{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,8]],"date-time":"2026-10-08T02:08:19Z","timestamp":1791425299482,"version":"4.3.3"},"reference-count":44,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2012,11,16]],"date-time":"2012-11-16T00:00:00Z","timestamp":1353024000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Develop Med Child Neuro"],"published-print":{"date-parts":[[2013,2]]},"abstract":"<jats:p><jats:bold>Aim\u2002<\/jats:bold> To investigate psychopathology in children with neurofibromatosis type 1 (NF1), particularly the prevalence of autism spectrum disorder (ASD) and attention\u2010deficit\u2013hyperactivity disorder (ADHD) symptomatology, using a population\u2010based sampling approach.<\/jats:p><jats:p><jats:bold>Method\u2002<\/jats:bold> Standard questionnaire screen reports were analysed for ASD (Social Responsiveness Scale, SRS), ADHD (Conners\u2019 Parent Rating Scale\u2010 Revised, CPRS\u2010R), and other psychiatric morbidity (Strengths and Difficulties Questionnaire, SDQ) from parents and teachers of children aged from 4 to 16\u2003years (112 females, 95 males) on the UK North West Regional Genetic Service register for NF1.<\/jats:p><jats:p><jats:bold>Results\u2002<\/jats:bold> Parental response rate was 52.7% (109\/207 children; 59 females, 50 males, mean age 9y 11mo, SD 3y 3mo). The SRS showed that in 29.4% (32\/109) of children, autism was in the severe, clinical range (<jats:italic>T<\/jats:italic>\u2010score&gt;75) and in 26.6% (29\/109) in the mild to moderate range (<jats:italic>T<\/jats:italic>\u2010score 60\u201375). CPRS\u2010R scores showed that in 53.8% (57\/106) of children autism was in the clinical ADHD range (ADHD index <jats:italic>T<\/jats:italic>\u2010score&gt;65). Based on their scores on the SDQ total difficulties scale, 41.5% (44\/106) of children were in the abnormal range and 14.2% (15\/106) were in the borderline range. Twenty\u2010five per cent (26\/104) of children met criteria for both clinical autism and ADHD.<\/jats:p><jats:p><jats:bold>Interpretation\u2002<\/jats:bold> This representative population\u2010based sample of children with NF1 indicates a high prevalence of ASD symptoms associated with NF1 as well as substantial co\u2010occurrence with ADHD symptoms. The findings clarify the psychopathology of NF1 and show the disorder as a potentially important single\u2010gene cause for autism symptoms.<\/jats:p>","DOI":"10.1111\/dmcn.12043","type":"journal-article","created":{"date-parts":[[2012,11,16]],"date-time":"2012-11-16T20:16:56Z","timestamp":1353097016000},"page":"139-145","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":173,"title":["Autism and other psychiatric comorbidity in neurofibromatosis type 1: evidence from a population\u2010based study"],"prefix":"10.1111","volume":"55","author":[{"given":"SHRUTI","family":"GARG","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"ANNUKKA","family":"LEHTONEN","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"SUSAN M","family":"HUSON","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"RICHARD","family":"EMSLEY","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"DOROTHY","family":"TRUMP","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"D GARETH","family":"EVANS","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"JONATHAN","family":"GREEN","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2012,11,16]]},"reference":[{"key":"e_1_2_8_2_2","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.33139"},{"key":"e_1_2_8_3_2","doi-asserted-by":"publisher","DOI":"10.1212\/01.wnl.0000179303.72345.ce"},{"key":"e_1_2_8_4_2","doi-asserted-by":"publisher","DOI":"10.1097\/00005053-199308000-00001"},{"key":"e_1_2_8_5_2","doi-asserted-by":"publisher","DOI":"10.2307\/3002000"},{"key":"e_1_2_8_6_2","first-page":"84","article-title":"Neurofibromatosis in Gothenburg, Sweden. III. Psychiatric and social aspects","volume":"2","author":"Samuelsson B","year":"1989","journal-title":"Neurofibromatosis"},{"key":"e_1_2_8_7_2","doi-asserted-by":"publisher","DOI":"10.1097\/00004703-199608000-00004"},{"key":"e_1_2_8_8_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3476(99)70296-9"},{"key":"e_1_2_8_9_2","doi-asserted-by":"publisher","DOI":"10.1111\/j.1469-8749.2005.tb01128.x"},{"key":"e_1_2_8_10_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3476(05)83163-4"},{"key":"e_1_2_8_11_2","doi-asserted-by":"publisher","DOI":"10.1177\/1087054707299398"},{"key":"e_1_2_8_12_2","doi-asserted-by":"publisher","DOI":"10.1177\/1359104503008001006"},{"key":"e_1_2_8_13_2","doi-asserted-by":"publisher","DOI":"10.1111\/j.1469-8749.2002.tb00780.x"},{"key":"e_1_2_8_14_2","volume-title":"Mental