Oral health-related quality of life in children and adolescents with osteogenesis imperfecta: Cross-sectional study

Mohammadamin Najirad, Mang Shin Ma, Frank Rauch, Vernon Reid Sutton, Brendan Lee, Jean Marc Retrouvey, Sandesh C.S. Nagamani, Francis Glorieux, Paul Esposito, Eric Rush, Michael Bober, David Eyre, Danielle Gomez, Gerald Harris, Tracy Hart, Mahim Jain, Deborah Krakow, Jeffrey Krischer, Eric Orwoll, Cathleen RaggioPeter Smith, Laura Tosi, Shahrokh Esfandiari

Research output: Contribution to journalArticle

1 Citation (Scopus)

Abstract

Background: Osteogenesis imperfecta (OI) affects dental and craniofacial development and may therefore impair Oral Health-Related Quality of Life (OHRQoL). However, little is known about OHRQoL in children and adolescents with OI. The aim of this study was to explore the influence of OI severity on oral health-related quality of life in children and adolescents. Methods: Children and adolescents aged 8-14 years were recruited in the context of a multicenter longitudinal study (Brittle Bone Disease Consortium) that enrolls individuals with OI in 10 centers across North America. OHRQoL was assessed using the Child Perceptions Questionnaire (CPQ) versions for 8 to 10-year-olds (CPQ8-10) and for 11 to 14-year-olds (CPQ11-14). Results: A total of 138 children and adolescents (62% girls) diagnosed with OI types I, III, IV, V and VI (n = 65, 30, 37, 4 and 2, respectively) participated in the study. CPQ8-10 scores were similar between OI types in children aged 8 to 10 years. In the 11 to 14-year-old group, CPQ11-14-scores were significantly higher (i.e. worse) for OI types III (24.7 [SD 12.5]) and IV (23.1 [SD 14.8]) than for OI type I (16.5 [SD 12.8]) (P < 0.05). The difference between OI types was due to the association between OI types and the functional limitations domain, as OI types III and IV were associated with significantly higher grade of functional limitations compared to OI type I. Conclusion: The severity of OI impacts OHRQoL in adolescents aged 11 to 14 years, but not in children age 8 to 10 years.

Original languageEnglish (US)
Article number187
JournalOrphanet Journal of Rare Diseases
Volume13
Issue number1
DOIs
StatePublished - Oct 25 2018

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Osteogenesis Imperfecta
Oral Health
Cross-Sectional Studies
Quality of Life
North America
Multicenter Studies
Longitudinal Studies
Tooth

Keywords

  • Child perceptions questionnaires
  • Oral health-related quality of life
  • Osteogenesis imperfecta

ASJC Scopus subject areas

  • Genetics(clinical)
  • Pharmacology (medical)

Cite this

Oral health-related quality of life in children and adolescents with osteogenesis imperfecta : Cross-sectional study. / Najirad, Mohammadamin; Ma, Mang Shin; Rauch, Frank; Sutton, Vernon Reid; Lee, Brendan; Retrouvey, Jean Marc; Nagamani, Sandesh C.S.; Glorieux, Francis; Esposito, Paul; Rush, Eric; Bober, Michael; Eyre, David; Gomez, Danielle; Harris, Gerald; Hart, Tracy; Jain, Mahim; Krakow, Deborah; Krischer, Jeffrey; Orwoll, Eric; Raggio, Cathleen; Smith, Peter; Tosi, Laura; Esfandiari, Shahrokh.

In: Orphanet Journal of Rare Diseases, Vol. 13, No. 1, 187, 25.10.2018.

