Outcomes of adrenal-sparing surgery or total adrenalectomy in phaeochromocytoma associated with multiple endocrine neoplasia type 2: an international retrospective population-based study

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dc.contributorSistema FMUSP-HC: Faculdade de Medicina da Universidade de São Paulo (FMUSP) e Hospital das Clínicas da FMUSP
dc.contributor.authorCASTINETTI, Frederic
dc.contributor.authorQI, Xiao-Ping
dc.contributor.authorWALZ, Martin K.
dc.contributor.authorMAIA, Ana Luiza
dc.contributor.authorSANSO, Gabriela
dc.contributor.authorPECZKOWSKA, Mariola
dc.contributor.authorHASSE-LAZAR, Kornelia
dc.contributor.authorLINKS, Thera P.
dc.contributor.authorDVORAKOVA, Sarka
dc.contributor.authorTOLEDO, Rodrigo A.
dc.contributor.authorMIAN, Caterina
dc.contributor.authorBUGALHO, Maria Joao
dc.contributor.authorWOHLLK, Nelson
dc.contributor.authorKOLLYUKH, Oleg
dc.contributor.authorCANU, Letizia
dc.contributor.authorLOLI, Paola
dc.contributor.authorBERGMANN, Simona R.
dc.contributor.authorCOSTA, Josefina Biarnes
dc.contributor.authorMAKAY, Ozer
dc.contributor.authorPATOCS, Attila
dc.contributor.authorPFEIFER, Marija
dc.contributor.authorSHAH, Nalini S.
dc.contributor.authorCUNY, Thomas
dc.contributor.authorBRAUCKHOFF, Michael
dc.contributor.authorBAUSCH, Birke
dc.contributor.authorDOBSCHUETZ, Ernst von
dc.contributor.authorLETIZIA, Claudio
dc.contributor.authorBARCZYNSKI, Marcin
dc.contributor.authorALEVIZAKI, Maria K.
dc.contributor.authorCZETWERTYNSKA, Malgorzata
dc.contributor.authorUGURLU, M. Umit
dc.contributor.authorVALK, Gerlof
dc.contributor.authorPLUKKER, John T. M.
dc.contributor.authorSARTORATO, Paola
dc.contributor.authorSIQUEIRA, Debora R.
dc.contributor.authorBARONTINI, Marta
dc.contributor.authorSZPERL, Malgorzata
dc.contributor.authorJARZAB, Barbara
dc.contributor.authorVERBEEK, Hans H. G.
dc.contributor.authorZELINKA, Tomas
dc.contributor.authorVLCEK, Petr
dc.contributor.authorTOLEDO, Sergio P. A.
dc.contributor.authorCOUTINHO, Flavia L.
dc.contributor.authorMANNELLI, Massimo
dc.contributor.authorRECASENS, Monica
dc.contributor.authorDEMARQUET, Lea
dc.contributor.authorPETRAMALA, Luigi
dc.contributor.authorYAREMCHUK, Svetlana
dc.contributor.authorZABOLOTNYI, Dmitry
dc.contributor.authorSCHIAVI, Francesca
dc.contributor.authorOPOCHER, Giuseppe
dc.contributor.authorRACZ, Karoly
dc.contributor.authorJANUSZEWICZ, Andrzej
dc.contributor.authorWERYHA, Georges
dc.contributor.authorHENRY, Jean-Francois
dc.contributor.authorBRUE, Thierry
dc.contributor.authorCONTE-DEVOLX, Bernard
dc.contributor.authorENG, Charis
dc.contributor.authorNEUMANN, Hartmut P. H.
