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The Benefits of a United Kingdom Multiple Sclerosis Register / RODDEN MIDDLETON

Swansea University Author: RODDEN MIDDLETON

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DOI (Published version): 10.23889/SUthesis.65409

Abstract

Background: In rare diseases such as multiple sclerosis (MS) the capture of data purely from clinical trials or hospital cohorts is insufficient for fully understanding the multifaceted impacts of the disease. MS is the most common non-traumatic cause of disability in young adults worldwide. The dis...

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Published: Swansea, Wales, UK 2023
Institution: Swansea University
Degree level: Doctoral
Degree name: Ph.D
Supervisor: Brophy, Sinead.
URI: https://cronfa.swan.ac.uk/Record/cronfa65409
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fullrecord <?xml version="1.0" encoding="utf-8"?><rfc1807 xmlns:xsi="http://www.w3.org/2001/XMLSchema-instance" xmlns:xsd="http://www.w3.org/2001/XMLSchema"><bib-version>v2</bib-version><id>65409</id><entry>2024-01-04</entry><title>The Benefits of a United Kingdom Multiple Sclerosis Register</title><swanseaauthors><author><sid>2a76cced6985381173d66002d3859019</sid><firstname>RODDEN</firstname><surname>MIDDLETON</surname><name>RODDEN MIDDLETON</name><active>true</active><ethesisStudent>false</ethesisStudent></author></swanseaauthors><date>2024-01-04</date><abstract>Background: In rare diseases such as multiple sclerosis (MS) the capture of data purely from clinical trials or hospital cohorts is insufficient for fully understanding the multifaceted impacts of the disease. MS is the most common non-traumatic cause of disability in young adults worldwide. The disease is multifactorial, profoundly impacts quality of life, and life span is affected by 7 to 14 years. Diagnosis has become easier and newer treatment options have proliferated but monitoring and researching the disease's various impacts remains challenging with a largely clinical focus on ‘hard outcomes’ such as imaging, biomarkers and in-person clinically assessed scales. Disease registers are best positioned to capture data about chronic disease such as MS as they allow for longitudinal capture from a variety of sources, including clinical outcomes but also data from patients/participants. Moving disease registers away from paper-based capture has allowed for easier, more accurate and rapid capture from these sources but can also add new data sources such as novel outcome measures, data from other devices and then facilitate linkage of that data across all these domains. Aim: To communicate the learning and experiences from building a UK-wide register which captures multifaceted MS data, in order to inform the development of similar registers for other conditions. Methods: Construction of an electronic platform sufficiently flexible to capture data from people with MS in the form of patient/participant reported outcomes, from the NHS as clinical datasets, and the technology to link these datasets together in a privacy protecting way to make these datasets available to other researchers. The data capture technology must be robust enough to add additional sources or datasets as needed whilst maintaining the core elements of reproducible research. Results: A robust, flexible, privacy-protecting secure research-ready disease register was constructed containing data directly captured from more than 20,000 participants, 50 NHS sites with more than 1 million completed Patient Reported Outcomes (PRO), clinically transmitted datasets and other diverse outcomes collected. Pseudonymised elements of that data, subject to robust governance and review, are released to appropriately qualified researchers to carry out their own research on the platform. A number of important lessons were learned in the construction of this research register. The most important being that involvement of people affected by the disease in all aspects of the project is crucial for enabling key aspects of a functional register, including the collection of varied and complementary data, the levels of engagement required for longitudinal research, and assisting with the direction of research. Feedback loops in this participant-register relationship create a more holistic research instrument. Another vital aspect is the ability to carry out data linkage both within the project and to outside routinely collected datasets, expanding the scope of potential research without adding burden to participants. Flexibility of approach is particularly important for chronic disease where aetiology is uncertain and impact of lifestyle elements on the disease and the person are evolving. Maintaining these core principles of participant engagement, flexibility, and the ability to include novel datasets allows the collection of real-world data from individuals, their clinicians, and other relevant sources and stakeholders, leading to diverse and significant research into the disease. 