Recurrence-Free Survival Analysis in Locally Advanced Pheochromocytoma: First Appraisal

Abstract Context The behavior of locally advanced pheochromocytoma (LAP) remains unknown. Objective We characterized the population with LAP and recurrence-free survival (RFS). Methods This retrospective multicentric study was run within the ENDOCAN-COMETE network and French Group of Endocrine Tumor...

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Veröffentlicht in:The journal of clinical endocrinology and metabolism 2021-09, Vol.106 (9), p.2726-2737
Hauptverfasser: Moog, Sophie, Castinetti, Frédéric, DoCao, Christine, Amar, Laurence, Hadoux, Julien, Lussey-Lepoutre, Charlotte, Borson-Chazot, Françoise, Vezzosi, Delphine, Drui, Delphine, Laboureau, Sandrine, Raffin Sanson, Marie-Laure, Lamartina, Livia, Pierre, Peggy, Batisse Ligner, Marie, Hescot, Ségolène, Al Ghuzlan, Abir, Renaudin, Karine, Libé, Rosella, Laroche, Suzanne, Deniziaut, Gabrielle, Gimenez-Roqueplo, Anne-Paule, Jannin, Arnaud, Leboulleux, Sophie, Guerin, Carole, Faron, Matthieu, Baudin, Eric
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container_issue 9
container_start_page 2726
container_title The journal of clinical endocrinology and metabolism
container_volume 106
creator Moog, Sophie
Castinetti, Frédéric
DoCao, Christine
Amar, Laurence
Hadoux, Julien
Lussey-Lepoutre, Charlotte
Borson-Chazot, Françoise
Vezzosi, Delphine
Drui, Delphine
Laboureau, Sandrine
Raffin Sanson, Marie-Laure
Lamartina, Livia
Pierre, Peggy
Batisse Ligner, Marie
Hescot, Ségolène
Al Ghuzlan, Abir
Renaudin, Karine
Libé, Rosella
Laroche, Suzanne
Deniziaut, Gabrielle
Gimenez-Roqueplo, Anne-Paule
Jannin, Arnaud
Leboulleux, Sophie
Guerin, Carole
Faron, Matthieu
Baudin, Eric
description Abstract Context The behavior of locally advanced pheochromocytoma (LAP) remains unknown. Objective We characterized the population with LAP and recurrence-free survival (RFS). Methods This retrospective multicentric study was run within the ENDOCAN-COMETE network and French Group of Endocrine Tumors (GTE) from 2003 to 2018, including patients from 11 French referral centers with LAP as defined by capsular invasion, vascular invasion, adipose tissue invasion, and/or positive locoregional lymph nodes at diagnosis without evidence of distant metastasis. The main outcome measure was recurrence, defined as tumor reappearance, including local site and/or distant metastasis. The primary endpoint was RFS analysis; secondary endpoints were characterization, overall survival (OS), and prognostic factors of recurrence. Results Among 950 patients, 90 (9%) exhibited LAP criteria and 55 met inclusion criteria (median age, 53 years; 61% males; 14% with germline mutation; 84% with catecholamine excess). LAP was defined by 31 (56%) capsular invasions, 27 (49%) fat invasions, 6 (11%) positive lymph nodes, and 22 (40%) vascular invasions. After median follow-up of 54 months (range, 6-180), 12 patients (22%) had recurrences and 3 (5%) died of metastatic disease. Median RFS was 115 months (range, 6-168). Recurrences were local in 2 patients, distant in 2, and both local and distant in 8 patients. Median OS of patients was not reached. Size above 6.5 cm (P = 0.019) and Ki-67 > 2% (P = 0.028) were identified as independent significant prognostic factors in multivariate analysis. Conclusion LAP represents 9% of pheochromocytoma’s population and has a metastatic behavior. This study paves the way for future pathological TNM classification.
