{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,17]],"date-time":"2026-07-17T05:10:20Z","timestamp":1784265020275,"version":"3.55.0"},"reference-count":28,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2021,1,31]],"date-time":"2021-01-31T00:00:00Z","timestamp":1612051200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/100001962","name":"A-T Children's Project","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100001962","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Develop Med Child Neuro"],"published-print":{"date-parts":[[2021,4]]},"abstract":"<jats:sec><jats:title>Aim<\/jats:title><jats:p>To investigate the characteristics and severity of dysarthria in children and adults with ataxia telangiectasia.<\/jats:p><\/jats:sec><jats:sec><jats:title>Method<\/jats:title><jats:p>All children and adults with ataxia telangiectasia who visited our multidisciplinary outpatient clinic for ataxia telangiectasia were asked to participate in this study, which took place in March 2019. To evaluate dysarthria, we used the Radboud Dysarthria Assessment in adults (older than 18y) and the paediatric Radboud Dysarthria Assessment in children (5\u201318y), including the observational tasks \u2018conversation\u2019 and \u2018reading\u2019, and the speech\u2010related maximum performance tasks \u2018repetition rate\u2019, \u2018phonation time\u2019, \u2018fundamental frequency range\u2019, and \u2018phonation volume\u2019. Speech intelligibility was measured using the Intelligibility in Context Scale.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Twenty\u2010two individuals (15 children [5\u201317y], seven adults [19\u201347y]; 14 males and eight females; mean age 19y, SD 15y 2mo) participated. Dysarthria was present in all participants and characterized by ataxic components in adults and similar uncontrolled movements in children. In most participants, speech was mildly to mildly\/severely affected. Almost all participants had an abnormal score for at least one maximum performance task.<\/jats:p><\/jats:sec><jats:sec><jats:title>Interpretation<\/jats:title><jats:p>Dysarthria in ataxia telangiectasia is characterized by uncontrolled, ataxic, and involuntary movements, resulting in monotonous, unstable, slow, hypernasal, and chanted speech.<\/jats:p><\/jats:sec><jats:sec><jats:title>What this paper adds<\/jats:title><jats:p>\n<jats:list list-type=\"bullet\">\n\n<jats:list-item><jats:p>Dysarthria in ataxia telangiectasia is characterized by uncontrolled, ataxic, and involuntary movements.<\/jats:p><\/jats:list-item>\n\n<jats:list-item><jats:p>Dysarthria in ataxia telangiectasia results in monotonous, unstable, slow, hypernasal, and chanted speech.<\/jats:p><\/jats:list-item>\n\n<jats:list-item><jats:p>Dysarthria in ataxia telangiectasia can be assessed using the Radboud Dysarthria Assessment and the paediatric Radboud Dysarthria Assessment.<\/jats:p><\/jats:list-item>\n<\/jats:list>\n<\/jats:p><\/jats:sec>","DOI":"10.1111\/dmcn.14811","type":"journal-article","created":{"date-parts":[[2021,2,1]],"date-time":"2021-02-01T07:38:18Z","timestamp":1612165098000},"page":"450-456","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":13,"title":["Dysarthria in children and adults with ataxia telangiectasia"],"prefix":"10.1111","volume":"63","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1361-5920","authenticated-orcid":false,"given":"Stefanie J G","family":"Veenhuis","sequence":"first","affiliation":[{"name":"Department of Pediatrics Amalia Children\u2019s Hospital Radboud University Medical Center Nijmegen the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nienke J H","family":"van OS","sequence":"additional","affiliation":[{"name":"Department of Pediatric Neurology Radboud University Medical Center Nijmegen the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marjo H J C","family":"van Gerven","sequence":"additional","affiliation":[{"name":"Donders Institute for Brain Cognition and Behaviour Department of Rehabilitation Radboud University Nijmegen the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Leenke","family":"van Haaften","sequence":"additional","affiliation":[{"name":"Donders Institute for Brain Cognition and Behaviour Department of Rehabilitation Radboud University Nijmegen the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elisabeth H","family":"Mulder","sequence":"additional","affiliation":[{"name":"Donders