Limitations of 6‐minute walk test reference values for spinal muscular atrophy

Introduction The 6‐minute walk test (6MWT) is a well‐established clinical assessment of functional endurance, validated as a measure of walking ability in spinal muscular atrophy (SMA). The current availability of disease‐modifying therapies for SMA indicates a growing need for normative reference d...

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Veröffentlicht in:Muscle & nerve 2020-03, Vol.61 (3), p.375-382
Hauptverfasser: Goodwin, Ashley M., Cornett, Kayla M.D., McKay, Marnee J., Burns, Joshua, Garber, Carol Ewing, De Vivo, Darryl C., Montes, Jacqueline
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container_end_page 382
container_issue 3
container_start_page 375
container_title Muscle & nerve
container_volume 61
creator Goodwin, Ashley M.
Cornett, Kayla M.D.
McKay, Marnee J.
Burns, Joshua
Garber, Carol Ewing
De Vivo, Darryl C.
Montes, Jacqueline
description Introduction The 6‐minute walk test (6MWT) is a well‐established clinical assessment of functional endurance, validated as a measure of walking ability in spinal muscular atrophy (SMA). The current availability of disease‐modifying therapies for SMA indicates a growing need for normative reference data to compare SMA patients with healthy controls. Methods The literature was searched in two scientific databases. Studies were evaluated and selected based on adherence to American Thoracic Society guidelines for administering the 6MWT. Reference equations from the selected studies were applied to 6MWT data collected from SMA patients to calculate and compare % predicted values. Results Three pediatric and six adult studies were selected for comparison. The % predicted values using the pediatric and adult equations ranged from 47.7 ± 18.2% to 67.6 ± 26.2% and 43.0 ± 17.9% to 59.5 ± 26.2%, respectively, and were significantly different (P 
doi_str_mv 10.1002/mus.26794
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The current availability of disease‐modifying therapies for SMA indicates a growing need for normative reference data to compare SMA patients with healthy controls. Methods The literature was searched in two scientific databases. Studies were evaluated and selected based on adherence to American Thoracic Society guidelines for administering the 6MWT. Reference equations from the selected studies were applied to 6MWT data collected from SMA patients to calculate and compare % predicted values. Results Three pediatric and six adult studies were selected for comparison. The % predicted values using the pediatric and adult equations ranged from 47.7 ± 18.2% to 67.6 ± 26.2% and 43.0 ± 17.9% to 59.5 ± 26.2%, respectively, and were significantly different (P &lt; 0.001). Discussion Results suggest significant variability between % predicted values derived from published reference equations in children and adults, despite adherence to 6MWT standardization.</description><identifier>ISSN: 0148-639X</identifier><identifier>EISSN: 1097-4598</identifier><identifier>DOI: 10.1002/mus.26794</identifier><identifier>PMID: 31884700</identifier><language>eng</language><publisher>Hoboken, USA: John Wiley &amp; Sons, Inc</publisher><subject>6‐minute walk test ; Adolescent ; Atrophy ; Child ; Child, Preschool ; Control methods ; Fatigue tests ; Female ; Humans ; Male ; Mathematical analysis ; Muscular Atrophy, Spinal - diagnosis ; Muscular Atrophy, Spinal - physiopathology ; neuromuscular disease ; Neuromuscular diseases ; normative reference ; outcome measure ; Patients ; Pediatrics ; Reference Values ; Spinal muscular atrophy ; Standardization ; Thorax ; Walk Test - standards ; Walking</subject><ispartof>Muscle &amp; nerve, 2020-03, Vol.61 (3), p.375-382</ispartof><rights>2019 Wiley Periodicals, Inc.</rights><rights>2020 Wiley Periodicals, Inc.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c4434-73fb4c243cd59110b62686550f152567f50a8b6215c950293cf258784cb90aeb3</citedby><cites>FETCH-LOGICAL-c4434-73fb4c243cd59110b62686550f152567f50a8b6215c950293cf258784cb90aeb3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://onlinelibrary.wiley.com/doi/pdf/10.1002%2Fmus.26794$$EPDF$$P50$$Gwiley$$H</linktopdf><linktohtml>$$Uhttps://onlinelibrary.wiley.com/doi/full/10.1002%2Fmus.26794$$EHTML$$P50$$Gwiley$$H</linktohtml><link.rule.ids>230,314,780,784,885,1417,27924,27925,45574,45575</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/31884700$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Goodwin, Ashley M.