Current management and outcome of pregnancies in women with adrenal insufficiency: experience from a multi-center survey

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  • Christina Bothou, Klinik für Endokrinologie, Diabetologie und Klinische Ernährung, Universitätsspital Zürich, Zürich, Switzerland.
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  • Gurpreet Anand, Klinik für Endokrinologie, Diabetologie und Klinische Ernährung, Universitätsspital Zürich, Zürich, Switzerland.
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  • Dingfeng Li, Division of Endocrinology, Diabetes, Metabolism and Nutrition, Mayo Clinic, Rochester, Minnesota, USA.
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  • Tina Kienitz, Endocrinology in Charlottenburg, Berlin, Germany.
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  • Khyatisha Seejore, Department of Endocrinology, Leeds Teaching Hospitals NHS Trust, St James's University Hospital, Leeds, U.K.
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  • Chiara Simeoli, Dipartimento di Medicina Clinica e Chirurgia, Sezione di Endocrinologia, Università Federico II di Napoli, Naples, Italy.
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  • Andreas Ebbehoj
  • Emma G Ward, Department of Endocrinology, Leeds Teaching Hospitals NHS Trust, St James's University Hospital, Leeds, U.K.
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  • Rosa Maria Paragliola, Unit of Endocrinology, Università Cattolica del Sacro Cuore - Fondazione Policlinico Universitario Agostino Gemelli IRCCS, Rome, Italy.
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  • Rosario Ferrigno, Dipartimento di Medicina Clinica e Chirurgia, Sezione di Endocrinologia, Università Federico II di Napoli, Naples, Italy.
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  • Klaus Badenhoop, Department of Internal Medicine I, Division of Endocrinology, Diabetes and Metabolism, University Hospital, Frankfurt, Germany.
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  • Sophie Bensing, Department of Molecular Medicine and Surgery, Karolinska Institutet and Department of Endocrinology, Inflammation and Infection Theme, Karolinska University Hospital, Stockholm, Sweden.
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  • Marianne Oksnes, Department of Clinical Science and K.G. Jebsen Center for Autoimmune Disorders, University of Bergen, Jonas Liesvei 65, N-5021 Bergen, Norway.
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  • Daniela Esposito, Department of Endocrinology, Sahlgrenska, University Hospital, 413 45 Gothenburg, Sweden.
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  • Ragnhildur Bergthorsdottir, Department of Endocrinology, Sahlgrenska, University Hospital, 413 45 Gothenburg, Sweden.
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  • William Drake, Department of Endocrinology, St Bartholomew's Hospital, London EC1A 7BE, UK.
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  • Jeanette Wahlberg, Department of Endocrinology, and Department of Health, Medicine and Caring Sciences, Linköping University, Linköping, Sweden.
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  • Nicole Reisch, Department of Endocrinology, Medizinische Klinik und Poliklinik IV, Klinikum der Universität München, Munich, Germany.
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  • Stefanie Hahner, Department of Internal Medicine I, Endocrinology and Diabetes Unit, University Hospital of Würzburg, University of Würzburg, Germany.
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  • Simon Pearce, Translational and Clinical Research Institute, Newcastle University, Newcastle upon Tyne, U.K.
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  • Peter Trainer, The Christie NHS Foundation, MAHSC, Wilmslow Road, Manchester M20 4BX, U.K.
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  • Gwendolin Etzrodt-Walter, Praxis Dr. Etzrodt-Walter Endokrinologiezentrum Ulm, Ulm, Germany.
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  • Sébastien P Thalmann, Ärztezentrum Sihlcity, Zurich, Switzerland.
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  • Åse B Sævik, Department of Clinical Science and K.G. Jebsen Center for Autoimmune Disorders, University of Bergen, Jonas Liesvei 65, N-5021 Bergen, Norway.
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  • Eystein Husebye, Department of Clinical Science and K.G. Jebsen Center for Autoimmune Disorders, University of Bergen, Jonas Liesvei 65, N-5021 Bergen, Norway.
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  • Andrea M Isidori, Department of Experimental Medicine, Sapienza University of Rome, Piazzale Aldo Moro 5, 00185, Roma, Italy.
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  • Henrik Falhammar, Department of Molecular Medicine and Surgery, Karolinska Institutet and Department of Endocrinology, Inflammation and Infection Theme, Karolinska University Hospital, Stockholm, Sweden.
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  • Gesine Meyer, Department of Internal Medicine I, Division of Endocrinology, Diabetes and Metabolism, University Hospital, Frankfurt, Germany.
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  • Salvatore M Corsello, Unit of Endocrinology, Università Cattolica del Sacro Cuore - Fondazione Policlinico Universitario Agostino Gemelli IRCCS, Rome, Italy.
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  • Rosario Pivonello, Dipartimento di Medicina Clinica e Chirurgia, Sezione di Endocrinologia, Università Federico II di Napoli, Naples, Italy.
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  • Robert Murray, Department of Endocrinology, Leeds Teaching Hospitals NHS Trust, St James's University Hospital, Leeds, U.K.
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  • Irina Bancos, Division of Endocrinology, Diabetes, Metabolism and Nutrition, Mayo Clinic, Rochester, Minnesota, USA.
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  • Marcus Quinkler, Endocrinology in Charlottenburg, Berlin, Germany.
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  • Felix Beuschlein, Department of Endocrinology, Medizinische Klinik und Poliklinik IV, Klinikum der Universität München, Munich, Germany.

CONTEXT: Appropriate management of adrenal insufficiency (AI) in pregnancy can be challenging due to the rarity of the disease and lack of evidence-based recommendations to guide glucocorticoid- and mineralocorticoid dosage adjustment.

OBJECTIVE: Multi-center survey on current clinical approaches in managing AI during pregnancy.

DESIGN: Retrospective anonymized data collection from 19 international centers from 2013-2019.

SETTING AND PATIENTS: 128 pregnancies in 113 women with different causes of AI: Addison disease (44%), secondary AI (25%), congenital adrenal hyperplasia (25%) and acquired AI due to bilateral adrenalectomy (6%).

RESULTS: Hydrocortisone (HC) was the most commonly used glucocorticoid in 82.9% (97/117) of pregnancies. Glucocorticoid dosage was increased at any time during pregnancy in 73/128 (57%) of cases. In these cases, difference of the daily dose of HC equivalent between baseline and the third trimester was 8.6±5.4 [range: 1, 30] mg. Fludrocortisone dosage was increased in fewer cases (7/54 during the first trimester, 9/64 during the second trimester and 9/62 cases during the last trimester). Overall, an adrenal crisis was reported in 9/128 (7%) pregnancies. Caesarian section was the most frequent mode of delivery at 58% (69/118). Fetal complications were reported in 3/120 (2.5%) and minor maternal complications in 15/120 (12.5%) pregnancies without fatal outcomes.

CONCLUSIONS: This survey confirms good maternal and fetal outcome in women with AI managed in specialized endocrine centers. An emphasis on careful endocrine follow up and repeated patient education is likely to have reduced the risk of adrenal crisis and resulted in positive outcomes.

Original languageEnglish
JournalThe Journal of clinical endocrinology and metabolism
Volume105
Issue8
ISSN0021-972X
DOIs
Publication statusPublished - 2020

    Research areas

  • Adrenal Insufficiency, Addison Disease, Pregnancy outcomes, Pregnancy Complications

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