{"id":"https://openalex.org/W7157689577","doi":"https://doi.org/10.1002/ajmg.a.70184","title":"Associations Between Sensory Processing and Irritability in Prader\u2013Willi Syndrome: Beyond Genetic Subtypes and Clinical Backgrounds","display_name":"Associations Between Sensory Processing and Irritability in Prader\u2013Willi Syndrome: Beyond Genetic Subtypes and Clinical Backgrounds","publication_year":2026,"publication_date":"2026-04-29","ids":{"openalex":"https://openalex.org/W7157689577","doi":"https://doi.org/10.1002/ajmg.a.70184","pmid":"https://pubmed.ncbi.nlm.nih.gov/42052935"},"language":"en","primary_location":{"id":"doi:10.1002/ajmg.a.70184","doi":"https://doi.org/10.1002/ajmg.a.70184","is_oa":false,"landing_page_url":"https://doi.org/10.1002/ajmg.a.70184","pdf_url":null,"source":{"id":"https://openalex.org/S180901878","display_name":"American Journal of Medical Genetics Part A","issn_l":"1552-4825","issn":["1552-4825","1552-4833"],"is_oa":false,"is_in_doaj":false,"is_core":true,"listed_in":["anvur-scientific","cwts-core","dongbi-b","doyens","jufo-1","ki-jl-1","medline","norway-1","poland-100","russia-white-list-3","vabb-shw"],"host_organization":"https://openalex.org/P4310320595","host_organization_name":"Wiley","host_organization_lineage":["https://openalex.org/P4310320595"],"host_organization_lineage_names":["Wiley"],"type":"journal"},"license":null,"license_id":null,"version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"American Journal of Medical Genetics Part A","raw_type":"journal-article"},"volume":null,"type":"article","is_traditional":true,"indexed_in":["crossref","pubmed"],"open_access":{"is_oa":false,"oa_status":"closed","oa_url":null,"any_repository_has_fulltext":false},"authorships":[{"author_position":"first","author":{"id":"https://openalex.org/A5118676465","display_name":"Erina Nakane","orcid":"https://orcid.org/0009-0007-5159-0572","observed_orcids":["https://orcid.org/0009-0007-5159-0572"]},"institutions":[{"id":"https://openalex.org/I4210141235","display_name":"Dokkyo Medical University Saitama Medical Center","ror":"https://ror.org/03fyvh407","country_code":"JP","type":"healthcare","lineage":["https://openalex.org/I4210141235"]}],"countries":["JP"],"is_corresponding":false,"raw_author_name":"Erina Nakane","raw_affiliation_strings":["Department of Psychiatry Dokkyo Medical University Saitama Medical Center  Koshigaya Saitama Japan"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Department of Psychiatry Dokkyo Medical University Saitama Medical Center  Koshigaya Saitama Japan","institution_ids":["https://openalex.org/I4210141235"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5035256309","display_name":"Hiroyuki Ogata","orcid":"https://orcid.org/0000-0002-2375-0467","observed_orcids":["https://orcid.org/0000-0002-2375-0467"]},"institutions":[{"id":"https://openalex.org/I4210141235","display_name":"Dokkyo Medical University Saitama Medical Center","ror":"https://ror.org/03fyvh407","country_code":"JP","type":"healthcare","lineage":["https://openalex.org/I4210141235"]}],"countries":["JP"],"is_corresponding":false,"raw_author_name":"Hiroyuki Ogata","raw_affiliation_strings":["Department of Psychiatry Dokkyo Medical University Saitama Medical Center  Koshigaya Saitama Japan"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Department of Psychiatry Dokkyo Medical University Saitama Medical Center  Koshigaya Saitama Japan","institution_ids":["https://openalex.org/I4210141235"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5059553309","display_name":"Sohei Saima","orcid":"https://orcid.org/0000-0002-1334-3892","observed_orcids":["https://orcid.org/0000-0002-1334-3892"]},"institutions":[{"id":"https://openalex.org/I4210141235","display_name":"Dokkyo Medical University Saitama Medical Center","ror":"https://ror.org/03fyvh407","country_code":"JP","type":"healthcare","lineage":["https://openalex.org/I4210141235"]}],"countries":["JP"],"is_corresponding":false,"raw_author_name":"Sohei Saima","raw_affiliation_strings":["Department