{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,3]],"date-time":"2026-04-03T12:52:53Z","timestamp":1775220773505,"version":"3.50.1"},"reference-count":25,"publisher":"FapUNIFESP (SciELO)","issue":"1","license":[{"start":{"date-parts":[[2026,1,1]],"date-time":"2026-01-01T00:00:00Z","timestamp":1767225600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2026,1,1]],"date-time":"2026-01-01T00:00:00Z","timestamp":1767225600000},"content-version":"am","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2026,1,1]],"date-time":"2026-01-01T00:00:00Z","timestamp":1767225600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Rev. Assoc. Med. Bras."],"abstract":"<jats:p>SUMMARY  OBJECTIVE: The aims of this study were to determine whether the six-minute walk test predicts muscular fatigability in ambulant children with congenital myasthenic syndrome and to examine the relationship between the six-minute walk test and mobility tests.  METHODS: Eight children (four girls and four boys) with congenital myasthenic syndrome (mean age: 11.75\u00b13.54 years) were included. Five children had COLQ variants. The distance of each minute for the six-minute walk test was determined to evaluate fatigability. The mobility tests included the 10-m walk\/run test, rising from the floor, and climbing up and down four stairs.  RESULTS: The mean six-minute walk test distance was 463.38\u00b1132.59 m. There was no difference between the first and last minute, between the first and last two minutes, and between any of the minutes during the six-minute walk test (p&gt;0.05). The mean times for performing the 10-m walk\/run test, rising from the floor, and climbing up and down four stairs were 8.59\u00b13.36, 6.38\u00b14.49, 4.38\u00b15.07, and 3.35\u00b13.69 s, respectively. The strong correlations were determined between six-minute walk test and all the mobility tests, except for climbing down four stairs (p&lt; 0.05, rs=-0.72 to -0.83).  CONCLUSION: The six-minute walk test did not provide sufficient evidence to predict muscular fatigability. However, this preliminary study suggested that the six-minute walk test may be used by clinicians and researchers to assess mobility in congenital myasthenic syndrome.<\/jats:p>","DOI":"10.1590\/1806-9282.20250307","type":"journal-article","created":{"date-parts":[[2026,4,3]],"date-time":"2026-04-03T11:49:58Z","timestamp":1775216998000},"source":"Crossref","is-referenced-by-count":0,"title":["Six-minute walk test to assess muscular fatigability and mobility in children with congenital myasthenic syndrome: a pilot study"],"prefix":"10.1590","volume":"72","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-5427-1103","authenticated-orcid":false,"given":"Numan","family":"Bulut","sequence":"first","affiliation":[{"name":"Hacettepe University,  Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1658-7697","authenticated-orcid":false,"given":"G\u00fcll\u00fc Ayd\u0131n","family":"Ya\u011fc\u0131o\u011flu","sequence":"additional","affiliation":[{"name":"University of Health Sciences,  Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8110-8720","authenticated-orcid":false,"given":"Aykut","family":"Demir","sequence":"additional","affiliation":[{"name":"Hacettepe University,  Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5556-6608","authenticated-orcid":false,"given":"\u0130pek","family":"Alemdaro\u011flu-G\u00fcrb\u00fcz","sequence":"additional","affiliation":[{"name":"Hacettepe University,  Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1813-275X","authenticated-orcid":false,"given":"G\u00f6knur","family":"Halilo\u011flu","sequence":"additional","affiliation":[{"name":"Hacettepe University,  Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0855-9541","authenticated-orcid":false,"given":"\u00d6znur","family":"Tunca","sequence":"additional","affiliation":[{"name":"Hacettepe University,  Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"530","published-online":{"date-parts":[[2026]]},"reference":[{"issue":"4","key":"ref1","doi-asserted-by":"crossref","first-page":"3730","DOI":"10.3390\/ijms24043730","article-title":"Clinical