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Comparison of Pheochromocytoma-Specific Morbidity and Mortality Among Adults With Bilateral Pheochromocytomas Undergoing Total Adrenalectomy vs Cortical-Sparing Adrenalectomy
HPH. Neumann, U. Tsoy, I. Bancos, V. Amodru, MK. Walz, A. Tirosh, RJ. Kaur, T. McKenzie, X. Qi, T. Bandgar, R. Petrov, MY. Yukina, A. Roslyakova, ANA. van der Horst-Schrivers, AMA. Berends, AO. Hoff, LA. Castroneves, AM. Ferrara, S. Rizzati, C....
Jazyk angličtina Země Spojené státy americké
Typ dokumentu srovnávací studie, časopisecké články, multicentrická studie, práce podpořená grantem
Grantová podpora
NV16-32665A
MZ0
CEP - Centrální evidence projektů
Digitální knihovna NLK
Plný text - Článek
NLK
Directory of Open Access Journals
od 2018
ProQuest Central
od 2018-01-01
Health & Medicine (ProQuest)
od 2018-01-01
- MeSH
- adrenalektomie škodlivé účinky metody mortalita MeSH
- dospělí MeSH
- feochromocytom mortalita chirurgie MeSH
- léčba šetřící orgány mortalita MeSH
- lidé MeSH
- lokální recidiva nádoru MeSH
- mladý dospělý MeSH
- morbidita MeSH
- nádory nadledvin mortalita chirurgie MeSH
- registrace MeSH
- retrospektivní studie MeSH
- Check Tag
- dospělí MeSH
- lidé MeSH
- mladý dospělý MeSH
- mužské pohlaví MeSH
- ženské pohlaví MeSH
- Publikační typ
- časopisecké články MeSH
- multicentrická studie MeSH
- práce podpořená grantem MeSH
- srovnávací studie MeSH
Importance: Large studies investigating long-term outcomes of patients with bilateral pheochromocytomas treated with either total or cortical-sparing adrenalectomies are needed to inform clinical management. Objective: To determine the association of total vs cortical-sparing adrenalectomy with pheochromocytoma-specific mortality, the burden of primary adrenal insufficiency after bilateral adrenalectomy, and the risk of pheochromocytoma recurrence. Design, Setting, and Participants: This cohort study used data from a multicenter consortium-based registry for 625 patients treated for bilateral pheochromocytomas between 1950 and 2018. Data were analyzed from September 1, 2018, to June 1, 2019. Exposures: Total or cortical-sparing adrenalectomy. Main Outcomes and Measures: Primary adrenal insufficiency, recurrent pheochromocytoma, and mortality. Results: Of 625 patients (300 [48%] female) with a median (interquartile range [IQR]) age of 30 (22-40) years at diagnosis, 401 (64%) were diagnosed with synchronous bilateral pheochromocytomas and 224 (36%) were diagnosed with metachronous pheochromocytomas (median [IQR] interval to second adrenalectomy, 6 [1-13] years). In 505 of 526 tested patients (96%), germline mutations were detected in the genes RET (282 patients [54%]), VHL (184 patients [35%]), and other genes (39 patients [7%]). Of 849 adrenalectomies performed in 625 patients, 324 (52%) were planned as cortical sparing and were successful in 248 of 324 patients (76.5%). Primary adrenal insufficiency occurred in all patients treated with total adrenalectomy but only in 23.5% of patients treated with attempted cortical-sparing adrenalectomy. A third of patients with adrenal insufficiency developed complications, such as adrenal crisis or iatrogenic Cushing syndrome. Of 377 patients who became steroid dependent, 67 (18%) developed at least 1 adrenal crisis and 50 (13%) developed iatrogenic Cushing syndrome during median (IQR) follow-up of 8 (3-25) years. Two patients developed recurrent pheochromocytoma in the adrenal bed despite total adrenalectomy. In contrast, 33 patients (13%) treated with successful cortical-sparing adrenalectomy developed another pheochromocytoma within the remnant adrenal after a median (IQR) of 8 (4-13) years, all of which were successfully treated with another surgery. Cortical-sparing surgery was not associated with survival. Overall survival was associated with comorbidities unrelated to pheochromocytoma: of 63 patients who died, only 3 (5%) died of metastatic pheochromocytoma. Conclusions and Relevance: Patients undergoing cortical-sparing adrenalectomy did not demonstrate decreased survival, despite development of recurrent pheochromocytoma in 13%. Cortical-sparing adrenalectomy should be considered in all patients with hereditary pheochromocytoma.
Department of Endocrinology Ospedale Niguarda Cà Granda Milan Italy
Department of Endocrinology Seth GS Medical College and KEM Hospital Mumbai India
Department of Medicine Division of Endocrinology Tufts Medical Center Boston Massachusetts
Department of Molecular Endocrinology Institute of Endocrinology Prague Czech Republic
Department of Molecular Medicine and Surgery Karolinska Institutet Stockholm Sweden
Department of Surgery Endocrinology Research Center Moscow Russia
Department of Surgery Huyssens Foundation Clinics Essen Germany
Department of Surgical Oncology Bakhrushin Brothers Moscow City Hospital Moscow Russia
Department of Urology Xiangya Hospital Central South University Changsha China
Division of Endocrinology and Metabolism Siriraj Hospital Mahidol University Bangkok Thailand
Division of Endocrinology Diabetes Metabolism and Nutrition Mayo Clinic Rochester Minnesota
Division of General Surgery Mayo Clinic Rochester Minnesota
Familial Cancer Clinic and Oncoendocrinology Veneto Institute of Oncology IOV IRCCS Padua Italy
Hospital Universitari de Girona Gerencia Territorial Girona Institut Català de la Salut Girona Spain
Institute of Cardiology Department of Hypertension Warsaw Poland
Institute of Endocrinology and Metabolism NAMS of Ukraine Kiev Ukraine
Operative Unit of the Endocrinology Department of Medicine University of Padua Padua Italy
Citace poskytuje Crossref.org
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