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spelling v2 65409 2024-01-04 The Benefits of a United Kingdom Multiple Sclerosis Register 2a76cced6985381173d66002d3859019 RODDEN MIDDLETON RODDEN MIDDLETON true false 2024-01-04 Background: In rare diseases such as multiple sclerosis (MS) the capture of data purely from clinical trials or hospital cohorts is insufficient for fully understanding the multifaceted impacts of the disease. MS is the most common non-traumatic cause of disability in young adults worldwide. The disease is multifactorial, profoundly impacts quality of life, and life span is affected by 7 to 14 years. Diagnosis has become easier and newer treatment options have proliferated but monitoring and researching the disease's various impacts remains challenging with a largely clinical focus on ‘hard outcomes’ such as imaging, biomarkers and in-person clinically assessed scales. Disease registers are best positioned to capture data about chronic disease such as MS as they allow for longitudinal capture from a variety of sources, including clinical outcomes but also data from patients/participants. Moving disease registers away from paper-based capture has allowed for easier, more accurate and rapid capture from these sources but can also add new data sources such as novel outcome measures, data from other devices and then facilitate linkage of that data across all these domains. Aim: To communicate the learning and experiences from building a UK-wide register which captures multifaceted MS data, in order to inform the development of similar registers for other conditions. Methods: Construction of an electronic platform sufficiently flexible to capture data from people with MS in the form of patient/participant reported outcomes, from the NHS as clinical datasets, and the technology to link these datasets together in a privacy protecting way to make these datasets available to other researchers. The data capture technology must be robust enough to add additional sources or datasets as needed whilst maintaining the core elements of reproducible research. Results: A robust, flexible, privacy-protecting secure research-ready disease register was constructed containing data directly captured from more than 20,000 participants, 50 NHS sites with more than 1 million completed Patient Reported Outcomes (PRO), clinically transmitted datasets and other diverse outcomes collected. Pseudonymised elements of that data, subject to robust governance and review, are released to appropriately qualified researchers to carry out their own research on the platform. A number of important lessons were learned in the construction of this research register. The most important being that involvement of people affected by the disease in all aspects of the project is crucial for enabling key aspects of a functional register, including the collection of varied and complementary data, the levels of engagement required for longitudinal research, and assisting with the direction of research. Feedback loops in this participant-register relationship create a more holistic research instrument. Another vital aspect is the ability to carry out data linkage both within the project and to outside routinely collected datasets, expanding the scope of potential research without adding burden to participants. Flexibility of approach is particularly important for chronic disease where aetiology is uncertain and impact of lifestyle elements on the disease and the person are evolving. Maintaining these core principles of participant engagement, flexibility, and the ability to include novel datasets allows the collection of real-world data from individuals, their clinicians, and other relevant sources and stakeholders, leading to diverse and significant research into the disease. Conclusion: The UK MS Register can serve as a model for the design and construction of disease registers, capturing validated data from diverse data sources with reference to patients’ requirements and desires and satisfying researchers and clinicians needs for an unbiased, varied research ready dataset. E-Thesis Swansea, Wales, UK Multiple Sclerosis, Disease Registers, Health Informatics, Epidemiology, PROS 4 12 2023 2023-12-04 10.23889/SUthesis.65409 A selection of third party content is redacted/removed or is partially redacted/removed from this thesis due to copyright restrictions. COLLEGE NANME COLLEGE CODE Swansea University Brophy, Sinead. Doctoral Ph.D Swansea University Swansea University 2024-01-04T16:55:12.1141500 2024-01-04T16:37:51.3041194 Faculty of Medicine, Health and Life Sciences Swansea University Medical School - Medicine RODDEN MIDDLETON 1 65409__29386__905c343335dd44459cac2cb2c2d04651.pdf 2023_Middleton_RM.final.65409.pdf 2024-01-04T16:53:15.8251526 Output 17241068 application/pdf E-Thesis – open access true Copyright: The Author, Rodden M. Middleton, 2023. true eng
title The Benefits of a United Kingdom Multiple Sclerosis Register
spellingShingle The Benefits of a United Kingdom Multiple Sclerosis Register
RODDEN MIDDLETON
title_short The Benefits of a United Kingdom Multiple Sclerosis Register
