{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,1]],"date-time":"2026-05-01T19:36:51Z","timestamp":1777664211813,"version":"3.51.4"},"reference-count":22,"publisher":"MDPI AG","issue":"4","license":[{"start":{"date-parts":[[2020,4,4]],"date-time":"2020-04-04T00:00:00Z","timestamp":1585958400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000030","name":"Centers for Disease Control and Prevention","doi-asserted-by":"publisher","award":["U01DD000231"],"award-info":[{"award-number":["U01DD000231"]}],"id":[{"id":"10.13039\/100000030","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000030","name":"Centers for Disease Control and Prevention","doi-asserted-by":"publisher","award":["U19DD000753"],"award-info":[{"award-number":["U19DD000753"]}],"id":[{"id":"10.13039\/100000030","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000030","name":"Centers for Disease Control and Prevention","doi-asserted-by":"publisher","award":["U01DD001189"],"award-info":[{"award-number":["U01DD001189"]}],"id":[{"id":"10.13039\/100000030","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["UL1 TR002535"],"award-info":[{"award-number":["UL1 TR002535"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Brain Sciences"],"abstract":"<jats:p>Health-related quality of life (HRQoL) is a multidimensional concept involving physical, psychological, social, and cognitive aspects of life. Individuals with Fragile X syndrome (FXS) experience a life-long disorder that impacts the HRQoL of the affected individual and their family. Thus, HRQoL may be an important outcome measure following intervention. However, it is yet not known whether HRQoL concerns relate to observed impairments in FXS. In the present study, we examined the nature and degree of association between HRQoL and established measures of functioning in FXS using the Parent Report for Children version of the PedsQL 4.0 Generic Core Scales and Cognitive Functioning Scale. We observed significant relationships between HRQoL a nd measures of adaptive behavior, maladaptive behaviors, and social functioning. The present study has implications for treatment outcomes for clinical trials in FXS.<\/jats:p>","DOI":"10.3390\/brainsci10040213","type":"journal-article","created":{"date-parts":[[2020,4,6]],"date-time":"2020-04-06T05:05:13Z","timestamp":1586149513000},"page":"213","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":14,"title":["Examination of Correlates to Health-Related Quality of Life in Individuals with Fragile X Syndrome"],"prefix":"10.3390","volume":"10","author":[{"given":"Marika C.","family":"Coffman","sequence":"first","affiliation":[{"name":"Division of Developmental and Behavioral Pediatrics, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati, OH 45229, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Rebecca C.","family":"Shaffer","sequence":"additional","affiliation":[{"name":"Division of Developmental and Behavioral Pediatrics, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati, OH 45229, USA"},{"name":"Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, OH 45267, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lauren M.","family":"Schmitt","sequence":"additional","affiliation":[{"name":"Division of Developmental and Behavioral Pediatrics, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati, OH 45229, USA"},{"name":"Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, OH 45267, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kelli C.","family":"Dominick","sequence":"additional","affiliation":[{"name":"Division of Child and Adolescent Psychiatry, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati, OH 45229, USA"},{"name":"Department of Psychiatry and Behavioral Neuroscience, University of Cincinnati College of Medicine, Cincinnati, OH 45267, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ernest","family":"Pedapati","sequence":"additional","affiliation":[{"name":"Division of Child and Adolescent Psychiatry, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati, OH 45229, USA"},{"name":"Department of Psychiatry and Behavioral Neuroscience, University of Cincinnati College of Medicine, Cincinnati, OH 45267, USA"},{"name":"Division of Child Neurology, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati, OH 45229, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Angel","family":"Wang","sequence":"additional","affiliation":[{"name":"Departments of Pediatrics, Rush University Medical Center, Chicago, IL 60612, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Elizabeth","family":"Berry-Kravis","sequence":"additional","affiliation":[{"name":"Departments of Pediatrics, Neurological Sciences, Biochemistry, Rush University Medical Center, Chicago, IL 60612, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nicole","family":"Tartaglia","sequence":"additional","affiliation":[{"name":"Developmental Pediatrics, Children\u2019s Hospital Colorado, Aurora, CO 80045, USA"},{"name":"Division of Developmental Pediatrics, Department of Pediatrics, University of Colorado School of Medicine, Aurora, CO 80045, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Craig A.","family":"Erickson","sequence":"additional","affiliation":[{"name":"Division of Child and Adolescent Psychiatry, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati, OH 45229, USA"},{"name":"Department of Psychiatry and Behavioral Neuroscience, University of Cincinnati College of Medicine, Cincinnati, OH 45267, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2020,4,4]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"42","DOI":"10.1002\/mrdd.20007","article-title":"Psychopharmacology in fragile X syndrome?Present and future","volume":"10","author":"Potanos","year":"2004","journal-title":"Ment. Retard. Dev. Disabil. Res. Rev."