Disease-specific wearable sensor algorithms for profiling activity, gait, and balance in individuals with Charcot-Marie-Tooth disease type 1A
Charcot-Marie-Tooth Disease type 1A (CMT1A), the most common inherited peripheral neuropathy, is characterized by progressive sensory loss and weakness, which results in impaired mobility. Increased understanding of the genetics and pathophysiology of CMT1A has led to development of potential therap...
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| Veröffentlicht in: | Journal of the peripheral nervous system 2023-09, Vol.28 (3), p.368-381 |
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| creator | Dinesh, K White, N Baker, L Sowden, J E Behrens-Spraggins, S Wood, E Charles, J Herrmann, D N Sharma, G Eichinger, K |
| description | Charcot-Marie-Tooth Disease type 1A (CMT1A), the most common inherited peripheral neuropathy, is characterized by progressive sensory loss and weakness, which results in impaired mobility. Increased understanding of the genetics and pathophysiology of CMT1A has led to development of potential therapeutic agents, necessitating clinical trial readiness. Wearable sensors may provide useful outcome measures for future trials.
Individuals with CMT1A and unaffected controls were recruited for this 12-month study. Participants wore sensors for in-clinic assessments and at-home, from which activity, gait, and balance metrics were derived. Mann-Whitney U tests were used to analyze group differences for activity, gait, and balance parameters. Test-retest reliability of gait and balance parameters and correlations of these parameters with clinical outcome assessments (COAs) were examined.
Thirty individuals, 15 CMT1A, and 15 controls, participated. Gait and balance metrics demonstrated moderate to excellent reliability. CMT1A participants had longer step durations (p |
| doi_str_mv | 10.1111/jns.12562 |
| format | Article |
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Individuals with CMT1A and unaffected controls were recruited for this 12-month study. Participants wore sensors for in-clinic assessments and at-home, from which activity, gait, and balance metrics were derived. Mann-Whitney U tests were used to analyze group differences for activity, gait, and balance parameters. Test-retest reliability of gait and balance parameters and correlations of these parameters with clinical outcome assessments (COAs) were examined.
Thirty individuals, 15 CMT1A, and 15 controls, participated. Gait and balance metrics demonstrated moderate to excellent reliability. CMT1A participants had longer step durations (p < .001), shorter step lengths (p = .03), slower gait speeds (p < .001), and greater postural sway (p < .001) than healthy controls. Moderate correlations were found between CMT-Functional Outcome Measure and step length (r = -0.59; p = .02), and gait speed (r = 0.64; p = .01); 11 out of 15 CMT1A participants demonstrated significant increases in stride duration between the first and last quarter of the 6-min walk test, suggesting fatigue.
In this initial study, gait and balance metrics derived from wearable sensors were reliable and associated with COAs in individuals with CMT1A. Larger longitudinal studies are needed to confirm our findings and evaluate sensitivity and utility of these disease-specific algorithms for clinical trial use.</description><identifier>ISSN: 1085-9489</identifier><identifier>EISSN: 1529-8027</identifier><identifier>DOI: 10.1111/jns.12562</identifier><identifier>PMID: 37209301</identifier><language>eng</language><publisher>United States: Wiley Subscription Services, Inc</publisher><subject>Algorithms ; Balance ; Charcot-Marie-Tooth disease ; Clinical trials ; Gait ; Pathophysiology ; Peripheral neuropathy ; Sensitivity analysis ; Sensors</subject><ispartof>Journal of the peripheral nervous system, 2023-09, Vol.28 (3), p.368-381</ispartof><rights>2023 Peripheral Nerve Society.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c348t-8f962f7327d5f14711dbee2907a233095e5defb8ca3dbdf9a8dc399c200406b23</citedby><cites>FETCH-LOGICAL-c348t-8f962f7327d5f14711dbee2907a233095e5defb8ca3dbdf9a8dc399c200406b23</cites><orcidid>0000-0001-6042-6079 ; 0000-0001-9735-9519 ; 0000-0003-4623-7014</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,777,781,27905,27906</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/37209301$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Dinesh, K</creatorcontrib><creatorcontrib>White, N</creatorcontrib><creatorcontrib>Baker, L</creatorcontrib><creatorcontrib>Sowden, J E</creatorcontrib><creatorcontrib>Behrens-Spraggins, S</creatorcontrib><creatorcontrib>Wood, E</creatorcontrib><creatorcontrib>Charles, J</creatorcontrib><creatorcontrib>Herrmann, D N</creatorcontrib><creatorcontrib>Sharma, G</creatorcontrib><creatorcontrib>Eichinger, K</creatorcontrib><title>Disease-specific wearable sensor algorithms for profiling activity, gait, and balance in individuals with Charcot-Marie-Tooth disease type 1A</title><title>Journal of the peripheral nervous system</title><addtitle>J Peripher Nerv Syst</addtitle><description>Charcot-Marie-Tooth Disease type 1A (CMT1A), the most common inherited peripheral neuropathy, is characterized by progressive sensory loss and weakness, which results in impaired mobility. Increased understanding of the genetics and pathophysiology of CMT1A has led to development of potential therapeutic agents, necessitating clinical trial readiness. Wearable sensors may provide useful outcome measures for future trials.