dc.date.accessioned2014-09-30T14:38:08Z
dc.date.available2014-09-30T14:38:08Z
dc.date.issued2014
dc.description.abstractBackground The prevention of medullary thyroid cancer in patients with multiple endocrine neoplasia type 2 syndrome has demonstrated the ability of molecular diagnosis and prophylactic surgery to improve patient outcomes. However, the other major neoplasia associated with multiple endocrine neoplasia type 2, phaeochromocytoma, is not as well characterised in terms of occurrence and treatment outcomes. In this study, we aimed to systematically characterise the outcomes of management of phaeochromocytoma associated with multiple endocrine neoplasia type 2. Methods This multinational observational retrospective population-based study compiled data on patients with multiple endocrine neoplasia type 2 from 30 academic medical centres across Europe, the Americas, and Asia. Patients were included if they were carriers of germline pathogenic mutations of the RET gene, or were first-degree relatives with histologically proven medullary thyroid cancer and phaeochromocytoma. We gathered clinical information about patients' RET genotype, type of treatment for phaeochromocytoma (ie, unilateral or bilateral operations as adrenalectomy or adrenal-sparing surgery, and as open or endoscopic operations), and postoperative outcomes (adrenal function, malignancy, and death). The type of surgery was decided by each investigator and the timing of surgery was patient driven. The primary aim of our analysis was to compare disease-free survival after either adrenal-sparing surgery or adrenalectomy. Findings 1210 patients with multiple endocrine neoplasia type 2 were included in our database, 563 of whom had phaeochromocytoma. Treatment was adrenalectomy in 438 (79%) of 552 operated patients, and adrenal-sparing surgery in 114 (21%). Phaeochromocytoma recurrence occurred in four (3%) of 153 of the operated glands after adrenal-sparing surgery after 6-13 years, compared with 11 (2%) of 717 glands operated by adrenalectomy (p=0.57). Postoperative adrenal insufficiency or steroid dependency developed in 292 (86%) of 339 patients with bilateral phaeochromocytoma who underwent surgery. However, 47 (57%) of 82 patients with bilateral phaeochromocytoma who underwent adrenal-sparing surgery did not become steroid dependent. Interpretation The treatment of multiple endocrine neoplasia type 2-related phaeochromocytoma continues to rely on adrenalectomies with their associated Addisonian-like complications and consequent lifelong dependency on steroids. Adrenal-sparing surgery, a highly successful treatment option in experienced centres, should be the surgical approach of choice to reduce these complications.
dc.description.indexMEDLINE
dc.description.sponsorshipEuropean Union
dc.description.sponsorshipGerman Cancer Foundation
dc.description.sponsorshipArthur Blank Foundation
dc.description.sponsorshipItalian Government
dc.description.sponsorshipCharles University
dc.description.sponsorshipCzech Ministry of Health
dc.description.sponsorshipNanjing Military Command
dc.description.sponsorshipNational Science Centre Poland
dc.description.sponsorshipNational Research Council for Scientific and Technological Development (CNPq) [401990/2010-9]
