{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T02:52:49Z","timestamp":1787021569300,"version":"build-2736575974"},"reference-count":43,"publisher":"Informa UK Limited","issue":"1","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2022,1,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Angelman Syndrome (AS) is a neurodevelopmental disorder most commonly caused by the impaired expression of the maternal UBE3A gene on chromosome 15. Though anxiety has been identified as a frequently present characteristic in AS, there are limited studies examining anxiety in this population. Studies of anxiety in other neurodevelopmental disorders have found disorder specific symptoms of anxiety and age specific displays of anxiety symptoms. However, there is a consistent challenge in identifying anxiety in people with neurodevelopmental disorders given the lack of measurement instruments specifically designed for this population. Given the limited information about AS and anxiety, the aims of the current project were to (a) examine symptoms of anxiety in children with AS and (b) determine the correlates of anxiety in children with AS. Participants included 42 adult caregivers of youth with AS in the AS Natural History study who completed the Developmental Behavior Checklist (DBC). The results found that 26% of the sample demonstrated elevated symptoms of anxiety and established a relationship between elevated anxiety in youth with AS and higher levels of irritability, hyperactivity, self-absorbed behaviors, and disruptive\/antisocial behaviors. Findings from this research provide a foundation for tailoring evidence-based assessments and treatments for youth with AS and anxiety.<\/jats:p>","DOI":"10.1352\/1944-7558-127.1.1","type":"journal-article","created":{"date-parts":[[2022,1,3]],"date-time":"2022-01-03T14:48:32Z","timestamp":1641221312000},"page":"1-10","source":"Crossref","is-referenced-by-count":8,"title":["Anxiety in Angelman Syndrome"],"prefix":"10.1080","volume":"127","author":[{"given":"Stacey C.","family":"Grebe","sequence":"first","affiliation":[{"name":"Stacey C. Grebe, Danica L. Limon, Morgan M. McNeel, and Andrew Guzick, Baylor College of Medicine"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Danica L.","family":"Limon","sequence":"additional","affiliation":[{"name":"Stacey C. Grebe, Danica L. Limon, Morgan M. McNeel, and Andrew Guzick, Baylor College of Medicine"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Morgan M.","family":"McNeel","sequence":"additional","affiliation":[{"name":"Stacey C. Grebe, Danica L. Limon, Morgan M. McNeel, and Andrew Guzick, Baylor College of Medicine"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrew","family":"Guzick","sequence":"additional","affiliation":[{"name":"Stacey C. Grebe, Danica L. Limon, Morgan M. McNeel, and Andrew Guzick, Baylor College of Medicine"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sarika U.","family":"Peters","sequence":"additional","affiliation":[{"name":"Sarika U. Peters, Vanderbilt Kennedy Center"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wen-Hann","family":"Tan","sequence":"additional","affiliation":[{"name":"Wen-Hann Tan and Anjali Sadhwani, Boston Children's Hospital"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anjali","family":"Sadhwani","sequence":"additional","affiliation":[{"name":"Wen-Hann Tan and Anjali Sadhwani, Boston Children's Hospital"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Carlos A.","family":"Bacino","sequence":"additional","affiliation":[{"name":"Carlos A. Bacino, Baylor College of Medicine and Texas Children's Hospital"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lynne M.","family":"Bird","sequence":"additional","affiliation":[{"name":"Lynne M. Bird, University of California and Boston Children's Hospital"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rodney C.","family":"Samaco","sequence":"additional","affiliation":[{"name":"Rodney C. Samaco, Baylor College of Medicine"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Leandra N.","family":"Berry","sequence":"additional","affiliation":[{"name":"Leandra N. Berry, Baylor College of Medicine and Texas Children's Hospital"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wayne K.","family":"Goodman","sequence":"additional","affiliation":[{"name":"Wayne