Harvard
Doser, K, Andersen, EW, Kenborg, L, Dalton, SO, Jepsen, JRM, Krøyer, A, Østergaard, J
, Hove, H, Sørensen, SA
, Johansen, C, Mulvihill, J, Winther, JF & Bidstrup, PE 2020, '
Clinical characteristics and quality of life, depression, and anxiety in adults with neurofibromatosis type 1: A nationwide study',
American Journal of Medical Genetics. Part A, vol. 182, no. 7, pp. 1704-1715.
https://doi.org/10.1002/ajmg.a.61627
APA
Doser, K., Andersen, E. W., Kenborg, L., Dalton, S. O., Jepsen, J. R. M., Krøyer, A., Østergaard, J.
, Hove, H., Sørensen, S. A.
, Johansen, C., Mulvihill, J., Winther, J. F., & Bidstrup, P. E. (2020).
Clinical characteristics and quality of life, depression, and anxiety in adults with neurofibromatosis type 1: A nationwide study.
American Journal of Medical Genetics. Part A,
182(7), 1704-1715.
https://doi.org/10.1002/ajmg.a.61627
CBE
Doser K, Andersen EW, Kenborg L, Dalton SO, Jepsen JRM, Krøyer A, Østergaard J
, Hove H, Sørensen SA
, Johansen C, Mulvihill J, Winther JF, Bidstrup PE. 2020.
Clinical characteristics and quality of life, depression, and anxiety in adults with neurofibromatosis type 1: A nationwide study.
American Journal of Medical Genetics. Part A. 182(7):1704-1715.
https://doi.org/10.1002/ajmg.a.61627
MLA
Vancouver
Author
Doser, Karoline ; Andersen, Elisabeth Wreford ; Kenborg, Line ; Dalton, Susanne Oksbjerg ; Jepsen, Jens Richardt Møllegaard ; Krøyer, Anja ; Østergaard, John
; Hove, Hanne ; Sørensen, Sven Asger
; Johansen, Christoffer ; Mulvihill, John ; Winther, Jeanette Falck ; Bidstrup, Pernille Envold. /
Clinical characteristics and quality of life, depression, and anxiety in adults with neurofibromatosis type 1 : A nationwide study. In:
American Journal of Medical Genetics. Part A. 2020 ; Vol. 182, No. 7. pp. 1704-1715.
Bibtex
@article{e381d53450be4acf889370928db875c8,
title = "Clinical characteristics and quality of life, depression, and anxiety in adults with neurofibromatosis type 1: A nationwide study",
abstract = "Neurofibromatosis type 1 (NF1) is a genetic condition characterized by numerous somatic manifestations. The psychosocial burden in adults has rarely been studied. We examined the prevalence of self-reported impairment of quality of life (QoL), symptoms of anxiety and depression and need for support, associated with disease severity and visibility. We conducted a nationwide cross-sectional study of all 467 adults with NF1 diagnosed between 1977 and 2016 at one of the two national centers for rare diseases in Denmark. A total of 244 (56% response rate) completed a questionnaire that included standard measures of QoL, symptoms of depression and anxiety, indicators of disease-related severity, visibility, and need for professional support. Associations between disease severity and visibility and psychosocial burden were analyzed in descriptive and multivariate models. We observed impaired QoL (mean = 81.3; 95% CI, 76.2; 86.4); 19% reported symptoms of depression (mean = 5.7; SD = 5.4), and 15% reported anxiety (mean = 5.1; SD = 5.2) at a clinical level. Adults with NF1 also reported requiring professional support for physical, psychological, and work-related problems. Disease severity and (partly) visibility were significantly (p < .0001) associated with psychosocial well-being and a requirement for support. This study provides new understanding of the factors associated with impaired QoL, indicating that follow-up care should be optimized into adult life.",
keywords = "adults, nationwide, need for support, neurofibromatosis type 1, population-based, psychosocial burden",
author = "Karoline Doser and Andersen, {Elisabeth Wreford} and Line Kenborg and Dalton, {Susanne Oksbjerg} and Jepsen, {Jens Richardt M{\o}llegaard} and Anja Kr{\o}yer and John {\O}stergaard and Hanne Hove and S{\o}rensen, {Sven Asger} and Christoffer Johansen and John Mulvihill and Winther, {Jeanette Falck} and Bidstrup, {Pernille Envold}",
note = "{\textcopyright} 2020 Wiley Periodicals, Inc.",
year = "2020",
month = jul,
doi = "10.1002/ajmg.a.61627",
language = "English",
volume = "182",
pages = "1704--1715",
journal = "American Journal of Medical Genetics, Part A",
issn = "1552-4825",
publisher = "JohnWiley & Sons, Inc",
number = "7",
}
RIS
TY - JOUR
T1 - Clinical characteristics and quality of life, depression, and anxiety in adults with neurofibromatosis type 1
T2 - A nationwide study
AU - Doser, Karoline
AU - Andersen, Elisabeth Wreford
AU - Kenborg, Line
AU - Dalton, Susanne Oksbjerg
AU - Jepsen, Jens Richardt Møllegaard
AU - Krøyer, Anja
AU - Østergaard, John
AU - Hove, Hanne
AU - Sørensen, Sven Asger
AU - Johansen, Christoffer
AU - Mulvihill, John
AU - Winther, Jeanette Falck
AU - Bidstrup, Pernille Envold
N1 - © 2020 Wiley Periodicals, Inc.
