TY - JOUR
T1 - Malocclusion traits and oral health–related quality of life in children with osteogenesis imperfecta
T2 - A cross-sectional study
AU - Members of the Brittle Bone Diseases Consortium
AU - Najirad, Mohammadamin
AU - Madathil, Sreenath Arekunnath
AU - Rauch, Frank
AU - Sutton, V. Reid
AU - Lee, Brendan
AU - Retrouvey, Jean Marc
AU - Nagamani, Sandesh C.S.
AU - Glorieux, Francis
AU - Esposito, Paul
AU - Wallace, Maegen
AU - Bober, Michael B.
AU - Eyre, David
AU - Gomez, Danielle
AU - Harris, Gerald
AU - Hart, Tracy
AU - Jain, Mahim
AU - Krakow, Deborah
AU - Krischer, Jeffrey
AU - Orwoll, Eric
AU - Nicol, Lindsey
AU - Raggio, Cathleen
AU - Smith, Peter
AU - Tosi, Laura
AU - Esfandiari, Shahrokh
N1 - Funding Information:
This study was funded by grant 1U54AR068069-0 from the Brittle Bone Disease Consortium, which is a part of the National Center for Advancing Translational Sciences Rare Diseases Clinical Research Network and is funded through the collaboration of the Office of Rare Diseases Research, National Center for Advancing, the National Institute of Arthritis and Musculoskeletal and Skin Diseases, and the National Institute of Dental and Craniofacial Research, and the Eunice Kennedy Shriver National Institutes of Child Health and Development. The content is solely the responsibility of the authors and does not necessarily represent the official views of the National Institutes of Health. The Brittle Bone Desease Consortium is also supported by the Osteogenesis Imperfecta Foundation.
Funding Information:
This study was funded by grant 1U54AR068069-0 from the Brittle Bone Disease Consortium , which is a part of the National Center for Advancing Translational Sciences Rare Diseases Clinical Research Network and is funded through the collaboration of the Office of Rare Diseases Research, National Center for Advancing, the National Institute of Arthritis and Musculoskeletal and Skin Diseases , and the National Institute of Dental and Craniofacial Research , and the Eunice Kennedy Shriver National Institutes of Child Health and Development . The content is solely the responsibility of the authors and does not necessarily represent the official views of the National Institutes of Health. The Brittle Bone Desease Consortium is also supported by the Osteogenesis Imperfecta Foundation .
Publisher Copyright:
© 2020 American Dental Association
PY - 2020/7
Y1 - 2020/7
N2 - Background: The incidence of malocclusion is higher among people with osteogenesis imperfecta (OI) than the general population, and treatment options are limited due to the weak structure of bones and teeth. Focusing on those malocclusion traits that might have a high impact on a patient's oral health–related quality of life (OHRQoL) is warranted. Methods: A total of 138 children and adolescents with OI were examined for malocclusion traits. OHRQoL was measured using age-specific versions (8 through 10 years and 11 through 14 years) of the Child Perceptions Questionnaire (CPQ), considering the following domains: oral symptoms, functional limitation, emotional well-being, and social well-being. Higher scores implied worse OHRQoL. Multivariable ordinal logistic regression was used to estimate the association between malocclusion traits and OHRQoL. Results: Among children aged 8 through 10 years (n = 56), the CPQ and its constituent domain scores were relatively similar between those with malocclusion (higher scores) and those without. In the adolescent (n = 82) group aged 11 through 14 years; however, those with posterior crossbite (odds ratio, 5.01; 95% confidence interval, 1.40 to 12.41) or open bite (odds ratio, 3.21; 95% confidence interval, 1.21 to 10.23) experienced statistically significantly higher degrees of functional limitations (a higher functional limitation score) than those without. Conclusions: Adolescents with OI and posterior open bites or crossbites have substantial self-reported functional limitations and worse oral symptoms, which warrants additional investigation and therapeutic trials in an attempt to improve the malocclusion. In addition, the authors found that the CPQ can be a useful tool in a clinical trial of orthodontic interventions in OI.
AB - Background: The incidence of malocclusion is higher among people with osteogenesis imperfecta (OI) than the general population, and treatment options are limited due to the weak structure of bones and teeth. Focusing on those malocclusion traits that might have a high impact on a patient's oral health–related quality of life (OHRQoL) is warranted. Methods: A total of 138 children and adolescents with OI were examined for malocclusion traits. OHRQoL was measured using age-specific versions (8 through 10 years and 11 through 14 years) of the Child Perceptions Questionnaire (CPQ), considering the following domains: oral symptoms, functional limitation, emotional well-being, and social well-being. Higher scores implied worse OHRQoL. Multivariable ordinal logistic regression was used to estimate the association between malocclusion traits and OHRQoL. Results: Among children aged 8 through 10 years (n = 56), the CPQ and its constituent domain scores were relatively similar between those with malocclusion (higher scores) and those without. In the adolescent (n = 82) group aged 11 through 14 years; however, those with posterior crossbite (odds ratio, 5.01; 95% confidence interval, 1.40 to 12.41) or open bite (odds ratio, 3.21; 95% confidence interval, 1.21 to 10.23) experienced statistically significantly higher degrees of functional limitations (a higher functional limitation score) than those without. Conclusions: Adolescents with OI and posterior open bites or crossbites have substantial self-reported functional limitations and worse oral symptoms, which warrants additional investigation and therapeutic trials in an attempt to improve the malocclusion. In addition, the authors found that the CPQ can be a useful tool in a clinical trial of orthodontic interventions in OI.
KW - Child Perceptions Questionnaires
KW - Osteogenesis imperfecta
KW - adolescent
KW - brittle bone disease
KW - child
KW - malocclusion
KW - oral health–related quality of life
UR - http://www.scopus.com/inward/record.url?scp=85086447473&partnerID=8YFLogxK
UR - http://www.scopus.com/inward/citedby.url?scp=85086447473&partnerID=8YFLogxK
U2 - 10.1016/j.adaj.2020.03.040
DO - 10.1016/j.adaj.2020.03.040
M3 - Article
C2 - 32593350
AN - SCOPUS:85086447473
SN - 0002-8177
VL - 151
SP - 480-490.e2
JO - Journal of the American Dental Association
JF - Journal of the American Dental Association
IS - 7
ER -