K. Goodman, Baylor College of Medicine"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sophie C.","family":"Schneider","sequence":"additional","affiliation":[{"name":"Sophie C. Schneider and Eric A. Storch, Baylor College of Medicine"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eric A.","family":"Storch","sequence":"additional","affiliation":[{"name":"Sophie C. Schneider and Eric A. Storch, Baylor College of Medicine"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"301","published-online":{"date-parts":[[2022,1,3]]},"reference":[{"key":"2022010319482435900_i1944-7558-127-1-1-Achenbach1","unstructured":"Achenbach,\n              T. M.,\n             &\n\t\t\t\t\t\tRescorla,L.\n          (2001).\n\t\t\t\t\tManual for the ASEBA school-age forms & profiles: An integrated system of multi-informant assessment.\n\t\t\t\t\tAseba."},{"key":"2022010319482435900_i1944-7558-127-1-1-Albrecht1","doi-asserted-by":"crossref","unstructured":"Albrecht,\n              U.,\n            \n            \n              Sutcliffe,\n              J. S.,\n            \n            \n              Cattanach,\n              B. M.,\n            \n            \n              Beechey,\n              C. V.,\n            \n            \n              Armstrong,\n              D.,\n            \n            \n              Eichele,\n              G.,\n             &\n\t\t\t\t\t\tBeaudet,A. L.\n          (1997).\n\t\t\t\t\tImprinted expression of the murine Angelman syndrome gene, Ube3a, in hippocampal and Purkinje neurons.\n\t\t\t\t\tNature Genetics, 17(1), 75.https:\/\/doi.org\/10.1038\/ng0997-75","DOI":"10.1038\/ng0997-75"},{"key":"2022010319482435900_i1944-7558-127-1-1-Aman1","doi-asserted-by":"crossref","unstructured":"Aman,\n              M. G.,\n            \n            \n              Singh,\n              N. N.,\n            \n            \n              Stewart,\n              A. W.,\n             &\n\t\t\t\t\t\tField,C. J.\n          (1985).\n\t\t\t\t\tThe aberrant behavior checklist: a behavior rating scale for the assessment of treatment effects.\n\t\t\t\t\tAmerican Journal of Mental Deficiency, 89(5),\n\t\t\t\t\t485\u2013\n\t\t\t\t\t91.\n\t\t\t\t\thttps:\/\/doi.org\/10.1037\/t10453-000","DOI":"10.1037\/t10453-000"},{"key":"2022010319482435900_i1944-7558-127-1-1-Bailey1","doi-asserted-by":"crossref","unstructured":"Bailey,\n              N.\n            \n          \n          (2007).\n\t\t\t\t\tPrevalence of psychiatric disorders in adults with moderate to profound learning disabilities.\n\t\t\t\t\tAdvances in Mental Health and Learning Disabilities, 1(2),\n\t\t\t\t\t36\u2013\n\t\t\t\t\t44.\n\t\t\t\t\thttps:\/\/doi.org\/10.1108\/17530180200700019","DOI":"10.1108\/17530180200700019"},{"key":"2022010319482435900_i1944-7558-127-1-1-Bayley1","doi-asserted-by":"crossref","unstructured":"Bayley,\n              N.\n            \n          \n          (2005).\n\t\t\t\t\tBayley Scales of Infant and Toddler Development, Third Edition (Bayley-III).\n\t\t\t\t\tHarcourt Assessment,\n\t\t\t\t\tInc.","DOI":"10.1037\/t14978-000"},{"key":"2022010319482435900_i1944-7558-127-1-1-Buiting1","doi-asserted-by":"crossref","unstructured":"Buiting,\n              K.,\n            \n            \n              Williams,\n              C.,\n             &\n\t\t\t\t\t\tHorsthemke,B.\n          (2016).\n\t\t\t\t\tAngelman syndrome\u2014insights into a rare neurogenetic disorder.\n\t\t\t\t\tNature Reviews Neurology, 12(10), 584.https:\/\/doi.org\/10.1038\/nrneurol.2016.133","DOI":"10.1038\/nrneurol.2016.133"},{"key":"2022010319482435900_i1944-7558-127-1-1-Cohen1","doi-asserted-by":"crossref","unstructured":"Cohen,\n              J.\n            \n          \n          (1992).\n\t\t\t\t\tA power primer.\n\t\t\t\t\tPsychological Bulletin,112(1),\n\t\t\t\t\t155.https:\/\/doi.org\/10.1037\/0033-2909.112.1.155","DOI":"10.1037\/0033-2909.112.1.155"},{"key":"2022010319482435900_i1944-7558-127-1-1-Crawford1","doi-asserted-by":"crossref","unstructured":"Crawford,\n              H.,\n            \n            \n              Waite,\n              J.,\n             &\n\t\t\t\t\t\tOliver,C.\n          (2017).\n\t\t\t\t\tDiverse profiles of anxiety related disorders in Fragile X, Cornelia de Lange and Rubinstein\u2013Taybi syndromes.