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The impact of craniofacial and dental osteogenesis imperfecta manifestations on oral health-related quality of life of children and adolescents

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Abstract

Objective

Craniofacial and oral manifestations of Osteogenesis Imperfecta (OI) can affect the functioning of the stomatognathic system and impact the patient’s quality of life. The objective of the study was to evaluate the relationship between craniofacial and oral manifestations and the Oral Health-related Quality of Life (OHRQoL) of OI children and adolescents. Material and Methods: A total of 30 OI patients aged eight to fourteen years old followed up at the Oral Care Center for Inherited Diseases were enrolled in the research. OHRQoL was assessed using the short form of the Child Perceptions Questionnaire (CPQ) for eight to ten-year-olds (CPQ8–10) and 11 to 14-year-olds (CPQ11–14). The relationship between the OHRQoL index and its assessment domains, OI types, and the presence of dentinogenesis imperfecta (DI), class III malocclusion, and dental agenesis were evaluated. Results: The median CPQ score of patients was 5, and there was no significant difference in OHRQoL between children and adolescents, nor associated with the disease severity or the presence of DI. The oral manifestations evaluated did not directly impact the patients’ OHRQoL. Conclusions: The study demonstrated that the perception of OHRQoL is similar for both adolescents and children. The oral symptom was the most relevant domain for the index among patients aged eight to fourteen years while the emotional well-being was the most impacted. Clinical Relevance: this study makes contributions by indicating that addressing dental care for children and adolescents with OI is important in clinical management and better OHRQoL for this population.

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Acknowledgements

The authors would like to thank patients and their families for their willingness to participate in the research.

Funding

The author Pantoja, LLQ was supported by the University of Brasília (UnB), Brazil. This study was supported by a Postgraduate Department of UnB grant (DPG/UnB #011/2022) that was used to afford statistical analysis. The funding agencies have no role in the submitted work.

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Authors and Affiliations

  1. Faculty of Health Sciences, Department of Dentistry, University of Brasília, Brasília, Brazil

    Leticia L. Quirino Pantoja, Mariana Candida Vaz Carvalho, Lilian Marly Paula & Ana Carolina Acevedo

  2. University Hospital of Brasilia, Oral Care Center for Inherited Diseases, Brasília, Brazil

    Leticia L. Quirino Pantoja, Mariana Candida Vaz Carvalho, Paulo Marcio Yamaguti, Lilian Marly Paula & Ana Carolina Acevedo

  3. Faculty of Medicine, Department of Pediatrics, University of Brasilia, Brasilia, Brazil

    Luiz Claudio Castro

  4. Faculty of Health Sciences, Department of Dentistry, Laboratory of Oral Histopathology, University of Brasíli, Brasília, Brazil

    Ana Carolina Acevedo

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  1. Leticia L. Quirino Pantoja
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  2. Mariana Candida Vaz Carvalho
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  3. Paulo Marcio Yamaguti
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  4. Luiz Claudio Castro
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Contributions

Pantoja, LLQ: Contributed to conception, design, data acquisition and interpretation, performed statistical analyses, drafted, and critically revised the manuscript. Carvalho, MCV: Contributed to data acquisition and interpretation, and critically revised the manuscript. Yamaguti, PM: Contributed to data interpretation, and critically revised the manuscript. Castro, LC: Contributed to design, data interpretation, and critically revised the manuscript. Paula, LM: Contributed to data interpretation, and critically revised the manuscript. Acevedo, AC: Contributed to conception, design, data analysis and interpretation, drafted and critically revised the manuscriptAll authors gave their final approval and agreed to be accountable for all aspects of the work.

Corresponding author

Correspondence to Leticia L. Quirino Pantoja.

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Ethics approval

This study was developed at the University Hospital of Brasília (HUB) and approved by the Human Research Ethics Committee of the University of Brasília School of Health Sciences (registration number 15367519.8.0000.0030).

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Parents or legal guardians of all subjects participating in the study signed an Informed Consent Form, in which they agreed to participate in the research.

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Pantoja, L.L., Carvalho, M.C., Yamaguti, P.M. et al. The impact of craniofacial and dental osteogenesis imperfecta manifestations on oral health-related quality of life of children and adolescents. Clin Oral Invest 28, 169 (2024). https://doi.org/10.1007/s00784-024-05568-8

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