Integrated Care for People Living With Rare Disease: A Scoping Review on Primary Care Models in Organization for Economic Cooperation and Development Countries
Introduction/Objectives: Individually rare, rare diseases are collectively common resulting in frequent health system use. Navigating the health system persists as a challenge. Primary care provides longitudinal contact with the health system and is placed to provide integrated rare-disease-care. Me...
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| Format: | Article |
| Language: | English |
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SAGE Publishing
2025-01-01
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| Series: | Journal of Primary Care & Community Health |
| Online Access: | https://doi.org/10.1177/21501319241311567 |
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| author | Nada Vidic Anna McGlynn Fatemeh Abdi Chun Wah Michael Tam Reginald Michael Crampton Kean-Seng Lim Elizabeth Emma Palmer Natalie Taylor Ben Harris-Roxas |
| author_facet | Nada Vidic Anna McGlynn Fatemeh Abdi Chun Wah Michael Tam Reginald Michael Crampton Kean-Seng Lim Elizabeth Emma Palmer Natalie Taylor Ben Harris-Roxas |
| author_sort | Nada Vidic |
| collection | DOAJ |
| description | Introduction/Objectives: Individually rare, rare diseases are collectively common resulting in frequent health system use. Navigating the health system persists as a challenge. Primary care provides longitudinal contact with the health system and is placed to provide integrated rare-disease-care. Methods: This scoping review used Joanna Briggs Institute and PRISMA methods with a Consolidated Framework for Implementation Research based data extraction tool to find how integrated rare-disease-care is delivered, enablers and barriers to the same, in primary care settings in contemporary literature in OECD countries. Results: The Primary Care Provider (PCP) role varies from routine primary care to shared-rare-disease-care models. In the 26 papers, the most frequently cited PCP roles included involvement in diagnosis (n = 14), care coordination (n = 16), primary and preventative care (n = 18), management of components of rare-disease-care (n = 13), and treatment monitoring (n = 10). Individuals whose PCP was actively involved in their care were reported to have shortened diagnostic delay, improved transitions of care across the lifespan, reduced unplanned utilization of emergency and hospital services, comprehensive psychosocial care, improved quality of life across environments including home, school and work and improved palliative care experiences. Conclusions: Sufficient communication from specialists, information, resources, time and reimbursement for complex care are still needed. Future integrated-rare-disease-care models should be developed by, or with, PCPs. |
| format | Article |
| id | doaj-art-f2a111c2ce334170aabb3e5c658898d3 |
| institution | Kabale University |
| issn | 2150-1327 |
| language | English |
| publishDate | 2025-01-01 |
| publisher | SAGE Publishing |
| record_format | Article |
| series | Journal of Primary Care & Community Health |
| spelling | doaj-art-f2a111c2ce334170aabb3e5c658898d32025-01-08T13:03:26ZengSAGE PublishingJournal of Primary Care & Community Health2150-13272025-01-011610.1177/21501319241311567Integrated Care for People Living With Rare Disease: A Scoping Review on Primary Care Models in Organization for Economic Cooperation and Development CountriesNada Vidic0Anna McGlynn1Fatemeh Abdi2Chun Wah Michael Tam3Reginald Michael Crampton4Kean-Seng Lim5Elizabeth Emma Palmer6Natalie Taylor7Ben Harris-Roxas8University of New South Wales, Sydney, NSW, AustraliaPopulation and Community Health Directorate, Sydney, AustraliaUniversity of New South Wales, Sydney, NSW, AustraliaPrimary and Integrated Care Unit, Liverpool, NSW, AustraliaWentWest Primary Health Network, Sydney, NSW, AustraliaMt Druitt Medical Practice, Sydney, NSW, AustraliaUniversity of New South Wales, Sydney, NSW, AustraliaUniversity of New South Wales, Sydney, NSW, AustraliaUniversity of Technology Sydney, Sydney, NSW, AustraliaIntroduction/Objectives: Individually rare, rare diseases are collectively common resulting in frequent health system use. Navigating the health system persists as a challenge. Primary care provides longitudinal contact with the health system and is placed to provide integrated rare-disease-care. Methods: This scoping review used Joanna Briggs Institute and PRISMA methods with a Consolidated Framework for Implementation Research based data extraction tool to find how integrated rare-disease-care is delivered, enablers and barriers to the same, in primary care settings in contemporary literature in OECD countries. Results: The Primary Care Provider (PCP) role varies from routine primary care to shared-rare-disease-care models. In the 26 papers, the most frequently cited PCP roles included involvement in diagnosis (n = 14), care coordination (n = 16), primary and preventative care (n = 18), management of components of rare-disease-care (n = 13), and treatment monitoring (n = 10). Individuals whose PCP was actively involved in their care were reported to have shortened diagnostic delay, improved transitions of care across the lifespan, reduced unplanned utilization of emergency and hospital services, comprehensive psychosocial care, improved quality of life across environments including home, school and work and improved palliative care experiences. Conclusions: Sufficient communication from specialists, information, resources, time and reimbursement for complex care are still needed. Future integrated-rare-disease-care models should be developed by, or with, PCPs.https://doi.org/10.1177/21501319241311567 |
| spellingShingle | Nada Vidic Anna McGlynn Fatemeh Abdi Chun Wah Michael Tam Reginald Michael Crampton Kean-Seng Lim Elizabeth Emma Palmer Natalie Taylor Ben Harris-Roxas Integrated Care for People Living With Rare Disease: A Scoping Review on Primary Care Models in Organization for Economic Cooperation and Development Countries Journal of Primary Care & Community Health |
| title | Integrated Care for People Living With Rare Disease: A Scoping Review on Primary Care Models in Organization for Economic Cooperation and Development Countries |
| title_full | Integrated Care for People Living With Rare Disease: A Scoping Review on Primary Care Models in Organization for Economic Cooperation and Development Countries |
| title_fullStr | Integrated Care for People Living With Rare Disease: A Scoping Review on Primary Care Models in Organization for Economic Cooperation and Development Countries |
| title_full_unstemmed | Integrated Care for People Living With Rare Disease: A Scoping Review on Primary Care Models in Organization for Economic Cooperation and Development Countries |
| title_short | Integrated Care for People Living With Rare Disease: A Scoping Review on Primary Care Models in Organization for Economic Cooperation and Development Countries |
| title_sort | integrated care for people living with rare disease a scoping review on primary care models in organization for economic cooperation and development countries |
| url | https://doi.org/10.1177/21501319241311567 |
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