\n\t\t\t\t\tJournal of Autism and Developmental Disorders,\n\t\t\t\t\t47(12),\n\t\t\t\t\t3728\u2013\n\t\t\t\t\t3740.\n\t\t\t\t\thttps:\/\/doi.org\/10.1007\/s10803-016-3015-y","DOI":"10.1007\/s10803-016-3015-y"},{"key":"2022010319482435900_i1944-7558-127-1-1-Dagli1","doi-asserted-by":"crossref","unstructured":"Dagli,\n              A.,\n            \n            \n              Buiting,\n              K.,\n             &\n\t\t\t\t\t\tWilliams,C.\n          (2012).\n\t\t\t\t\tMolecular and clinical aspects of Angelman Syndrome.\n\t\t\t\t\tMolecular Syndromology,\n\t\t\t\t\t2(3-5),\n\t\t\t\t\t100\u2013\n\t\t\t\t\t112.\n\t\t\t\t\thttps:\/\/doi.org\/10.1159\/000328837","DOI":"10.1159\/000328837"},{"key":"2022010319482435900_i1944-7558-127-1-1-Dykens1","unstructured":"Dykens,\n              E. M.,\n            \n            \n              Hodapp,\n              R. M.,\n             &\n\t\t\t\t\t\tEvans,D. W.\n          (1994).\n\t\t\t\t\tProfiles and development of adaptive behavior in children with Down Syndrome.\n\t\t\t\t\tAmerican Journal on Mental Retardation, 98(5),\n\t\t\t\t\t580\u2013\n\t\t\t\t\t587."},{"key":"2022010319482435900_i1944-7558-127-1-1-Einfeld1","unstructured":"Einfeld,\n              S. L.,\n             &\n\t\t\t\t\t\tTonge,B. J.\n          (2002).\n\t\t\t\t\tManual for the Developmental Behaviour Checklist: Primary carer version (DBC-P) and teacher version (DBC-T).\n\t\t\t\t\tMonash University Centre for Developmental Psychiatry and Psychology."},{"key":"2022010319482435900_i1944-7558-127-1-1-Ezell1","doi-asserted-by":"crossref","unstructured":"Ezell,\n              J.,\n            \n            \n              Hogan,\n              A.,\n            \n            \n              Fairchild,\n              A.,\n            \n            \n              Hills,\n              K.,\n            \n            \n              Klusek,\n              J.,\n            \n            \n              Abbeduto,\n              L.,\n             &\n\t\t\t\t\t\tRoberts,J.\n          (2019).\n\t\t\t\t\tPrevalence and predictors of anxiety disorders in adolescent and adult males with autism spectrum disorder and fragile X syndrome.\n\t\t\t\t\tJournal of Autism and Developmental Disorders,\n\t\t\t\t\t49(3),\n\t\t\t\t\t1131\u2013\n\t\t\t\t\t1141.\n\t\t\t\t\thttps:\/\/doi.org\/10.1007\/s10803-018-3804-6","DOI":"10.1007\/s10803-018-3804-6"},{"key":"2022010319482435900_i1944-7558-127-1-1-Freund1","doi-asserted-by":"crossref","unstructured":"Freund,\n              L. S.,\n             &\n\t\t\t\t\t\tReiss,A. L.\n          (1991).\n\t\t\t\t\tRating problem behaviors in outpatients with mental retardation: Use of the Aberrant Behavior Checklist.\n\t\t\t\t\tResearch in Developmental Disabilities, 12(4),\n\t\t\t\t\t435\u2013\n\t\t\t\t\t451.\n\t\t\t\t\thttps:\/\/doi.org\/10.1016\/0891-4222(91)90037-S","DOI":"10.1016\/0891-4222(91)90037-S"},{"key":"2022010319482435900_i1944-7558-127-1-1-Gentile1","doi-asserted-by":"crossref","unstructured":"Gentile,\n              J. K.,\n            \n            \n              Tan,\n              W.-H.,\n            \n            \n              Horowitz,\n              L. T.,\n            \n            \n              Bacino,\n              C. A.,\n            \n            \n              Skinner,\n              S. A.,\n            \n            \n              Barbieri-Welge,\n              R.,\n            \n            \n              Bauer-Carlin,\n              A.,\n            \n            \n              Beaudet,\n              A. L.,\n            \n            \n              Bichell,\n              T. J.,\n             &\n\t\t\t\t\t\tLee,H.-S.\n          (2010).\n\t\t\t\t\tA neurodevelopmental survey of Angelman syndrome with genotype-phenotype correlations.\n\t\t\t\t\tJournal of Developmental and Behavioral Pediatrics: \n\t\t\t\t\tJDBP,31(7), 592.\n\t\t\t\t\thttps:\/\/doi.org\/10.1097\/DBP.0b013e3181ee408e","DOI":"10.1097\/DBP.0b013e3181ee408e"},{"key":"2022010319482435900_i1944-7558-127-1-1-Godavarthi1","doi-asserted-by":"crossref","unstructured":"Godavarthi,\n              S. K.,\n            \n            \n              Dey,\n              P.,\n            \n            \n              Maheshwari,\n              M.,\n             &\n\t\t\t\t\t\tRanjan Jana,N.\n          (2012).\n\t\t\t\t\tDefective glucocorticoid hormone receptor signaling leads to increased stress and anxiety in a mouse model of Angelman syndrome.