{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,9]],"date-time":"2026-10-09T08:26:28Z","timestamp":1791534388795,"version":"4.3.4"},"reference-count":47,"publisher":"Oxford University Press (OUP)","issue":"1","license":[{"start":{"date-parts":[[2020,12,14]],"date-time":"2020-12-14T00:00:00Z","timestamp":1607904000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/journals\/pages\/open_access\/funder_policies\/chorus\/standard_publication_model"}],"funder":[{"name":"Clinical and Translational Intramural"},{"DOI":"10.13039\/100011500","name":"Research Institute at Nationwide Children\u2019s Hospital","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100011500","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100003690","name":"Cleft Palate Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100003690","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"NIH","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2021,1,20]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Objective<\/jats:title><jats:p>\u2003To assess health-related quality of life (HRQoL), psychosocial adjustment, and family functioning of children with differences of sex development (DSD) or cleft lip and\/or palate (CL\/P).<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>\u2003In this cross-sectional study, parents of children with DSD (n\u2009=\u200967), CL\/P (n\u2009=\u2009121), and a comparison group of unaffected youth (n\u2009=\u2009126) completed standardized measures assessing family functioning and their children\u2019s HRQoL and psychosocial adjustment. Medical charts were abstracted for youth with either congenital condition.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>\u2003Children with DSD were rated as having significantly lower HRQoL and greater internalizing problems compared to youth with CL\/P and unaffected youth. Children in the DSD group were also significantly more likely to fall into the clinical risk categories for total and internalizing problems relative to the CL\/P and unaffected groups. Caregivers of children with DSD were significantly more likely to endorse items about child suicidality compared with caregivers in the CL\/P and unaffected groups. No significant differences were found between groups for externalizing problems or the expressiveness domain of family functioning; parents of children with DSD reported significantly less family conflict relative to the other groups and greater cohesion relative to the unaffected group. Conclusions\u2003Youth with DSD appear to be at greater risk for psychosocial problems relative to children with CL\/P and unaffected peers. Results underscore the need for integrated interdisciplinary care and ongoing psychosocial risk monitoring in youth with DSD.<\/jats:p><\/jats:sec>","DOI":"10.1093\/jpepsy\/jsaa089","type":"journal-article","created":{"date-parts":[[2020,9,13]],"date-time":"2020-09-13T11:07:29Z","timestamp":1599995249000},"page":"69-79","source":"Crossref","is-referenced-by-count":16,"title":["Psychosocial Functioning Among Children With and Without Differences of Sex Development"],"prefix":"10.1093","volume":"46","author":[{"given":"Jennifer A","family":"Hansen-Moore","sequence":"first","affiliation":[{"name":"Nationwide Children\u2019s Hospital"},{"name":"Department of Pediatrics, The Ohio State University College of Medicine"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hillary M","family":"Kapa","sequence":"additional","affiliation":[{"name":"Center for Biobehavioral Health, The Abigail Wexner Research Institute at Nationwide Children\u2019s Hospital"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jennifer L","family":"Litteral","sequence":"additional","affiliation":[{"name":"Center for Biobehavioral Health, The Abigail Wexner Research Institute at Nationwide Children\u2019s Hospital"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Leena","family":"Nahata","sequence":"additional","affiliation":[{"name":"Nationwide Children\u2019s Hospital"},{"name":"Department of Pediatrics, The Ohio State University College of Medicine"},{"name":"Center for Biobehavioral Health, The Abigail Wexner Research Institute at Nationwide Children\u2019s Hospital"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Justin