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Patient-reported outcomes in multiple sclerosis: a prospective registry cohort study

Annalaura Lerede Orcid Logo, Jeff Rodgers Orcid Logo, Rod Middleton Orcid Logo, Adam Hampshire Orcid Logo, Richard Nicholas Orcid Logo, Alasdair Coles, Jeremy Chataway, Martin Duddy, Hedley Emsley, Helen Ford, Leonora Fisniku, Ian Galea, Timothy Harrower, Jeremy Hobart, Huseyin Huseyin, Christopher M Kipps, Monica Marta, Gavin V McDonnell, Brendan McLean, Owen R Pearson, David Rog, Klaus Schmierer, Basil Sharrack, Agne Straukiene, David Ford Orcid Logo, (The UK MS Register Research Group)

Brain Communications, Volume: 5, Issue: 4

Swansea University Authors: Rod Middleton Orcid Logo, David Ford Orcid Logo

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DOI (Published version): 10.1093/braincomms/fcad199

Abstract

Registries have the potential to tackle some of the current limitations in determining the long-term impact of multiple sclerosis. Online assessments using patient-reported outcomes can streamline follow-up enabling large-scale, long-term, cost-effective, home-based, and patient-focused data collect...

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Published in: Brain Communications
ISSN: 2632-1297
Published: Oxford University Press (OUP) 2023
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URI: https://cronfa.swan.ac.uk/Record/cronfa66407
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Online assessments using patient-reported outcomes can streamline follow-up enabling large-scale, long-term, cost-effective, home-based, and patient-focused data collection. However, registry data are sparsely sampled and the sensitivity of patient-reported outcomes relative to clinician-reported scales is unknown, making it hard to fully leverage their unique scope and scale to derive insights. This retrospective and prospective cohort study over 11 years involved 15 976 patients with multiple sclerosis from the United Kingdom Multiples Sclerosis Register. Primary outcomes were changes in two patient-reported outcomes: Multiple Sclerosis Impact Scale motor component, and Multiple Sclerosis Walking Scale. First, we investigated their validity in measuring the impact of physical disability in multiple sclerosis, by looking at their sensitivity to disease subtype and duration. We grouped the available records (91 351 for Multiple Sclerosis Impact Scale motor and 68 092 for Multiple Sclerosis Walking Scale) by these two factors, and statistically compared the resulting groups using a novel approach based on Monte Carlo permutation analysis that was designed to cope with the intrinsic sparsity of registry data. Next, we used the patient-reported outcomes to draw novel insights into the developmental time course of subtypes; in particular, the period preceding the transition from relapsing to progressive forms. We report a robust main effect of disease subtype on the patient-reported outcomes and interactions of disease subtype with duration (all P &lt; 0.0001). Specifically, patient-reported outcomes worsen with disease duration for all subtypes (all P &lt; 0.0001) apart from benign multiple sclerosis (Multiple Sclerosis Impact Scale motor: P = 0.796; Multiple Sclerosis Walking Scale: P = 0.983). Furthermore, the patient-reported outcomes of each subtype are statistically different from those of the other subtypes at all time bins (Multiple Sclerosis Impact Scale motor: all P &lt; 0.05; Multiple Sclerosis Walking Scale: all P &lt; 0.01) except when comparing relapsing-remitting multiple sclerosis with benign multiple sclerosis and primary progressive multiple sclerosis with secondary progressive multiple sclerosis. Notably, there were statistically significant differences between relapsing-remitting and progressive subtypes at disease onset. Critically, the patient-reported outcomes are sensitive to future transitions to progressive subtypes, with individuals who transition presenting with higher patient-reported outcomes in their relapsing-remitting phase compared to individuals who don’t transition since onset (all P &lt; 0.0001). Patient-reported outcomes capture different patterns of physical worsening over disease length and across subtypes; therefore, they are a valid tool to measure the physical impact of multiple sclerosis over the long-term and cost-effectively. Furthermore, more advanced physical disability manifests years before clinical detection of progressive subtypes, adding evidence to the presence of a multiple sclerosis prodrome.