doi_str_mv 10.1210/clinem/dgab202
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Objective We characterized the population with LAP and recurrence-free survival (RFS). Methods This retrospective multicentric study was run within the ENDOCAN-COMETE network and French Group of Endocrine Tumors (GTE) from 2003 to 2018, including patients from 11 French referral centers with LAP as defined by capsular invasion, vascular invasion, adipose tissue invasion, and/or positive locoregional lymph nodes at diagnosis without evidence of distant metastasis. The main outcome measure was recurrence, defined as tumor reappearance, including local site and/or distant metastasis. The primary endpoint was RFS analysis; secondary endpoints were characterization, overall survival (OS), and prognostic factors of recurrence. Results Among 950 patients, 90 (9%) exhibited LAP criteria and 55 met inclusion criteria (median age, 53 years; 61% males; 14% with germline mutation; 84% with catecholamine excess). LAP was defined by 31 (56%) capsular invasions, 27 (49%) fat invasions, 6 (11%) positive lymph nodes, and 22 (40%) vascular invasions. After median follow-up of 54 months (range, 6-180), 12 patients (22%) had recurrences and 3 (5%) died of metastatic disease. Median RFS was 115 months (range, 6-168). Recurrences were local in 2 patients, distant in 2, and both local and distant in 8 patients. Median OS of patients was not reached. Size above 6.5 cm (P = 0.019) and Ki-67 &gt; 2% (P = 0.028) were identified as independent significant prognostic factors in multivariate analysis. Conclusion LAP represents 9% of pheochromocytoma’s population and has a metastatic behavior. This study paves the way for future pathological TNM classification.</description><identifier>ISSN: 0021-972X</identifier><identifier>EISSN: 1945-7197</identifier><identifier>DOI: 10.1210/clinem/dgab202</identifier><identifier>PMID: 33782697</identifier><language>eng</language><publisher>US: Oxford University Press</publisher><subject>Adipose tissue ; Adipose tissues ; Catecholamines ; Gene mutations ; Human health and pathology ; Invasions ; Life Sciences ; Lymph nodes ; Lymphatic system ; Medical prognosis ; Metastases ; Metastasis ; Multivariate analysis ; Pheochromocytoma ; Survival ; Survival analysis</subject><ispartof>The journal of clinical endocrinology and metabolism, 2021-09, Vol.106 (9), p.2726-2737</ispartof><rights>The Author(s) 2021. Published by Oxford University Press on behalf of the Endocrine Society. All rights reserved. For permissions, please e-mail: journals.permissions@oup.com 2021</rights><rights>The Author(s) 2021. Published by Oxford University Press on behalf of the Endocrine Society. All rights reserved. For permissions, please e-mail: journals.permissions@oup.com.</rights><rights>COPYRIGHT 2021 Oxford University Press</rights><rights>The Author(s) 2021. Published by Oxford University Press on behalf of the Endocrine Society. All rights reserved. For permissions, please e-mail: journals.permissions@oup.com</rights><rights>Distributed under a Creative Commons Attribution 4.0 International License</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c498t-9232ef29c4123fe4a11ad3c685b5874f7ef3b083ed9c76c46ac2ca84f1d59bfc3</citedby><cites>FETCH-LOGICAL-c498t-9232ef29c4123fe4a11ad3c685b5874f7ef3b083ed9c76c46ac2ca84f1d59bfc3</cites><orcidid>0000-0002-2198-3582 ; 0000-0003-2515-7840 ; 0000-0003-2228-0106 ; 0000-0002-1808-8800 ; 0000-0003-3942-4276 ; 0000-0002-4816-670X ; 0000-0002-4824-8502 ; 0000-0002-4247-3039</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>230,314,780,784,885,27924,27925</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/33782697$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink><backlink>$$Uhttps://amu.hal.science/hal-03662818$$DView record in HAL$$Hfree_for_read</backlink></links><search><creatorcontrib>Moog, Sophie</creatorcontrib><creatorcontrib>Castinetti, Frédéric</creatorcontrib><creatorcontrib>DoCao, Christine</creatorcontrib><creatorcontrib>Amar, Laurence</creatorcontrib><creatorcontrib>Hadoux, Julien</creatorcontrib><creatorcontrib>Lussey-Lepoutre, Charlotte</creatorcontrib><creatorcontrib>Borson-Chazot, Françoise</creatorcontrib><creatorcontrib>Vezzosi, Delphine</creatorcontrib><creatorcontrib>Drui, Delphine</creatorcontrib><creatorcontrib>Laboureau, Sandrine</creatorcontrib><creatorcontrib>Raffin Sanson, Marie-Laure</creatorcontrib><creatorcontrib>Lamartina, Livia</creatorcontrib><creatorcontrib>Pierre, Peggy</creatorcontrib><creatorcontrib>Batisse Ligner, Marie</creatorcontrib><creatorcontrib>Hescot, Ségolène</creatorcontrib><creatorcontrib>Al Ghuzlan, Abir</creatorcontrib><creatorcontrib>Renaudin, Karine</creatorcontrib><creatorcontrib>Libé, Rosella</creatorcontrib><creatorcontrib>Laroche, Suzanne</creatorcontrib><creatorcontrib>Deniziaut, Gabrielle</creatorcontrib><creatorcontrib>Gimenez-Roqueplo, Anne-Paule</creatorcontrib><creatorcontrib>Jannin, Arnaud</creatorcontrib><creatorcontrib>Leboulleux, Sophie</creatorcontrib><creatorcontrib>Guerin, Carole</creatorcontrib><creatorcontrib>Faron, Matthieu</creatorcontrib><creatorcontrib>Baudin, Eric</creatorcontrib><title>Recurrence-Free Survival Analysis in Locally Advanced Pheochromocytoma: First Appraisal</title><title>The journal of clinical endocrinology and metabolism</title><addtitle>J Clin Endocrinol Metab</addtitle><description>Abstract Context The behavior of locally advanced pheochromocytoma (LAP) remains unknown. Objective We characterized the population with LAP and recurrence-free survival (RFS). Methods This retrospective multicentric study was run within the ENDOCAN-COMETE network and French Group of Endocrine Tumors (GTE) from 2003 to 2018, including patients from 11 French referral centers with LAP as defined by capsular invasion, vascular invasion, adipose tissue invasion, and/or positive locoregional lymph nodes at diagnosis without evidence of distant metastasis. The main outcome measure was recurrence, defined as tumor reappearance, including local site and/or distant metastasis. The primary endpoint was RFS analysis; secondary endpoints were characterization, overall survival (OS), and prognostic factors of recurrence. Results Among 950 patients, 90 (9%) exhibited LAP criteria and 55 met inclusion criteria (median age, 53 years; 61% males; 14% with germline mutation; 84% with catecholamine excess). LAP was defined by 31 (56%) capsular invasions, 27 (49%) fat invasions, 6 (11%) positive lymph nodes, and 22 (40%) vascular invasions. After median follow-up of 54 months (range, 6-180), 12 patients (22%) had recurrences and 3 (5%) died of metastatic disease. Median RFS was 115 months (range, 6-168). Recurrences were local in 2 patients, distant in 2, and both local and distant in 8 patients. Median OS of patients was not reached. Size above 6.5 cm (P = 0.019) and Ki-67 &gt; 2% (P = 0.028) were identified as independent significant prognostic factors in multivariate analysis. Conclusion LAP represents 9% of pheochromocytoma’s population and has a metastatic behavior. This study paves the way for future pathological TNM classification.