Individuals with CMT1A and unaffected controls were recruited for this 12-month study. Participants wore sensors for in-clinic assessments and at-home, from which activity, gait, and balance metrics were derived. Mann-Whitney U tests were used to analyze group differences for activity, gait, and balance parameters. Test-retest reliability of gait and balance parameters and correlations of these parameters with clinical outcome assessments (COAs) were examined.
Thirty individuals, 15 CMT1A, and 15 controls, participated. Gait and balance metrics demonstrated moderate to excellent reliability. CMT1A participants had longer step durations (p < .001), shorter step lengths (p = .03), slower gait speeds (p < .001), and greater postural sway (p < .001) than healthy controls. Moderate correlations were found between CMT-Functional Outcome Measure and step length (r = -0.59; p = .02), and gait speed (r = 0.64; p = .01); 11 out of 15 CMT1A participants demonstrated significant increases in stride duration between the first and last quarter of the 6-min walk test, suggesting fatigue.
In this initial study, gait and balance metrics derived from wearable sensors were reliable and associated with COAs in individuals with CMT1A. Larger longitudinal studies are needed to confirm our findings and evaluate sensitivity and utility of these disease-specific algorithms for clinical trial use.</description><subject>Algorithms</subject><subject>Balance</subject><subject>Charcot-Marie-Tooth disease</subject><subject>Clinical trials</subject><subject>Gait</subject><subject>Pathophysiology</subject><subject>Peripheral neuropathy</subject><subject>Sensitivity analysis</subject><subject>Sensors</subject><issn>1085-9489</issn><issn>1529-8027</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2023</creationdate><recordtype>article</recordtype><recordid>eNpdkclOwzAQhi0EomwHXgBZ4gISAS9JHB9RWSUQl3KOHHvcukrjYiegPgTvjKGFA6ORxmN_nkU_QseUXNJkV_MuXlJWlGwL7dGCyawiTGynM6mKTOaVHKH9GOeEUCGp3EUjLhiRnNA99HnjIqgIWVyCdtZp_AEqqKYFHKGLPmDVTn1w_WwRsU3pMnjrWtdNsdK9e3f96gJPlesvsOoMblSrOg3YdclNejaDaiP-SP_xeKaC9n32rIKDbOJ9ujPr7rhfLQHT60O0YxMPR5t4gF7vbifjh-zp5f5xfP2UaZ5XfVZZWTIrOBOmsDQXlJoGgEkiFOOcyAIKA7aptOKmMVaqymgupWaE5KRsGD9AZ-u6aZu3AWJfL1zU0KbhwQ-xZhUtRSFzKRN6-g-d-yF0abpElZTkORM0UedrSgcfYwBbL4NbqLCqKam_NaqTRvWPRok92VQcmgWYP_JXFP4FO8yNzQ</recordid><startdate>20230901</startdate><enddate>20230901</enddate><creator>Dinesh, K</creator><creator>White, N</creator><creator>Baker, L</creator><creator>Sowden, J E</creator><creator>Behrens-Spraggins, S</creator><creator>Wood, E</creator><creator>Charles, J</creator><creator>Herrmann, D N</creator><creator>Sharma, G</creator><creator>Eichinger, K</creator><general>Wiley Subscription Services, Inc</general><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7TK</scope><scope>K9.</scope><scope>7X8</scope><orcidid>https://orcid.org/0000-0001-6042-6079</orcidid><orcidid>https://orcid.org/0000-0001-9735-9519</orcidid><orcidid>https://orcid.org/0000-0003-4623-7014</orcidid></search><sort><creationdate>20230901</creationdate><title>Disease-specific wearable sensor algorithms for profiling activity, gait, and balance in individuals with Charcot-Marie-Tooth disease type 1A</title><author>Dinesh, K ; White, N ; Baker, L ; Sowden, J E ; Behrens-Spraggins, S ; Wood, E ; Charles, J ; Herrmann, D N ; Sharma, G ; Eichinger, K</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c348t-8f962f7327d5f14711dbee2907a233095e5defb8ca3dbdf9a8dc399c200406b23</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2023</creationdate><topic>Algorithms</topic><topic>Balance</topic><topic>Charcot-Marie-Tooth disease</topic><topic>Clinical trials</topic><topic>Gait</topic><topic>Pathophysiology</topic><topic>Peripheral neuropathy</topic><topic>Sensitivity analysis</topic><topic>Sensors</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Dinesh, K</creatorcontrib><creatorcontrib>White, N</creatorcontrib><creatorcontrib>Baker, L</creatorcontrib><creatorcontrib>Sowden, J