</abstract><type>Journal Article</type><journal>Brain Communications</journal><volume>5</volume><journalNumber>4</journalNumber><paginationStart/><paginationEnd/><publisher>Oxford University Press (OUP)</publisher><placeOfPublication/><isbnPrint/><isbnElectronic/><issnPrint/><issnElectronic>2632-1297</issnElectronic><keywords>multiple sclerosis, patient-reported outcomes, online registry</keywords><publishedDay>20</publishedDay><publishedMonth>8</publishedMonth><publishedYear>2023</publishedYear><publishedDate>2023-08-20</publishedDate><doi>10.1093/braincomms/fcad199</doi><url/><notes/><college>COLLEGE NANME</college><department>Medical School</department><CollegeCode>COLLEGE CODE</CollegeCode><DepartmentCode>MEDS</DepartmentCode><institution>Swansea University</institution><apcterm>Another institution paid the OA fee</apcterm><funders>A.L. is supported by the UK Research and Innovation Centre for Doctoral Training in Artificial Intelligence for Healthcare http://ai4health.io (grant number EP/S023283/1). A.L. and A.H. are supported by the UK Dementia Research Institute Care Research and Technology Centre. A.L., J.R., R.M. and R.N. are supported by the UK Multiple Sclerosis Society. R.N. is supported by the Multiple Sclerosis Trials Collaboration (MSTC) and the Berkeley Foundation.</funders><projectreference/><lastEdited>2024-06-19T15:19:09.7428711</lastEdited><Created>2024-05-14T10:01:55.7589744</Created><path><level id="1">Faculty of Medicine, Health and Life Sciences</level><level id="2">Swansea University Medical School - Health Data Science</level></path><authors><author><firstname>Annalaura</firstname><surname>Lerede</surname><orcid>0000-0003-0903-8606</orcid><order>1</order></author><author><firstname>Jeff</firstname><surname>Rodgers</surname><orcid>0000-0003-0757-3664</orcid><order>2</order></author><author><firstname>Rod</firstname><surname>Middleton</surname><orcid>0000-0002-2130-4420</orcid><order>3</order></author><author><firstname>Adam</firstname><surname>Hampshire</surname><orcid>0000-0002-5176-5420</orcid><order>4</order></author><author><firstname>Richard</firstname><surname>Nicholas</surname><orcid>0000-0003-0414-1225</orcid><order>5</order></author><author><firstname>Alasdair</firstname><surname>Coles</surname><order>6</order></author><author><firstname>Jeremy</firstname><surname>Chataway</surname><order>7</order></author><author><firstname>Martin</firstname><surname>Duddy</surname><order>8</order></author><author><firstname>Hedley</firstname><surname>Emsley</surname><order>9</order></author><author><firstname>Helen</firstname><surname>Ford</surname><order>10</order></author><author><firstname>Leonora</firstname><surname>Fisniku</surname><order>11</order></author><author><firstname>Ian</firstname><surname>Galea</surname><order>12</order></author><author><firstname>Timothy</firstname><surname>Harrower</surname><order>13</order></author><author><firstname>Jeremy</firstname><surname>Hobart</surname><order>14</order></author><author><firstname>Huseyin</firstname><surname>Huseyin</surname><order>15</order></author><author><firstname>Christopher M</firstname><surname>Kipps</surname><order>16</order></author><author><firstname>Monica</firstname><surname>Marta</surname><order>17</order></author><author><firstname>Gavin V</firstname><surname>McDonnell</surname><order>18</order></author><author><firstname>Brendan</firstname><surname>McLean</surname><order>19</order></author><author><firstname>Owen R</firstname><surname>Pearson</surname><order>20</order></author><author><firstname>David</firstname><surname>Rog</surname><order>21</order></author><author><firstname>Klaus</firstname><surname>Schmierer</surname><order>22</order></author><author><firstname>Basil</firstname><surname>Sharrack</surname><order>23</order></author><author><firstname>Agne</firstname><surname>Straukiene</surname><order>24</order></author><author><firstname>David</firstname><surname>Ford</surname><orcid>0000-0001-6551-721X</orcid><order>25</order></author><author><firstname>(The UK MS Register Research</firstname><surname>Group)</surname><order>26</order></author></authors><documents><document><filename>66407__30347__c85a75cc7ec544f194c54e48d1627d4c.pdf</filename><originalFilename>66407.pdf</originalFilename><uploaded>2024-05-14T10:18:45.1135149</uploaded><type>Output</type><contentLength>1932550</contentLength><contentType>application/pdf</contentType><version>Version of Record</version><cronfaStatus>true</cronfaStatus><documentNotes>© The Author(s) 2023. 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spelling v2 66407 2024-05-14 Patient-reported outcomes in multiple sclerosis: a prospective registry cohort study 005518f819ef1a2a13fdf438529bdfcd 0000-0002-2130-4420 Rod Middleton Rod Middleton true false 52fc0c473b0da1b7218d87f9fc68a3e6 0000-0001-6551-721X David Ford David Ford true false 2024-05-14 MEDS Registries have the potential to tackle some of the current limitations in determining the long-term impact of multiple sclerosis. Online assessments using patient-reported outcomes can streamline follow-up enabling large-scale, long-term, cost-effective, home-based, and patient-focused data collection. However, registry data are sparsely sampled and the sensitivity of patient-reported outcomes relative to clinician-reported scales is unknown, making it hard to fully leverage their unique scope and scale to derive insights. This retrospective and