and pathologic features of congenital myasthenic syndromes caused by 35 genes-a comprehensive review","volume":"24","author":"Ohno K","year":"2023","journal-title":"Int J Mol Sci"},{"key":"ref2","doi-asserted-by":"crossref","first-page":"5","DOI":"10.1016\/j.ejpn.2020.04.011","article-title":"Prevelance in congenital myasthenic syndrome","volume":"26","author":"Finsterer J","year":"2020","journal-title":"Eur J Paediatr Neurol"},{"issue":"10","key":"ref3","doi-asserted-by":"crossref","first-page":"943","DOI":"10.1016\/j.nmd.2021.07.400","article-title":"Congenital myasthenic syndromes: where do we go from here?","volume":"31","author":"Ramdas S","year":"2021","journal-title":"Neuromuscul Disord"},{"issue":"5","key":"ref4","doi-asserted-by":"crossref","first-page":"5005","DOI":"10.3390\/ijms24055005","article-title":"Pathophysiology and management of fatigue in neuromuscular diseases","volume":"24","author":"Torri F","year":"2023","journal-title":"Int J Mol Sci"},{"issue":"5","key":"ref5","first-page":"769","article-title":"A pediatric case of COLQ-related congenital myasthenic syndrome with marked fatigue","volume":"10","author":"Horibe T","year":"2023","journal-title":"Children (Basel)"},{"issue":"1","key":"ref6","doi-asserted-by":"crossref","first-page":"111","DOI":"10.1164\/ajrccm.166.1.at1102","article-title":"ATS statement: guidelines for the six-minute walk test","volume":"166","year":"2002","journal-title":"Am J Respir Crit Care Med"},{"issue":"2","key":"ref7","doi-asserted-by":"crossref","first-page":"581","DOI":"10.3390\/s22020581","article-title":"Development technologies for the monitoring of six-minute walk test: a systematic review","volume":"22","author":"Pires IM","year":"2022","journal-title":"Sensors (Basel)"},{"issue":"1","key":"ref8","doi-asserted-by":"crossref","first-page":"297","DOI":"10.1186\/s12890-021-01666-5","article-title":"Normative data for the 6-min walk test in 11-14 year-olds: a population-based study","volume":"21","author":"Kasovi\u0107 M","year":"2021","journal-title":"BMC Pulm Med"},{"issue":"1","key":"ref9","doi-asserted-by":"crossref","first-page":"81","DOI":"10.1007\/s10072-020-04829-0","article-title":"Reliability of the six-minute walk test in individuals with stroke: systematic review and meta-analysis","volume":"42","author":"Macchiavelli A","year":"2021","journal-title":"Neurol Sci"},{"issue":"2","key":"ref10","doi-asserted-by":"crossref","first-page":"208","DOI":"10.1002\/mus.26345","article-title":"Functional exercise capacity evaluated by timed walk tests in myasthenia gravis","volume":"59","author":"Salci Y","year":"2019","journal-title":"Muscle Nerve"},{"issue":"3","key":"ref11","doi-asserted-by":"crossref","first-page":"333","DOI":"10.1002\/mus.25251","article-title":"Validity of the 6 minute walk test in facioscapulohumeral muscular dystrophy","volume":"55","author":"Eichinger K","year":"2017","journal-title":"Muscle Nerve"},{"issue":"10","key":"ref12","doi-asserted-by":"crossref","first-page":"833","DOI":"10.1212\/WNL.0b013e3181d3e308","article-title":"Six-minute walk test demonstrates motor fatigue in spinal muscular atrophy","volume":"74","author":"Montes J","year":"2010","journal-title":"Neurology"},{"issue":"1","key":"ref13","doi-asserted-by":"crossref","first-page":"105","DOI":"10.1186\/s13023-018-0848-9","article-title":"6-minute walk test as a measure of disease progression and fatigability in a cohort of individuals with RYR1-related myopathies","volume":"13","author":"Witherspoon JW","year":"2018","journal-title":"Orphanet J Rare Dis"},{"issue":"Supp 1","key":"ref14","doi-asserted-by":"crossref","first-page":"104441.628","DOI":"10.1016\/j.nmd.2024.07.637","article-title":"Exploring the relationship between an instrumented walking test and community physical activity in individuals with congenital myasthenic syndrome","volume":"43","author":"Ramjattan H","year":"2024","journal-title":"Neuromuscul Disord"},{"issue":"9","key":"ref15","doi-asserted-by":"crossref","first-page":"111","DOI":"10.1016\/j.nmd.2022.12.006","article-title":"260th ENMC international workshop: congenital myasthenic