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"1056","DOI":"10.1007\/s10803-019-04292-7","article-title":"Pediatric Quality of Life Inventory (PedsQL) in Fragile X Syndrome","volume":"50","author":"Fitzpatrick","year":"2019","journal-title":"J. Autism Dev. Disord."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"699","DOI":"10.1111\/j.1365-2788.2005.00738.x","article-title":"Moving from the quality of life concept to a theory","volume":"49","author":"Cummins","year":"2005","journal-title":"J. Intellect. Disabil. Res."},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"457","DOI":"10.1352\/0047-6765(2002)040<0457:CMAAOQ>2.0.CO;2","article-title":"Conceptualization, Measurement, and Application of Quality of Life for Persons with Intellectual Disabilities: Report of an International Panel of Experts","volume":"40","author":"Schalock","year":"2002","journal-title":"Ment. Retard."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"800","DOI":"10.1097\/00005650-200108000-00006","article-title":"PedsQL\u2122 4.0: Reliability and Validity of the Pediatric Quality of Life Inventory\u2122 Version 4.0 Generic Core Scales in Healthy and Patient Populations","volume":"39","author":"Varni","year":"2001","journal-title":"Med Care"},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"126","DOI":"10.1097\/00005650-199902000-00003","article-title":"The PedsQL\u2122: Measurement Model for the Pediatric Quality of Life Inventory","volume":"37","author":"Varni","year":"1999","journal-title":"Med Care"},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"714","DOI":"10.1002\/art.10095","article-title":"The PedsQL in pediatric rheumatology: Reliability, validity, and responsiveness of the Pediatric Quality of Life Inventory Generic Core Scales and Rheumatology Module","volume":"46","author":"Varni","year":"2002","journal-title":"Arthritis Rheum."},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"1599","DOI":"10.1007\/s11136-004-7711-2","article-title":"Determinants of quality of life in children with psychiatric disorders","volume":"14","author":"Bastiaansen","year":"2005","journal-title":"Qual. Life Res."},{"key":"ref_9","first-page":"641","article-title":"The relationship of health-related quality of life and treatment outcome during inpatient treatment of depression","volume":"24","author":"Unger","year":"2014","journal-title":"Qual. Life Res."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"183","DOI":"10.1159\/000091776","article-title":"Quality of Life and Treatment Outcome in Panic Disorder: Cognitive Behavior Group Therapy Effects in Patients Refractory to Medication Treatment","volume":"75","author":"Heldt","year":"2006","journal-title":"Psychother. Psychosom."},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"2060","DOI":"10.1002\/ajmg.a.32439","article-title":"Co-occurring conditions associated withFMR1gene variations: Findings from a national parent survey","volume":"146","author":"Bailey","year":"2008","journal-title":"Am. J. Med Genet. Part A"},{"key":"ref_12","unstructured":"Sparrow, S.S., Cicchetti, D.V., and Saulnier, C.A. (2016). Vineland adaptive behavior scales, (Vineland-3), Psychological Corporation."},{"key":"ref_13","unstructured":"Constantino, J., and Gruber, C. (2012). Social Responsiveness Scale, Western Psychological Services. [2nd ed.]."},{"key":"ref_14","first-page":"485","article-title":"The aberrant behavior checklist: A behavior rating scale for the assessment of treatment effects","volume":"89","author":"Aman","year":"1985","journal-title":"Am. J. Ment. Defic."},{"key":"ref_15","doi-asserted-by":"crossref","first-page":"1143","DOI":"10.1097\/CHI.0b013e3181bfd669","article-title":"Medication and parent training in children with pervasive developmental disorders and serious behavior problems: Results from a randomized clinical trial","volume":"48","author":"Aman","year":"2009","journal-title":"J. Am. Acad. Child Adolesc. Psychiatry"},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"1377","DOI":"10.1007\/s10803-011-1370-2","article-title":"Psychometric Study of the Aberrant Behavior Checklist in Fragile X Syndrome and Implications for Targeted Treatment","volume":"42","author":"Sansone","year":"2011","journal-title":"J. Autism Dev. Disord."},{"key":"ref_17","unstructured":"Allaire, J. (2020, April 03). RStudio: Integrated Development Environment for R. Available online: https:\/\/support.rstudio.com\/hc\/en-us\/articles\/206212048-Citing-RStudio."},{"key":"ref_18","doi-asserted-by":"crossref","first-page":"551","DOI":"10.1037\/a0021268","article-title":"Trajectories of emotional well-being in mothers of adolescents and adults with autism","volume":"47","author":"Barker","year":"2011","journal-title":"Dev. Psychol."},{"key":"ref_19","first-page":"2","article-title":"Maternal burden in families with children with autistic spectrum disorder","volume":"33","author":"Fitzgerald","year":"2002","journal-title":"Irish J. Psychol. Med."},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"215","DOI":"10.1080\/1366825021000008639","article-title":"Ten years on: A longitudinal study of families of children with autism","volume":"27","author":"Gray","year":"2002","journal-title":"J. Intellect. Dev. Disabil."},{"key":"ref_21","doi-asserted-by":"crossref","first-page":"401","DOI":"10.1352\/0895-8017(2007)112[401:TACIAA]2.0.CO;2","article-title":"Transition and change in adolescents and young adults with autism: Longitudinal effects on maternal well-being","volume":"112","author":"Lounds","year":"2007","journal-title":"Am. J. Ment. Retard."},{"key":"ref_22","doi-asserted-by":"crossref","first-page":"113","DOI":"10.1111\/jir.12238","article-title":"Aggression in fragile X syndrome","volume":"60","author":"Wheeler","year":"2015","journal-title":"J. Intellect. Disabil. 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