of Psychiatry Dokkyo Medical University Saitama Medical Center  Koshigaya Saitama Japan"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Department of Psychiatry Dokkyo Medical University Saitama Medical Center  Koshigaya Saitama Japan","institution_ids":["https://openalex.org/I4210141235"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5084369127","display_name":"Chuichi Kondo","orcid":"https://orcid.org/0009-0001-0707-1405","observed_orcids":["https://orcid.org/0009-0001-0707-1405"]},"institutions":[],"countries":[],"is_corresponding":false,"raw_author_name":"Chuichi Kondo","raw_affiliation_strings":["Department of Psychiatry Ikezawa Shinkeika Hospital  Hanyuu Saitama Japan"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Department of Psychiatry Ikezawa Shinkeika Hospital  Hanyuu Saitama Japan","institution_ids":[]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5008300031","display_name":"Masaki SEKI","orcid":"https://orcid.org/0009-0008-7654-0085","observed_orcids":["https://orcid.org/0009-0008-7654-0085"]},"institutions":[],"countries":[],"is_corresponding":false,"raw_author_name":"Masaki Seki","raw_affiliation_strings":["Department of Psychiatry Okute Hospital  Mizunami Gifu Japan"],"raw_orcid":"https://orcid.org/0009-0008-7654-0085","affiliations":[{"raw_affiliation_string":"Department of Psychiatry Okute Hospital  Mizunami Gifu Japan","institution_ids":[]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5035760327","display_name":"Yuji Oto","orcid":"https://orcid.org/0000-0003-0130-0585","observed_orcids":["https://orcid.org/0000-0003-0130-0585"]},"institutions":[{"id":"https://openalex.org/I4210141235","display_name":"Dokkyo Medical University Saitama Medical Center","ror":"https://ror.org/03fyvh407","country_code":"JP","type":"healthcare","lineage":["https://openalex.org/I4210141235"]}],"countries":["JP"],"is_corresponding":false,"raw_author_name":"Yuji Oto","raw_affiliation_strings":["Department of Pediatrics Dokkyo Medical University Saitama Medical Center  Koshigaya Saitama Japan"],"raw_orcid":"https://orcid.org/0000-0003-0130-0585","affiliations":[{"raw_affiliation_string":"Department of Pediatrics Dokkyo Medical University Saitama Medical Center  Koshigaya Saitama Japan","institution_ids":["https://openalex.org/I4210141235"]}]},{"author_position":"last","author":{"id":"https://openalex.org/A5102762421","display_name":"Hiroshi Ihara","orcid":"https://orcid.org/0000-0002-6888-7141","observed_orcids":["https://orcid.org/0000-0002-6888-7141"]},"institutions":[{"id":"https://openalex.org/I4210141235","display_name":"Dokkyo Medical University Saitama Medical Center","ror":"https://ror.org/03fyvh407","country_code":"JP","type":"healthcare","lineage":["https://openalex.org/I4210141235"]}],"countries":["JP"],"is_corresponding":false,"raw_author_name":"Hiroshi Ihara","raw_affiliation_strings":["Department of Psychiatry Dokkyo Medical University Saitama Medical Center  Koshigaya Saitama Japan"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Department of Psychiatry Dokkyo Medical University Saitama Medical Center  Koshigaya Saitama Japan","institution_ids":["https://openalex.org/I4210141235"]}]}],"institutions":[],"countries_distinct_count":1,"institutions_distinct_count":1,"corresponding_author_ids":[],"corresponding_institution_ids":[],"apc_list":{"value":4710,"currency":"USD","value_usd":4710},"apc_paid":null,"fwci":0.0,"has_fulltext":false,"cited_by_count":0,"citation_normalized_percentile":{"value":0.0,"is_in_top_1_percent":false,"is_in_top_10_percent":false},"cited_by_percentile_year":null,"biblio":{"volume":"200","issue":"9","first_page":"2061","last_page":"2066"},"is_retracted":false,"is_paratext":false,"is_xpac":false,"primary_topic":{"id":"https://openalex.org/T11928","display_name":"Genetic Syndromes and Imprinting","score":0.9628999829292297,"subfield":{"id":"https://openalex.org/subfields/1311","display_name":"Genetics"},"field":{"id":"https://openalex.org/fields/13","display_name":"Biochemistry, Genetics and