prospective cohort study over 11 years involved 15 976 patients with multiple sclerosis from the United Kingdom Multiples Sclerosis Register. Primary outcomes were changes in two patient-reported outcomes: Multiple Sclerosis Impact Scale motor component, and Multiple Sclerosis Walking Scale. First, we investigated their validity in measuring the impact of physical disability in multiple sclerosis, by looking at their sensitivity to disease subtype and duration. We grouped the available records (91 351 for Multiple Sclerosis Impact Scale motor and 68 092 for Multiple Sclerosis Walking Scale) by these two factors, and statistically compared the resulting groups using a novel approach based on Monte Carlo permutation analysis that was designed to cope with the intrinsic sparsity of registry data. Next, we used the patient-reported outcomes to draw novel insights into the developmental time course of subtypes; in particular, the period preceding the transition from relapsing to progressive forms. We report a robust main effect of disease subtype on the patient-reported outcomes and interactions of disease subtype with duration (all P < 0.0001). Specifically, patient-reported outcomes worsen with disease duration for all subtypes (all P < 0.0001) apart from benign multiple sclerosis (Multiple Sclerosis Impact Scale motor: P = 0.796; Multiple Sclerosis Walking Scale: P = 0.983). Furthermore, the patient-reported outcomes of each subtype are statistically different from those of the other subtypes at all time bins (Multiple Sclerosis Impact Scale motor: all P < 0.05; Multiple Sclerosis Walking Scale: all P < 0.01) except when comparing relapsing-remitting multiple sclerosis with benign multiple sclerosis and primary progressive multiple sclerosis with secondary progressive multiple sclerosis. Notably, there were statistically significant differences between relapsing-remitting and progressive subtypes at disease onset. Critically, the patient-reported outcomes are sensitive to future transitions to progressive subtypes, with individuals who transition presenting with higher patient-reported outcomes in their relapsing-remitting phase compared to individuals who don’t transition since onset (all P < 0.0001). Patient-reported outcomes capture different patterns of physical worsening over disease length and across subtypes; therefore, they are a valid tool to measure the physical impact of multiple sclerosis over the long-term and cost-effectively. Furthermore, more advanced physical disability manifests years before clinical detection of progressive subtypes, adding evidence to the presence of a multiple sclerosis prodrome. Journal Article Brain Communications 5 4 Oxford University Press (OUP) 2632-1297 multiple sclerosis, patient-reported outcomes, online registry 20 8 2023 2023-08-20 10.1093/braincomms/fcad199 COLLEGE NANME Medical School COLLEGE CODE MEDS Swansea University Another institution paid the OA fee A.L. is supported by the UK Research and Innovation Centre for Doctoral Training in Artificial Intelligence for Healthcare http://ai4health.io (grant number EP/S023283/1). A.L. and A.H. are supported by the UK Dementia Research Institute Care Research and Technology Centre. A.L., J.R., R.M. and R.N. are supported by the UK Multiple Sclerosis Society. R.N. is supported by the Multiple Sclerosis Trials Collaboration (MSTC) and the Berkeley Foundation. 2024-06-19T15:19:09.7428711 2024-05-14T10:01:55.7589744 Faculty of Medicine, Health and Life Sciences Swansea University Medical School - Health Data Science Annalaura Lerede 0000-0003-0903-8606 1 Jeff Rodgers 0000-0003-0757-3664 2 Rod Middleton 0000-0002-2130-4420 3 Adam Hampshire 0000-0002-5176-5420 4 Richard Nicholas 0000-0003-0414-1225 5 Alasdair Coles 6 Jeremy Chataway 7 Martin Duddy 8 Hedley Emsley 9 Helen Ford 10 Leonora Fisniku 11 Ian Galea 12 Timothy Harrower 13 Jeremy Hobart 14 Huseyin Huseyin 15 Christopher M Kipps 16 Monica Marta 17 Gavin V McDonnell 18 Brendan McLean 19 Owen R Pearson 20 David Rog 21 Klaus Schmierer 22 Basil Sharrack 23 Agne Straukiene 24 David Ford 0000-0001-6551-721X 25 (The UK MS Register Research Group) 26 66407__30347__c85a75cc7ec544f194c54e48d1627d4c.pdf 66407.pdf 2024-05-14T10:18:45.1135149 Output 1932550 application/pdf Version of Record true © The Author(s) 2023. This is an Open Access article distributed under the terms of the Creative Commons Attribution License. true eng https://creativecommons.org/licenses/by/4.0/
title Patient-reported outcomes in multiple sclerosis: a prospective registry cohort study
spellingShingle Patient-reported outcomes in multiple sclerosis: a prospective registry cohort study
Rod Middleton
David Ford
title_short Patient-reported outcomes in multiple sclerosis: a prospective registry cohort study
title_full Patient-reported outcomes in multiple sclerosis: a prospective registry cohort study
title_fullStr Patient-reported outcomes in multiple sclerosis: a prospective registry cohort study
