1. Maternal and fetal outcomes in phaeochromocytoma and pregnancy: a multicentre retrospective cohort study and systematic review of literature
- Author
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Andreas Ebbehoj, Eleonora P M Corssmit, Jan Calissendorff, Thera P. Links, Özer Makay, Maximilien Rappaport, Petr Vlĕek, Tushar Bandgar, Catharina Larsson, Elena N. Grineva, Luigi Petramala, Ravinder Kaur, Viacheslav I. Egorov, Hartmut P. H. Neumann, Heather Wachtel, Tobias Else, Francesca Boaretto, Xiao-Ping Qi, Henri J L M Timmers, Anna Roslyakova, M. Umit Ugurlu, Ronald M. Lechan, Anand Vaidya, Kornelia Hasse-Lazar, Claudio E. Kater, Esben Søndergaard, Zhi-xian Yu, Rene Eduardo Diaz, Mohammad Hassan Murad, Ruth T Casey, Debbie L. Cohen, Roman Petrov, Lucinda Gruber, C Christofer Juhlin, Claudio Letizia, Maria Adelaide Albergaria Pereira, Inna Stepanovna Kudlai, Bernadette Jenner, Sergiy Cherenko, Lauren Fishbein, William F. Young, Ya-Sheng Huang, Marina Y. Yukina, Scott A Akker, Andrey Y Kovalenko, Uma Kaimal Saikia, Minghao Li, Silvia Rizzati, Stefania Zovato, Xu-dong Fang, Nelson Wohllk, Charis Eng, Mariola Pęczkowska, Martin Fassnacht, Sanjeet Kumar Jaiswal, Oliver Gimm, Gianluca Donatini, Milan Jovanovic, Robin P.F Dullaart, Ilgin Yildirim Simsir, Helen Simpson, Maciej Robaczyk, Marcin Barczyński, Steven G. Waguespack, Katharina Langton, Martin K. Walz, Paul Skierczynski, Alfonso Massimiliano Ferrara, Dipti Sarma, Irina Bancos, Vishnu Garla, Birke Bausch, Maria João Bugalho, Merav Fraenkel, Joanne Ngeow Yuen Yie, Flavia A Costa-Barbosa, Giuseppe Opocher, Camilo Jimenez, Tada Kunavisarut, Larry J. Prokop, Lawrence S Kirschner, Longfei Liu, Feyza Erenler, Elisa Taschin, Valentina Morelli, Per Løgstrup Poulsen, Marcus Quinkler, Natalia Valeryevna Khudiakova, Åse Krogh Rasmussen, Volha Vasilkova, Nicola Tufton, Nikita V. Ivanov, William Drake, Maryna Bobryk, Eric Jonasch, Swati Ramteke-Jadhav, Aviva Cohn, Diane Donegan, Sarka Dvorakova, Elizabeth J. Atkinson, Dmitry Beltsevich, Emma Hodson, Uliana Tsoy, Nino Zavrashvili, Jochen Seufert, Zulfiya Shafigullina, Xin He, Utku E Soyaltin, Nicole M. Iñiguez-Ariza, Timo Deutschbein, Francesca Schiavi, Mark Sherlock, Stefan Zschiedrich, Jes Sloth Mathiesen, Bonita Bennett, Anna Riester, Nalini S. Shah, Giovanni Barbon, and Julie A Miller
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Male ,Endocrinology, Diabetes and Metabolism ,Adrenal Gland Neoplasms ,hemodynamics ,Infant, Newborn, Diseases ,Cohort Studies ,paraganglioma ,0302 clinical medicine ,Endocrinology ,Pregnancy ,030212 general & internal medicine ,resection ,Young adult ,Obstetrics ,Incidence ,Incidence (epidemiology) ,endocrine neoplasia type-2 ,case series ,mutations ,Pregnancy Outcome ,Vascular damage Radboud Institute for Molecular Life Sciences [Radboudumc 16] ,Prenatal Care ,Fear ,Middle Aged ,Multicenter study ,Fetal Diseases ,Prenatal Exposure Delayed Effects ,Cohort ,Female ,Pregnancy Complications, Neoplastic ,Cohort study ,Adult ,medicine.medical_specialty ,Adolescent ,030209 endocrinology & metabolism ,Pheochromocytoma ,Article ,Young Adult ,03 medical and health sciences ,Internal Medicine ,medicine ,Humans ,Retrospective Studies ,Literature review ,business.industry ,Infant, Newborn ,Retrospective cohort study ,Odds ratio ,medicine.disease ,business - Abstract
Contains fulltext : 245825.pdf (Publisher’s version ) (Closed access) BACKGROUND: Phaeochromocytoma or paraganglioma (collectively known as PPGL) in pregnant women can lead to severe complications and death due to associated catecholamine excess. We aimed to identify factors associated with maternal and fetal outcomes in women with PPGL during pregnancy. METHODS: We did a multicentre, retrospective study of patients with PPGL and pregnancy between Jan 1, 1980, and Dec 31, 2019, in the International Pheochromocytoma and Pregnancy Registry and a systematic review of studies published between Jan 1, 2005, and Dec 27, 2019 reporting on at least five cases. The inclusion criteria were pregnancy after 1980 and PPGL before or during pregnancy or within 12 months post partum. Eligible patients from the retrospective study and systematic review were included in the analysis. Outcomes of interest were maternal or fetal death and maternal severe cardiovascular complications of catecholamine excess. Potential variables associated with these outcomes were evaluated by logistic regression. FINDINGS: The systematic review identified seven studies (reporting on 63 pregnancies in 55 patients) that met the eligibility criteria and were of adequate quality. A further 197 pregnancies in 186 patients were identified in the International Pheochromocytoma and Pregnancy Registry. After excluding 11 pregnancies due to potential overlap, the final cohort included 249 pregnancies in 232 patients with PPGL. The diagnosis of PPGL was made before pregnancy in 37 (15%) pregnancies, during pregnancy in 134 (54%), and after delivery in 78 (31%). Of 144 patients evaluated for genetic predisposition for phaeochromocytoma, 95 (66%) were positive. Unrecognised PPGL during pregnancy (odds ratio 27·0; 95% CI 3·5-3473·1), abdominal or pelvic tumour location (11·3; 1·5-1440·5), and catecholamine excess at least ten-times the upper limit of the normal range (4·7; 1·8-13·8) were associated with adverse outcomes. For patients diagnosed during pregnancy, α-adrenergic blockade therapy was associated with fewer adverse outcomes (3·6; 1·1-13·2 for no α-adrenergic blockade vs α-adrenergic blockade), whereas surgery during pregnancy was not associated with better outcomes (0·9; 0·3-3·9 for no surgery vs surgery). INTERPRETATION: Unrecognised and untreated PPGL was associated with a substantially higher risk of either maternal or fetal complications. Appropriate case detection and counselling for premenopausal women at risk for PPGL could prevent adverse pregnancy-related outcomes. FUNDING: US National Institutes of Health.
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- 2021
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