A","family":"Indyk","sequence":"additional","affiliation":[{"name":"Nationwide Children\u2019s Hospital"},{"name":"Department of Pediatrics, The Ohio State University College of Medicine"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Venkata R","family":"Jayanthi","sequence":"additional","affiliation":[{"name":"Nationwide Children\u2019s Hospital"},{"name":"Department of Urology, The Ohio State University College of Medicine"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yee-Ming","family":"Chan","sequence":"additional","affiliation":[{"name":"Division of Endocrinology, Department of Pediatrics, Boston Children\u2019s Hospital"},{"name":"Department of Pediatrics, Harvard Medical School"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1467-1732","authenticated-orcid":false,"given":"Amy C","family":"Tishelman","sequence":"additional","affiliation":[{"name":"Division of Endocrinology, Department of Pediatrics, Boston Children\u2019s Hospital"},{"name":"Department of Psychiatry, Harvard Medical School"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Canice E","family":"Crerand","sequence":"additional","affiliation":[{"name":"Nationwide Children\u2019s Hospital"},{"name":"Department of Pediatrics, The Ohio State University College of Medicine"},{"name":"Center for Biobehavioral Health, The Abigail Wexner Research Institute at Nationwide Children\u2019s Hospital"},{"name":"Department of Plastic Surgery, The Ohio State University College of Medicine"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2020,12,14]]},"reference":[{"key":"2022110403593006900_jsaa089-B1","volume-title":"Manual for the ASEBA school-age forms & profiles","author":"Achenbach","year":"2001"},{"key":"2022110403593006900_jsaa089-B2","doi-asserted-by":"crossref","first-page":"137","DOI":"10.1177\/1055665617739564","article-title":"Parameters for evaluation and treatment of patients with cleft lip\/palate or other craniofacial anomalies","volume":"55","year":"2018","journal-title":"The Cleft Palate-Craniofacial Journal"},{"key":"2022110403593006900_jsaa089-B3","doi-asserted-by":"crossref","first-page":"634","DOI":"10.1111\/cen.13296","article-title":"Health-related quality of life and psychological well-being in adults with differences\/disorders of sex development","volume":"86","author":"Bennecke","year":"2017","journal-title":"Clinical Endocrinology"},{"key":"2022110403593006900_jsaa089-B4","doi-asserted-by":"crossref","first-page":"369","DOI":"10.1111\/j.1545-5300.1997.00369.x","article-title":"The Family Environment Scale: Reliability and normative data for an adolescent sample","volume":"36","author":"Boyd","year":"1997","journal-title":"Family Process"},{"key":"2022110403593006900_jsaa089-B5","doi-asserted-by":"crossref","first-page":"339","DOI":"10.1007\/s11136-013-0450-5","article-title":"Evaluation of the similarities and differences in response patterns to the pediatric quality of life inventory and the child oral health impact scores among youth with cleft","volume":"23","author":"Broder","year":"2014","journal-title":"Quality of Life Research"},{"key":"2022110403593006900_jsaa089-B6","doi-asserted-by":"crossref","first-page":"828","DOI":"10.2105\/AJPH.2012.300654","article-title":"Health disparities among children with cleft","volume":"102","author":"Broder","year":"2012","journal-title":"American Journal of Public Health"},{"issue":"1","key":"2022110403593006900_jsaa089-B7","first-page":"1","article-title":"Update on overall prevalence of major birth defects\u2014Atlanta, Georgia, 1978\u20132005","volume":"57","year":"2008","journal-title":"Morbidity and Mortality Weekly Report"},{"key":"2022110403593006900_jsaa089-B8","doi-asserted-by":"crossref","first-page":"75","DOI":"10.1080\/01973533.2012.746612","article-title":"Discredited\u201d versus \u201cdiscreditable\u201d: Understanding how shared and unique stigma mechanisms affect psychological and physical health disparities","volume":"35","author":"Chaudoir","year":"2013","journal-title":"Basic and Applied Social Psychology"},{"key":"2022110403593006900_jsaa089-B9","doi-asserted-by":"crossref","first-page":"397","DOI":"10.1597\/10-007","article-title":"Psychosocial functioning in children