John Rosendahl Østergaard

Multisystem burden of neurofibromatosis 1 in Denmark: registry- and population-based rates of hospitalizations over the life span

Research output: Contribution to journal/Conference contribution in journal/Contribution to newspaperJournal articleResearchpeer-review

Standard

Multisystem burden of neurofibromatosis 1 in Denmark : registry- and population-based rates of hospitalizations over the life span. / Kenborg, Line; Duun-Henriksen, Anne Katrine; Dalton, Susanne O.; Bidstrup, Pernille E.; Doser, Karoline; Rugbjerg, Kathrine; Pedersen, Camilla; Krøyer, Anja; Johansen, Christoffer; Andersen, Klaus Kaae; Østergaard, John R.; Hove, Hanne; Sørensen, Sven Asger; Riccardi, Vincent M.; Mulvihill, John J.; Winther, Jeanette F.

In: Genetics in Medicine, Vol. 22, No. 6, 06.2020, p. 1069-1078.

Research output: Contribution to journal/Conference contribution in journal/Contribution to newspaperJournal articleResearchpeer-review

Harvard

Kenborg, L, Duun-Henriksen, AK, Dalton, SO, Bidstrup, PE, Doser, K, Rugbjerg, K, Pedersen, C, Krøyer, A, Johansen, C, Andersen, KK, Østergaard, JR, Hove, H, Sørensen, SA, Riccardi, VM, Mulvihill, JJ & Winther, JF 2020, 'Multisystem burden of neurofibromatosis 1 in Denmark: registry- and population-based rates of hospitalizations over the life span', Genetics in Medicine, vol. 22, no. 6, pp. 1069-1078. https://doi.org/10.1038/s41436-020-0769-6

APA

Kenborg, L., Duun-Henriksen, A. K., Dalton, S. O., Bidstrup, P. E., Doser, K., Rugbjerg, K., Pedersen, C., Krøyer, A., Johansen, C., Andersen, K. K., Østergaard, J. R., Hove, H., Sørensen, S. A., Riccardi, V. M., Mulvihill, J. J., & Winther, J. F. (2020). Multisystem burden of neurofibromatosis 1 in Denmark: registry- and population-based rates of hospitalizations over the life span. Genetics in Medicine, 22(6), 1069-1078. https://doi.org/10.1038/s41436-020-0769-6

CBE

Kenborg L, Duun-Henriksen AK, Dalton SO, Bidstrup PE, Doser K, Rugbjerg K, Pedersen C, Krøyer A, Johansen C, Andersen KK, Østergaard JR, Hove H, Sørensen SA, Riccardi VM, Mulvihill JJ, Winther JF. 2020. Multisystem burden of neurofibromatosis 1 in Denmark: registry- and population-based rates of hospitalizations over the life span. Genetics in Medicine. 22(6):1069-1078. https://doi.org/10.1038/s41436-020-0769-6

MLA

Vancouver

Kenborg L, Duun-Henriksen AK, Dalton SO, Bidstrup PE, Doser K, Rugbjerg K et al. Multisystem burden of neurofibromatosis 1 in Denmark: registry- and population-based rates of hospitalizations over the life span. Genetics in Medicine. 2020 Jun;22(6):1069-1078. https://doi.org/10.1038/s41436-020-0769-6

Author

Kenborg, Line ; Duun-Henriksen, Anne Katrine ; Dalton, Susanne O. ; Bidstrup, Pernille E. ; Doser, Karoline ; Rugbjerg, Kathrine ; Pedersen, Camilla ; Krøyer, Anja ; Johansen, Christoffer ; Andersen, Klaus Kaae ; Østergaard, John R. ; Hove, Hanne ; Sørensen, Sven Asger ; Riccardi, Vincent M. ; Mulvihill, John J. ; Winther, Jeanette F. / Multisystem burden of neurofibromatosis 1 in Denmark : registry- and population-based rates of hospitalizations over the life span. In: Genetics in Medicine. 2020 ; Vol. 22, No. 6. pp. 1069-1078.