Molecular Biology"},"domain":{"id":"https://openalex.org/domains/1","display_name":"Life Sciences"}},"topics":[{"id":"https://openalex.org/T11928","display_name":"Genetic Syndromes and Imprinting","score":0.9628999829292297,"subfield":{"id":"https://openalex.org/subfields/1311","display_name":"Genetics"},"field":{"id":"https://openalex.org/fields/13","display_name":"Biochemistry, Genetics and Molecular Biology"},"domain":{"id":"https://openalex.org/domains/1","display_name":"Life Sciences"}},{"id":"https://openalex.org/T13258","display_name":"Williams Syndrome Research","score":0.0071000000461936,"subfield":{"id":"https://openalex.org/subfields/2806","display_name":"Developmental Neuroscience"},"field":{"id":"https://openalex.org/fields/28","display_name":"Neuroscience"},"domain":{"id":"https://openalex.org/domains/1","display_name":"Life Sciences"}},{"id":"https://openalex.org/T10106","display_name":"Autism Spectrum Disorder Research","score":0.0044999998062849045,"subfield":{"id":"https://openalex.org/subfields/2805","display_name":"Cognitive Neuroscience"},"field":{"id":"https://openalex.org/fields/28","display_name":"Neuroscience"},"domain":{"id":"https://openalex.org/domains/1","display_name":"Life Sciences"}}],"keywords":[{"id":"https://openalex.org/keywords/prader-willi-syndrome","display_name":"Prader-Willi syndrome","score":0.9509999752044678},{"id":"https://openalex.org/keywords/irritability","display_name":"irritability","score":0.9570000171661377},{"id":"https://openalex.org/keywords/sensory-processing","display_name":"sensory processing","score":0.9980000257492065},{"id":"https://openalex.org/keywords/auditory-filtering","display_name":"auditory filtering","score":0.7319999933242798},{"id":"https://openalex.org/keywords/sensory-sensitivity","display_name":"sensory sensitivity","score":0.6060000061988831},{"id":"https://openalex.org/keywords/maternal-uniparental-disomy","display_name":"maternal uniparental disomy","score":0.9340000152587891},{"id":"https://openalex.org/keywords/genetic-subtypes","display_name":"genetic subtypes","score":0.6150000095367432},{"id":"https://openalex.org/keywords/psychotropic-drugs","display_name":"psychotropic drugs","score":0.9139999747276306},{"id":"https://openalex.org/keywords/behavioral-interventions","display_name":"behavioral interventions","score":0.4129999876022339}],"concepts":[{"id":"https://openalex.org/C2780820931","wikidata":"https://www.wikidata.org/wiki/Q29299","display_name":"Irritability","level":3,"score":0.7646999955177307},{"id":"https://openalex.org/C94487597","wikidata":"https://www.wikidata.org/wiki/Q11101","display_name":"Sensory system","level":2,"score":0.7271999716758728},{"id":"https://openalex.org/C2776861996","wikidata":"https://www.wikidata.org/wiki/Q241849","display_name":"Sensory processing","level":3,"score":0.6194999814033508},{"id":"https://openalex.org/C130093455","wikidata":"https://www.wikidata.org/wiki/Q173253","display_name":"Sensation","level":2,"score":0.571399986743927},{"id":"https://openalex.org/C15744967","wikidata":"https://www.wikidata.org/wiki/Q9418","display_name":"Psychology","level":0,"score":0.4203000068664551},{"id":"https://openalex.org/C169760540","wikidata":"https://www.wikidata.org/wiki/Q207011","display_name":"Neuroscience","level":1,"score":0.36739999055862427},{"id":"https://openalex.org/C548259974","wikidata":"https://www.wikidata.org/wiki/Q569965","display_name":"Audiology","level":1,"score":0.3540000021457672},{"id":"https://openalex.org/C71924100","wikidata":"https://www.wikidata.org/wiki/Q11190","display_name":"Medicine","level":0,"score":0.3276999890804291},{"id":"https://openalex.org/C67636389","wikidata":"https://www.wikidata.org/wiki/Q784237","display_name":"Genetic predisposition","level":3,"score":0.2962999939918518},{"id":"https://openalex.org/C115816405","wikidata":"https://www.wikidata.org/wiki/Q73822","display_name":"Sensation seeking","level":3,"score":0.2759000062942505},{"id":"https://openalex.org/C27415008","wikidata":"https://www.wikidata.org/wiki/Q7256382","display_name":"Psychological