\n\t\t\t\t\tHuman Molecular Genetics,\n\t\t\t\t\t21(8),\n\t\t\t\t\t1824\u2013\n\t\t\t\t\t1834.\n\t\t\t\t\thttps:\/\/doi.org\/10.1093\/hmg\/ddr614","DOI":"10.1093\/hmg\/ddr614"},{"key":"2022010319482435900_i1944-7558-127-1-1-Hair1","unstructured":"Hair,\n              J. F.,\n            \n            \n              Black,\n              W. C.,\n            \n            \n              Babin,\n              B. J.,\n            \n            \n              Anderson,\n              R. E.,\n             &\n\t\t\t\t\t\tTatham,R. L.\n          (1998).\n\t\t\t\t\tMultivariate data analysis(Vol. 5,\n\t\t\t\t\tNo. 3,\n\t\t\t\t\tpp. 207\u2013219).\n\t\t\t\t\tPrentice Hall."},{"key":"2022010319482435900_i1944-7558-127-1-1-Hardiman1","doi-asserted-by":"crossref","unstructured":"Hardiman,\n              R.,\n             &\n\t\t\t\t\t\tBratt,A.\n          (2016).\n\t\t\t\t\tHypothalamic-pituitary-adrenal axis function in Fragile X Syndrome and its relationship to behaviour: A systematic review.\n\t\t\t\t\tPhysiology & Behavior,\n\t\t\t\t\t167,\n\t\t\t\t\t341\u2013\n\t\t\t\t\t353.\n\t\t\t\t\thttps:\/\/doi.org\/10.1016\/j.physbeh.2016.09.030","DOI":"10.1016\/j.physbeh.2016.09.030"},{"key":"2022010319482435900_i1944-7558-127-1-1-Kerns1","doi-asserted-by":"crossref","unstructured":"Kerns,\n              C. M.,\n            \n            \n              Kendall,\n              P. C.,\n            \n            \n              Berry,\n              L.,\n            \n            \n              Souders,\n              M. C.,\n            \n            \n              Franklin,\n              M. E.,\n            \n            \n              Schultz,\n              R. T.,\n            \n            et al.\n\t\t\t\t\t\n          (2014).\n\t\t\t\t\tTraditional and atypical presentations of anxiety in youth with autism spectrum disorder.\n\t\t\t\t\tJournal of Autism and Developmental Disorders,\n\t\t\t\t\t44(11),\n\t\t\t\t\t2851\u2013\n\t\t\t\t\t2861.\n\t\t\t\t\thttps:\/\/doi.org\/10.1007\/s10803-014-2141-7.","DOI":"10.1007\/s10803-014-2141-7"},{"key":"2022010319482435900_i1944-7558-127-1-1-Larson1","doi-asserted-by":"crossref","unstructured":"Larson,\n              A. M.,\n            \n            \n              Shinnick,\n              J. E.,\n            \n            \n              Shaaya,\n              E. A.,\n            \n            \n              Thiele,\n              E. A.,\n             &\n\t\t\t\t\t\tThibert,R. L.\n          (2015).\n\t\t\t\t\tAngelman syndrome in adulthood. American Journal of Medical Genetics.\n\t\t\t\t\tPart A, 167(2),\n\t\t\t\t\t331\u2013\n\t\t\t\t\t344.\n\t\t\t\t\thttps:\/\/doi.org\/10.1002\/ajmg.a.36864","DOI":"10.1002\/ajmg.a.36864"},{"key":"2022010319482435900_i1944-7558-127-1-1-Lossie1","doi-asserted-by":"crossref","unstructured":"Lossie,\n              A. C.,\n            \n            \n              Whitney,\n              M. M.,\n            \n            \n              Amidon,\n              D.,\n            \n            \n              Dong,\n              H. J.,\n            \n            \n              Chen,\n              P.,\n            \n            \n              Theriaque,\n              D.,\n            \n            \n              Hutson,\n              A.,\n            \n            \n              Nichols,\n              R.D.,\n            \n            \n              Zori,\n              R.T.,\n            \n            \n              Williams,\n              C.A.,\n             &\n\t\t\t\t\t\tDriscoll,D. J.\n          (2001).\n\t\t\t\t\tDistinct phenotypes distinguish the molecular classes of Angelman syndrome.\n\t\t\t\t\tJournal of Medical Genetics,\n\t\t\t\t\t38(12),\n\t\t\t\t\t834\u2013\n\t\t\t\t\t845.\n\t\t\t\t\thttps:\/\/doi.org\/10.1136\/jmg.38.12.834","DOI":"10.1136\/jmg.38.12.834"},{"key":"2022010319482435900_i1944-7558-127-1-1-March1","doi-asserted-by":"crossref","unstructured":"March,\n              J. S.,\n            \n            \n              Parker,\n              J. D.,\n            \n            \n              Sullivan,\n              K.,\n            \n            \n              Stallings,\n              P.,\n             &\n\t\t\t\t\t\tConners,C. K.\n          (1997).\n\t\t\t\t\tThe Multidimensional Anxiety Scale for Children (MASC): Factor structure, reliability, and validity.