PY - 2020/7
Y1 - 2020/7
N2 - Neurofibromatosis type 1 (NF1) is a genetic condition characterized by numerous somatic manifestations. The psychosocial burden in adults has rarely been studied. We examined the prevalence of self-reported impairment of quality of life (QoL), symptoms of anxiety and depression and need for support, associated with disease severity and visibility. We conducted a nationwide cross-sectional study of all 467 adults with NF1 diagnosed between 1977 and 2016 at one of the two national centers for rare diseases in Denmark. A total of 244 (56% response rate) completed a questionnaire that included standard measures of QoL, symptoms of depression and anxiety, indicators of disease-related severity, visibility, and need for professional support. Associations between disease severity and visibility and psychosocial burden were analyzed in descriptive and multivariate models. We observed impaired QoL (mean = 81.3; 95% CI, 76.2; 86.4); 19% reported symptoms of depression (mean = 5.7; SD = 5.4), and 15% reported anxiety (mean = 5.1; SD = 5.2) at a clinical level. Adults with NF1 also reported requiring professional support for physical, psychological, and work-related problems. Disease severity and (partly) visibility were significantly (p < .0001) associated with psychosocial well-being and a requirement for support. This study provides new understanding of the factors associated with impaired QoL, indicating that follow-up care should be optimized into adult life.
AB - Neurofibromatosis type 1 (NF1) is a genetic condition characterized by numerous somatic manifestations. The psychosocial burden in adults has rarely been studied. We examined the prevalence of self-reported impairment of quality of life (QoL), symptoms of anxiety and depression and need for support, associated with disease severity and visibility. We conducted a nationwide cross-sectional study of all 467 adults with NF1 diagnosed between 1977 and 2016 at one of the two national centers for rare diseases in Denmark. A total of 244 (56% response rate) completed a questionnaire that included standard measures of QoL, symptoms of depression and anxiety, indicators of disease-related severity, visibility, and need for professional support. Associations between disease severity and visibility and psychosocial burden were analyzed in descriptive and multivariate models. We observed impaired QoL (mean = 81.3; 95% CI, 76.2; 86.4); 19% reported symptoms of depression (mean = 5.7; SD = 5.4), and 15% reported anxiety (mean = 5.1; SD = 5.2) at a clinical level. Adults with NF1 also reported requiring professional support for physical, psychological, and work-related problems. Disease severity and (partly) visibility were significantly (p < .0001) associated with psychosocial well-being and a requirement for support. This study provides new understanding of the factors associated with impaired QoL, indicating that follow-up care should be optimized into adult life.
KW - adults
KW - nationwide
KW - need for support
KW - neurofibromatosis type 1
KW - population-based
KW - psychosocial burden
U2 - 10.1002/ajmg.a.61627
DO - 10.1002/ajmg.a.61627
M3 - Journal article
C2 - 32484306
VL - 182
SP - 1704
EP - 1715
JO - American Journal of Medical Genetics, Part A
JF - American Journal of Medical Genetics, Part A
SN - 1552-4825
IS - 7
ER -