Families' healthcare experiences for children with inherited metabolic diseases: Protocol for a mixed methods cohort study

Andrea J. Chow, Ryan Iverson, Monica Lamoureux, Kylie Tingley, Isabel Jordan, Nicole Pallone, Maureen Smith, Zobaida Al-Baldawi, Pranesh Chakraborty, Jamie Brehaut, Alicia Chan, Eyal Cohen, Sarah Dyack, Lisa Jane Gillis, Sharan Goobie, Ian D. Graham, Cheryl R. Greenberg, Jeremy M. Grimshaw, Robin Z. Hayeems, Shailly Jain-GhaiAnn Jolly, Sara Khangura, Jennifer J. MacKenzie, Nathalie Major, John J. Mitchell, Stuart G. Nicholls, Amy Pender, Murray Potter, Chitra Prasad, Lisa A. Prosser, Andreas Schulze, Komudi Siriwardena, Rebecca Sparkes, Kathy Speechley, Sylvia Stockler, Monica Taljaard, Mari Teitelbaum, Yannis Trakadis, Clara Van Karnebeek, Jagdeep S. Walia, Brenda J. Wilson, Kumanan Wilson, Beth K. Potter

Résultat de recherche: Articleexamen par les pairs

1 Citation (Scopus)

Résumé

Introduction Children with inherited metabolic diseases (IMDs) often have complex and intensive healthcare needs and their families face challenges in receiving high-quality, family centred health services. Improvement in care requires complex interventions involving multiple components and stakeholders, customised to specific care contexts. This study aims to comprehensively understand the healthcare experiences of children with IMDs and their families across Canada. Methods and analysis A two-stage explanatory sequential mixed methods design will be used. Stage 1: quantitative data on healthcare networks and encounter experiences will be collected from 100 parent/guardians through a care map, 2 baseline questionnaires and 17 weekly diaries over 5-7 months. Care networks will be analysed using social network analysis. Relationships between demographic or clinical variables and ratings of healthcare experiences across a range of family centred care dimensions will be analysed using generalised linear regression. Other quantitative data related to family experiences and healthcare experiences will be summarised descriptively. Ongoing analysis of quantitative data and purposive, maximum variation sampling will inform sample selection for stage 2: a subset of stage 1 participants will participate in one-on-one videoconference interviews to elaborate on the quantitative data regarding care networks and healthcare experiences. Interview data will be analysed thematically. Qualitative and quantitative data will be merged during analysis to arrive at an enhanced understanding of care experiences. Quantitative and qualitative data will be combined and presented narratively using a weaving approach (jointly on a theme-by-theme basis) and visually in a side-by-side joint display. Ethics and dissemination The study protocol and procedures were approved by the Children's Hospital of Eastern Ontario's Research Ethics Board, the University of Ottawa Research Ethics Board and the research ethics boards of each participating study centre. Findings will be published in peer-reviewed journals and presented at scientific conferences.

Langue d'origineEnglish
Numéro d'articlee055664
JournalBMJ Open
Volume12
Numéro de publication2
DOI
Statut de publicationPublished - févr. 22 2022

Note bibliographique

Funding Information:
Competing interests SD has been or is a member of advisory boards for, received indirect educational grants from, and/or received indirect speakers’ fees from Sanofi-Genzyme, Takeda, and Horizon Therapeutics. IDG is a recipient of a CIHR Foundation Grant (FDN# 143237). MP has been an advisory board member with honoraria from Ultragenyx (Mar/Apr 2021) and Horizon Therapeutics (Oct 2020), and received a speakers’ honorarium (Sep 2020) and small investigator grant ($6,000, 2019) from Horizon Therapeutics. SS received educational grants from Biomarin, Shire, Recordati and serves/served as PI in clinical trials and postmarketing registries sponsored by Actelion, Biomarin, Shire, Ultragenyx. KW is the CEO of CANImmunize Inc.

Publisher Copyright:
© 2022 BMJ Publishing Group. All rights reserved.

ASJC Scopus Subject Areas

  • General Medicine

PubMed: MeSH publication types

  • Journal Article
  • Research Support, Non-U.S. Gov't

Empreinte numérique

Plonger dans les sujets de recherche 'Families' healthcare experiences for children with inherited metabolic diseases: Protocol for a mixed methods cohort study'. Ensemble, ils forment une empreinte numérique unique.

Citer