</creatorcontrib><creatorcontrib>Cornett, Kayla M.D.</creatorcontrib><creatorcontrib>McKay, Marnee J.</creatorcontrib><creatorcontrib>Burns, Joshua</creatorcontrib><creatorcontrib>Garber, Carol Ewing</creatorcontrib><creatorcontrib>De Vivo, Darryl C.</creatorcontrib><creatorcontrib>Montes, Jacqueline</creatorcontrib><title>Limitations of 6‐minute walk test reference values for spinal muscular atrophy</title><title>Muscle &amp; nerve</title><addtitle>Muscle Nerve</addtitle><description>Introduction The 6‐minute walk test (6MWT) is a well‐established clinical assessment of functional endurance, validated as a measure of walking ability in spinal muscular atrophy (SMA). The current availability of disease‐modifying therapies for SMA indicates a growing need for normative reference data to compare SMA patients with healthy controls. Methods The literature was searched in two scientific databases. Studies were evaluated and selected based on adherence to American Thoracic Society guidelines for administering the 6MWT. Reference equations from the selected studies were applied to 6MWT data collected from SMA patients to calculate and compare % predicted values. Results Three pediatric and six adult studies were selected for comparison. The % predicted values using the pediatric and adult equations ranged from 47.7 ± 18.2% to 67.6 ± 26.2% and 43.0 ± 17.9% to 59.5 ± 26.2%, respectively, and were significantly different (P &lt; 0.001). 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Medical Complete (Alumni)</collection><collection>Nursing &amp; Allied Health Premium</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>Muscle &amp; nerve</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Goodwin, Ashley M.</au><au>Cornett, Kayla M.D.</au><au>McKay, Marnee J.</au><au>Burns, Joshua</au><au>Garber, Carol Ewing</au><au>De Vivo, Darryl C.</au><au>Montes, Jacqueline</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Limitations of 6‐minute walk test reference values for spinal muscular atrophy</atitle><jtitle>Muscle &amp; nerve</jtitle><addtitle>Muscle Nerve</addtitle><date>2020-03</date><risdate>2020</risdate><volume>61</volume><issue>3</issue><spage>375</spage><epage>382</epage><pages>375-382</pages><issn>0148-639X</issn><eissn>1097-4598</eissn><abstract>Introduction The 6‐minute walk test (6MWT) is a well‐established clinical assessment of functional endurance, validated as a measure of walking ability in spinal muscular atrophy (SMA). The current availability of disease‐modifying therapies for SMA indicates a growing need for normative reference data to compare SMA patients with healthy controls. Methods The literature was searched in two scientific databases. Studies were evaluated and selected based on adherence to American Thoracic Society guidelines for administering the 6MWT. Reference equations from the selected studies were applied to 6MWT data collected from SMA patients to calculate and compare % predicted values. Results Three pediatric and six adult studies were selected for comparison. The % predicted values using the pediatric and adult equations ranged from 47.7 ± 18.2% to 67.6 ± 26.2% and 43.0 ± 17.9% to 59.5 ± 26.2%, respectively, and were significantly different (P &lt; 0.001). 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subjects 6‐minute walk test
Adolescent
Atrophy
Child
Child, Preschool
Control methods
Fatigue tests
Female
Humans
Male
Mathematical analysis
Muscular Atrophy, Spinal - diagnosis
Muscular Atrophy, Spinal - physiopathology
neuromuscular disease
Neuromuscular diseases
normative reference
outcome measure
Patients
Pediatrics
Reference Values
Spinal muscular atrophy
Standardization
Thorax
Walk Test - standards
Walking
title Limitations of 6‐minute walk test reference values for spinal muscular atrophy
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