Evidence map›Paper›PMID 34570215›Full record

ArticleThe Journal of clinical endocrinology and metabolism2022

Pregnancy Outcome in Women With APECED (APS-1): A Multicenter Study on 43 Females With 83 Pregnancies.

Saila Laakso, Elina Holopainen, Corrado Betterle, Viivi Saari, Elinor Vogt, Monica M Schmitt, Karen K Winer, Maria Kareva, Chiara Sabbadin, Eystein S Husebye and 3 more

Open access · hybridAbstract readMulticenter Study
PubMed Full text at PMC Publisher
In one paragraph

Article in The Journal of clinical endocrinology and metabolism, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
6citing papers in PubMed, 1 pooled it
1.6field-weighted citation impact, top 15% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

6 citing papers in PubMed, 1 synthesis or guideline pooled it, 16 citations in OpenAlex.

  1. Pooled it
  2. Review
  3. Article
  4. Approach to the Patient: Management of Parathyroid Diseases Across Pregnancy.The Journal of clinical endocrinology and metabolism · 2023
    Article
  5. Addison's Disease: Diagnosis and Management Strategies.International journal of general medicine · 2023
    Review
  6. Pregnancy Outcome in Poly-Autoimmune Disorders: The Case of APECED Syndrome.The Journal of clinical endocrinology and metabolism · 2022
    Article
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

13 authors at 5 institutions in 5 countries.

Saila LaaksoChildren's Hospital and Pediatric Research Center, University of Helsinki and Helsinki University Hospital, Helsinki, Finland.ORCID 0000-0002-5588-7855
Elina HolopainenResearch Program for Clinical and Molecular Metabolism, Faculty of Medicine, University of Helsinki, Helsinki, Finland.
Corrado BetterleEndocrine Unit, Department of Medicine (DIMED), University of Padua, Padua,Italy.
Viivi SaariChildren's Hospital and Pediatric Research Center, University of Helsinki and Helsinki University Hospital, Helsinki, Finland.
Elinor VogtDepartment of Clinical Science and K.G. Jebsen Center for Autoimmune Diseases, University of Bergen, Norway.
Monica M SchmittLaboratory of Clinical Immunology and Microbiology, National Institute of Allergy & Infectious Diseases (NIAID), National Institutes of Health (NIH), Bethesda, Maryland, USA.
Karen K WinerEunice Kennedy Shriver National Institutes of Child Health and Human Development (NICHD), National Institutes of Health, Bethesda, MD,USA.
Maria KarevaEndocrinology Research Centre, Moscow, Russia.
Chiara SabbadinEndocrine Unit, Department of Medicine (DIMED), University of Padua, Padua,Italy.
Eystein S HusebyeDepartment of Clinical Science and K.G. Jebsen Center for Autoimmune Diseases, University of Bergen, Norway.
Elizaveta OrlovaEndocrinology Research Centre, Moscow, Russia.
Michail S LionakisLaboratory of Clinical Immunology and Microbiology, National Institute of Allergy & Infectious Diseases (NIAID), National Institutes of Health (NIH), Bethesda, Maryland, USA.
Outi MäkitieChildren's Hospital and Pediatric Research Center, University of Helsinki and Helsinki University Hospital, Helsinki, Finland.ORCID 0000-0002-4547-001X
University of Helsinki · FINational Institutes of Health · USEndocrinology Research Center · RUHaukeland University Hospital · NOUniversity of Padua · IT

Funding

Immunopathogenesis of Fungal InfectionsZIAAI001175 · NIAID · NATIONAL INSTITUTE OF ALLERGY AND INFECTIOUS DISEASES · PI LIONAKIS, MICHAIL · 2012 to 2025
$23.9M
Intramural NIH HHS
6 · The paper itself

Abstract

contextAutoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy (APECED; also known as autoimmune polyendocrine syndrome type 1) has a severe, unpredictable course. Autoimmunity and disease components may affect fertility and predispose to maternal and fetal complications, but pregnancy outcomes remain unknown.

objectiveTo assess fetal and maternal outcomes and course of clinical APECED manifestations during pregnancy in women with APECED. DESIGN AND

settingA multicenter registry-based study including 5 national patient cohorts. PATIENTS: 321 females with APECED.

main outcome measureNumber of pregnancies, miscarriages, and deliveries.

resultsForty-three patients had altogether 83 pregnancies at median age of 27 years (range, 17-39). Sixty (72%) pregnancies led to a delivery, including 2 stillbirths (2.4%) and 5 (6.0%) preterm livebirths. Miscarriages, induced abortions, and ectopic pregnancies were observed in 14 (17%), 8 (10%), and 1 (1.2%) pregnancies, respectively. Ovum donation resulted in 5 (6.0%) pregnancies. High maternal age, premature ovarian insufficiency, primary adrenal insufficiency, or hypoparathyroidism did not associate with miscarriages. Women with livebirth had, on average, 4 APECED manifestations (range 0-10); 78% had hypoparathyroidism, and 36% had primary adrenal insufficiency. APECED manifestations remained mostly stable during pregnancy, but in 1 case, development of primary adrenal insufficiency led to adrenal crisis and stillbirth. Birth weights were normal in >80% and apart from 1 neonatal death of a preterm baby, no serious perinatal complications occurred.

conclusionsOutcome of pregnancy in women with APECED was generally favorable. However, APECED warrants careful maternal multidisciplinary follow-up from preconceptual care until puerperium.

Indexed as

StillbirthAbortion, SpontaneousAdolescentAdultAge FactorsFemaleHumansMaternal AgePolyendocrinopathies, AutoimmunePregnancyPremature BirthRegistriesRetrospective StudiesRisk FactorsYoung Adultautoimmunitydeliveryfertilitypremature ovarian insufficiency

Identifiers

PMID34570215
PMCPMC8764323
OpenAlexW3203885377

What OpenQuestion holds

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LicenceCC BY
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Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.