{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,7]],"date-time":"2026-02-07T21:19:02Z","timestamp":1770499142103,"version":"3.49.0"},"reference-count":52,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2004,11,25]],"date-time":"2004-11-25T00:00:00Z","timestamp":1101340800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Child Psychology Psychiatry"],"published-print":{"date-parts":[[2005,5]]},"abstract":"<jats:p>\n                    <jats:bold>Background:\u2002<\/jats:bold>\n                    Williams syndrome (WS), a neurodevelopmental disorder, is characterized by pervasive cognitive deficits alongside a relative sparing of auditory perception and cognition. A frequent characteristic of the phenotype is adverse reactions to, and\/or fascination with, certain sounds. Previously published reports indicate that people with WS experience hyperacusis, yet careful examination reveals that the term \u2018hyperacusis\u2019 has been used indiscriminately in the literature to describe quite different auditory abnormalities.\n                  <\/jats:p>\n                  <jats:p>\n                    <jats:bold>Method:\u2002<\/jats:bold>\n                    In an effort to clarify and document the incidence of auditory abnormalities in and among people with WS we collected data from parents of people with WS (\n                    <jats:italic>n<\/jats:italic>\n                    \u2003=\u2003118) and comparison groups of people with Down syndrome, autism, and normal controls.\n                  <\/jats:p>\n                  <jats:p>\n                    <jats:bold>Results:\u2002<\/jats:bold>\n                    Our findings revealed four phenomenologically separate auditory abnormalities, all of which were significantly more prevalent in WS than the three comparison groups. Among people with WS, we found relatively few reports of true hyperacusis (lowered threshold for soft sounds) or auditory fascinations\/fixations, whereas 80% reported fearfulness to idiosyncratically particular sounds, and 91% reported lowered uncomfortable loudness levels, or \u2018odynacusis.\u2019\n                  <\/jats:p>\n                  <jats:p>\n                    <jats:bold>Conclusions:\u2002<\/jats:bold>\n                    Our results confirm anecdotal reports of an unusual auditory phenotype in WS, and provide an important foundation for understanding the nature of auditory experience and pathology in WS. We conclude by reviewing the ways in which the present findings extend and complement recent neuroanatomical and neurophysiological findings on auditory function in people with WS.\n                  <\/jats:p>","DOI":"10.1111\/j.1469-7610.2004.00376.x","type":"journal-article","created":{"date-parts":[[2005,4,21]],"date-time":"2005-04-21T04:07:20Z","timestamp":1114056440000},"page":"514-523","source":"Crossref","is-referenced-by-count":115,"title":["Aversion, awareness, and attraction: investigating claims of hyperacusis in the Williams syndrome phenotype"],"prefix":"10.1111","volume":"46","author":[{"given":"Daniel J.","family":"Levitin","sequence":"first","affiliation":[]},{"given":"Kristen","family":"Cole","sequence":"additional","affiliation":[]},{"given":"Alan","family":"Lincoln","sequence":"additional","affiliation":[]},{"given":"Ursula","family":"Bellugi","sequence":"additional","affiliation":[]}],"member":"311","published-online":{"date-parts":[[2005,4,21]]},"reference":[{"key":"e_1_2_18_2_1","first-page":"1192","volume-title":"Health care supervision for children with Williams Syndrome","author":"American Academy of Pediatrics Committee on