with and without orofacial clefts and their parents","volume":"49","author":"Collett","year":"2012","journal-title":"The Cleft Palate-Craniofacial Journal"},{"key":"2022110403593006900_jsaa089-B10","doi-asserted-by":"crossref","first-page":"415","DOI":"10.1038\/s41574-018-0010-8","article-title":"Caring for individuals with a difference of sex development (DSD): A consensus statement","volume":"14","author":"Cools","year":"2018","journal-title":"Nature Reviews Endocrinology"},{"key":"2022110403593006900_jsaa089-B11","doi-asserted-by":"crossref","first-page":"536","DOI":"10.1177\/1055665617748010","article-title":"Identifying psychosocial risk factors among families of children with craniofacial conditions: Validation of the psychosocial assessment tool-craniofacial version","volume":"55","author":"Crerand","year":"2018","journal-title":"The Cleft Palate-Craniofacial Journal"},{"key":"2022110403593006900_jsaa089-B12","doi-asserted-by":"crossref","first-page":"651","DOI":"10.1597\/14-050","article-title":"Parent-reported family functioning among children with cleft lip\/palate","volume":"52","author":"Crerand","year":"2015","journal-title":"The Cleft Palate-Craniofacial Journal"},{"key":"2022110403593006900_jsaa089-B13","doi-asserted-by":"crossref","first-page":"2","DOI":"10.1597\/15-167","article-title":"Body image and quality of life in adolescents with craniofacial conditions","volume":"54","author":"Crerand","year":"2017","journal-title":"The Cleft Palate-Craniofacial Journal"},{"key":"2022110403593006900_jsaa089-B14","doi-asserted-by":"crossref","first-page":"e1920956","DOI":"10.1001\/jamanetworkopen.2019.20956","article-title":"Prevalence and family-related factors associated with suicidal ideation, suicide attempts, and self-injury in children aged 9 to 10 years","volume":"3","author":"DeVille","year":"2020","journal-title":"JAMA Network Open"},{"key":"2022110403593006900_jsaa089-B15","doi-asserted-by":"crossref","first-page":"629","DOI":"10.1097\/PSY.0000000000000718","article-title":"Mental health of a large group of adults with disorders of sex development in six European countries","volume":"81","author":"de Vries","year":"2019","journal-title":"Psychosomatic Medicine"},{"key":"2022110403593006900_jsaa089-B16","doi-asserted-by":"crossref","first-page":"380","DOI":"10.1002\/bdrc.21144","article-title":"Sociocultural aspects of disorders of sex development","volume":"108","author":"Ediati","year":"2016","journal-title":"Birth Defects Research Part C, Embryo Today: Reviews"},{"key":"2022110403593006900_jsaa089-B17","doi-asserted-by":"crossref","first-page":"368","DOI":"10.1159\/000496114","article-title":"Psychosocial screening in disorders\/differences of sex development: Psychometric evaluation of the psychosocial assessment tool","volume":"90","author":"Ernst","year":"2018","journal-title":"Hormone Research in Paediatrics"},{"key":"2022110403593006900_jsaa089-B18","doi-asserted-by":"crossref","first-page":"pii: e20174045","DOI":"10.1542\/peds.2017-4045","article-title":"Disorders of sex development\/intersex: Gaps in psychosocial care for children","volume":"142","author":"Ernst","year":"2018","journal-title":"Pediatrics"},{"key":"2022110403593006900_jsaa089-B19","doi-asserted-by":"crossref","first-page":"5","DOI":"10.1597\/12-328","article-title":"When there is more than a cleft: Psychological adjustment when a cleft is associated with an additional condition","volume":"51","author":"Feragen","year":"2014","journal-title":"The Cleft Palate-Craniofacial Journal"},{"key":"2022110403593006900_jsaa089-B20","doi-asserted-by":"crossref","first-page":"377","DOI":"10.1016\/j.jbi.2008.08.010","article-title":"Research Electronic Data Capture (REDCap)\u2014A metadata-driven methodology and workflow process for providing translational research informatics support","volume":"42","author":"Harris","year":"2009","journal-title":"Journal of Biomedical Informatics"},{"key":"2022110403593006900_jsaa089-B21","doi-asserted-by":"crossref","first-page":"26","DOI":"10.1097\/DBP.0b013e3181c7226b","article-title":"Family functioning in the context of pediatric chronic conditions","volume":"31","author":"Herzer","year":"2010","journal-title":"Journal of Developmental and Behavioral