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Journal Articles The Journal of Pediatrics Year : 2022

Determinants of Quality of Life in Children with Inborn Errors of Metabolism Receiving a Restricted Diet

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Abdoulaye Ouattara
Noemie Resseguier
  • Function : Author
Aline Cano
  • Function : Author
Pascale de Lonlay
  • Function : Author
Jean-Baptiste Arnoux
  • Function : Author
Anais Brassier
  • Function : Author
Manuel Schiff
  • Function : Author
Samia Pichard
  • Function : Author
Alexandre Fabre
  • Function : Author
Celia Hoebeke
  • Function : Author
Nathalie Guffon
  • Function : Author
Alain Fouilhoux
  • Function : Author
Pierre Broué
  • Function : Author
Guy Touati
  • Function : Author
Dries Dobbelaere
  • Function : Author
Karine Mention
  • Function : Author
Francois Labarthe
  • Function : Author
Marine Tardieu
  • Function : Author
Loïc de Parscau
  • Function : Author
Francois Feillet
  • Function : Author
Chrystèle Bonnemains
  • Function : Author
Philippe Labrune
  • Function : Author
Magalie Barth
Lena Damaj
Delphine Lamireau
  • Function : Author
Julie Berbis
  • Function : Author
Pascal Auquier
  • Function : Author
Brigitte Chabrol
  • Function : Author

Abstract

Objective To investigate the determinants of quality of life (QoL) in children with inborn errors of metabolism with restricted diet (IEMRDs) using a single theory-based multidimensional model. Study design In this multicenter cross-sectional study, data from children aged 8-17 years with IEMRDs (except phenylketonuria) and their parents were collected from January 2015 to December 2017. Measurements included a child's self-reported QoL, self-rated behavioral problems and anxiety, and parental anxiety. Based on hypotheses from a literature-built theoretical model linking demographic, clinical, family environment, and psychosocial characteristics to QoL either directly or indirectly, associations of these factors with a child's self-rated QoL were examined using a structural equation modeling approach. Results A total of 312 children (mean [SD] age, 12.2 [2.6] years; 51% boys [n = 160]) were included. Higher levels of trait anxiety and behavioral problems in children were the most important factors associated with poorer QoL (standardized path coefficients, À0.71 and À0.23, respectively). In addition, higher parent trait anxiety, younger age at diagnosis, and a disease requiring an emergency diet were associated with poorer QoL in these children. The final model fit the data closely according to conventional goodness-of-fit statistics and explained 86% of the QoL variance.
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Dates and versions

hal-03797484 , version 1 (04-10-2022)

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Abdoulaye Ouattara, Noemie Resseguier, Aline Cano, Pascale de Lonlay, Jean-Baptiste Arnoux, et al.. Determinants of Quality of Life in Children with Inborn Errors of Metabolism Receiving a Restricted Diet. The Journal of Pediatrics, 2022, 242, pp.192-200.e3. ⟨10.1016/j.jpeds.2021.11.021⟩. ⟨hal-03797484⟩

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