\n\t\t\t\t\tJournal of the American Academy of Child & Adolescent Psychiatry,\n\t\t\t\t\t36(4),\n\t\t\t\t\t554\u2013\n\t\t\t\t\t565.\n\t\t\t\t\thttps:\/\/doi.org\/10.1097\/00004583-199704000-00019","DOI":"10.1097\/00004583-199704000-00019"},{"key":"2022010319482435900_i1944-7558-127-1-1-Margolis1","doi-asserted-by":"crossref","unstructured":"Margolis,\n              S. S.,\n            \n            \n              Sell,\n              G.,\n            \n            \n              Zbinden,\n              M. A.,\n             &\n\t\t\t\t\t\tBird,L.\n          (2015).\n\t\t\t\t\tAngelman Syndrome.\n\t\t\t\t\tNeurotherapeutics, 12(3),\n\t\t\t\t\t641\u2013\n\t\t\t\t\t650.\n\t\t\t\t\thttps:\/\/doi.org\/10.1007\/s13311-015-0361-y","DOI":"10.1007\/s13311-015-0361-y"},{"key":"2022010319482435900_i1944-7558-127-1-1-Matherly1","doi-asserted-by":"crossref","unstructured":"Matherly,\n              S.,\n            \n            \n              Klusek,\n              J.,\n            \n            \n              Thurman,\n              A.,\n            \n            \n              Mcduffie,\n              A.,\n            \n            \n              Abbeduto,\n              L.,\n             &\n\t\t\t\t\t\tRoberts,J.\n          (2018).\n\t\t\t\t\tCortisol profiles differentiated in adolescents and young adult males with Fragile X syndrome versus autism spectrum disorder.\n\t\t\t\t\tDevelopmental Psychobiology,\n\t\t\t\t\t60(1),\n\t\t\t\t\t78\u2013\n\t\t\t\t\t89.\n\t\t\t\t\thttps:\/\/doi.org\/10.1002\/dev.21578","DOI":"10.1002\/dev.21578"},{"key":"2022010319482435900_i1944-7558-127-1-1-Mattila1","doi-asserted-by":"crossref","unstructured":"Mattila,\n              M.-L.,\n            \n            \n              Hurtig,\n              T.,\n            \n            \n              Haapsamo,\n              H.,\n            \n            \n              Jussila,\n              K.,\n            \n            \n              Kuusikko-Gauffin,\n              S.,\n            \n            \n              Kielinen,\n              M.,\n            \n            \n              Linna,\n              S.-L.,\n            \n            \n              Ebeling,\n              H.,\n            \n            \n              Bloigu,\n              R.,\n            \n            \n              Joskitt,\n              L.,\n            \n            \n              Pauls,\n              D.,\n             &\n\t\t\t\t\t\tMoilanen,I.\n          (2010).\n\t\t\t\t\tComorbid psychiatric disorders associated with Asperger Syndrome\/High-Functioning Autism: A community- and clinic-based study.\n\t\t\t\t\tJournal of Autism and Developmental Disorders, 40(9),\n\t\t\t\t\t1080\u2013\n\t\t\t\t\t1093.\n\t\t\t\t\thttps:\/\/doi.org\/10.1007\/s10803-010-0958-2","DOI":"10.1007\/s10803-010-0958-2"},{"key":"2022010319482435900_i1944-7558-127-1-1-McDougall1","doi-asserted-by":"crossref","unstructured":"McDougall,\n              J.,\n             &\n\t\t\t\t\t\tWright,V.\n          (2017).\n\t\t\t\t\tFactors related to symptoms of social anxiety in youth with cerebral palsy.\n\t\t\t\t\tDevelopmental Medicine & Child Neurology, 59,\n\t\t\t\t\t37\u2013\n\t\t\t\t\t37.\n\t\t\t\t\thttps:\/\/doi.org\/10.1111\/dmcn.52_13511","DOI":"10.1111\/dmcn.52_13511"},{"key":"2022010319482435900_i1944-7558-127-1-1-Miodrag1","doi-asserted-by":"crossref","unstructured":"Miodrag,\n              N.,\n             &\n\t\t\t\t\t\tPeters,S.\n          (2015).\n\t\t\t\t\tParent stress across molecular subtypes of children with Angelman syndrome.\n\t\t\t\t\tJournal of Intellectual Disability Research, 59(9),\n\t\t\t\t\t816\u2013\n\t\t\t\t\t826.\n\t\t\t\t\thttps:\/\/doi.org\/10.1111\/jir.12195","DOI":"10.1111\/jir.12195"},{"key":"2022010319482435900_i1944-7558-127-1-1-Prasad1","doi-asserted-by":"crossref","unstructured":"Prasad,\n              A.,\n            \n            \n              Grocott,\n              O.,\n            \n            \n              Parkin,\n              K.,\n            \n            \n              Larson,\n              A.,\n             &\n\t\t\t\t\t\tThibert,R. L.\n          (2018).\n\t\t\t\t\tAngelman syndrome in adolescence and adulthood: A retrospective chart review of 53 cases.\n\t\t\t\t\tAmerican Journal of Medical Genetics Part A, 176(6),\n\t\t\t\t\t1327\u2013\n\t\t\t\t\t1334.