dc.description.sponsorshipState of Sao Paulo Research Foundation - FAPESP
dc.identifier.citationLANCET ONCOLOGY, v.15, n.6, p.648-655, 2014
dc.identifier.doi10.1016/S1470-2045(14)70154-8
dc.identifier.eissn1474-5488
dc.identifier.issn1470-2045
dc.identifier.urihttps://observatorio.fm.usp.br/handle/OPI/7346
dc.language.isoeng
dc.publisherELSEVIER SCIENCE INC
dc.relation.ispartofLancet Oncology
dc.rightsrestrictedAccess
dc.rights.holderCopyright ELSEVIER SCIENCE INC
dc.subject.othermedullary-thyroid carcinoma
dc.subject.otherret protooncogene
dc.subject.othergermline mutations
dc.subject.otherdisease
dc.subject.other2a
dc.subject.othermanagement
dc.subject.wosOncology
dc.titleOutcomes of adrenal-sparing surgery or total adrenalectomy in phaeochromocytoma associated with multiple endocrine neoplasia type 2: an international retrospective population-based study
dc.typearticle
dc.type.categoryoriginal article
dc.type.versionpublishedVersion
dspace.entity.typePublication
hcfmusp.affiliation.countryTurquia
hcfmusp.affiliation.countryFrança
hcfmusp.affiliation.countryChina
hcfmusp.affiliation.countryAlemanha
hcfmusp.affiliation.countryArgentina
hcfmusp.affiliation.countryPolônia
hcfmusp.affiliation.countryHolanda
hcfmusp.affiliation.countryRepública Tcheca
hcfmusp.affiliation.countryItália
hcfmusp.affiliation.countryPortugal
hcfmusp.affiliation.countryChile
hcfmusp.affiliation.countryUcrânia
hcfmusp.affiliation.countryEspanha
hcfmusp.affiliation.countryHungria
hcfmusp.affiliation.countryEslovênia
hcfmusp.affiliation.countryÍndia
hcfmusp.affiliation.countryNoruega
hcfmusp.affiliation.countryGrécia
hcfmusp.affiliation.countryEstados Unidos
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hcfmusp.affiliation.countryisous
hcfmusp.author.externalCASTINETTI, Frederic:Aix Marseille Univ, La Timone Hosp, Hop Marseille, Dept Endocrinol, F-13005 Marseille, France; Aix Marseille Univ, Ctr Rech Neurobiol & Neurophysiol Marseille, F-13005 Marseille, France
hcfmusp.author.externalQI, Xiao-Ping:Anhui Med Univ, PLA Hangzhou Clin Coll, PLA Hosp 117, Dept Oncol, Hangzhou, Zhejiang, Peoples R China; Anhui Med Univ, PLA Hangzhou Clin Coll, PLA Hosp 117, Dept Urol Surg, Hangzhou, Zhejiang, Peoples R China
hcfmusp.author.externalWALZ, Martin K.:Kliniken Essen Mitte, Dept Surg, Essen, Germany; Kliniken Essen Mitte, Ctr Minimally Invas Surg, Essen, Germany
hcfmusp.author.externalMAIA, Ana Luiza:Univ Fed Rio Grande do Sul, Hosp Clin Porto Alegre, Div Endocrinol, Thyroid Sect, BR-90046900 Porto Alegre, RS, Brazil
hcfmusp.author.externalSANSO, Gabriela:Hosp Ninos Dr Ricardo Gutierrez, Ctr Endocrinol Invest, Buenos Aires, DF, Argentina
hcfmusp.author.externalPECZKOWSKA, Mariola:Inst Cardiol, Dept Hypertens, Warsaw, Poland
hcfmusp.author.externalHASSE-LAZAR, Kornelia:Maria Sklodowska Curie Mem Ctr & Inst Oncol, Dept Nucl Med & Endocrine Oncol, Gliwice Branch, Gliwice, Poland
hcfmusp.author.externalLINKS, Thera P.:Univ Groningen, Univ Med Ctr Groningen, Dept Endocrinol, NL-9700 AB Groningen, Netherlands
hcfmusp.author.externalDVORAKOVA, Sarka:Res Inst Endocrinol, Dept Mol Endocrinol, CR-11694 Prague, Czech Republic