</description><subject>Adipose tissue</subject><subject>Adipose tissues</subject><subject>Catecholamines</subject><subject>Gene mutations</subject><subject>Human health and pathology</subject><subject>Invasions</subject><subject>Life Sciences</subject><subject>Lymph nodes</subject><subject>Lymphatic system</subject><subject>Medical prognosis</subject><subject>Metastases</subject><subject>Metastasis</subject><subject>Multivariate analysis</subject><subject>Pheochromocytoma</subject><subject>Survival</subject><subject>Survival analysis</subject><issn>0021-972X</issn><issn>1945-7197</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2021</creationdate><recordtype>article</recordtype><recordid>eNqF0c-L1DAUB_AiijuuXj1KwYseuptfzQ9vZXFcYUDxB3oLafqykyVtukk7MP-9HWZcRRYkh8Dj8x7J-xbFS4wuMMHo0gY_QH_Z3ZiWIPKoWGHF6kpgJR4XK4QIrpQgP8-KZznfIoQZq-nT4oxSIQlXYlX8-AJ2TgkGC9U6AZRf57TzOxPKZjBhn30u_VBuojUh7Mum25lFduXnLUS7TbGPdj_F3rwr1z7lqWzGMRmfTXhePHEmZHhxus-L7-v3366uq82nDx-vmk1lmZJTpQgl4IiyDBPqgBmMTUctl3VbS8GcAEdbJCl0ygpuGTeWWCOZw12tWmfpefH2OHdrgh6T703a62i8vm42-lBDlHMisdzhxb452jHFuxnypHufLYRgBohz1qRGAjNOqVzo63_obZzTspFFqZpjilT9l7oxAbQfXJySsYehuuFS8hoLrhZ18YBaTge9t3EA55f6Qw02xZwTuPuPYaQPqetj6vqU-tLw6vTaue2hu-e_Y_6zpTiP_xv2C5lwtkg</recordid><startdate>20210901</startdate><enddate>20210901</enddate><creator>Moog, Sophie</creator><creator>Castinetti, Frédéric</creator><creator>DoCao, Christine</creator><creator>Amar, Laurence</creator><creator>Hadoux, Julien</creator><creator>Lussey-Lepoutre, Charlotte</creator><creator>Borson-Chazot, Françoise</creator><creator>Vezzosi, Delphine</creator><creator>Drui, Delphine</creator><creator>Laboureau, Sandrine</creator><creator>Raffin Sanson, Marie-Laure</creator><creator>Lamartina, Livia</creator><creator>Pierre, Peggy</creator><creator>Batisse Ligner, Marie</creator><creator>Hescot, Ségolène</creator><creator>Al Ghuzlan, Abir</creator><creator>Renaudin, Karine</creator><creator>Libé, Rosella</creator><creator>Laroche, Suzanne</creator><creator>Deniziaut, Gabrielle</creator><creator>Gimenez-Roqueplo, Anne-Paule</creator><creator>Jannin, Arnaud</creator><creator>Leboulleux, Sophie</creator><creator>Guerin, Carole</creator><creator>Faron, Matthieu</creator><creator>Baudin, Eric</creator><general>Oxford University Press</general><general>Endocrine Society</general><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7QP</scope><scope>7T5</scope><scope>7TM</scope><scope>H94</scope><scope>K9.</scope><scope>7X8</scope><scope>1XC</scope><orcidid>https://orcid.org/0000-0002-2198-3582</orcidid><orcidid>https://orcid.org/0000-0003-2515-7840</orcidid><orcidid>https://orcid.org/0000-0003-2228-0106</orcidid><orcidid>https://orcid.org/0000-0002-1808-8800</orcidid><orcidid>https://orcid.org/0000-0003-3942-4276</orcidid><orcidid>https://orcid.org/0000-0002-4816-670X</orcidid><orcidid>https://orcid.org/0000-0002-4824-8502</orcidid><orcidid>https://orcid.org/0000-0002-4247-3039</orcidid></search><sort><creationdate>20210901</creationdate><title>Recurrence-Free Survival Analysis in Locally Advanced Pheochromocytoma: First Appraisal</title><author>Moog, Sophie ; Castinetti, Frédéric ; DoCao, Christine ; Amar, Laurence ; Hadoux, Julien ; Lussey-Lepoutre, Charlotte ; Borson-Chazot, Françoise ; Vezzosi, Delphine ; Drui, Delphine ; Laboureau, Sandrine ; Raffin