{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,8]],"date-time":"2026-07-08T22:08:48Z","timestamp":1783548528256,"version":"3.55.0"},"reference-count":33,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2016,12,19]],"date-time":"2016-12-19T00:00:00Z","timestamp":1482105600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Research Intellect Disabil"],"published-print":{"date-parts":[[2018,1]]},"abstract":"<jats:sec><jats:title>Background<\/jats:title><jats:p>Angelman syndrome is a rare disorder in which most individuals do not develop speech. Testing of communication ability using traditional neuropsychological measures reveals a performance level at or near the floor of the instrument resulting in an inability to detect change when experimental therapeutics are applied.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Nine individuals, with molecularly confirmed <jats:styled-content style=\"fixed-case\">AS<\/jats:styled-content>, ranging in age from 34 to 126\u00a0months, and a single healthy control child (age 16\u00a0months) were audio and video\u2010recorded while interacting with a licensed speech\u2010language pathologist in an attempt to elicit vocalization and non\u2010verbal communication. Thirty\u2010minute audio recordings were transcribed and categorized per the Stark Assessment of Early Vocal Development\u2010Revised and a phonetic inventory was created. Using video recordings, gestures were classified by function, either behavioral regulation or social interaction and further categorized as deictic or representational (i.e., behavioral regulation) and joint attention or shared engagement (i.e., social interaction).<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>The range of vocalizations produced by the children with <jats:styled-content style=\"fixed-case\">AS<\/jats:styled-content> was characteristic of children between 0\u20136\u00a0months and none of the children with <jats:styled-content style=\"fixed-case\">AS<\/jats:styled-content> used advanced forms of vocalizations. The mean frequency of reflexive vocalizations, control of phonation and expansion far exceeded the number of uses of canonical syllables, consistant with the characteristics of children around 12\u00a0months of age. Most vocalizations were either laughter or isolated vowels, only three children with <jats:styled-content style=\"fixed-case\">AS<\/jats:styled-content> produced consonant\u2010vowel combinations. Children with <jats:styled-content style=\"fixed-case\">AS<\/jats:styled-content> tended to use central and low vowels with few producing high vowels, suggesting the presence of childhood apraxia of speech.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Our results show the utilization of video\u2010recorded behavioral observations provides a feasible and reliable alternative for quantification of communication ability in this patient population and may be employed during future clinical studies of potential therapeutics.<\/jats:p><\/jats:sec>","DOI":"10.1111\/jar.12305","type":"journal-article","created":{"date-parts":[[2016,12,19]],"date-time":"2016-12-19T05:52:42Z","timestamp":1482126762000},"source":"Crossref","is-referenced-by-count":23,"title":["Quantitative Measurement of Communication Ability in Children with Angelman Syndrome"],"prefix":"10.1111","volume":"31","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-7680-3008","authenticated-orcid":false,"given":"Joseph C.","family":"Grieco","sequence":"first","affiliation":[{"name":"Department of Molecular Pharmacology and Physiology Morsani College of Medicine University of South Florida Tampa FL USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ruth H.","family":"Bahr","sequence":"additional","affiliation":[{"name":"Department of Communication Sciences and Disorders University of South Florida Tampa FL USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mike R.","family":"Schoenberg","sequence":"additional","affiliation":[{"name":"Department of Neuropsychology and Psychiatry University of South Florida Tampa FL USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laura","family":"Conover","sequence":"additional","affiliation":[{"name":"Department of Communication Sciences and Disorders University of South Florida Tampa FL USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lauren N.","family":"Mackie","sequence":"additional","affiliation":[{"name":"Department of Molecular Pharmacology and Physiology Morsani College of Medicine University of South Florida Tampa FL USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Edwin J.","family":"Weeber","sequence":"additional","affiliation":[{"name":"Department of Molecular Pharmacology and Physiology Morsani College of Medicine University of South Florida Tampa FL USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2016,12,19]]},"reference":[{"key":"e_1_2_6_2_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng0997-75"},{"key":"e_1_2_6_3_1","first-page":"158","article-title":"Gaze\u2010shift patterns of young children with developmental disabilities who are at risk for being nonspeaking","volume":"40","author":"Arens K.","year":"2005","journal-title":"Education and Training in Developmental Disabilities"},{"key":"e_1_2_6_4_1","doi-asserted-by":"crossref","unstructured":"BayleyN.