hcfmusp.author.externalTOLEDO, Rodrigo A.:Univ Sao Paulo, Sch Med, Dept Endocrinol, Sao Paulo, Brazil
hcfmusp.author.externalMIAN, Caterina:Univ Padua, Endocrinol Dept Med DIMED, Operat Unit, Padua, Italy
hcfmusp.author.externalBUGALHO, Maria Joao:Univ Nova Lisboa, Inst Portugues Oncol Lisboa Francisco Gentil EPE, Serv Endocrinol, P-1200 Lisbon, Portugal; Univ Nova Lisboa, Fac Ciencias Med, P-1200 Lisbon, Portugal
hcfmusp.author.externalWOHLLK, Nelson:Univ Chile, Hosp Salvador, Endocrine Sect, Santiago, Chile
hcfmusp.author.externalKOLLYUKH, Oleg:Inst Endocrinol, Kiev, Ukraine
hcfmusp.author.externalCANU, Letizia:Univ Florence, Dept Expt & Clin Biomed Sci, Endocrinol Unit, Florence, Italy
hcfmusp.author.externalLOLI, Paola:Osped Niguarda Ca Granda, Dept Endocrinol, Milan, Italy
hcfmusp.author.externalBERGMANN, Simona R.:Univ Marburg, Fac Med, Div Endocrinol & Diabetol, Marburg, Germany
hcfmusp.author.externalCOSTA, Josefina Biarnes:Hosp Univ Girona, Gerencia Terr Girona, Inst Catala Salut, Girona, Spain
hcfmusp.author.externalMAKAY, Ozer:Ege Univ Hosp, Div Endocrine Surg, Dept Gen Surg, Izmir, Turkey
hcfmusp.author.externalPATOCS, Attila:Hungarian Acad Sci, HSA SE Lendulet Hereditary Endocrine Tumor Res Gr, Mol Med Res Grp, Budapest, Hungary; Semmelweis Univ, H-1085 Budapest, Hungary
hcfmusp.author.externalPFEIFER, Marija:Univ Med Ctr Ljubljana, Dept Endocrinol, Ljubljana, Slovenia
hcfmusp.author.externalSHAH, Nalini S.:Kings Edward Mem Hosp, Seth GS Med Coll, Mumbai, Maharashtra, India
hcfmusp.author.externalCUNY, Thomas:Univ Hosp, Dept Endocrinol, Nancy, France
hcfmusp.author.externalBRAUCKHOFF, Michael:Univ Bergen, Dept Endocrine Surg, Bergen, Norway
hcfmusp.author.externalBAUSCH, Birke:Univ Freiburg, Univ Med Ctr, Dept Med 2, D-79106 Freiburg, Germany
hcfmusp.author.externalDOBSCHUETZ, Ernst von:Univ Freiburg, Univ Med Ctr, Dept Visceral Surg, D-79106 Freiburg, Germany
hcfmusp.author.externalLETIZIA, Claudio:Univ Roma La Sapienza, Dept Internal Med & Med Specialties, Rome, Italy
hcfmusp.author.externalBARCZYNSKI, Marcin:Jagiellonian Univ, Coll Med, Chair Gen Surg 3, Dept Endocrine Surg, Krakow, Poland
hcfmusp.author.externalALEVIZAKI, Maria K.:Univ Athens, Sch Med, Evgenide Hosp, Endocrine Unit, GR-11527 Athens, Greece; Univ Athens, Sch Med, Alexandra Hosp, Dept Med Therapeut, GR-11527 Athens, Greece
hcfmusp.author.externalCZETWERTYNSKA, Malgorzata:Maria Sklodowska Curie Mem Ctr & Inst Oncol, Warsaw, Poland
hcfmusp.author.externalUGURLU, M. Umit:Marmara Univ, Dept Gen Surg, Breast & Endocrine Surg Unit, Istanbul, Turkey
hcfmusp.author.externalVALK, Gerlof:Univ Med Ctr Utrecht, Dept Internal Med, Utrecht, Netherlands
hcfmusp.author.externalPLUKKER, John T. M.:Univ Groningen, Univ Med Ctr Groningen, Dept Surg, NL-9713 AV Groningen, Netherlands
hcfmusp.author.externalSARTORATO, Paola:Gen Hosp, Dept Internal Med, Treviso, Italy
hcfmusp.author.externalSIQUEIRA, Debora R.:Univ Fed Rio Grande do Sul, Hosp Clin Porto Alegre, Div Endocrinol, Thyroid Sect, BR-90046900 Porto Alegre, RS, Brazil
hcfmusp.author.externalBARONTINI, Marta:Hosp Ninos Dr Ricardo Gutierrez, Ctr Endocrinol Invest, Buenos Aires, DF, Argentina
hcfmusp.author.externalSZPERL, Malgorzata:Inst Cardiol, Dept Hypertens, Warsaw, Poland