Institute for Brain Cognition and Behaviour Department of Rehabilitation Radboud University Nijmegen the Netherlands"},{"name":"Faculty of Arts at Radboud University Nijmegen the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Corry M R","family":"Weemaes","sequence":"additional","affiliation":[{"name":"Department of Pediatric Infectious Diseases and Immunology Radboud University Medical Center Nijmegen the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mich\u00e8l A A P","family":"Willemsen","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Amalia Children\u2019s Hospital Radboud University Medical Center Nijmegen the Netherlands"},{"name":"Department of Pediatric Neurology Radboud University Medical Center Nijmegen the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2021,1,31]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.7792600"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrm3546"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.21.4.526"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.clim.2017.01.009"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/cge.12453"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1002\/ana.25394"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1002\/humu.22016"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpeds.2011.02.005"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.3109\/17518423.2011.603368"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1007\/978-90-368-0533-9"},{"key":"e_1_2_8_12_1","doi-asserted-by":"publisher","DOI":"10.1159\/000484556"},{"key":"e_1_2_8_13_1","unstructured":"RuessinkM Engel\u2010HoekLL deSwartB SpekB vanGervenM KalfH.Validation of the pediatric Radboud Dysarthria Assessment. Forthcoming."},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.1044\/jshr.1202.246"},{"key":"e_1_2_8_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neubiorev.2011.07.011"},{"key":"e_1_2_8_16_1","doi-asserted-by":"publisher","DOI":"10.1044\/1092-4388(2011\/10-0130)"},{"key":"e_1_2_8_17_1","volume-title":"Algemene Fonetiek (3rd completely revised edition)","author":"Rietveld T","year":"2009"},{"key":"e_1_2_8_18_1","doi-asserted-by":"publisher","DOI":"10.1044\/jshr.3603.452"},{"key":"e_1_2_8_19_1","doi-asserted-by":"publisher","DOI":"10.1080\/17549507.2017.1380227"},{"key":"e_1_2_8_20_1","doi-asserted-by":"publisher","DOI":"10.3109\/02699209608985178"},{"key":"e_1_2_8_21_1","first-page":"146","article-title":"Obtaining and interpreting maximum performance tasks from children: a tutorial","volume":"29","author":"Rvachew S","year":"2005","journal-title":"J Speech Lang Path Audiol"},{"key":"e_1_2_8_22_1","doi-asserted-by":"publisher","DOI":"10.1191\/0265659006ct307xx"},{"key":"e_1_2_8_23_1","doi-asserted-by":"publisher","DOI":"10.1044\/jshd.5204.367"},{"key":"e_1_2_8_24_1","doi-asserted-by":"publisher","DOI":"10.1016\/0093-934X(82)90060-8"},{"key":"e_1_2_8_25_1","doi-asserted-by":"publisher","DOI":"10.1002\/ppul.20738"},{"key":"e_1_2_8_26_1","doi-asserted-by":"publisher","DOI":"10.3109\/17549507.2013.876667"},{"key":"e_1_2_8_27_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0167-6393(97)00026-5"},{"key":"e_1_2_8_28_1","doi-asserted-by":"publisher","DOI":"10.1080\/17549507.2018.1472808"},{"key":"e_1_2_8_29_1","doi-asserted-by":"publisher","DOI":"10.3109\/17549507.2013.876662"}],"container-title":["Developmental Medicine &amp; Child Neurology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/dmcn.14811","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/dmcn.14811","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/dmcn.14811","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,1]],"date-time":"2023-09-01T08:11:50Z","timestamp":1693555910000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/dmcn.14811"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,1,31]]},"references-count":28,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2021,4]]}},"alternative-id":["10.1111\/dmcn.14811"],"URL":"https:\/\/doi.org\/10.1111\/dmcn.14811","archive":["Portico"],"relation":{},"ISSN":["0012-1622","1469-8749"],"issn-type":[{"value":"0012-1622","type":"print"},{"value":"1469-8749","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,1,31]]},"assertion":[{"value":"2020-12-08","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2021-01-31","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}