Genetics","year":"2001"},{"key":"e_1_2_18_3_1","doi-asserted-by":"publisher","DOI":"10.1080\/010503999424653"},{"key":"e_1_2_18_4_1","doi-asserted-by":"publisher","DOI":"10.1177\/014107680309601203"},{"key":"e_1_2_18_5_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.1320370621"},{"key":"e_1_2_18_6_1","volume-title":"Neural correlates of higher cortical functioning in a neurodevelopmental disorder","author":"Bellugi U.","year":"1989"},{"key":"e_1_2_18_7_1","volume-title":"Cognitive and neural development: Clues from genetically based syndromes","author":"Bellugi U.","year":"1996"},{"key":"e_1_2_18_8_1","doi-asserted-by":"publisher","DOI":"10.1162\/089892900561959"},{"key":"e_1_2_18_9_1","first-page":"2163","volume-title":"Encyclopedia of neuroscience","author":"Bellugi U.","year":"1999"},{"key":"e_1_2_18_10_1","volume-title":"Pitch and rhythmic abilities in adults with Williams\u2013Beuren syndrome","author":"Brochard R.","year":"2003"},{"key":"e_1_2_18_11_1","volume-title":"Stedman's concise medical dictionary for the health professions","author":"Dirckx J.H.","year":"2001"},{"key":"e_1_2_18_12_1","doi-asserted-by":"publisher","DOI":"10.1076\/chin.5.3.154.7337"},{"key":"e_1_2_18_13_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.20416"},{"key":"e_1_2_18_14_1","doi-asserted-by":"publisher","DOI":"10.1352\/0895-8017(1997)102<0045:BAEDII>2.0.CO;2"},{"key":"e_1_2_18_15_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/8.10.1947"},{"key":"e_1_2_18_16_1","doi-asserted-by":"publisher","DOI":"10.1162\/089892900561995"},{"key":"e_1_2_18_17_1","doi-asserted-by":"publisher","DOI":"10.3109\/03005368609079002"},{"key":"e_1_2_18_18_1","volume-title":"Neurodevelopmental disorders: Diagnosis and treatment","author":"Hagerman R.J.","year":"1999"},{"key":"e_1_2_18_19_1","volume-title":"Cartoon sound fx. Audio compact disc recording","author":"Hanna\u2010Barbera","year":"1994"},{"key":"e_1_2_18_20_1","first-page":"66","article-title":"Electrophysiological and quantitative MR analysis of the cortical auditory system in Williams syndrome","volume":"2","author":"Hickok G.","year":"1995","journal-title":"Cognitive Neuroscience Society Abstracts"},{"key":"e_1_2_18_21_1","volume-title":"Williams syndrome: Neuronal size and packing density in primary auditory cortext","author":"Holinger D.P.","year":"2002"},{"key":"e_1_2_18_22_1","unstructured":"HolingerD.P. BellugiU. Korenberg J.R. Mills D.L. Reiss A.L. Sherman G.F. &Galaburda A.M.(submitted).Neuronal measurements in primary auditory cortex of postmorten Williams Syndrome brains."},{"key":"e_1_2_18_23_1","doi-asserted-by":"publisher","DOI":"10.1076\/chin.7.1.42.3147"},{"key":"e_1_2_18_24_1","doi-asserted-by":"publisher","DOI":"10.1111\/1469-7610.00312"},{"key":"e_1_2_18_25_1","doi-asserted-by":"publisher","DOI":"10.1111\/1469-7610.00347"},{"key":"e_1_2_18_26_1","volume-title":"Greatest Hits$ Audio compact disc recording","author":"Jones S.","year":"1999"},{"key":"e_1_2_18_27_1","doi-asserted-by":"publisher","DOI":"10.1097\/00129492-200105000-00009"},{"key":"e_1_2_18_28_1","first-page":"436","article-title":"Psychometric normalization of a hyperacusis questionnaire","volume":"64","author":"Khalfa S.","year":"2002","journal-title":"Journal for Oto-Rhino-Laryngology"},{"key":"e_1_2_18_29_1","doi-asserted-by":"publisher","DOI":"10.1044\/jshd.5502.339"},{"key":"e_1_2_18_30_1","doi-asserted-by":"publisher","DOI":"10.1162\/089892900562002"},{"key":"e_1_2_18_31_1","doi-asserted-by":"publisher","DOI":"10.2307\/40300863"},{"key":"e_1_2_18_32_1","doi-asserted-by":"publisher","DOI":"10.1121\/1.427612"},{"key":"e_1_2_18_33_1","unstructured":"Levitin D.J. Bellugi U. &Cole K.