title_full_unstemmed Patient-reported outcomes in multiple sclerosis: a prospective registry cohort study
title_sort Patient-reported outcomes in multiple sclerosis: a prospective registry cohort study
author_id_str_mv 005518f819ef1a2a13fdf438529bdfcd
52fc0c473b0da1b7218d87f9fc68a3e6
author_id_fullname_str_mv 005518f819ef1a2a13fdf438529bdfcd_***_Rod Middleton
52fc0c473b0da1b7218d87f9fc68a3e6_***_David Ford
author Rod Middleton
David Ford
author2 Annalaura Lerede
Jeff Rodgers
Rod Middleton
Adam Hampshire
Richard Nicholas
Alasdair Coles
Jeremy Chataway
Martin Duddy
Hedley Emsley
Helen Ford
Leonora Fisniku
Ian Galea
Timothy Harrower
Jeremy Hobart
Huseyin Huseyin
Christopher M Kipps
Monica Marta
Gavin V McDonnell
Brendan McLean
Owen R Pearson
David Rog
Klaus Schmierer
Basil Sharrack
Agne Straukiene
David Ford
(The UK MS Register Research Group)
format Journal article
container_title Brain Communications
container_volume 5
container_issue 4
publishDate 2023
institution Swansea University
issn 2632-1297
doi_str_mv 10.1093/braincomms/fcad199
publisher Oxford University Press (OUP)
college_str Faculty of Medicine, Health and Life Sciences
hierarchytype
hierarchy_top_id facultyofmedicinehealthandlifesciences
hierarchy_top_title Faculty of Medicine, Health and Life Sciences
hierarchy_parent_id facultyofmedicinehealthandlifesciences
hierarchy_parent_title Faculty of Medicine, Health and Life Sciences
department_str Swansea University Medical School - Health Data Science{{{_:::_}}}Faculty of Medicine, Health and Life Sciences{{{_:::_}}}Swansea University Medical School - Health Data Science
document_store_str 1
active_str 0
description Registries have the potential to tackle some of the current limitations in determining the long-term impact of multiple sclerosis. Online assessments using patient-reported outcomes can streamline follow-up enabling large-scale, long-term, cost-effective, home-based, and patient-focused data collection. However, registry data are sparsely sampled and the sensitivity of patient-reported outcomes relative to clinician-reported scales is unknown, making it hard to fully leverage their unique scope and scale to derive insights. This retrospective and prospective cohort study over 11 years involved 15 976 patients with multiple sclerosis from the United Kingdom Multiples Sclerosis Register. Primary outcomes were changes in two patient-reported outcomes: Multiple Sclerosis Impact Scale motor component, and Multiple Sclerosis Walking Scale. First, we investigated their validity in measuring the impact of physical disability in multiple sclerosis, by looking at their sensitivity to disease subtype and duration. We grouped the available records (91 351 for Multiple Sclerosis Impact Scale motor and 68 092 for Multiple Sclerosis Walking Scale) by these two factors, and statistically compared the resulting groups using a novel approach based on Monte Carlo permutation analysis that was designed to cope with the intrinsic sparsity of registry data. Next, we used the patient-reported outcomes to draw novel insights into the developmental time course of subtypes; in particular, the period preceding the transition from relapsing to progressive forms. We report a robust main effect of disease subtype on the patient-reported outcomes and interactions of disease subtype with duration (all P < 0.0001). Specifically, patient-reported outcomes worsen with disease duration for all subtypes (all P < 0.0001) apart from benign multiple sclerosis (Multiple Sclerosis Impact Scale motor: P = 0.796; Multiple Sclerosis Walking Scale: P = 0.983). Furthermore, the patient-reported outcomes of each subtype are statistically different from those of the other subtypes at all time bins (Multiple Sclerosis Impact Scale motor: all P < 0.05; Multiple Sclerosis Walking Scale: all P < 0.01) except when comparing relapsing-remitting multiple sclerosis with benign multiple sclerosis and primary progressive multiple sclerosis with secondary progressive multiple sclerosis. Notably, there were statistically significant differences between relapsing-remitting and progressive subtypes at disease onset. Critically, the patient-reported outcomes are sensitive to future transitions to progressive subtypes, with individuals who transition presenting with higher patient-reported outcomes in their relapsing-remitting phase compared to individuals who don’t transition since onset (all P < 0.0001). Patient-reported outcomes capture different patterns of physical worsening over disease length and across subtypes; therefore, they are a valid tool to measure the physical impact of multiple sclerosis over the long-term and cost-effectively. Furthermore, more advanced physical disability manifests years before clinical detection of progressive subtypes, adding evidence to the presence of a multiple sclerosis prodrome.
published_date 2023-08-20T15:19:08Z
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score 11.016235

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