Sanson, Marie-Laure ; Lamartina, Livia ; Pierre, Peggy ; Batisse Ligner, Marie ; Hescot, Ségolène ; Al Ghuzlan, Abir ; Renaudin, Karine ; Libé, Rosella ; Laroche, Suzanne ; Deniziaut, Gabrielle ; Gimenez-Roqueplo, Anne-Paule ; Jannin, Arnaud ; Leboulleux, Sophie ; Guerin, Carole ; Faron, Matthieu ; Baudin, Eric</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c498t-9232ef29c4123fe4a11ad3c685b5874f7ef3b083ed9c76c46ac2ca84f1d59bfc3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2021</creationdate><topic>Adipose tissue</topic><topic>Adipose tissues</topic><topic>Catecholamines</topic><topic>Gene mutations</topic><topic>Human health and pathology</topic><topic>Invasions</topic><topic>Life Sciences</topic><topic>Lymph nodes</topic><topic>Lymphatic system</topic><topic>Medical prognosis</topic><topic>Metastases</topic><topic>Metastasis</topic><topic>Multivariate analysis</topic><topic>Pheochromocytoma</topic><topic>Survival</topic><topic>Survival analysis</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Moog, Sophie</creatorcontrib><creatorcontrib>Castinetti, Frédéric</creatorcontrib><creatorcontrib>DoCao, Christine</creatorcontrib><creatorcontrib>Amar, Laurence</creatorcontrib><creatorcontrib>Hadoux, Julien</creatorcontrib><creatorcontrib>Lussey-Lepoutre, Charlotte</creatorcontrib><creatorcontrib>Borson-Chazot, Françoise</creatorcontrib><creatorcontrib>Vezzosi, Delphine</creatorcontrib><creatorcontrib>Drui, Delphine</creatorcontrib><creatorcontrib>Laboureau, Sandrine</creatorcontrib><creatorcontrib>Raffin Sanson, Marie-Laure</creatorcontrib><creatorcontrib>Lamartina, Livia</creatorcontrib><creatorcontrib>Pierre, Peggy</creatorcontrib><creatorcontrib>Batisse Ligner, Marie</creatorcontrib><creatorcontrib>Hescot, Ségolène</creatorcontrib><creatorcontrib>Al Ghuzlan, Abir</creatorcontrib><creatorcontrib>Renaudin, Karine</creatorcontrib><creatorcontrib>Libé, Rosella</creatorcontrib><creatorcontrib>Laroche, Suzanne</creatorcontrib><creatorcontrib>Deniziaut, Gabrielle</creatorcontrib><creatorcontrib>Gimenez-Roqueplo, Anne-Paule</creatorcontrib><creatorcontrib>Jannin, Arnaud</creatorcontrib><creatorcontrib>Leboulleux, Sophie</creatorcontrib><creatorcontrib>Guerin, Carole</creatorcontrib><creatorcontrib>Faron, Matthieu</creatorcontrib><creatorcontrib>Baudin, Eric</creatorcontrib><collection>PubMed</collection><collection>CrossRef</collection><collection>Calcium &amp; Calcified Tissue Abstracts</collection><collection>Immunology Abstracts</collection><collection>Nucleic Acids Abstracts</collection><collection>AIDS and Cancer Research Abstracts</collection><collection>ProQuest Health &amp; Medical Complete (Alumni)</collection><collection>MEDLINE - Academic</collection><collection>Hyper Article en Ligne (HAL)</collection><jtitle>The journal of clinical endocrinology and metabolism</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Moog, Sophie</au><au>Castinetti, Frédéric</au><au>DoCao, Christine</au><au>Amar, Laurence</au><au>Hadoux, Julien</au><au>Lussey-Lepoutre, Charlotte</au><au>Borson-Chazot, Françoise</au><au>Vezzosi, Delphine</au><au>Drui, Delphine</au><au>Laboureau, Sandrine</au><au>Raffin Sanson, Marie-Laure</au><au>Lamartina, Livia</au><au>Pierre, Peggy</au><au>Batisse Ligner, Marie</au><au>Hescot, Ségolène</au><au>Al Ghuzlan, Abir</au><au>Renaudin, Karine</au><au>Libé, Rosella</au><au>Laroche, Suzanne</au><au>Deniziaut, Gabrielle</au><au>Gimenez-Roqueplo, Anne-Paule</au><au>Jannin, Arnaud</au><au>Leboulleux, Sophie</au><au>Guerin, Carole</au><au>Faron, Matthieu</au><au>Baudin, Eric</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Recurrence-Free