\n\t\t\t\t\thttps:\/\/doi.org\/10.1002\/ajmg.a.38694","DOI":"10.1002\/ajmg.a.38694"},{"key":"2022010319482435900_i1944-7558-127-1-1-Pruijssers1","doi-asserted-by":"crossref","unstructured":"Pruijssers,\n              A. C.,\n            \n            \n              Meijel,\n              B.,\n            \n            \n              Maaskant,\n              M.,\n            \n            \n              Nijssen,\n              W.,\n             &\n\t\t\t\t\t\tAchterberg,T.\n          (2014).\n\t\t\t\t\tThe relationship between challenging behaviour and anxiety in adults with intellectual disabilities: a literature review.\n\t\t\t\t\tJournal of Intellectual Disability Research, 58(2),\n\t\t\t\t\t162\u2013\n\t\t\t\t\t171.\n\t\t\t\t\thttps:\/\/doi.org\/10.1111\/jir.12012","DOI":"10.1111\/jir.12012"},{"key":"2022010319482435900_i1944-7558-127-1-1-Reynolds1","doi-asserted-by":"crossref","unstructured":"Reynolds,\n              C. R.,\n             &\n\t\t\t\t\t\tRichmond,B. O.\n          (1978).\n\t\t\t\t\tWhat I Think and Feel: A revised measure of children's manifest anxiety.\n\t\t\t\t\tJournal of Abnormal Child Psychology,\n\t\t\t\t\t6(2),\n\t\t\t\t\t271\u2013\n\t\t\t\t\t280.\n\t\t\t\t\thttps:\/\/doi.org\/10.1007\/BF00919131","DOI":"10.1007\/BF00919131"},{"key":"2022010319482435900_i1944-7558-127-1-1-Royston1","doi-asserted-by":"crossref","unstructured":"Royston,\n              R.,\n            \n            \n              Howlin,\n              P.,\n            \n            \n              Waite,\n              J.,\n             &\n\t\t\t\t\t\tOliver,C.\n          (2017).\n\t\t\t\t\tAnxiety disorders in Williams syndrome contrasted with intellectual disability and the general population: A systematic review and meta-analysis.\n\t\t\t\t\tJournal of Autism and Developmental Disorders,\n\t\t\t\t\t47(12),\n\t\t\t\t\t3765\u2013\n\t\t\t\t\t3777.\n\t\t\t\t\thttps:\/\/doi.org\/10.1007\/s10803-016-2909-z","DOI":"10.1007\/s10803-016-2909-z"},{"key":"2022010319482435900_i1944-7558-127-1-1-Sadhwani1","doi-asserted-by":"crossref","unstructured":"Sadhwani,\n              A.,\n            \n            \n              Willen,\n              J. M.,\n            \n            \n              LaVallee,\n              N.,\n            \n            \n              Stepanians,\n              M.,\n            \n            \n              Miller,\n              H.,\n            \n            \n              Peters,\n              S. U.,\n            \n            \n              Barbieri-Welge,\n              R. L.,\n            \n            \n              Horowitz,\n              L. T.,\n            \n            \n              Noll,\n              L. M.,\n            \n            \n              Hundley,\n              R. J.,\n            \n            \n              Bird,\n              L. M.,\n             &\n\t\t\t\t\t\tTan,W.\n          (2019).\n\t\t\t\t\tMaladaptive behaviors in individuals with Angelman syndrome. American Journal of Medical Genetics.\n\t\t\t\t\tPart A,\n\t\t\t\t\t179(6),\n\t\t\t\t\t983\u2013\n\t\t\t\t\t992.\n\t\t\t\t\thttps:\/\/doi.org\/10.1002\/ajmg.a.61140","DOI":"10.1002\/ajmg.a.61140"},{"key":"2022010319482435900_i1944-7558-127-1-1-Sansone1","doi-asserted-by":"crossref","unstructured":"Sansone,\n              S.,\n            \n            \n              Widaman,\n              K.,\n            \n            \n              Hall,\n              S.,\n            \n            \n              Reiss,\n              A.,\n            \n            \n              Lightbody,\n              A.,\n            \n            \n              Kaufmann,\n              W.,\n            \n            \n              Berry-Kravis,\n              E.,\n            \n            \n              Lachiewicz,\n              A.,\n            \n            \n              Brown,\n              E.,\n             &\n\t\t\t\t\t\tHessl,D.\n          (2012).\n\t\t\t\t\tPsychometric study of the Aberrant Behavior Checklist in Fragile X Syndrome and implications for targeted treatment.\n\t\t\t\t\tJournal of Autism and Developmental Disorders, 42(7),\n\t\t\t\t\t1377\u2013\n\t\t\t\t\t1392.