Adverse pregnancy outcomes and effect of treatment in Wilson disease during pregnancy: Systematic review and meta‐analysis

Author:

Brown Ashley N.1ORCID,Lange M. Marcia12ORCID,Aliasi‐Sinai Lital3,Zhang Xiaotao4,Kogan Sasha5,Martin Lily6,Kushner Tatyana4

Affiliation:

1. Department of Medicine Icahn School of Medicine at Mount Sinai New York New York USA

2. Department of Medicine Brigham and Women's Hospital Boston Massachusetts USA

3. Department of Medicine Icahn School of Medicine at Mount Sinai Morningside/West New York USA

4. Division of Liver Diseases Department of Medicine, Icahn School of Medicine at Mount Sinai New York New York USA

5. Jacob's School of Medicine at the University of Buffalo Buffalo New York USA

6. Levy Library Icahn School of Medicine at Mount Sinai New York New York USA

Abstract

AbstractBackground and AimsWilson disease (WD) is a rare disorder of copper metabolism, leading to liver and neurological disease. Existing literature on WD in pregnancy is scarce, limiting preconception and obstetrical counselling. In this systematic review with meta‐analysis, we determine the prevalence of various adverse pregnancy and neonatal outcomes in WD, as well as evaluate the impact of WD treatment on these outcomes.MethodsScopus, MEDLINE and EMBASE were searched until 12 May 2023, for studies of pregnant individuals with WD and at least one pregnancy or neonatal outcome of interest. Meta‐analysis of single proportions was conducted to pool prevalence data for each outcome. Outcome rates were compared between treated and untreated groups in a meta‐analysis of dichotomous events.ResultsSixteen studies, published from 1975 to 2022, were included in the systematic review. Thirty‐seven percent of pregnancies reported at least one adverse pregnancy outcome. Spontaneous abortions (20%), liver diseases of pregnancy (4.5%) and preterm births (2%) were the most frequent adverse pregnancy outcomes in patients with WD. The prevalence of spontaneous abortions was significantly lower in pregnant individuals with WD who received treatment during pregnancy (OR: .47, 95% CI: 35%–63%). The prevalence of any adverse pregnancy outcome was also significantly lower with treatment (OR: .53, 95% CI: .37–.76), which appears to be mostly driven by the reduction of spontaneous abortions.ConclusionsThere is low to moderate quality evidence to suggest that preconception and obstetrical counselling for patients with WD should include a discussion on the potentially high frequency of adverse pregnancy outcomes in this population, as well as the importance of continuing WD treatment during pregnancy to ensure satisfactory pregnancy course and potentially minimize the risk of spontaneous abortions.

Publisher

Wiley

Reference49 articles.

1. Pregnancy in Wilson's disease: Management and outcome

2. The Prevalence of Wilson’s Disease: An Update

3. A multidisciplinary approach to the diagnosis and management of Wilson disease: 2022 practice guidance on Wilson disease from the American Association for the Study of Liver Diseases;Schilsky ML;Hepatology,2022

4. Declaración PRISMA 2020: una guía actualizada para la publicación de revisiones sistemáticas

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