Disorders: A Glossary and Guide to their Classification in Accordance with the 10th Revision of the International Classification of Diseases: Research Diagnostic Criteria (ICD\u201010)","author":"World Health Organization","year":"1993"},{"key":"e_1_2_8_15_2","first-page":"15","article-title":"Neurofibromatosis in infantile autism and other types of childhood psychoses","volume":"55","author":"Mouridsen S","year":"1992","journal-title":"Acta Paedopsychiatr"},{"key":"e_1_2_8_16_2","doi-asserted-by":"publisher","DOI":"10.1007\/BF01487072"},{"key":"e_1_2_8_17_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(06)69041-7"},{"key":"e_1_2_8_18_2","doi-asserted-by":"publisher","DOI":"10.1007\/BF02408551"},{"key":"e_1_2_8_19_2","doi-asserted-by":"publisher","DOI":"10.1097\/00004583-199711000-00019"},{"key":"e_1_2_8_20_2","doi-asserted-by":"publisher","DOI":"10.1017\/S0012162204000921"},{"key":"e_1_2_8_21_2","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.31923"},{"key":"e_1_2_8_22_2","doi-asserted-by":"publisher","DOI":"10.1111\/j.1469-8749.2010.03639.x"},{"key":"e_1_2_8_23_2","doi-asserted-by":"publisher","DOI":"10.1007\/s10519-010-9430-5"},{"key":"e_1_2_8_24_2","doi-asserted-by":"publisher","DOI":"10.1038\/ejhg.2011.113"},{"key":"e_1_2_8_25_2","doi-asserted-by":"publisher","DOI":"10.1001\/archneur.1988.00520290115023"},{"key":"e_1_2_8_26_2","volume-title":"Neurofibromatosis: Phenotype, Natural History and Pathogenesis","author":"Riccardi V","year":"1992"},{"key":"e_1_2_8_27_2","volume-title":"The Social Responsiveness Scale","author":"Constantino J","year":"2002"},{"key":"e_1_2_8_28_2","doi-asserted-by":"publisher","DOI":"10.1023\/A:1025014929212"},{"key":"e_1_2_8_29_2","doi-asserted-by":"publisher","DOI":"10.1023\/A:1022602400621"},{"key":"e_1_2_8_30_2","doi-asserted-by":"publisher","DOI":"10.1111\/j.1469-7610.1997.tb01545.x"},{"key":"e_1_2_8_31_2","doi-asserted-by":"publisher","DOI":"10.1192\/bjp.177.6.534"},{"key":"e_1_2_8_32_2","volume-title":"The English Indices of Deprivation 2007","author":"Noble M","year":"2008"},{"key":"e_1_2_8_33_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.ridd.2009.06.003"},{"key":"e_1_2_8_34_2","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2788.2003.00567.x"},{"key":"e_1_2_8_35_2","doi-asserted-by":"publisher","DOI":"10.1037\/0033-2909.101.2.213"},{"key":"e_1_2_8_36_2","doi-asserted-by":"publisher","DOI":"10.1111\/j.1469-7610.1991.tb00350.x"},{"key":"e_1_2_8_37_2","doi-asserted-by":"publisher","DOI":"10.1111\/1469-7610.00424"},{"key":"e_1_2_8_38_2","volume-title":"Diagnostic and Statistical Manual of Mental Disorder","author":"American Psychiatric Association","year":"2000"},{"key":"e_1_2_8_39_2","doi-asserted-by":"publisher","DOI":"10.1007\/s10803-006-0125-y"},{"key":"e_1_2_8_40_2","doi-asserted-by":"publisher","DOI":"10.1097\/CHI.0b013e318179964f"},{"key":"e_1_2_8_41_2","first-page":"570","article-title":"A genomewide screen for autism: strong evidence for linkage to chromosomes 2q, 7q, and 16p","volume":"69","author":"International Molecular Genetic Study of Autism Consortium (IMGSAC)","year":"2001","journal-title":"Am J Med Genet"},{"key":"e_1_2_8_42_2","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.b.20119"},{"key":"e_1_2_8_43_2","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096-8628(19991215)88:6<729::AID-AJMG26>3.0.CO;2-Q"},{"key":"e_1_2_8_44_2","doi-asserted-by":"publisher","DOI":"10.1001\/archpsyc.60.5.524"},{"key":"e_1_2_8_45_2","doi-asserted-by":"publisher","DOI":"10.1111\/1469-7610.00742"}],"container-title":["Developmental Medicine &amp; Child Neurology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fdmcn.12043","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/dmcn.12043","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/dmcn.12043","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/dmcn.12043","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,8,18]],"date-time":"2023-08-18T07:33:15Z","timestamp":1692343995000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/dmcn.12043"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2012,11,16]]},"references-count":44,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2013,2]]}},"alternative-id":["10.1111\/dmcn.12043"],"URL":"https:\/\/doi.org\/10.1111\/dmcn.12043","archive":["Portico"],"relation":{},"ISSN":["0012-1622","1469-8749"],"issn-type":[{"value":"0012-1622","type":"print"},{"value":"1469-8749","type":"electronic"}],"subject":[],"published":{"date-parts":[[2012,11,16]]},"assertion":[{"value":"2012-11-16","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}