{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,3]],"date-time":"2026-10-03T18:19:00Z","timestamp":1791051540122,"version":"4.1.0"},"reference-count":24,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2013,12,19]],"date-time":"2013-12-19T00:00:00Z","timestamp":1387411200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["American J of Med Genetics Pt A"],"published-print":{"date-parts":[[2014,3]]},"abstract":"<jats:sec>\n                    <jats:label\/>\n                    <jats:p>The developmental course of cognitive deficits in individuals with neurofibromatosis type 1 (NF1) is unclear. The objectives of this study were to determine the natural history of cognitive function and MRI T2\u2010hyperintesities (T2H) from childhood to adulthood and to examine whether the presence of discrete T2H in childhood can predict cognitive performance in adulthood. We present cognitive and structural neuroimaging data from 18 patients with NF1 and five sibling controls assessed prospectively across an 18\u2010year period. Longitudinal analyses revealed a significant increase in general cognitive function in patients with NF1 over the study period. Improvements were limited to individuals with discrete T2H in childhood. Patients without lesions in childhood exhibited a stable profile. The number of T2H decreased over time, particularly discrete lesions. Lesions located within the cerebral hemispheres and deep white matter were primarily stable, whereas those located in the basal ganglia, thalamus and brainstem tended to resolve. Our results support the hypothesis that resolution of T2H is accompanied by an improvement in general cognitive performance, possibly as a result of increased efficiency within white matter tracts. \u00a9 2013 Wiley Periodicals, Inc.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/ajmg.a.36338","type":"journal-article","created":{"date-parts":[[2013,12,19]],"date-time":"2013-12-19T15:15:04Z","timestamp":1387466104000},"page":"661-665","source":"Crossref","is-referenced-by-count":51,"title":["Longitudinal assessment of cognition and T2\u2010hyperintensities in NF1: An 18\u2010year study"],"prefix":"10.1002","volume":"164","author":[{"given":"Jonathan M.","family":"Payne","sequence":"first","affiliation":[{"name":"Institute for Neuroscience and Muscle Research The Children's Hospital at Westmead Sydney Australia"},{"name":"Discipline of Pediatrics and Child Health, Faculty of Medicine University of Sydney Sydney Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tania","family":"Pickering","sequence":"additional","affiliation":[{"name":"Department of Psychology, Faculty of Human Sciences Macquarie University Sydney Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Melanie","family":"Porter","sequence":"additional","affiliation":[{"name":"Department of Psychology, Faculty of Human Sciences Macquarie University Sydney Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emily C.","family":"Oates","sequence":"additional","affiliation":[{"name":"Institute for Neuroscience and Muscle Research The Children's Hospital at Westmead Sydney Australia"},{"name":"Discipline of Pediatrics and Child Health, Faculty of Medicine University of Sydney Sydney Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Navdeep","family":"Walia","sequence":"additional","affiliation":[{"name":"TY Nelson Department of Neurology The Children's Hospital at Westmead Sydney Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kristina","family":"Prelog","sequence":"additional","affiliation":[{"name":"Department of Medical Imaging The Children's Hospital at Westmead Sydney Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kathryn N.","family":"North","sequence":"additional","affiliation":[{"name":"Institute for Neuroscience and Muscle Research The Children's Hospital at Westmead Sydney Australia"},{"name":"Discipline of Pediatrics and Child Health, Faculty of Medicine University of Sydney Sydney Australia"},{"name":"Murdoch Children's Research Institute Melbourne Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2013,12,19]]},"reference":[{"key":"e_1_2_5_2_1","doi-asserted-by":"publisher","DOI":"10.1080\/87565640903265137"},{"key":"e_1_2_5_3_1","doi-asserted-by":"publisher","DOI":"10.1017\/S135561770286012X"},{"key":"e_1_2_5_4_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096-8628(19960216)67:1<98::AID-AJMG17>3.0.CO;2-K"},{"key":"e_1_2_5_5_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.33139"},{"key":"e_1_2_5_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/0022-510X(96)00022-6"},{"key":"e_1_2_5_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejpn.2011.10.002"},{"key":"e_1_2_5_8_1","doi-asserted-by":"publisher","DOI":"10.1038\/13158"},{"key":"e_1_2_5_9_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00247-006-0267-2"},{"key":"e_1_2_5_10_1","doi-asserted-by":"publisher","DOI":"10.1177\/08830738040190110201"},{"key":"e_1_2_5_11_1","doi-asserted-by":"publisher","DOI":"10.1212\/01.WNL.0000055090.78351.C1"},{"key":"e_1_2_5_12_1","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp.2006.108134"},{"key":"e_1_2_5_13_1","doi-asserted-by":"publisher","DOI":"10.1212\/01.wnl.0000179303.72345.ce"},{"key":"e_1_2_5_14_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1469-8749.2006.tb01268.x"},{"key":"e_1_2_5_15_1","doi-asserted-by":"publisher","DOI":"10.1037\/0022-006X.59.1.12"},{"key":"e_1_2_5_16_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1469-8749.2012.04399.x"},{"key":"e_1_2_5_17_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.46.6.1660"},{"key":"e_1_2_5_18_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.31923"},{"key":"e_1_2_5_19_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.44.5.878"},{"key":"e_1_2_5_20_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.35840"},{"key":"e_1_2_5_21_1","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp.2009.179630"},{"key":"e_1_2_5_22_1","volume-title":"Neurofibromatosis. Phenotype, natural history and pathogenesis","author":"Riccardi VM","year":"1986"},{"key":"e_1_2_5_23_1","doi-asserted-by":"publisher","DOI":"10.2214\/ajr.159.1.1609692"},{"key":"e_1_2_5_24_1","volume-title":"Wechsler adult intelligence scale","author":"Wechsler D","year":"1997"},{"key":"e_1_2_5_25_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.2007-3204"}],"container-title":["American Journal of Medical Genetics Part A"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fajmg.a.36338","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/ajmg.a.36338","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,10,6]],"date-time":"2023-10-06T16:10:44Z","timestamp":1696608644000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/ajmg.a.36338"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2013,12,19]]},"references-count":24,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2014,3]]}},"alternative-id":["10.1002\/ajmg.a.36338"],"URL":"https:\/\/doi.org\/10.1002\/ajmg.a.36338","archive":["Portico"],"relation":{},"ISSN":["1552-4825","1552-4833"],"issn-type":[{"value":"1552-4825","type":"print"},{"value":"1552-4833","type":"electronic"}],"subject":[],"published":{"date-parts":[[2013,12,19]]}}}