intervention","level":2,"score":0.27079999446868896},{"id":"https://openalex.org/C110235638","wikidata":"https://www.wikidata.org/wiki/Q17119820","display_name":"Sensory threshold","level":2,"score":0.2685999870300293},{"id":"https://openalex.org/C180747234","wikidata":"https://www.wikidata.org/wiki/Q23373","display_name":"Cognitive psychology","level":1,"score":0.2599000036716461},{"id":"https://openalex.org/C8868529","wikidata":"https://www.wikidata.org/wiki/Q124794","display_name":"Taste","level":2,"score":0.2572999894618988},{"id":"https://openalex.org/C26760741","wikidata":"https://www.wikidata.org/wiki/Q160402","display_name":"Perception","level":2,"score":0.2542000114917755},{"id":"https://openalex.org/C25457674","wikidata":"https://www.wikidata.org/wiki/Q17163316","display_name":"Sensory stimulation therapy","level":3,"score":0.2538999915122986}],"mesh":[{"descriptor_ui":"D000293","descriptor_name":"Adolescent","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D000328","descriptor_name":"Adult","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D002648","descriptor_name":"Child","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D002675","descriptor_name":"Child, Preschool","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D005260","descriptor_name":"Female","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D006801","descriptor_name":"Humans","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D007508","descriptor_name":"Irritable Mood","qualifier_ui":"Q000502","qualifier_name":"physiology","is_major_topic":true},{"descriptor_ui":"D008297","descriptor_name":"Male","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D011218","descriptor_name":"Prader-Willi Syndrome","qualifier_ui":"Q000235","qualifier_name":"genetics","is_major_topic":true},{"descriptor_ui":"D011218","descriptor_name":"Prader-Willi Syndrome","qualifier_ui":"Q000503","qualifier_name":"physiopathology","is_major_topic":true},{"descriptor_ui":"D024182","descriptor_name":"Uniparental Disomy","qualifier_ui":"Q000235","qualifier_name":"genetics","is_major_topic":false},{"descriptor_ui":"D055815","descriptor_name":"Young Adult","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false}],"locations_count":2,"locations":[{"id":"doi:10.1002/ajmg.a.70184","doi":"https://doi.org/10.1002/ajmg.a.70184","is_oa":false,"landing_page_url":"https://doi.org/10.1002/ajmg.a.70184","pdf_url":null,"source":{"id":"https://openalex.org/S180901878","display_name":"American Journal of Medical Genetics Part A","issn_l":"1552-4825","issn":["1552-4825","1552-4833"],"is_oa":false,"is_in_doaj":false,"is_core":true,"listed_in":["anvur-scientific","cwts-core","dongbi-b","doyens","jufo-1","ki-jl-1","medline","norway-1","poland-100","russia-white-list-3","vabb-shw"],"host_organization":"https://openalex.org/P4310320595","host_organization_name":"Wiley","host_organization_lineage":["https://openalex.org/P4310320595"],"host_organization_lineage_names":["Wiley"],"type":"journal"},"license":null,"license_id":null,"version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"American Journal of Medical Genetics Part A","raw_type":"journal-article"},{"id":"pmid:42052935","doi":null,"is_oa":false,"landing_page_url":"https://pubmed.ncbi.nlm.nih.gov/42052935","pdf_url":null,"source":{"id":"https://openalex.org/S4306525036","display_name":"PubMed","issn_l":null,"issn":null,"is_oa":false,"is_in_doaj":false,"is_core":false,"listed_in":[],"host_organization":"https://openalex.org/I1299303238","host_organization_name":"National Institutes of Health","host_organization_lineage":["https://openalex.org/I1299303238"],"host_organization_lineage_names":[],"type":"repository"},"license":null,"license_id":null,"version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"American journal of medical genetics. Part A","raw_type":"Journal Article"}],"best_oa_location":null,"sustainable_development_goals":[{"display_name":"Good health and