(in preparation).Quantifying musicality in people with WS."},{"key":"e_1_2_18_34_1","article-title":"Characterizing the musical phenotype in individuals with WS","author":"Levitin D.J.","journal-title":"Child Neuropsychology"},{"key":"e_1_2_18_35_1","doi-asserted-by":"publisher","DOI":"10.1006\/nimg.2002.1297"},{"key":"e_1_2_18_36_1","first-page":"152","article-title":"Central hyperacusis in Williams syndrome","volume":"6","author":"Marriage J.","year":"1995","journal-title":"Genetic Counseling"},{"key":"e_1_2_18_37_1","doi-asserted-by":"publisher","DOI":"10.1017\/S0022215100131676"},{"key":"e_1_2_18_38_1","doi-asserted-by":"publisher","DOI":"10.1136\/adc.59.7.605"},{"key":"e_1_2_18_39_1","first-page":"337","volume-title":"Textbook of pain","author":"Melzack R.","year":"1984"},{"key":"e_1_2_18_40_1","volume-title":"Neurodevelopmental disorders: Contributions to a new framework from the cognitive neurosciences","author":"Mervis C.B.","year":"1999"},{"key":"e_1_2_18_41_1","doi-asserted-by":"publisher","DOI":"10.1017\/S0022215100127203"},{"key":"e_1_2_18_42_1","doi-asserted-by":"publisher","DOI":"10.1080\/87565649909540742"},{"key":"e_1_2_18_43_1","first-page":"371","article-title":"Disturbances of loudness perception","volume":"9","author":"Phillips D.P.","year":"1998","journal-title":"Journal of the American Academy of Audiology"},{"key":"e_1_2_18_44_1","doi-asserted-by":"publisher","DOI":"10.1016\/0304-3959(77)90035-5"},{"key":"e_1_2_18_45_1","doi-asserted-by":"publisher","DOI":"10.1023\/A:1023022709710"},{"key":"e_1_2_18_46_1","first-page":"367","volume-title":"Language, learning, and behavior disorders: Developmental, biological, and clinical perspectives","author":"Rossen M.L.","year":"1996"},{"key":"e_1_2_18_47_1","volume-title":"The Carl Stalling Project: Music from Warner Brothers Cartoons 1936\u20131958. Audio compact disc recording","author":"Stalling C.","year":"1990"},{"key":"e_1_2_18_48_1","doi-asserted-by":"publisher","DOI":"10.1017\/S0264180100001119"},{"key":"e_1_2_18_49_1","volume-title":"Social cognitive abilities in children and adolescents with Williams syndrome","author":"Tager\u2010Flusberg H.","year":"1996"},{"key":"e_1_2_18_50_1","doi-asserted-by":"publisher","DOI":"10.1016\/0022-460X(84)90582-0"},{"key":"e_1_2_18_51_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1469-8749.1990.tb16912.x"},{"key":"e_1_2_18_52_1","doi-asserted-by":"publisher","DOI":"10.1080\/01688639108401040"},{"key":"e_1_2_18_53_1","volume-title":"Taber's cyclopedic medical dictionary","author":"Venes D.","year":"2001"}],"container-title":["Journal of Child Psychology and Psychiatry"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fj.1469-7610.2004.00376.x","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/acamh.onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/j.1469-7610.2004.00376.x","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,11,3]],"date-time":"2025-11-03T13:08:28Z","timestamp":1762175308000},"score":1,"resource":{"primary":{"URL":"https:\/\/acamh.onlinelibrary.wiley.com\/doi\/10.1111\/j.1469-7610.2004.00376.x"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2005,4,21]]},"references-count":52,"journal-issue":{"issue":"5","published-print":{"date-parts":[[2005,5]]}},"alternative-id":["10.1111\/j.1469-7610.2004.00376.x"],"URL":"https:\/\/doi.org\/10.1111\/j.1469-7610.2004.00376.x","archive":["Portico"],"relation":{},"ISSN":["0021-9630","1469-7610"],"issn-type":[{"value":"0021-9630","type":"print"},{"value":"1469-7610","type":"electronic"}],"subject":[],"published":{"date-parts":[[2005,4,21]]}}}