Pregnancy outcomes in women with neurofibromatosis 1: a Danish population-based cohort study.

Autor:	Kenborg L; Childhood Cancer Research Group, Danish Cancer Society Research Center, Copenhagen, Denmark kenborg@cancer.dk., Boschini C; Statistics and Data Analysis, Danish Cancer Society Research Center, Copenhagen, Denmark., Bidstrup PE; Psychological Aspects of Cancer, Danish Cancer Society Research Center, Copenhagen, Denmark., Dalton SO; Survivorship and Inequality in Cancer, Danish Cancer Society Research Center, Copenhagen, Denmark., Doser K; Childhood Cancer Research Group, Danish Cancer Society Research Center, Copenhagen, Denmark., Nielsen TT; Childhood Cancer Research Group, Danish Cancer Society Research Center, Copenhagen, Denmark., Krøyer A; Childhood Cancer Research Group, Danish Cancer Society Research Center, Copenhagen, Denmark., Johansen C; Psychological Aspects of Cancer, Danish Cancer Society Research Center, Copenhagen, Denmark.; Department of Oncology, Rigshospitalet, Copenhagen University Hospital, Copenhagen, Denmark., Frederiksen K; Statistics and Data Analysis, Danish Cancer Society Research Center, Copenhagen, Denmark., Sørensen SA; Department of Neurogenetics, Institute of Cellular and Molecular Medicine, University of Copenhagen, Copenhagen, Denmark., Hove H; Department of Clinical Genetics, Rigshospitalet, Copenhagen University Hospital, Copenhagen, Denmark., Østergaard JR; Center for Rare Diseases, Department of Pediatrics and Adolescent Medicine, Aarhus University Hospital, Aarhus, Denmark., Mulvihill JJ; Department of Pediatrics, University of Oklahoma, Oklahoma City, OK, USA., Winther JF; Childhood Cancer Research Group, Danish Cancer Society Research Center, Copenhagen, Denmark.; Department of Clinical Medicine, Faculty of Health, Aarhus University, Aarhus, Denmark.
Jazyk:	angličtina
Zdroj:	Journal of medical genetics [J Med Genet] 2022 Mar; Vol. 59 (3), pp. 237-242. Date of Electronic Publication: 2021 Jan 25.
DOI:	10.1136/jmedgenet-2020-107201
Abstrakt:	Background: The probability of a pregnancy, live birth, stillbirth and abortion has never been assessed in women with neurofibromatosis 1 (NF1) in a large population-based study. Methods: We included 1006 women (15-49 years) registered with NF1 in the Danish National Patient Registry or followed in two national Centers for Rare Diseases and 10 020 women from the Danish population. Information on pregnancy outcomes was ascertained from health registries. Cumulative incidence, mean cumulative count, hazard ratios (HRs) and proportion ratios (PRs) with 95% CIs were calculated. Results: The cumulative incidence of a first pregnancy at age 50 years was slightly lower in women with NF1 (74%; 95% CI 70 to 77) than in population comparisons (78%; 95% CI 77 to 79). When all pregnancies were included, two pregnancies were expected per woman at age of 50 years, irrespective of a NF1 diagnosis. The hazard of a pregnancy did not differ between women with NF1 (HR 1.03; 95% CI 0.95 to 1.11) and the comparisons after adjustment for somatic and psychiatric disease. The proportion of pregnancies that resulted in a live birth was 63% (783/1252) among women NF1 and 68% (8432/12 465) among the comparisons, yielding a PR of 0.95 (95% CI 0.90 to 1.00). The proportions of stillbirths (PR 2.83; 95% CI 1.63 to 4.93) and spontaneous abortions (PR 1.40; 95% CI 1.09 to 1.79) were increased in women with NF1. Conclusions: A similar hazard for pregnancy was observed for women with NF1 and population comparisons after adjustment for potential medical consequences of NF1. However, women with NF1 experienced more spontaneous abortions and stillbirths. Competing Interests: Competing interests: None declared. (© Author(s) (or their employer(s)) 2022. No commercial re-use. See rights and permissions. Published by BMJ.)
Databáze:	MEDLINE
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