(2005)Bayley scales of infant and toddler development.San Antonio TX NCS Pearson.","DOI":"10.1037\/t14978-000"},{"key":"e_1_2_6_5_1","doi-asserted-by":"publisher","DOI":"10.1002\/mrdd.20036"},{"key":"e_1_2_6_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/jar.12110"},{"key":"e_1_2_6_7_1","doi-asserted-by":"crossref","first-page":"101","DOI":"10.1177\/002383098302600201","article-title":"The acquisition of communicative intentions in infants eight\u00a0to fifteen months of age","volume":"26","author":"Carpenter R. L.","year":"1983","journal-title":"Language and Speech"},{"key":"e_1_2_6_8_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.1320460105"},{"key":"e_1_2_6_9_1","doi-asserted-by":"publisher","DOI":"10.1044\/1058-0360(2008\/07-0041)"},{"key":"e_1_2_6_10_1","first-page":"152","article-title":"Comparison of engagement patterns of young children with developmental disabilities between structured and free play","volume":"42","author":"Cress C. J.","year":"2007","journal-title":"Education and Training in Developmental Disabilities"},{"key":"e_1_2_6_11_1","doi-asserted-by":"publisher","DOI":"10.1177\/1525740111416644"},{"key":"e_1_2_6_12_1","doi-asserted-by":"publisher","DOI":"10.1044\/1092-4388(2011\/10-0148)"},{"key":"e_1_2_6_13_1","doi-asserted-by":"publisher","DOI":"10.1080\/15475441.2011.564569"},{"key":"e_1_2_6_14_1","doi-asserted-by":"crossref","first-page":"50","DOI":"10.1044\/lle22.2.50","article-title":"Gesture as a window onto communicative abilities: implications for diagnosis and intervention","volume":"22","author":"Goldin\u2010Meadow S.","year":"2015","journal-title":"Perspectives on Language Learning and Education"},{"key":"e_1_2_6_15_1","doi-asserted-by":"publisher","DOI":"10.1186\/s12883-014-0232-x"},{"key":"e_1_2_6_16_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.nbd.2010.04.012"},{"key":"e_1_2_6_17_1","doi-asserted-by":"publisher","DOI":"10.1086\/302473"},{"key":"e_1_2_6_18_1","doi-asserted-by":"publisher","DOI":"10.1044\/1092-4388(2009\/08-0234)"},{"key":"e_1_2_6_19_1","doi-asserted-by":"publisher","DOI":"10.1136\/adc.69.1.148"},{"key":"e_1_2_6_20_1","doi-asserted-by":"publisher","DOI":"10.1111\/cdev.12249"},{"key":"e_1_2_6_21_1","first-page":"335","volume-title":"Routledge Handbook of Communication Disorders","author":"Longard J.","year":"2015"},{"key":"e_1_2_6_22_1","volume-title":"Qualitative Data Analysis: An Expanded Sourcebook","author":"Miles M. B.","year":"1994"},{"key":"e_1_2_6_23_1","doi-asserted-by":"publisher","DOI":"10.1080\/02699200500211451"},{"key":"e_1_2_6_24_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0021-9924(99)00013-1"},{"key":"e_1_2_6_25_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.1320460108"},{"key":"e_1_2_6_26_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.30065"},{"key":"e_1_2_6_27_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.33509"},{"key":"e_1_2_6_28_1","doi-asserted-by":"publisher","DOI":"10.1017\/S0142716400010237"},{"key":"e_1_2_6_29_1","doi-asserted-by":"publisher","DOI":"10.1136\/adc.64.1.83"},{"key":"e_1_2_6_30_1","doi-asserted-by":"crossref","first-page":"61","DOI":"10.1044\/lle22.2.61","article-title":"Co\u2010speech gesture input as a support for language learning in children with and without early language delay","volume":"22","author":"Singleton N. C.","year":"2015","journal-title":"Perspectives on Language Learning and Education"},{"key":"e_1_2_6_31_1","doi-asserted-by":"publisher","DOI":"10.1016\/B978-0-12-770601-6.50010-3"},{"key":"e_1_2_6_32_1","doi-asserted-by":"publisher","DOI":"10.1177\/014272378900900607"},{"key":"e_1_2_6_33_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1468-3148.2011.00625.x"},{"key":"e_1_2_6_34_1","doi-asserted-by":"publisher","DOI":"10.1055\/s-0028-1082491"}],"container-title":["Journal of Applied Research in Intellectual Disabilities"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fjar.12305","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fjar.12305","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/jar.12305","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,29]],"date-time":"2023-09-29T19:38:40Z","timestamp":1696016320000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/jar.12305"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2016,12,19]]},"references-count":33,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2018,1]]}},"alternative-id":["10.1111\/jar.12305"],"URL":"https:\/\/doi.org\/10.1111\/jar.12305","archive":["Portico"],"relation":{},"ISSN":["1360-2322","1468-3148"],"issn-type":[{"value":"1360-2322","type":"print"},{"value":"1468-3148","type":"electronic"}],"subject":[],"published":{"date-parts":[[2016,12,19]]}}}