{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,10]],"date-time":"2026-09-10T14:55:49Z","timestamp":1789052149272,"version":"build-2803163510"},"reference-count":41,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2022,2,28]],"date-time":"2022-02-28T00:00:00Z","timestamp":1646006400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/100005614","name":"Biogen","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100005614","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100005202","name":"Muscular Dystrophy Association","doi-asserted-by":"publisher","award":["MDA627906"],"award-info":[{"award-number":["MDA627906"]}],"id":[{"id":"10.13039\/100005202","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100003975","name":"Myotonic Dystrophy Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100003975","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000065","name":"National Institute of Neurological Disorders and Stroke","doi-asserted-by":"publisher","award":["P50NS048843"],"award-info":[{"award-number":["P50NS048843"]}],"id":[{"id":"10.13039\/100000065","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Muscle and Nerve"],"published-print":{"date-parts":[[2022,5]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Introduction\/Aims<\/jats:title><jats:p>Myotonic dystrophy type 1 (DM1) is known to affect cognitive function, but the best methods to assess central nervous system involvement in multicenter studies have not been determined. In this study our primary aim was to evaluate the potential of computerized cognitive tests to assess cognition in DM1.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>We conducted a prospective, longitudinal, observational study of 113 adults with DM1 at six sites. Psychomotor speed, attention, working memory, and executive functioning were assessed at baseline, 3 months, and 12 months using computerized cognitive tests. Results were compared with assessments of muscle function and patient reported outcomes (PROs), including the Myotonic Dystrophy Health Index (MDHI) and the 5\u2010dimension EuroQol (EQ\u20105D\u20105L) questionnaire.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Based on intraclass correlation coefficients, computerized cognitive tests had moderate to good reliability for psychomotor speed (0.76), attention (0.82), working memory speed (0.79), working memory accuracy (0.65), and executive functioning (0.87). Performance at baseline was lowest for working memory accuracy (<jats:italic>P<\/jats:italic>\u00a0&lt;\u2009.0001). Executive function performance improved from baseline to 3 months (<jats:italic>P<\/jats:italic>\u2009&lt;\u2009.0001), without further changes over 1 year. There was a moderate correlation between poorer executive function and larger CTG repeat size (r\u00a0=\u00a0\u22120.433). There were some weak associations between PROs and cognitive performance.<\/jats:p><\/jats:sec><jats:sec><jats:title>Discussion<\/jats:title><jats:p>Computerized tests of cognition are feasible in multicenter studies of DM1. Poor performance was exhibited in working memory, which may be a useful variable in clinical trials. Learning effects may have contributed to the improvement in executive functioning. The relationship between PROs and cognitive impairment in DM1 requires further study.<\/jats:p><\/jats:sec>","DOI":"10.1002\/mus.27520","type":"journal-article","created":{"date-parts":[[2022,2,18]],"date-time":"2022-02-18T10:40:37Z","timestamp":1645180837000},"page":"560-567","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":8,"title":["Brief assessment of cognitive function in myotonic dystrophy: Multicenter longitudinal study using computer\u2010assisted evaluation"],"prefix":"10.1002","volume":"65","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-6886-4356","authenticated-orcid":false,"given":"Gayle K.","family":"Deutsch","sequence":"first","affiliation":[{"name":"Department of Neurology and Neurological Sciences Stanford University School of Medicine  Stanford CA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Katharine A.","family":"Hagerman","sequence":"additional","affiliation":[{"name":"Department of Neurology and Neurological Sciences Stanford University School of Medicine  Stanford CA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jacinda","family":"Sampson","sequence":"additional","affiliation":[{"name":"Department of Neurology and Neurological Sciences Stanford University School of Medicine  Stanford CA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gersham","family":"Dent","sequence":"additional","affiliation":[{"name":"Biogen  Cambridge MA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jeanne","family":"Dekdebrun","sequence":"additional","affiliation":[{"name":"Department of Neurology The University of Rochester Medical Center  Rochester NY"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dana M.","family":"Parker","sequence":"additional","affiliation":[{"name":"Department of Neurology and Neurological Sciences Stanford University School of Medicine  Stanford CA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Charles A.","family":"Thornton","sequence":"additional","affiliation":[{"name":"Department of Neurology The University of Rochester Medical Center  Rochester NY"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chad R.","family":"Heatwole","sequence":"additional","affiliation":[{"name":"Department of Neurology The University of Rochester Medical Center  Rochester NY"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sub H.","family":"Subramony","sequence":"additional","affiliation":[{"name":"Department of Neurology, Norman Fixel Center for Neurological Disorders University of Florida College of Medicine  Gainesville FL"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ami K.","family":"Mankodi","sequence":"additional","affiliation":[{"name":"National Institute of Neurological Disorders and Stroke  Rockville