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Outcomes of adrenal-sparing surgery or total adrenalectomy in phaeochromocytoma associated with multiple endocrine neoplasia type 2: an international retrospective population-based study
F. Castinetti, XP. Qi, MK. Walz, AL. Maia, G. Sansó, M. Peczkowska, K. Hasse-Lazar, TP. Links, S. Dvorakova, RA. Toledo, C. Mian, MJ. Bugalho, N. Wohllk, O. Kollyukh, L. Canu, P. Loli, SR. Bergmann, J. Biarnes Costa, O. Makay, A. Patocs, M....
Jazyk angličtina Země Anglie, Velká Británie
Typ dokumentu časopisecké články, pozorovací studie, práce podpořená grantem
Grantová podpora
NT12336
MZ0
CEP - Centrální evidence projektů
NT13901
MZ0
CEP - Centrální evidence projektů
Digitální knihovna NLK
Plný text - Článek
Plný text - Článek
Zdroj
Zdroj
NLK
ProQuest Central
od 2000-09-01 do Před 2 měsíci
Nursing & Allied Health Database (ProQuest)
od 2000-09-01 do Před 2 měsíci
Health & Medicine (ProQuest)
od 2000-09-01 do Před 2 měsíci
Public Health Database (ProQuest)
od 2000-09-01 do Před 2 měsíci
- MeSH
- adrenalektomie mortalita MeSH
- dítě MeSH
- dospělí MeSH
- feochromocytom etiologie mortalita chirurgie MeSH
- Kaplanův-Meierův odhad MeSH
- lidé středního věku MeSH
- lidé MeSH
- mladiství MeSH
- mladý dospělý MeSH
- mnohočetná endokrinní neoplazie typ 2A komplikace mortalita chirurgie MeSH
- nádory nadledvin etiologie mortalita chirurgie MeSH
- přežití bez známek nemoci MeSH
- retrospektivní studie MeSH
- senioři nad 80 let MeSH
- senioři MeSH
- výsledek terapie MeSH
- Check Tag
- dítě MeSH
- dospělí MeSH
- lidé středního věku MeSH
- lidé MeSH
- mladiství MeSH
- mladý dospělý MeSH
- mužské pohlaví MeSH
- senioři nad 80 let MeSH
- senioři MeSH
- ženské pohlaví MeSH
- Publikační typ
- časopisecké články MeSH
- pozorovací studie MeSH
- práce podpořená grantem MeSH
BACKGROUND: 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.
2nd Department of Internal Medicine Semmelweis University Budapest Hungary
Aix Marseille University Department of Endocrine Surgery La Timone Hospital Marseille France
Center for Endocrinological Investigations Hospital de Ninos R Gutierrez Buenos Aires Argentina
Department of Endocrine Surgery University of Bergen Bergen Norway
Department of Endocrinology Ospedale Niguarda Cà Granda Milan Italy
Department of Endocrinology University Hospital Nancy France
Department of Endocrinology University Medical Center Groningen University of Groningen Netherlands
Department of Endocrinology University Medical Center Ljubljana Ljubljana Slovenia
Department of Endocrinology University of Sao Paulo School of Medicine Sao Paulo Brazil
Department of General Surgery Breast and Endocrine Surgery Unit Marmara University Istanbul Turkey
Department of General Surgery Division of Endocrine Surgery Ege University Hospital Izmir Turkey
Department of Hypertension Institute of Cardiology Warsaw Poland
Department of Internal Medicine and Medical Specialties University La Sapienza Rome Italy
Department of Internal Medicine General Hospital Montebelluna Treviso Italy
Department of Internal Medicine University Medical Centre Utrecht Utrecht Netherlands
Department of Medicine University of Padova Padova Italy
Department of Molecular Endocrinology Institute of Endocrinology Prague Czech Republic
Department of Surgery and Center of Minimally Invasive Surgery Kliniken Essen Mitte Essen Germany
Department of Surgery University Medical Centre Groningen Netherlands
Endocrine Section Universidad de Chile Hospital del Salvador Santiago de Chile Chile
Familial Cancer Clinic and Oncoendocrinology Veneto Institute of Oncology IRCCS Padova Padova Italy
Hospital Universitari de Girona Gerencia Territorial Girona Institut Català de la Salut Girona Spain
Institute of Endocrinology Kiev Ukraine
Institute of Otolaryngology NAMS of Ukraine Kiev Ukraine
Operative Unit of the Endocrinology Department of Medicine University of Padova Padova Italy
Citace poskytuje Crossref.org
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- $a Castinetti, Frederic $u Department of Endocrinology, La Timone Hospital, Hopitaux de Marseille and Centre de Recherche en Neurobiologie et Neurophysiologie de Marseille, Aix-Marseille University, Marseille, France. Electronic address: frederic.castinetti@ap-hm.fr.
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