Research output: Contribution to journalArticle

Najirad, M, Ma, MS, Rauch, F, Sutton, VR, Lee, B, Retrouvey, JM, Nagamani, SCS, Glorieux, F, Esposito, P, Rush, E, Bober, M, Eyre, D, Gomez, D, Harris, G, Hart, T, Jain, M, Krakow, D, Krischer, J, Orwoll, E, Raggio, C, Smith, P, Tosi, L & Esfandiari, S 2018, 'Oral health-related quality of life in children and adolescents with osteogenesis imperfecta: Cross-sectional study', Orphanet Journal of Rare Diseases, vol. 13, no. 1, 187. https://doi.org/10.1186/s13023-018-0935-y
Najirad, Mohammadamin ; Ma, Mang Shin ; Rauch, Frank ; Sutton, Vernon Reid ; Lee, Brendan ; Retrouvey, Jean Marc ; Nagamani, Sandesh C.S. ; Glorieux, Francis ; Esposito, Paul ; Rush, Eric ; Bober, Michael ; Eyre, David ; Gomez, Danielle ; Harris, Gerald ; Hart, Tracy ; Jain, Mahim ; Krakow, Deborah ; Krischer, Jeffrey ; Orwoll, Eric ; Raggio, Cathleen ; Smith, Peter ; Tosi, Laura ; Esfandiari, Shahrokh. / Oral health-related quality of life in children and adolescents with osteogenesis imperfecta : Cross-sectional study. In: Orphanet Journal of Rare Diseases. 2018 ; Vol. 13, No. 1.
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abstract = "Background: Osteogenesis imperfecta (OI) affects dental and craniofacial development and may therefore impair Oral Health-Related Quality of Life (OHRQoL). However, little is known about OHRQoL in children and adolescents with OI. The aim of this study was to explore the influence of OI severity on oral health-related quality of life in children and adolescents. Methods: Children and adolescents aged 8-14 years were recruited in the context of a multicenter longitudinal study (Brittle Bone Disease Consortium) that enrolls individuals with OI in 10 centers across North America. OHRQoL was assessed using the Child Perceptions Questionnaire (CPQ) versions for 8 to 10-year-olds (CPQ8-10) and for 11 to 14-year-olds (CPQ11-14). Results: A total of 138 children and adolescents (62{\%} girls) diagnosed with OI types I, III, IV, V and VI (n = 65, 30, 37, 4 and 2, respectively) participated in the study. CPQ8-10 scores were similar between OI types in children aged 8 to 10 years. In the 11 to 14-year-old group, CPQ11-14-scores were significantly higher (i.e. worse) for OI types III (24.7 [SD 12.5]) and IV (23.1 [SD 14.8]) than for OI type I (16.5 [SD 12.8]) (P < 0.05). The difference between OI types was due to the association between OI types and the functional limitations domain, as OI types III and IV were associated with significantly higher grade of functional limitations compared to OI type I. Conclusion: The severity of OI impacts OHRQoL in adolescents aged 11 to 14 years, but not in children age 8 to 10 years.",
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T2 - Cross-sectional study

AU - Najirad, Mohammadamin

AU - Ma, Mang Shin

AU - Rauch, Frank

AU - Sutton, Vernon Reid

AU - Lee, Brendan

AU - Retrouvey, Jean Marc

AU - Nagamani, Sandesh C.S.

AU - Glorieux, Francis

AU - Esposito, Paul

AU - Rush, Eric

AU - Bober, Michael

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 - Raggio, Cathleen

AU - Smith, Peter

AU - Tosi, Laura

AU - Esfandiari, Shahrokh

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N2 - Background: Osteogenesis imperfecta (OI) affects dental and craniofacial development and may therefore impair Oral Health-Related Quality of Life (OHRQoL). However, little is known about OHRQoL in children and adolescents with OI. The aim of this study was to explore the influence of OI severity on oral health-related quality of life in children and adolescents. Methods: Children and adolescents aged 8-14 years were recruited in the context of a multicenter longitudinal study (Brittle Bone Disease Consortium) that enrolls individuals with OI in 10 centers across North America. OHRQoL was assessed using the Child Perceptions Questionnaire (CPQ) versions for 8 to 10-year-olds (CPQ8-10) and for 11 to 14-year-olds (CPQ11-14). Results: A total of 138 children and adolescents (62% girls) diagnosed with OI types I, III, IV, V and VI (n = 65, 30, 37, 4 and 2, respectively) participated in the study. CPQ8-10 scores were similar between OI types in children aged 8 to 10 years. In the 11 to 14-year-old group, CPQ11-14-scores were significantly higher (i.e. worse) for OI types III (24.7 [SD 12.5]) and IV (23.1 [SD 14.8]) than for OI type I (16.5 [SD 12.8]) (P < 0.05). The difference between OI types was due to the association between OI types and the functional limitations domain, as OI types III and IV were associated with significantly higher grade of functional limitations compared to OI type I. Conclusion: The severity of OI impacts OHRQoL in adolescents aged 11 to 14 years, but not in children age 8 to 10 years.

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