Pediatrics"},{"key":"2022110403593006900_jsaa089-B22","doi-asserted-by":"publisher","first-page":"306","DOI":"10.1016\/S1474-4422(13)70302-8","article-title":"Cognitive and neurological aspects of sex chromosome aneuploidies","volume":"13","author":"Hong","year":"2014","journal-title":"The Lancet Neurology"},{"key":"2022110403593006900_jsaa089-B23","doi-asserted-by":"crossref","first-page":"148","DOI":"10.1016\/j.jpurol.2006.03.004","article-title":"Consensus statement on management of intersex disorders","volume":"2","author":"Hughes","year":"2006","journal-title":"Journal of Pediatric Urology"},{"key":"2022110403593006900_jsaa089-B24","doi-asserted-by":"crossref","first-page":"317","DOI":"10.1016\/S2215-0366(20)30049-3","article-title":"Risk and protective factors for childhood suicidality: A US population-based study","volume":"7","author":"Janiri","year":"2020","journal-title":"The Lancet Psychiatry"},{"key":"2022110403593006900_jsaa089-B25","doi-asserted-by":"crossref","first-page":"pii: e20181771","DOI":"10.1542\/peds.2018-1771","article-title":"Parent-adolescent agreement about adolescents\u2019 suicidal thoughts","volume":"143","author":"Jones","year":"2019","journal-title":"Pediatrics"},{"key":"2022110403593006900_jsaa089-B26","doi-asserted-by":"crossref","first-page":"893","DOI":"10.1007\/s00431-014-2264-z","article-title":"Health-related quality of life in children with disorders of sex development (DSD)","volume":"173","author":"J\u00fcrgensen","year":"2014","journal-title":"European Journal of Pediatrics"},{"key":"2022110403593006900_jsaa089-B27","doi-asserted-by":"crossref","first-page":"463","DOI":"10.1016\/j.jadohealth.2010.03.007","article-title":"Psychological adjustment and sexual development of adolescents with disorders of sex development","volume":"47","author":"Kleinemeier","year":"2010","journal-title":"Journal of Adolescent Health"},{"key":"2022110403593006900_jsaa089-B28","doi-asserted-by":"crossref","first-page":"199","DOI":"10.1111\/j.1545-5300.1990.00199.x","article-title":"Conceptual and empirical approaches to developing family-based assessment procedures: Resolving the case of the Family Environment Scale","volume":"29","author":"Moos","year":"1990","journal-title":"Family Process"},{"key":"2022110403593006900_jsaa089-B29","volume-title":"Family Environment Scale manual","author":"Moos","year":"2002","edition":"3rd ed."},{"key":"2022110403593006900_jsaa089-B30","doi-asserted-by":"crossref","first-page":"6","DOI":"10.1111\/j.1365-2214.2011.01244.x","article-title":"Parents\u2019 experiences of caring for a child with a cleft lip and\/or palate: A review of the literature","volume":"38","author":"Nelson","year":"2012","journal-title":"Child: Care, Health and Development"},{"key":"2022110403593006900_jsaa089-B31","doi-asserted-by":"crossref","first-page":"42","DOI":"10.1111\/cch.12276","article-title":"Psychosocial well-being of parents of children with oral clefts","volume":"42","author":"Nidey","year":"2016","journal-title":"Child: Care, Health and Development"},{"key":"2022110403593006900_jsaa089-B32","doi-asserted-by":"publisher","first-page":"143","DOI":"10.1515\/jpem.2009.22.2.143","article-title":"Variables related to behavioral and emotional problems and gender typed behaviors in female patients with congenital adrenal hyperplasia","volume":"22","author":"Oner","year":"2009","journal-title":"Journal of Pediatric Endocrinology & Metabolism"},{"key":"2022110403593006900_jsaa089-B33","doi-asserted-by":"crossref","first-page":"1008","DOI":"10.1002\/bdra.20735","article-title":"Updated national birth prevalence estimates for selected birth defects in the United States, 2004\u20132006","volume":"88","author":"Parker","year":"2010","journal-title":"Birth Defects Research Part A: Clinical and Molecular Teratology"},{"key":"2022110403593006900_jsaa089-B34","doi-asserted-by":"crossref","first-page":"556","DOI":"10.1597\/04-066r.1","article-title":"Parenting stress in infancy and psychosocial adjustment in toddlerhood: A longitudinal study of children with craniofacial anomalies","volume":"42","author":"Pope","year":"2005","journal-title":"The Cleft Palate-Craniofacial