well-being","score":0.9476000070571899,"id":"https://metadata.un.org/sdg/3"}],"study_designs":[{"display_name":"Observational Study","id":"https://openalex.org/study-designs/observational-study"}],"awards":[{"id":"https://openalex.org/G2298376937","display_name":null,"funder_award_id":"JP24K10739","funder_id":"https://openalex.org/F4320320912","funder_display_name":"Ministry of Education, Culture, Sports, Science and Technology"}],"funders":[{"id":"https://openalex.org/F4320320912","display_name":"Ministry of Education, Culture, Sports, Science and Technology","ror":"https://ror.org/048rj2z13"}],"has_content":{"grobid_xml":false,"pdf":false},"content_urls":null,"referenced_works_count":19,"referenced_works":["https://openalex.org/W1570088680","https://openalex.org/W1665473994","https://openalex.org/W1971733522","https://openalex.org/W1986686546","https://openalex.org/W1992468072","https://openalex.org/W2056469339","https://openalex.org/W2058393559","https://openalex.org/W2139694871","https://openalex.org/W2550915126","https://openalex.org/W2799289346","https://openalex.org/W2895325993","https://openalex.org/W2898518884","https://openalex.org/W2950616289","https://openalex.org/W3177473377","https://openalex.org/W4220808049","https://openalex.org/W4224227142","https://openalex.org/W4238168833","https://openalex.org/W4311664551","https://openalex.org/W4391126435"],"related_works":["https://openalex.org/W7208071242","https://openalex.org/W3134107483","https://openalex.org/W4286835651","https://openalex.org/W4401716031","https://openalex.org/W2142669696","https://openalex.org/W7171653037","https://openalex.org/W1968607589","https://openalex.org/W3092031756","https://openalex.org/W4247888154","https://openalex.org/W2019984583"],"abstract_inverted_index":{"ABSTRACT":[0],"Irritability":[1],"is":[2,162,177],"a":[3,29,163],"core":[4],"challenge":[5],"in":[6,28,97,185],"Prader\u2013Willi":[7],"syndrome":[8],"(PWS).":[9],"This":[10],"study":[11],"investigated":[12],"the":[13,45,49,55,59,147,150],"independent":[14,140,168],"contribution":[15],"of":[16,31,48,58,83,149,166,169],"sensory":[17,95,158,175],"processing":[18,161],"to":[19],"irritability":[20,56,84],"while":[21],"controlling":[22],"for":[23,157,179],"clinical":[24],"and":[25,54,77,131,172,182],"genetic":[26,75,170],"backgrounds":[27],"cohort":[30],"101":[32],"individuals":[33],"with":[34],"PWS":[35],"(77":[36],"deletion,":[37],"24":[38],"maternal":[39],"uniparental":[40],"disomy":[41],"[mUPD]).":[42],"Caregivers":[43],"completed":[44],"Japanese":[46],"version":[47],"Short":[50],"Sensory":[51,160],"Profile":[52],"(SSP\u2010J)":[53],"subscale":[57],"Aberrant":[60],"Behavior":[61],"Checklist":[62],"(ABC\u2010J).":[63],"Hierarchical":[64],"multiple":[65],"regression":[66],"analysis":[67],"was":[68],"performed.":[69],"In":[70],"Step":[71,98],"1,":[72],"age,":[73],"sex,":[74],"subtype,":[76],"psychotropic":[78],"medication":[79],"use":[80],"explained":[81,100],"22.3%":[82],"variance":[85],"(":[86,114,121,127,134,142],"R":[87,105,115],"2":[88,99,106,116],"=":[89,107,117,123,129,136],"0.223,":[90],"p":[91,109,143],"<":[92,110,144],"0.001).":[93],"Adding":[94],"variables":[96],"an":[101],"additional":[102],"38.2%":[103],"(\u0394":[104],"0.382,":[108],"0.001),":[111],"totaling":[112],"60.6%":[113],"0.606).":[118],"Auditory":[119],"filtering":[120],"\u03b2":[122,128,135],"0.262),":[124],"under\u2010responsiveness/seeking":[125],"sensation":[126],"0.201),":[130],"visual/auditory":[132],"sensitivity":[133],"0.194)":[137],"were":[138],"significant":[139],"predictors":[141],"0.05).":[145],"Notably,":[146],"effect":[148],"mUPD":[151],"subtype":[152,171],"became":[153],"non\u2010significant":[154],"after":[155],"accounting":[156],"variables.":[159],"robust":[164],"predictor":[165],"irritability,":[167],"medication.":[173],"Evaluating":[174],"profiles":[176],"essential":[178],"environmental":[180],"adaptations":[181],"behavioral":[183],"interventions":[184],"PWS.":[186]},"counts_by_year":[],"updated_date":"2026-11-04T10:54:05.112534","created_date":"2026-04-30T00:00:00"}