title_full The Benefits of a United Kingdom Multiple Sclerosis Register
title_fullStr The Benefits of a United Kingdom Multiple Sclerosis Register
title_full_unstemmed The Benefits of a United Kingdom Multiple Sclerosis Register
title_sort The Benefits of a United Kingdom Multiple Sclerosis Register
author_id_str_mv 2a76cced6985381173d66002d3859019
author_id_fullname_str_mv 2a76cced6985381173d66002d3859019_***_RODDEN MIDDLETON
author RODDEN MIDDLETON
author2 RODDEN MIDDLETON
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publishDate 2023
institution Swansea University
doi_str_mv 10.23889/SUthesis.65409
college_str Faculty of Medicine, Health and Life Sciences
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hierarchy_top_title Faculty of Medicine, Health and Life Sciences
hierarchy_parent_id facultyofmedicinehealthandlifesciences
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department_str Swansea University Medical School - Medicine{{{_:::_}}}Faculty of Medicine, Health and Life Sciences{{{_:::_}}}Swansea University Medical School - Medicine
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description Background: In rare diseases such as multiple sclerosis (MS) the capture of data purely from clinical trials or hospital cohorts is insufficient for fully understanding the multifaceted impacts of the disease. MS is the most common non-traumatic cause of disability in young adults worldwide. The disease is multifactorial, profoundly impacts quality of life, and life span is affected by 7 to 14 years. Diagnosis has become easier and newer treatment options have proliferated but monitoring and researching the disease's various impacts remains challenging with a largely clinical focus on ‘hard outcomes’ such as imaging, biomarkers and in-person clinically assessed scales. Disease registers are best positioned to capture data about chronic disease such as MS as they allow for longitudinal capture from a variety of sources, including clinical outcomes but also data from patients/participants. Moving disease registers away from paper-based capture has allowed for easier, more accurate and rapid capture from these sources but can also add new data sources such as novel outcome measures, data from other devices and then facilitate linkage of that data across all these domains. Aim: To communicate the learning and experiences from building a UK-wide register which captures multifaceted MS data, in order to inform the development of similar registers for other conditions. Methods: Construction of an electronic platform sufficiently flexible to capture data from people with MS in the form of patient/participant reported outcomes, from the NHS as clinical datasets, and the technology to link these datasets together in a privacy protecting way to make these datasets available to other researchers. The data capture technology must be robust enough to add additional sources or datasets as needed whilst maintaining the core elements of reproducible research. Results: A robust, flexible, privacy-protecting secure research-ready disease register was constructed containing data directly captured from more than 20,000 participants, 50 NHS sites with more than 1 million completed Patient Reported Outcomes (PRO), clinically transmitted datasets and other diverse outcomes collected. Pseudonymised elements of that data, subject to robust governance and review, are released to appropriately qualified researchers to carry out their own research on the platform. A number of important lessons were learned in the construction of this research register. The most important being that involvement of people affected by the disease in all aspects of the project is crucial for enabling key aspects of a functional register, including the collection of varied and complementary data, the levels of engagement required for longitudinal research, and assisting with the direction of research. Feedback loops in this participant-register relationship create a more holistic research instrument. Another vital aspect is the ability to carry out data linkage both within the project and to outside routinely collected datasets, expanding the scope of potential research without adding burden to participants. Flexibility of approach is particularly important for chronic disease where aetiology is uncertain and impact of lifestyle elements on the disease and the person are evolving. Maintaining these core principles of participant engagement, flexibility, and the ability to include novel datasets allows the collection of real-world data from individuals, their clinicians, and other relevant sources and stakeholders, leading to diverse and significant research into the disease. Conclusion: The UK MS Register can serve as a model for the design and construction of disease registers, capturing validated data from diverse data sources with reference to patients’ requirements and desires and satisfying researchers and clinicians needs for an unbiased, varied research ready dataset.
published_date 2023-12-04T16:55:13Z
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