TY - JOUR

T1 - Psychiatric disorders in individuals with neurofibromatosis 1 in Denmark

T2 - A nationwide register-based cohort study

AU - Kenborg, Line

AU - Andersen, Elisabeth W.

AU - Duun-Henriksen, Anne Katrine

AU - Jepsen, Jens R.M.

AU - Doser, Karoline

AU - Dalton, Susanne O.

AU - Bidstrup, Pernille E.

AU - Krøyer, Anja

AU - Frederiksen, Line E.

AU - Johansen, Christoffer

AU - Østergaard, John R.

AU - Hove, Hanne

AU - Sørensen, Sven Asger

AU - Riccardi, Vincent M.

AU - Mulvihill, John J.

AU - Winther, Jeanette F.

N1 - Publisher Copyright: © 2021 Wiley Periodicals LLC.

PY - 2021

Y1 - 2021

N2 - The aim of this study was to assess the risks of psychiatric disorders in a large cohort of 905 individuals with NF1 and 7614 population comparisons matched on sex and year of birth. The cohort was linked to the Danish Psychiatric Central Research Register to ascertain information on hospital contacts for psychiatric disorders based on the International Classification of Diseases version 8 and 10. The hazard ratio (HR) for a first psychiatric hospital contact was higher in girls (4.19, 95% confidence interval [CI] 1.81–9.69) and boys with NF1 (5.02, 95% CI 3.27–7.69) <7 years of age than in the population comparisons. Both sexes had increased HRs for developmental disorders, including attention deficit/hyperactivity disorders, autism spectrum disorders, and intellectual disabilities in childhood. Females with NF1 had also increased HRs for unipolar depression, other emotional and behavioral disorders, and severe stress reaction and adjustment disorders in early adulthood. The HRs for psychoses, schizophrenia, bipolar disorders, and substance abuse were similar in individuals with NF1 and the population comparisons. Finally, the cumulative incidence of a first hospital contact due to any psychiatric disorder by age 30 years was 35% (95% CI 29–41) in females and 28% (95% CI 19–37) in males with NF1. Thus, screening for psychiatric disorders may be important for early diagnosis and facilitation of appropriate and effective treatment in individuals with NF1.

AB - The aim of this study was to assess the risks of psychiatric disorders in a large cohort of 905 individuals with NF1 and 7614 population comparisons matched on sex and year of birth. The cohort was linked to the Danish Psychiatric Central Research Register to ascertain information on hospital contacts for psychiatric disorders based on the International Classification of Diseases version 8 and 10. The hazard ratio (HR) for a first psychiatric hospital contact was higher in girls (4.19, 95% confidence interval [CI] 1.81–9.69) and boys with NF1 (5.02, 95% CI 3.27–7.69) <7 years of age than in the population comparisons. Both sexes had increased HRs for developmental disorders, including attention deficit/hyperactivity disorders, autism spectrum disorders, and intellectual disabilities in childhood. Females with NF1 had also increased HRs for unipolar depression, other emotional and behavioral disorders, and severe stress reaction and adjustment disorders in early adulthood. The HRs for psychoses, schizophrenia, bipolar disorders, and substance abuse were similar in individuals with NF1 and the population comparisons. Finally, the cumulative incidence of a first hospital contact due to any psychiatric disorder by age 30 years was 35% (95% CI 29–41) in females and 28% (95% CI 19–37) in males with NF1. Thus, screening for psychiatric disorders may be important for early diagnosis and facilitation of appropriate and effective treatment in individuals with NF1.

KW - cohort study

KW - neurofibromatosis 1

KW - population-based

KW - psychiatric disorders

U2 - 10.1002/ajmg.a.62436

DO - 10.1002/ajmg.a.62436

M3 - Journal article

C2 - 34327813

AN - SCOPUS:85111539113

VL - 185

SP - 3706

EP - 3716

JO - American Journal of Medical Genetics, Part A

JF - American Journal of Medical Genetics, Part A

SN - 1552-4825

IS - 12

ER -