients with PPGLs and 57 with HNPGLs. DSS was assessed according to age, location and size of tumours, recurrent/metastatic disease, genetics, plasma metanephrines and methoxytyramine.
RESULTS
Among all patients with PPGLs, multivariable analysis indicated that apart from older age (HR = 5.4, CI = 2.93-10.29, P < 0.0001) and presence of metastases (HR = 4.8, CI = 2.41-9.94, P < 0.0001), shorter DSS was also associated with extra-adrenal tumour location (HR = 2.6, CI = 1.32-5.23, P = 0.0007) and higher plasma methoxytyramine (HR = 1.8, CI = 1.11-2.85, P = 0.0170) and normetanephrine (HR = 1.8, CI = 1.12-2.91, P = 0.0160). Among patients with HNPGLs, those with metastases presented with longer DSS compared to patients with metastatic PPGLs (33.4 versus 20.2 years, P < 0.0001) and only plasma methoxytyramine (HR = 13, CI = 1.35-148, P = 0.0380) was an independent predictor of DSS. For patients with metastatic PPGLs, multivariable analysis revealed that apart from older age (HR = 6.2, CI = 3.20-12.20, P < 0.0001), shorter DSS was associated with the presence of synchronous metastases (HR = 4.9, CI = 2.78-8.80, P < 0.0001), higher plasma methoxytyramine (HR = 2.4, CI = 1.44-4.14, P = 0.0010) and extensive metastatic burden (HR = 2.1, CI = 1.07-3.79, P = 0.0290).
CONCLUSIONS
DSS among patients with PPGLs/HNPGLs relates to several presentations of the disease that may provide prognostic markers. In particular, the independent associations of higher methoxytyramine with shorter DSS in patients with HNPGLs and metastatic PPGLs suggest the utility of this biomarker to guide individualized management and follow-up strategies in affected patients.

Identifiants

DOI: 10.1016/j.ejca.2022.03.032 PMID: 35500459
pubmed: 35500459
pii: S0959-8049(22)00180-0
doi: 10.1016/j.ejca.2022.03.032
pii:
doi:

Substances chimiques

Metanephrine 5001-33-2

Types de publication

Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

32-41

Informations de copyright

Copyright © 2022 The Author(s). Published by Elsevier Ltd.. All rights reserved.

Déclaration de conflit d'intérêts

Conflict of Interest statement The authors declare that they have no financial relationships that could be broadly relevant to the work.

Auteurs

Christina Pamporaki (C)

Department of Medicine ΙΙI, University Hospital Carl Gustav Carus at the TU Dresden, Dresden, Germany. Electronic address: Christina.Pamporaki@uniklinikum-dresden.de.

Tamara Prodanov (T)

National Institutes of Health (NIH), Bethesda, USA.

Leah Meuter (L)

National Institutes of Health (NIH), Bethesda, USA.

Annika M A Berends (AMA)

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

Nicole Bechmann (N)

Department of Medicine ΙΙI, University Hospital Carl Gustav Carus at the TU Dresden, Dresden, Germany; Institute of Clinical Chemistry and Laboratory Medicine, University Hospital Carl Gustav Carus at the TU Dresden, Germany.

Georgiana Constantinescu (G)

Department of Medicine ΙΙI, University Hospital Carl Gustav Carus at the TU Dresden, Dresden, Germany.

Felix Beuschlein (F)

Department of Internal Medicine, University Hospital of Munich, Germany; Department of Endocrinology, Diabetology, and Clinical Nutrition, University Hospital, Zurich, Switzerland.

Hanna Remde (H)

Department of Internal Medicine, University Hospital of Würzburg, Germany.

Andrzej Januszewicz (A)

Department of Hypertension, Institute of Cardiology, Warsaw, Poland.

Michiel N Kerstens (MN)

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

Henri J L M Timmers (HJLM)

Department of Internal Medicine, Radboud University Hospital, Nijmegen, the Netherlands.

David Taïeb (D)

Department of Nuclear Medicine, La Timone University Hospital, CERIMED, Aix-Marseille University, France.

Mercedes Robledo (M)

Hereditary Endocrine Cancer Group, Human Cancer Genetics Programme, Spanish National Cancer Research Centre, Spain; Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER), Spain.

Jacques W M Lenders (JWM)

Department of Medicine ΙΙI, University Hospital Carl Gustav Carus at the TU Dresden, Dresden, Germany; Department of Internal Medicine, Radboud University Hospital, Nijmegen, the Netherlands.

Karel Pacak (K)

National Institutes of Health (NIH), Bethesda, USA.

Graeme Eisenhofer (G)

Department of Medicine ΙΙI, University Hospital Carl Gustav Carus at the TU Dresden, Dresden, Germany; Institute of Clinical Chemistry and Laboratory Medicine, University Hospital Carl Gustav Carus at the TU Dresden, Germany.

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Classifications MeSH

Recherchemedicale.com › Maladies endocriniennes › Tumeurs des glandes endocrines › Tumeurs de la surrénale › Determinants of disease-specific survival in...
Recherchemedicale.com › Eucaryotes › Animaux › Chordés › Vertébrés › Mammifères › Eutheria › Primates › Haplorhini › Catarrhini › Hominidae › Humains › Determinants of disease-specific survival in...
Recherchemedicale.com › Composés chimiques organiques › Hydrocarbures › Hydrocarbures cycliques › Hydrocarbures aromatiques › Dérivés du benzène › Phénols › Catéchols › Catécholamines › Épinéphrine › Métanéphrine › Determinants of disease-specific survival in...
Recherchemedicale.com › Tumeurs › Seconde tumeur primitive › Determinants of disease-specific survival in...
Recherchemedicale.com › Tumeurs › Tumeurs par type histologique › Tumeurs du tissu nerveux › Tumeurs neuroectodermiques › Tumeurs neuroendocrines › Paragangliome › Determinants of disease-specific survival in...
Recherchemedicale.com › Tumeurs › Tumeurs par type histologique › Tumeurs du tissu nerveux › Tumeurs neuroectodermiques › Tumeurs neuroendocrines › Paragangliome › Phéochromocytome › Determinants of disease-specific survival in...
Recherchemedicale.com › Environnement et santé publique › Santé publique › Méthodes épidémiologiques › Caractéristiques des études épidémiologiques › Études épidémiologiques › Études de cohortes › Études rétrospectives › Determinants of disease-specific survival in...