hcfmusp.author.externalJARZAB, Barbara:Maria Sklodowska Curie Mem Ctr & Inst Oncol, Dept Nucl Med & Endocrine Oncol, Gliwice Branch, Gliwice, Poland
hcfmusp.author.externalVERBEEK, Hans H. G.:Univ Groningen, Univ Med Ctr Groningen, Dept Endocrinol, NL-9700 AB Groningen, Netherlands
hcfmusp.author.externalZELINKA, Tomas:Charles Univ Prague, Fac Med 1, Dept Endocrinol & Metab, Dept Med, Prague, Czech Republic; Gen Univ Hosp, Prague, Czech Republic
hcfmusp.author.externalVLCEK, Petr:Charles Univ Prague, Fac Med 2, Dept Nucl Med & Endocrinol, Prague, Czech Republic
hcfmusp.author.externalMANNELLI, Massimo:Univ Florence, Dept Expt & Clin Biomed Sci, Endocrinol Unit, Florence, Italy
hcfmusp.author.externalRECASENS, Monica:Hosp Univ Girona, Gerencia Terr Girona, Inst Catala Salut, Girona, Spain
hcfmusp.author.externalDEMARQUET, Lea:Univ Hosp, Dept Endocrinol, Nancy, France
hcfmusp.author.externalPETRAMALA, Luigi:Univ Roma La Sapienza, Dept Internal Med & Med Specialties, Rome, Italy
hcfmusp.author.externalYAREMCHUK, Svetlana:Inst Otolaryngol NAMS Ukraine, Kiev, Ukraine
hcfmusp.author.externalZABOLOTNYI, Dmitry:Inst Otolaryngol NAMS Ukraine, Kiev, Ukraine
hcfmusp.author.externalSCHIAVI, Francesca:IRCCS Padova, Veneto Inst Oncol, Padua, Italy
hcfmusp.author.externalOPOCHER, Giuseppe:Univ Padua, Dept Med DIMED, Padua, Italy
hcfmusp.author.externalRACZ, Karoly:Semmelweis Univ, Dept Internal Med 2, H-1085 Budapest, Hungary
hcfmusp.author.externalJANUSZEWICZ, Andrzej:Inst Cardiol, Dept Hypertens, Warsaw, Poland
hcfmusp.author.externalWERYHA, Georges:Univ Hosp, Dept Endocrinol, Nancy, France
hcfmusp.author.externalHENRY, Jean-Francois:Aix Marseille Univ, La Timone Hosp, Dept Endocrine Surg, F-13005 Marseille, France
hcfmusp.author.externalBRUE, Thierry:Aix Marseille Univ, La Timone Hosp, Hop Marseille, Dept Endocrinol, F-13005 Marseille, France; Aix Marseille Univ, Ctr Rech Neurobiol & Neurophysiol Marseille, F-13005 Marseille, France
hcfmusp.author.externalCONTE-DEVOLX, Bernard:Aix Marseille Univ, La Timone Hosp, Hop Marseille, Dept Endocrinol, F-13005 Marseille, France; Aix Marseille Univ, Ctr Rech Neurobiol & Neurophysiol Marseille, F-13005 Marseille, France
hcfmusp.author.externalENG, Charis:Cleveland Clin, Lerner Res Inst, Genom Med Inst, Cleveland, OH 44106 USA; Cleveland Clin, Taussig Canc Inst, Cleveland, OH 44106 USA
hcfmusp.author.externalNEUMANN, Hartmut P. H.:Univ Freiburg, Univ Med Ctr, Dept Nephrol & Gen Med, Sect Prevent Med, D-79106 Freiburg, Germany
hcfmusp.citation.scopus131
hcfmusp.contributor.author-fmusphcSERGIO PEREIRA DE ALMEIDA TOLEDO
hcfmusp.contributor.author-fmusphcFLAVIA LIMA COUTINHO
hcfmusp.description.beginpage648
hcfmusp.description.endpage655
hcfmusp.description.issue6
hcfmusp.description.volume15
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hcfmusp.origem.pubmed24745698
hcfmusp.origem.scopus2-s2.0-84899970790
hcfmusp.origem.wosWOS:000335512100052
hcfmusp.publisher.cityNEW YORK
hcfmusp.publisher.countryUSA
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hcfmusp.remissive.sponsorshipCNPq
hcfmusp.remissive.sponsorshipFAPESP
hcfmusp.remissive.sponsorshipEuropean Union
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