Faculty Collaboration Database

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

Date

10/26/2018

Pubmed ID

30359278

Pubmed Central ID

PMC6202869

DOI

10.1186/s13023-018-0935-y

Scopus ID

2-s2.0-85055618264 (requires institutional sign-in at Scopus site)   28 Citations

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.

Author List

Najirad M, Ma MS, Rauch F, Sutton VR, Lee B, Retrouvey JM, Members of the BBD, Esfandiari S

Author

Gerald Harris PhD Director in the Orthopaedic Research Engineering Center (OREC) department at Marquette University

MESH terms used to index this publication - Major topics in bold

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