syndromes 11-13 March 2022, hoofddorp, The Netherlands","volume":"33","author":"Spendiff S","year":"2023","journal-title":"Neuromuscul Disord"},{"issue":"6","key":"ref16","doi-asserted-by":"crossref","first-page":"939","DOI":"10.3233\/JND-210646","article-title":"The Minimal Clinical Important Difference (MCID) in annual rate of change of timed function tests in boys with DMD","volume":"8","author":"Duong T","year":"2021","journal-title":"J Neuromuscul Dis"},{"issue":"5","key":"ref17","doi-asserted-by":"crossref","first-page":"644","DOI":"10.1111\/dmcn.15788","article-title":"Reference curves of motor function outcomes in young steroid-na\u00efve males with Duchenne muscular dystrophy","volume":"66","author":"Hoskens J","year":"2024","journal-title":"Dev Med Child Neurol"},{"issue":"3","key":"ref18","doi-asserted-by":"crossref","first-page":"91","DOI":"10.1016\/j.tjem.2018.08.001","article-title":"User#8217;s guide to correlation coefficients","volume":"18","author":"Akoglu H","year":"2018","journal-title":"Turk J Emerg Med"},{"issue":"4","key":"ref19","doi-asserted-by":"crossref","first-page":"315","DOI":"10.1016\/j.nmd.2017.11.013","article-title":"Congenital myasthenic syndromes in Turkey: clinical clues and prognosis with long term follow-up","volume":"28","author":"Durmus H","year":"2018","journal-title":"Neuromuscul Disord"},{"issue":"2","key":"ref20","doi-asserted-by":"crossref","first-page":"529","DOI":"10.1007\/s13760-019-01246-9","article-title":"Congenital myasthenic syndrome in Turkey: clinical and genetic features in the long-term follow-up of patients","volume":"121","author":"G\u00fcl Mert G","year":"2021","journal-title":"Acta Neurol Belg"},{"issue":"4","key":"ref21","first-page":"816","article-title":"Six-minute walk test as outcome measure of fatigability in adults with spinal muscular atrophy treated with nusinersen","volume":"70","author":"Govoni A","year":"2024","journal-title":"Muscle Nerve"},{"issue":"2","key":"ref22","doi-asserted-by":"crossref","first-page":"147","DOI":"10.1136\/archdischild-2014-307396","article-title":"Timed motor function tests capacity in healthy children","volume":"101","author":"Pereira AC","year":"2016","journal-title":"Arch Dis Child"},{"issue":"6","key":"ref23","doi-asserted-by":"crossref","first-page":"640","DOI":"10.1016\/j.ejpn.2015.08.002","article-title":"Six-minute walk test versus two-minute walk test in children with Duchenne muscular dystrophy: is more time more information?","volume":"19","author":"Vill K","year":"2015","journal-title":"Eur J Paediatr Neurol"},{"key":"ref24","doi-asserted-by":"crossref","first-page":"379","DOI":"10.1007\/s10694-021-01140-y","article-title":"A review of the biomechanics of staircase descent: implications for building fire evacuations","volume":"58","author":"Agyemang C","year":"2022","journal-title":"Fire Technol"},{"issue":"11","key":"ref25","doi-asserted-by":"crossref","first-page":"e0294161","DOI":"10.1371\/journal.pone.0294161","article-title":"Joint power, joint work and lower limb muscle activity for transitions between level walking and stair ambulation at three inclinations","volume":"18","author":"Grimmer M","year":"2023","journal-title":"PLoS One"}],"container-title":["Revista da Associa\u00e7\u00e3o M\u00e9dica Brasileira"],"original-title":[],"link":[{"URL":"http:\/\/www.scielo.br\/scielo.php?script=sci_pdf&pid=S0104-42302026000102201&tlng=en","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,4,3]],"date-time":"2026-04-03T11:50:01Z","timestamp":1775217001000},"score":1,"resource":{"primary":{"URL":"http:\/\/www.scielo.br\/scielo.php?script=sci_arttext&pid=S0104-42302026000102201&tlng=en"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026]]},"references-count":25,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2026]]}},"alternative-id":["S0104-42302026000102201"],"URL":"https:\/\/doi.org\/10.1590\/1806-9282.20250307","relation":{},"ISSN":["1806-9282","0104-4230"],"issn-type":[{"value":"1806-9282","type":"electronic"},{"value":"0104-4230","type":"print"}],"subject":[],"published":{"date-parts":[[2026]]},"article-number":"e20250307"}}