E</creatorcontrib><creatorcontrib>Behrens-Spraggins, S</creatorcontrib><creatorcontrib>Wood, E</creatorcontrib><creatorcontrib>Charles, J</creatorcontrib><creatorcontrib>Herrmann, D N</creatorcontrib><creatorcontrib>Sharma, G</creatorcontrib><creatorcontrib>Eichinger, K</creatorcontrib><collection>PubMed</collection><collection>CrossRef</collection><collection>Neurosciences Abstracts</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>MEDLINE - Academic</collection><jtitle>Journal of the peripheral nervous system</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Dinesh, K</au><au>White, N</au><au>Baker, L</au><au>Sowden, J E</au><au>Behrens-Spraggins, S</au><au>Wood, E</au><au>Charles, J</au><au>Herrmann, D N</au><au>Sharma, G</au><au>Eichinger, K</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Disease-specific wearable sensor algorithms for profiling activity, gait, and balance in individuals with Charcot-Marie-Tooth disease type 1A</atitle><jtitle>Journal of the peripheral nervous system</jtitle><addtitle>J Peripher Nerv Syst</addtitle><date>2023-09-01</date><risdate>2023</risdate><volume>28</volume><issue>3</issue><spage>368</spage><epage>381</epage><pages>368-381</pages><issn>1085-9489</issn><eissn>1529-8027</eissn><abstract>Charcot-Marie-Tooth Disease type 1A (CMT1A), the most common inherited peripheral neuropathy, is characterized by progressive sensory loss and weakness, which results in impaired mobility. Increased understanding of the genetics and pathophysiology of CMT1A has led to development of potential therapeutic agents, necessitating clinical trial readiness. Wearable sensors may provide useful outcome measures for future trials.
Individuals with CMT1A and unaffected controls were recruited for this 12-month study. Participants wore sensors for in-clinic assessments and at-home, from which activity, gait, and balance metrics were derived. Mann-Whitney U tests were used to analyze group differences for activity, gait, and balance parameters. Test-retest reliability of gait and balance parameters and correlations of these parameters with clinical outcome assessments (COAs) were examined.
Thirty individuals, 15 CMT1A, and 15 controls, participated. Gait and balance metrics demonstrated moderate to excellent reliability. CMT1A participants had longer step durations (p < .001), shorter step lengths (p = .03), slower gait speeds (p < .001), and greater postural sway (p < .001) than healthy controls. Moderate correlations were found between CMT-Functional Outcome Measure and step length (r = -0.59; p = .02), and gait speed (r = 0.64; p = .01); 11 out of 15 CMT1A participants demonstrated significant increases in stride duration between the first and last quarter of the 6-min walk test, suggesting fatigue.
In this initial study, gait and balance metrics derived from wearable sensors were reliable and associated with COAs in individuals with CMT1A. Larger longitudinal studies are needed to confirm our findings and evaluate sensitivity and utility of these disease-specific algorithms for clinical trial use.</abstract><cop>United States</cop><pub>Wiley Subscription Services, Inc</pub><pmid>37209301</pmid><doi>10.1111/jns.12562</doi><tpages>14</tpages><orcidid>https://orcid.org/0000-0001-6042-6079</orcidid><orcidid>https://orcid.org/0000-0001-9735-9519</orcidid><orcidid>https://orcid.org/0000-0003-4623-7014</orcidid><oa>free_for_read</oa></addata></record> |
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| subjects | Algorithms Balance Charcot-Marie-Tooth disease Clinical trials Gait Pathophysiology Peripheral neuropathy Sensitivity analysis Sensors |
| title | Disease-specific wearable sensor algorithms for profiling activity, gait, and balance in individuals with Charcot-Marie-Tooth disease type 1A |
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