MD"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tetsuo","family":"Ashizawa","sequence":"additional","affiliation":[{"name":"Department of Neurology, Weill Cornell Medicine at Houston Methodist Hospital  Houston TX"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0790-5315","authenticated-orcid":false,"given":"Jeffrey M.","family":"Statland","sequence":"additional","affiliation":[{"name":"Department of Neurology, University of Kansas Medical Center  Kansas City KS"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"W. David","family":"Arnold","sequence":"additional","affiliation":[{"name":"Departments of Neurology, PM&amp;R, Neuroscience and Physiology and Cell Biology The Ohio State University  Columbus OH"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"suffix":"3rd","given":"Richard T.","family":"Moxley","sequence":"additional","affiliation":[{"name":"Department of Neurology The University of Rochester Medical Center  Rochester NY"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"John W.","family":"Day","sequence":"additional","affiliation":[{"name":"Department of Neurology and Neurological Sciences Stanford University School of Medicine  Stanford CA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"name":"Myotonic Dystrophy Clinical Research Network","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2022,2,28]]},"reference":[{"key":"e_1_2_9_2_1","volume-title":"GeneReviews(R)","author":"Bird TD","year":"1993"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1111\/ene.13062"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.nmd.2016.10.003"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00415-008-0017-5"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0213381"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1111\/jnp.12192"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10072-016-2778-4"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.1002\/mus.20800"},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.1212\/CPJ.0000000000000531"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.1002\/hup.799"},{"key":"e_1_2_9_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cct.2006.10.010"},{"key":"e_1_2_9_13_1","doi-asserted-by":"publisher","DOI":"10.1093\/arclin\/acp010"},{"key":"e_1_2_9_14_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jalz.2015.01.008"},{"key":"e_1_2_9_15_1","unstructured":"Cogstate Normative Data.2018.www.cogstate.com"},{"key":"e_1_2_9_16_1","volume-title":"Comprehensive Norms for an Expanded Halstead\u2010Reitan Battery: Demographic Corrections, Research Findings, and Clinical Applications","author":"Heaton RK","year":"1991"},{"key":"e_1_2_9_17_1","doi-asserted-by":"publisher","DOI":"10.1002\/mus.24097"},{"key":"e_1_2_9_18_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0b013e318260cbe6"},{"key":"e_1_2_9_19_1","doi-asserted-by":"publisher","DOI":"10.1002\/mus.24725"},{"key":"e_1_2_9_20_1","doi-asserted-by":"publisher","DOI":"10.1002\/mus.26066"},{"key":"e_1_2_9_21_1","doi-asserted-by":"publisher","DOI":"10.1007\/s11136-011-9903-x"},{"key":"e_1_2_9_22_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.56.3.336"},{"key":"e_1_2_9_23_1","doi-asserted-by":"publisher","DOI":"10.1002\/ana.23992"},{"key":"e_1_2_9_24_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jbi.2008.08.010"},{"key":"e_1_2_9_25_1","volume-title":"R: A Language and Environment for Statistical Computing","author":"R Development Core Team","year":"2018"},{"key":"e_1_2_9_26_1","volume-title":"IBM SPSS Statistics for Mac, version 26.0","author":"IBM Corporation","year":"2019"},{"key":"e_1_2_9_27_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jcm.2016.02.012"},{"key":"e_1_2_9_28_1","doi-asserted-by":"publisher","DOI":"10.1213\/ANE.0000000000002864"},{"key":"e_1_2_9_29_1","doi-asserted-by":"publisher","DOI":"10.1001\/archneur.61.12.1943"},{"key":"e_1_2_9_30_1","doi-asserted-by":"publisher","DOI":"10.1186\/1744-9081-2-16"},{"key":"e_1_2_9_31_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.nmd.2013.07.004"},{"key":"e_1_2_9_32_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cortex.2017.08.008"},{"key":"e_1_2_9_33_1","doi-asserted-by":"publisher","DOI":"10.1006\/cogp.1999.0734"},{"key":"e_1_2_9_34_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jns.2014.04.005"},{"key":"e_1_2_9_35_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00415-018-8898-4"},{"key":"e_1_2_9_36_1","doi-asserted-by":"publisher","DOI":"10.1159\/000116989"},{"key":"e_1_2_9_37_1","doi-asserted-by":"publisher","DOI":"10.1159\/000264823"},{"key":"e_1_2_9_38_1","doi-asserted-by":"publisher","DOI":"10.1093\/arclin\/act021"},{"key":"e_1_2_9_39_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.acn.2008.03.002"},{"key":"e_1_2_9_40_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-016-0417-z"},{"key":"e_1_2_9_41_1","doi-asserted-by":"publisher","DOI":"10.3389\/fneur.2018.00780"},{"key":"e_1_2_9_42_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.gde.2017.01.007"}],"container-title":["Muscle &amp; Nerve"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/mus.27520","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/mus.27520","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/mus.27520","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,8,25]],"date-time":"2023-08-25T20:08:41Z","timestamp":1692994121000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/mus.27520"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,2,28]]},"references-count":41,"journal-issue":{"issue":"5","published-print":{"date-parts":[[2022,5]]}},"alternative-id":["10.1002\/mus.27520"],"URL":"https:\/\/doi.org\/10.1002\/mus.27520","archive":["Portico"],"relation":{},"ISSN":["0148-639X","1097-4598"],"issn-type":[{"value":"0148-639X","type":"print"},{"value":"1097-4598","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,2,28]]},"assertion":[{"value":"2021-06-20","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2022-02-12","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2022-02-28","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}