{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,19]],"date-time":"2026-08-19T17:50:23Z","timestamp":1787161823090,"version":"build-2736575974"},"reference-count":106,"publisher":"BMJ","issue":"2","license":[{"start":{"date-parts":[[2022,2,21]],"date-time":"2022-02-21T00:00:00Z","timestamp":1645401600000},"content-version":"unspecified","delay-in-days":20,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100000024","name":"Canadian Institutes of Health Research","doi-asserted-by":"publisher","award":["PJT-153230"],"award-info":[{"award-number":["PJT-153230"]}],"id":[{"id":"10.13039\/501100000024","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Open"],"accepted":{"date-parts":[[2022,1,23]]},"published-print":{"date-parts":[[2022,2]]},"abstract":"<jats:sec>\n                    <jats:title>Introduction<\/jats:title>\n                    <jats:p>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.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods and analysis<\/jats:title>\n                    <jats:p>\n                      A two-stage explanatory sequential mixed methods design will be used.\n                      <jats:italic>Stage 1:<\/jats:italic>\n                      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\u20137 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\n                      <jats:italic>stage 2:<\/jats:italic>\n                      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.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Ethics and dissemination<\/jats:title>\n                    <jats:p>The study protocol and procedures were approved by the Children\u2019s Hospital of Eastern Ontario\u2019s 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.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1136\/bmjopen-2021-055664","type":"journal-article","created":{"date-parts":[[2022,2,22]],"date-time":"2022-02-22T12:44:06Z","timestamp":1645533846000},"page":"e055664","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":1,"title":["Families\u2019 healthcare experiences for children with inherited metabolic diseases: protocol for a mixed methods cohort study"],"prefix":"10.1136","volume":"12","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1683-2912","authenticated-orcid":false,"given":"Andrea J","family":"Chow","sequence":"first","affiliation":[{"name":"School of Epidemiology and Public Health, University of Ottawa, Ottawa, Ontario, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ryan","family":"Iverson","sequence":"additional","affiliation":[{"name":"Children's Hospital of Eastern Ontario, Ottawa, Ontario, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Monica","family":"Lamoureux","sequence":"additional","affiliation":[{"name":"Children's Hospital of Eastern Ontario, Ottawa, Ontario, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kylie","family":"Tingley","sequence":"additional","affiliation":[{"name":"School of Epidemiology and Public Health, University of Ottawa, Ottawa, Ontario, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Isabel","family":"Jordan","sequence":"additional","affiliation":[{"name":"Patient Partner, Squamish, British Columbia, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nicole","family":"Pallone","sequence":"additional","affiliation":[{"name":"Patient Partner, Canadian PKU & Allied Disorders Inc, Toronto, Ontario, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maureen","family":"Smith","sequence":"additional","affiliation":[{"name":"Patient Partner, Canadian Organization for Rare Disorders, Toronto, Ontario, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zobaida","family":"Al-Baldawi","sequence":"additional","affiliation":[{"name":"School of Epidemiology and Public Health, University of Ottawa, Ottawa, Ontario, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pranesh","family":"Chakraborty","sequence":"additional","affiliation":[{"name":"Children's Hospital of Eastern Ontario, Ottawa, Ontario, Canada"},{"name":"Department of Pediatrics, University of Ottawa, Ottawa, Ontario, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jamie","family":"Brehaut","sequence":"additional","affiliation":[{"name":"School of Epidemiology and Public Health, University of 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