Current management and outcome of pregnancies in women with adrenal insufficiency: experience from a multicenter survey
Open accessAbstract
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: Multicenter survey on current clinical approaches in managing AI during pregnancy. Design: Retrospective anonymized data collection from 19 international centers from 2013 to 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 83% (97/117) of pregnancies. Glucocorticoid dosage was increased at any time during pregnancy in 73/128 (57%) of cases. In these cases, the difference in 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 third trimester). Overall, an adrenal crisis was reported in 9/128 (7%) pregnancies. Cesarean section was the most frequent mode of delivery at 58% (69/118). Fetal complications were reported in 3/120 (3%) and minor maternal complications in 15/120 (13%) 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.
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Most (53-68%) women with NC CAH conceive spontaneously without any treatment.
“Most (53-68%) women with NC CAH conceive spontaneously without any treatment (650,666).”
From Congenital Adrenal Hyperplasia—Current Insights in Pathophysiology, Diagnostics, and Management (2022) · cites this paper as [666]
The miscarriage rate in women with NC CAH is substantially higher (25%) than in the general population (6%) in some but not all studies.
“The course of pregnancy is usually uneventful; however, the miscarriage rate in women with NC CAH is substantially higher (25%) than in the general population (6%) in some (650,666) but not all (667) studies.”
From Congenital Adrenal Hyperplasia—Current Insights in Pathophysiology, Diagnostics, and Management (2022) · cites this paper as [666]
The course of pregnancy is usually uneventful in women with NC CAH.
“The course of pregnancy is usually uneventful; however, the miscarriage rate in women with NC CAH is substantially higher (25%) than in the general population (6%) in some (650,666) but not all (667) studies.”
From Congenital Adrenal Hyperplasia—Current Insights in Pathophysiology, Diagnostics, and Management (2022) · cites this paper as [666]
The miscarriage rate can be reduced to normal in women treated with low to moderate doses of HC, prednisolone or prednisone prior to and during pregnancy.
“The miscarriage rate can be reduced to normal in women treated with low to moderate doses of HC (650), prednisolone or prednisone (666) prior to and during pregnancy.”
From Congenital Adrenal Hyperplasia—Current Insights in Pathophysiology, Diagnostics, and Management (2022) · cites this paper as [666]
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- Authors
- Bothou, C., Anand, G., Li, D., Kienitz, T., Seejore, K., Simeoli, C., Ebbehoj, A., Ward, E. G., Paragliola, R. M., Ferrigno, R., Badenhoop, K., Bensing, S., Oksnes, M., Esposito, D., Bergthorsdottir, R., Drake, W., Wahlberg, J., Reisch, N., Hahner, S., Pearce, S., Trainer, P., Etzrodt-Walter, G., Thalmann, S. P., Sævik, Å. B., Husebye, E., Isidori, A. M., Falhammar, H., Meyer, G., Corsello, S. M., Pivonello, R., Murray, R., Bancos, I., Quinkler, M., & Beuschlein, F.
- Affiliations
- University Hospital Zurich (CH)
- Mayo Clinic (US)
- Endocrinology in Charlottenburg, Berlin, Germany
- Leeds Teaching Hospitals NHS Trust (GB)
- University of Naples Federico II (IT)
- Department of Clinical Medicine, Department of Endocrinology and Diabetes, Aarhus University, Aarhus, Denmark
- Università Cattolica del Sacro Cuore (IT)
- University Hospital Frankfurt (DE)
- Karolinska University Hospital (SE)
- Department of Internal Medicine and Clinical Nutrition, Institute of Medicine, Sahlgrenska Academy, University of Gothenburg, Gothenburg, Sweden
- Sahlgrenska University Hospital (SE)
- St Bartholomew's Hospital (GB)
- Linköping University (SE)
- LMU Klinikum (DE)
- University of Würzburg (DE)
- Newcastle University (GB)
- The Christie NHS Foundation Trust (GB)
- Praxis Dr. Etzrodt-Walter Endokrinologiezentrum Ulm, Ulm, Germany
- Ärztezentrum Sihlcity, Zurich, Switzerland
- University of Bergen (NO)
- Sapienza University of Rome (IT)
- Corresponding author
- Beuschlein, Felix
- ORCID
- Bothou, Christina
- Anand, Gurpreet
- Li, Dingfeng
- Kienitz, Tina
- Seejore, Khyatisha
- Simeoli, Chiara
- Paragliola, Rosa Maria
- Ferrigno, Rosario
- Bensing, Sophie
- Bergthorsdottir, Ragnhildur
- Wahlberg, Jeanette
- Reisch, Nicole
- Hahner, Stefanie
- Pearce, Simon
- Trainer, Peter
- Etzrodt-Walter, Gwendolin
- Thalmann, Sébastien P
- Sævik, Åse B
- Husebye, Eystein
- Isidori, Andrea M
- Falhammar, Henrik
- Meyer, Gesine
- Corsello, Salvatore M
- Pivonello, Rosario
- Murray, Robert
- Bancos, Irina
- Quinkler, Marcus
- Beuschlein, Felix
- Venue
- The Journal of Clinical Endocrinology & Metabolism
- Publisher
- The Endocrine Society
- Published
- 2020-05-19
- Volume
- 105
- Issue
- 8
- Pages
- e2853-e2863
- ISSN
- 0021-972X (ISSN-L)
- 1945-7197
- PMC
- PMC7320831
- OpenAlex
- W3027092105
- Cited by
- 44
- References
- 29
- Citations by year
- 2026: 5 · 2025: 6 · 2024: 3 · 2023: 8 · 2022: 10 · 2021: 11 · 2020: 1
- FWCI
- 5.66
- Type
- journal article
- Language
- en
- Open access
- bronze
- doi.org
- Licence
- academic.oup.com/journals/pages/open_access/funder_policies/chorus/standard_publication_model (vor)
- Locations
- The Journal of Clinical Endocrinology & Metabolism (publishedVersion)
- PubMed Central (publishedVersion)
- PubMed (publishedVersion)
- Zurich Open Repository and Archive (University of Zurich) (acceptedVersion, other-oa)
- IRIS Research product catalog (Sapienza University of Rome) (submittedVersion, other-oa)
- Zurich Open Repository and Archive (University of Zurich) (submittedVersion, other-oa)
- Zurich Open Repository and Archive (University of Zurich)
- Funders
- Deutsche Forschungsgemeinschaft
- James A. Ruppe Career Development Award in Endocrinology
- National Institute of Diabetes and Digestive and Kidney Diseases
- National Institutes of Health (K23DK121888)
- Topics
- Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Adrenal Hormones and Disorders
- Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Thyroid Disorders and Treatments
- Health Sciences › Medicine › Endocrinology, Diabetes and Metabolism › Hormonal Regulation and Hypertension
- Keywords
- adrenal insufficiency, pregnancy, Addison's disease, secondary adrenal insufficiency, congenital adrenal hyperplasia, bilateral adrenalectomy, cortisol, fludrocortisone, mineralocorticoid therapy, adrenal crisis, cesarean section, maternal and fetal outcomes
- MeSH terms
- Adrenal Insufficiency, Adult, Cesarean Section, Dose-Response Relationship, Drug, Female, Fludrocortisone, Glucocorticoids, Humans, Hydrocortisone, Mineralocorticoids, Pregnancy, Pregnancy Complications, Pregnancy Outcome, Retrospective Studies, Severity of Illness Index, Treatment Outcome, Hormone Replacement Therapy
About these checks
Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.
Statistics and arithmetic are recomputed, not judged.
Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.
What counts as a claim ›How we measure confidence ›How we check this ›
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