\n\t\t\t\t\thttps:\/\/doi.org\/10.1007\/s10803-011-1370-2","DOI":"10.1007\/s10803-011-1370-2"},{"key":"2022010319482435900_i1944-7558-127-1-1-Smith1","doi-asserted-by":"crossref","unstructured":"Smith,\n              K. J.,\n            \n            \n              Peterson,\n              M. D.,\n            \n            \n              O'Connell,\n              N. E.,\n            \n            \n              Victor,\n              C.,\n            \n            \n              Liverani,\n              S.,\n            \n            \n              Anokye,\n              N.,\n             &\n\t\t\t\t\t\tRyan,J. M.\n          (2019).\n\t\t\t\t\tRisk of depression and anxiety in adults with cerebral palsy.\n\t\t\t\t\tJAMA Neurology, 76(3),\n\t\t\t\t\t294\u2013\n\t\t\t\t\t300.\n\t\t\t\t\thttps:\/\/doi.org\/10.1001\/jamaneurol.2018.4147","DOI":"10.1001\/jamaneurol.2018.4147"},{"key":"2022010319482435900_i1944-7558-127-1-1-Sparrow1","doi-asserted-by":"crossref","unstructured":"Sparrow,\n              S.,\n            \n            \n              Cicchetti,\n              D.,\n             &\n\t\t\t\t\t\tBalla,D.\n          (2005).\n\t\t\t\t\tVineland Adaptive Behavior Scales, 2nd Edition.\n\t\t\t\t\tPearson Assessments.","DOI":"10.1037\/t15164-000"},{"key":"2022010319482435900_i1944-7558-127-1-1-Tan1","doi-asserted-by":"crossref","unstructured":"Tan,\n              W.,\n             &\n\t\t\t\t\t\tBird,L.\n          (2016).\n\t\t\t\t\tAngelman syndrome: Current and emerging therapies in 2016.\n\t\t\t\t\tAmerican Journal Of Medical Genetics Part C-Seminars In Medical Genetics, 172(4),\n\t\t\t\t\t384\u2013\n\t\t\t\t\t401.\n\t\t\t\t\thttps:\/\/doi.org\/10.1002\/ajmg.c.31536","DOI":"10.1002\/ajmg.c.31536"},{"key":"2022010319482435900_i1944-7558-127-1-1-vanLieshout1","doi-asserted-by":"crossref","unstructured":"van Lieshout,\n              C. F.,\n            \n            \n              de Meyer,\n              R. E.,\n            \n            \n              Curfs,\n              L. M.,\n            \n            \n              Koot,\n              H. M.,\n             &\n\t\t\t\t\t\tFryns,J. P.\n          (1998).\n\t\t\t\t\tProblem behaviors and personality of children and adolescents with Prader-Willi syndrome.\n\t\t\t\t\tJournal of Pediatric Psychology, 23(2),\n\t\t\t\t\t111\u2013\n\t\t\t\t\t120.\n\t\t\t\t\thttps:\/\/doi.org\/10.1093\/jpepsy\/23.2.111","DOI":"10.1093\/jpepsy\/23.2.111"},{"key":"2022010319482435900_i1944-7558-127-1-1-vanSteensel1","doi-asserted-by":"crossref","unstructured":"van Steensel,\n              F. J. A.,\n             &\n\t\t\t\t\t\tHeeman,E. J.\n          (2017).\n\t\t\t\t\tAnxiety levels in children with autism spectrum disorder: A meta-analysis.\n\t\t\t\t\tJournal of Child and Family Studies, 26(7),\n\t\t\t\t\t1753\u2013\n\t\t\t\t\t1767.\n\t\t\t\t\thttps:\/\/doi.org\/10.1007\/s10826-017-0687-7","DOI":"10.1007\/s10826-017-0687-7"},{"key":"2022010319482435900_i1944-7558-127-1-1-Wheeler1","doi-asserted-by":"crossref","unstructured":"Wheeler,\n              A. C.,\n            \n            \n              Okoniewski,\n              K. C.,\n            \n            \n              Wylie,\n              A.,\n            \n            \n              Deramus,\n              M.,\n            \n            \n              Hiruma,\n              L. S.,\n            \n            \n              Toth,\n              D.,\n             &\n\t\t\t\t\t\tChristian,R. B.\n          (2019).\n\t\t\t\t\tAnxiety-associated and separation distress-associated behaviours in Angelman syndrome.\n\t\t\t\t\tJournal of Intellectual Disability Research, 63(10),\n\t\t\t\t\t1234\u2013\n\t\t\t\t\t1247.\n\t\t\t\t\thttps:\/\/doi.org\/10.1111\/jir.12635","DOI":"10.1111\/jir.12635"},{"key":"2022010319482435900_i1944-7558-127-1-1-Wheeler2","doi-asserted-by":"crossref","unstructured":"Wheeler,\n              A. C.,\n            \n            \n              Sacco,\n              P.,\n             &\n\t\t\t\t\t\tCabo,R.\n          (2017).\n\t\t\t\t\tUnmet clinical needs and burden in Angelman syndrome: a review of the literature.\n\t\t\t\t\tOrphanet Journal Of Rare Diseases, 12(1).https:\/\/doi.org\/10.1186\/s13023-017-0716-z","DOI":"10.1186\/s13023-017-0716-z"},{"key":"2022010319482435900_i1944-7558-127-1-1-White1","doi-asserted-by":"crossref","unstructured":"White,\n              S. W.,\n            \n            \n              Oswald,\n              D.,\n            \n            \n              Ollendick,\n              T.,\n             &\n\t\t\t\t\t\tScahill,L.