Bibtex

@article{6a68d9ee5e46427492672f3cabbfa2c3,
title = "Multisystem burden of neurofibromatosis 1 in Denmark: registry- and population-based rates of hospitalizations over the life span",
abstract = "Purpose: The aim was to assess lifetime risk for hospitalization in individuals with neurofibromatosis 1 (NF1). Methods: The 2467 individuals discharged with a diagnosis indicating NF1 or followed in a clinical center for NF1 were matched to 20,132 general population comparisons. Based on diagnoses in 12 main diagnostic groups and 146 subcategories, we calculated rate ratios (RRs), absolute excess risks (AERs), and hazard ratios for hospitalizations. Results: The RR for any first hospitalization among individuals with NF1 was 2.3 (95% confidence interval 2.2–2.5). A high AER was seen for all 12 main diagnostic groups, dominated by disorders of the nervous system (14.5% of all AERs), benign (13.6%) and malignant neoplasms (13.4%), and disorders of the digestive (10.5%) and respiratory systems (10.3%). Neoplasms, nerve and peripheral ganglia disease, pneumonia, epilepsy, bone and joint disorders, and intestinal infections were major contributors to the excess disease burden caused by NF1. Individuals with NF1 had more hospitalizations and spent more days in hospital than the comparisons. The increased risk for any hospitalization was observed for both children and adults, with or without an associated cancer. Conclusion: NF1 causes an overall greater likelihood of hospitalization, with frequent and longer hospitalizations involving all organ systems throughout life.",
keywords = "cohort study, hospitalizations, neurofibromatosis 1, population-based",
author = "Line Kenborg and Duun-Henriksen, {Anne Katrine} and Dalton, {Susanne O.} and Bidstrup, {Pernille E.} and Karoline Doser and Kathrine Rugbjerg and Camilla Pedersen and Anja Kr{\o}yer and Christoffer Johansen and Andersen, {Klaus Kaae} and {\O}stergaard, {John R.} and Hanne Hove and S{\o}rensen, {Sven Asger} and Riccardi, {Vincent M.} and Mulvihill, {John J.} and Winther, {Jeanette F.}",
year = "2020",
month = jun,
doi = "10.1038/s41436-020-0769-6",
language = "English",
volume = "22",
pages = "1069--1078",
journal = "Genetics in Medicine",
issn = "1098-3600",
publisher = "Nature Publishing Group",
number = "6",

}

RIS

TY - JOUR

T1 - Multisystem burden of neurofibromatosis 1 in Denmark

T2 - registry- and population-based rates of hospitalizations over the life span

AU - Kenborg, Line

AU - Duun-Henriksen, Anne Katrine

AU - Dalton, Susanne O.

AU - Bidstrup, Pernille E.

AU - Doser, Karoline

AU - Rugbjerg, Kathrine

AU - Pedersen, Camilla

AU - Krøyer, Anja

AU - Johansen, Christoffer

AU - Andersen, Klaus Kaae

AU - Østergaard, John R.

AU - Hove, Hanne

AU - Sørensen, Sven Asger

AU - Riccardi, Vincent M.

AU - Mulvihill, John J.

AU - Winther, Jeanette F.

PY - 2020/6

Y1 - 2020/6

N2 - Purpose: The aim was to assess lifetime risk for hospitalization in individuals with neurofibromatosis 1 (NF1). Methods: The 2467 individuals discharged with a diagnosis indicating NF1 or followed in a clinical center for NF1 were matched to 20,132 general population comparisons. Based on diagnoses in 12 main diagnostic groups and 146 subcategories, we calculated rate ratios (RRs), absolute excess risks (AERs), and hazard ratios for hospitalizations. Results: The RR for any first hospitalization among individuals with NF1 was 2.3 (95% confidence interval 2.2–2.5). A high AER was seen for all 12 main diagnostic groups, dominated by disorders of the nervous system (14.5% of all AERs), benign (13.6%) and malignant neoplasms (13.4%), and disorders of the digestive (10.5%) and respiratory systems (10.3%). Neoplasms, nerve and peripheral ganglia disease, pneumonia, epilepsy, bone and joint disorders, and intestinal infections were major contributors to the excess disease burden caused by NF1. Individuals with NF1 had more hospitalizations and spent more days in hospital than the comparisons. The increased risk for any hospitalization was observed for both children and adults, with or without an associated cancer. Conclusion: NF1 causes an overall greater likelihood of hospitalization, with frequent and longer hospitalizations involving all organ systems throughout life.

AB - Purpose: The aim was to assess lifetime risk for hospitalization in individuals with neurofibromatosis 1 (NF1). Methods: The 2467 individuals discharged with a diagnosis indicating NF1 or followed in a clinical center for NF1 were matched to 20,132 general population comparisons. Based on diagnoses in 12 main diagnostic groups and 146 subcategories, we calculated rate ratios (RRs), absolute excess risks (AERs), and hazard ratios for hospitalizations. Results: The RR for any first hospitalization among individuals with NF1 was 2.3 (95% confidence interval 2.2–2.5). A high AER was seen for all 12 main diagnostic groups, dominated by disorders of the nervous system (14.5% of all AERs), benign (13.6%) and malignant neoplasms (13.4%), and disorders of the digestive (10.5%) and respiratory systems (10.3%). Neoplasms, nerve and peripheral ganglia disease, pneumonia, epilepsy, bone and joint disorders, and intestinal infections were major contributors to the excess disease burden caused by NF1. Individuals with NF1 had more hospitalizations and spent more days in hospital than the comparisons. The increased risk for any hospitalization was observed for both children and adults, with or without an associated cancer. Conclusion: NF1 causes an overall greater likelihood of hospitalization, with frequent and longer hospitalizations involving all organ systems throughout life.

KW - cohort study

KW - hospitalizations

KW - neurofibromatosis 1

KW - population-based

UR - http://www.scopus.com/inward/record.url?scp=85081012624&partnerID=8YFLogxK

U2 - 10.1038/s41436-020-0769-6

DO - 10.1038/s41436-020-0769-6

M3 - Journal article

C2 - 32107470

AN - SCOPUS:85081012624

VL - 22

SP - 1069

EP - 1078

JO - Genetics in Medicine

JF - Genetics in Medicine

SN - 1098-3600

IS - 6

ER -