Survival Analysis in Locally Advanced Pheochromocytoma: First Appraisal</atitle><jtitle>The journal of clinical endocrinology and metabolism</jtitle><addtitle>J Clin Endocrinol Metab</addtitle><date>2021-09-01</date><risdate>2021</risdate><volume>106</volume><issue>9</issue><spage>2726</spage><epage>2737</epage><pages>2726-2737</pages><issn>0021-972X</issn><eissn>1945-7197</eissn><abstract>Abstract Context The behavior of locally advanced pheochromocytoma (LAP) remains unknown. Objective We characterized the population with LAP and recurrence-free survival (RFS). Methods This retrospective multicentric study was run within the ENDOCAN-COMETE network and French Group of Endocrine Tumors (GTE) from 2003 to 2018, including patients from 11 French referral centers with LAP as defined by capsular invasion, vascular invasion, adipose tissue invasion, and/or positive locoregional lymph nodes at diagnosis without evidence of distant metastasis. The main outcome measure was recurrence, defined as tumor reappearance, including local site and/or distant metastasis. The primary endpoint was RFS analysis; secondary endpoints were characterization, overall survival (OS), and prognostic factors of recurrence. Results Among 950 patients, 90 (9%) exhibited LAP criteria and 55 met inclusion criteria (median age, 53 years; 61% males; 14% with germline mutation; 84% with catecholamine excess). LAP was defined by 31 (56%) capsular invasions, 27 (49%) fat invasions, 6 (11%) positive lymph nodes, and 22 (40%) vascular invasions. After median follow-up of 54 months (range, 6-180), 12 patients (22%) had recurrences and 3 (5%) died of metastatic disease. Median RFS was 115 months (range, 6-168). Recurrences were local in 2 patients, distant in 2, and both local and distant in 8 patients. Median OS of patients was not reached. Size above 6.5 cm (P = 0.019) and Ki-67 &gt; 2% (P = 0.028) were identified as independent significant prognostic factors in multivariate analysis. Conclusion LAP represents 9% of pheochromocytoma’s population and has a metastatic behavior. This study paves the way for future pathological TNM classification.</abstract><cop>US</cop><pub>Oxford University Press</pub><pmid>33782697</pmid><doi>10.1210/clinem/dgab202</doi><tpages>12</tpages><orcidid>https://orcid.org/0000-0002-2198-3582</orcidid><orcidid>https://orcid.org/0000-0003-2515-7840</orcidid><orcidid>https://orcid.org/0000-0003-2228-0106</orcidid><orcidid>https://orcid.org/0000-0002-1808-8800</orcidid><orcidid>https://orcid.org/0000-0003-3942-4276</orcidid><orcidid>https://orcid.org/0000-0002-4816-670X</orcidid><orcidid>https://orcid.org/0000-0002-4824-8502</orcidid><orcidid>https://orcid.org/0000-0002-4247-3039</orcidid><oa>free_for_read</oa></addata></record>
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source Oxford University Press Journals All Titles (1996-Current); EZB-FREE-00999 freely available EZB journals; Alma/SFX Local Collection
subjects Adipose tissue
Adipose tissues
Catecholamines
Gene mutations
Human health and pathology
Invasions
Life Sciences
Lymph nodes
Lymphatic system
Medical prognosis
Metastases
Metastasis
Multivariate analysis
Pheochromocytoma
Survival
Survival analysis
title Recurrence-Free Survival Analysis in Locally Advanced Pheochromocytoma: First Appraisal
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