\n          (2009).\n\t\t\t\t\tAnxiety in children and adolescents with autism spectrum disorders.\n\t\t\t\t\tClinical Psychology Review, 29(3),\n\t\t\t\t\t216\u2013\n\t\t\t\t\t229.\n\t\t\t\t\thttps:\/\/doi.org\/10.1016\/j.cpr.2009.01.003","DOI":"10.1016\/j.cpr.2009.01.003"},{"key":"2022010319482435900_i1944-7558-127-1-1-Williams1","doi-asserted-by":"crossref","unstructured":"Williams,\n              C. A.,\n            \n            \n              Beaudet,\n              A. L.,\n            \n            \n              Clayton-Smith,\n              J.,\n            \n            \n              Knoll,\n              J. H.,\n            \n            \n              Kyllerman,\n              M.,\n            \n            \n              Laan,\n              L. A.,\n            \n            \n              Magenis,\n              R. E.,\n            \n            \n              Moncla,\n              A.,\n            \n            \n              Schinzel,\n              A. A.,\n            \n            \n              Summers,\n              J. A.,\n             &\n\t\t\t\t\t\tWagstaff,J.\n          (2006).\n\t\t\t\t\tAngelman syndrome 2005: Updated consensus for diagnostic criteria.\n\t\t\t\t\tAmerican Journal of Medical Genetics Part A, 140(5),\n\t\t\t\t\t413\u2013\n\t\t\t\t\t418.\n\t\t\t\t\thttps:\/\/doi.org\/10.1002\/ajmg.a.31074","DOI":"10.1002\/ajmg.a.31074"},{"key":"2022010319482435900_i1944-7558-127-1-1-Wink1","doi-asserted-by":"crossref","unstructured":"Wink,\n              L. K.,\n            \n            \n              Fitzpatrick,\n              S.,\n            \n            \n              Shaffer,\n              R.,\n            \n            \n              Melnyk,\n              S.,\n            \n            \n              Begtrup,\n              A. H.,\n            \n            \n              Fox,\n              E.,\n            \n            \n              Schaefer,\n              T. L.,\n            \n            \n              Mathieu-Frasier,\n              L.,\n            \n            \n              Ray,\n              B.,\n            \n            \n              Lahiri,\n              D.,\n            \n            \n              Horn,\n              P. A.,\n             &\n\t\t\t\t\t\tErickson,C. A.\n          (2015).\n\t\t\t\t\tThe neurobehavioral and molecular phenotype of Angelman Syndrome.\n\t\t\t\t\tAmerican Journal of Medical Genetics Part A, 167(11),\n\t\t\t\t\t2623\u2013\n\t\t\t\t\t2628.\n\t\t\t\t\thttps:\/\/doi.org\/10.1002\/ajmg.a.37254","DOI":"10.1002\/ajmg.a.37254"},{"key":"2022010319482435900_i1944-7558-127-1-1-Woodcock1","doi-asserted-by":"crossref","unstructured":"Woodcock,\n              K.,\n            \n            \n              Oliver,\n              C.,\n             &\n\t\t\t\t\t\tHumphreys,G.\n          (2009).\n\t\t\t\t\tAssociations between repetitive questioning, resistance to change, temper outbursts and anxiety in Prader\u2013Willi and Fragile-X syndromes.\n\t\t\t\t\tJournal of Intellectual Disability Research, 53(3),\n\t\t\t\t\t265\u2013\n\t\t\t\t\t278.\n\t\t\t\t\thttps:\/\/doi.org\/10.1111\/j.1365-2788.2008.01122.x","DOI":"10.1111\/j.1365-2788.2008.01122.x"}],"container-title":["American Journal on Intellectual and Developmental Disabilities"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/meridian.allenpress.com\/ajidd\/article-pdf\/127\/1\/1\/2995746\/i1944-7558-127-1-1.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/meridian.allenpress.com\/ajidd\/article-pdf\/127\/1\/1\/2995746\/i1944-7558-127-1-1.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,1,13]],"date-time":"2026-01-13T16:34:56Z","timestamp":1768322096000},"score":1,"resource":{"primary":{"URL":"https:\/\/meridian.allenpress.com\/ajidd\/article\/127\/1\/1\/475762\/Anxiety-in-Angelman-Syndrome"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,1,1]]},"references-count":43,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2022,1,3]]},"published-print":{"date-parts":[[2022,1,1]]}},"URL":"https:\/\/doi.org\/10.1352\/1944-7558-127.1.1","relation":{},"ISSN":["1944-7558","1944-7515"],"issn-type":[{"value":"1944-7558","type":"electronic"},{"value":"1944-7515","type":"print"}],"subject":[],"published":{"date-parts":[[2022,1,1]]}}}