PMID- 33495305
OWN - NLM
STAT- MEDLINE
DCOM- 20220509
LR  - 20220509
IS  - 1468-6244 (Electronic)
IS  - 0022-2593 (Linking)
VI  - 59
IP  - 3
DP  - 2022 Mar
TI  - Pregnancy outcomes in women with neurofibromatosis 1: a Danish population-based 
      cohort study.
PG  - 237-242
LID - 10.1136/jmedgenet-2020-107201 [doi]
AB  - 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.
CI  - (c) Author(s) (or their employer(s)) 2022. No commercial re-use. See rights and 
      permissions. Published by BMJ.
FAU - Kenborg, Line
AU  - Kenborg L
AUID- ORCID: 0000-0002-3584-6570
AD  - Childhood Cancer Research Group, Danish Cancer Society Research Center, 
      Copenhagen, Denmark kenborg@cancer.dk.
FAU - Boschini, Cristina
AU  - Boschini C
AD  - Statistics and Data Analysis, Danish Cancer Society Research Center, Copenhagen, 
      Denmark.
FAU - Bidstrup, Pernille E
AU  - Bidstrup PE
AD  - Psychological Aspects of Cancer, Danish Cancer Society Research Center, 
      Copenhagen, Denmark.
FAU - Dalton, Susanne O
AU  - Dalton SO
AD  - Survivorship and Inequality in Cancer, Danish Cancer Society Research Center, 
      Copenhagen, Denmark.
FAU - Doser, Karoline
AU  - Doser K
AD  - Childhood Cancer Research Group, Danish Cancer Society Research Center, 
      Copenhagen, Denmark.
FAU - Nielsen, Thomas T
AU  - Nielsen TT
AD  - Childhood Cancer Research Group, Danish Cancer Society Research Center, 
      Copenhagen, Denmark.
FAU - Kroyer, Anja
AU  - Kroyer A
AD  - Childhood Cancer Research Group, Danish Cancer Society Research Center, 
      Copenhagen, Denmark.
FAU - Johansen, Christoffer
AU  - Johansen C
AD  - Psychological Aspects of Cancer, Danish Cancer Society Research Center, 
      Copenhagen, Denmark.
AD  - Department of Oncology, Rigshospitalet, Copenhagen University Hospital, 
      Copenhagen, Denmark.
FAU - Frederiksen, Kirsten
AU  - Frederiksen K
AD  - Statistics and Data Analysis, Danish Cancer Society Research Center, Copenhagen, 
      Denmark.
FAU - Sorensen, Sven Asger
AU  - Sorensen SA
AD  - Department of Neurogenetics, Institute of Cellular and Molecular Medicine, 
      University of Copenhagen, Copenhagen, Denmark.
FAU - Hove, Hanne
AU  - Hove H
AD  - Department of Clinical Genetics, Rigshospitalet, Copenhagen University Hospital, 
      Copenhagen, Denmark.
FAU - Ostergaard, John R
AU  - Ostergaard JR
AD  - Center for Rare Diseases, Department of Pediatrics and Adolescent Medicine, 
      Aarhus University Hospital, Aarhus, Denmark.
FAU - Mulvihill, John J
AU  - Mulvihill JJ
AD  - Department of Pediatrics, University of Oklahoma, Oklahoma City, OK, USA.
FAU - Winther, Jeanette F
AU  - Winther JF
AD  - Childhood Cancer Research Group, Danish Cancer Society Research Center, 
      Copenhagen, Denmark.
AD  - Department of Clinical Medicine, Faculty of Health, Aarhus University, Aarhus, 
      Denmark.
LA  - eng
PT  - Journal Article
PT  - Research Support, U.S. Gov't, Non-P.H.S.
DEP - 20210125
PL  - England
TA  - J Med Genet
JT  - Journal of medical genetics
JID - 2985087R
SB  - IM
MH  - *Abortion, Spontaneous/epidemiology
MH  - Cohort Studies
MH  - Denmark/epidemiology
MH  - Female
MH  - Humans
MH  - Male
MH  - Middle Aged
MH  - *Neurofibromatosis 1/complications/epidemiology/genetics
MH  - Pregnancy
MH  - Pregnancy Outcome
MH  - Registries
MH  - Stillbirth/epidemiology
OTO - NOTNLM
OT  - genetic fitness
OT  - genetics
OT  - obstetrics
OT  - population
COIS- Competing interests: None declared.
EDAT- 2021/01/27 06:00
MHDA- 2022/05/10 06:00
CRDT- 2021/01/26 05:37
PHST- 2020/05/21 00:00 [received]
PHST- 2020/10/21 00:00 [revised]
PHST- 2020/12/21 00:00 [accepted]
PHST- 2021/01/27 06:00 [pubmed]
PHST- 2022/05/10 06:00 [medline]
PHST- 2021/01/26 05:37 [entrez]
AID - jmedgenet-2020-107201 [pii]
AID - 10.1136/jmedgenet-2020-107201 [doi]
PST - ppublish
SO  - J Med Genet. 2022 Mar;59(3):237-242. doi: 10.1136/jmedgenet-2020-107201. Epub 
      2021 Jan 25.