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; Members of the BBD; Shahrokh Esfandiari

doi:10.1186/s13023-018-0935-y

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

Orphanet J Rare Dis. 2018 Oct 25;13(1):187. doi: 10.1186/s13023-018-0935-y.

Authors

Mohammadamin Najirad¹, Mang Shin Ma², Frank Rauch³, Vernon Reid Sutton⁴, Brendan Lee⁴, Jean-Marc Retrouvey⁵; Members of the BBD; Shahrokh Esfandiari²

Collaborators

Members of the BBD:
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 Raggio, Peter Smith, Laura Tosi

Affiliations

¹ Division of Oral Health and Society, Faculty of Dentistry, McGill University, 2001 McGill College, Suite 500, Montreal, Quebec, H3A 1G1, Canada. Mohammadamin.najirad@mail.mcgill.ca.
² Division of Oral Health and Society, Faculty of Dentistry, McGill University, 2001 McGill College, Suite 500, Montreal, Quebec, H3A 1G1, Canada.
³ Shriners Hospital for Children, Mount Royal, 1529 Cedar Avenue, Montreal, Quebec, H3G 1A6, Canada.
⁴ Molecular and Human Genetics, Baylor College of Medicine, 6701 Fannin CC1560, Houston, TX, 77030, USA.
⁵ Department of Orthodontics, Faculty of Dentistry, McGill University, 2001 McGill College, Suite 500, Montreal, Quebec, H3A 1G1, Canada.

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 (CPQ_8-10) and for 11 to 14-year-olds (CPQ_11-14).

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. CPQ_8-10 scores were similar between OI types in children aged 8 to 10 years. In the 11 to 14-year-old group, CPQ_11-14-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.

Keywords: Child perceptions questionnaires; Oral health-related quality of life; Osteogenesis imperfecta.

Publication types

Multicenter Study
Research Support, N.I.H., Extramural
Research Support, Non-U.S. Gov't

MeSH terms

Adolescent
Child
Cross-Sectional Studies
Female
Humans
Male
Oral Health*
Osteogenesis Imperfecta / complications*
Quality of Life*

Abstract

Publication types

MeSH terms

Grants and funding