Journal"},{"key":"2022110403593006900_jsaa089-B35","doi-asserted-by":"crossref","first-page":"203","DOI":"10.1111\/j.1545-5300.1987.00203.x","article-title":"Chronic illness and the life cycle: A conceptual framework","volume":"26","author":"Rolland","year":"1987","journal-title":"Family Process"},{"key":"2022110403593006900_jsaa089-B36","volume-title":"The volunteer subject","author":"Rosenthal","year":"1975"},{"key":"2022110403593006900_jsaa089-B37","doi-asserted-by":"crossref","first-page":"279","DOI":"10.1002\/ajmg.c.31561","article-title":"Interdisciplinary care in disorders\/differences of sex development (DSD): The psychosocial component of the DSD-translational research network","volume":"175","author":"Sandberg","year":"2017","journal-title":"American Journal of Medical Genetics Part C: Seminars in Medical Genetics"},{"key":"2022110403593006900_jsaa089-B38","doi-asserted-by":"crossref","first-page":"443","DOI":"10.1055\/s-0032-1324729","article-title":"Psychological aspects of the treatment of patients with disorders of sex development","volume":"30","author":"Sandberg","year":"2012","journal-title":"Seminars in Reproductive Medicine"},{"key":"2022110403593006900_jsaa089-B39","doi-asserted-by":"crossref","first-page":"520","DOI":"10.1093\/jpepsy\/jsm098","article-title":"Psychosocial adjustment, health-related quality of life, and psychosexual development of boys with hypospadias: A systematic review","volume":"33","author":"Schonbucher","year":"2007","journal-title":"Journal of Pediatric Psychology"},{"key":"2022110403593006900_jsaa089-B40","doi-asserted-by":"crossref","first-page":"16","DOI":"10.1007\/s10508-007-9241-9","article-title":"Psychological distress, self-harming behavior, and suicidal tendencies in adults with disorders of sex development","volume":"38","author":"Sch\u00fctzmann","year":"2009","journal-title":"Archives of Sexual Behavior"},{"key":"2022110403593006900_jsaa089-B41","first-page":"504","article-title":"Coping with diverse sex development: Treatment experiences and psychosocial support during childhood and adolescence and adult well-being","volume":"42","author":"Schweizer","year":"2017","journal-title":"Journal of Pediatric Psychology"},{"key":"2022110403593006900_jsaa089-B42","first-page":"893","article-title":"Quality of life in children with disorders of sex development","volume":"173","author":"Selveindran","year":"2017","journal-title":"Hormone Research in Paediatrics"},{"key":"2022110403593006900_jsaa089-B43","doi-asserted-by":"crossref","first-page":"777","DOI":"10.1080\/08870446.2016.1143944","article-title":"Psychological adjustment to cleft lip and\/or palate: A narrative review of the literature","volume":"31","author":"Stock","year":"2016","journal-title":"Psychology & Health"},{"key":"2022110403593006900_jsaa089-B44","doi-asserted-by":"crossref","first-page":"800","DOI":"10.1097\/00005650-200108000-00006","article-title":"PedsQLTM 4.0: Reliability and validity of the Pediatric Quality of Life InventoryTM Version 4.0 Generic Core Scales in healthy and patient populations","volume":"39","author":"Varni","year":"2001","journal-title":"Medical Care"},{"key":"2022110403593006900_jsaa089-B45","doi-asserted-by":"crossref","first-page":"11","DOI":"10.1097\/MOU.0000000000000344","article-title":"Psychosocial implications of disorders of sex development treatment for parents","volume":"27","author":"Wisniewski","year":"2017","journal-title":"Current Opinion in Urology"},{"key":"2022110403593006900_jsaa089-B46","doi-asserted-by":"crossref","first-page":"185","DOI":"10.1597\/03-118.1","article-title":"Family environment of individuals with oral clefts in Argentina","volume":"42","author":"Wyszynski","year":"2005","journal-title":"The Cleft Palate-Craniofacial Journal"},{"key":"2022110403593006900_jsaa089-B47","doi-asserted-by":"crossref","first-page":"165","DOI":"10.1159\/000435855","article-title":"Health-related quality of life in children with congenital adrenal hyperplasia","volume":"84","author":"Yau","year":"2015","journal-title":"Hormone Research in Paediatrics"}],"container-title":["Journal of Pediatric Psychology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/jpepsy\/article-pdf\/46\/1\/69\/46783429\/jsaa089.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/jpepsy\/article-pdf\/46\/1\/69\/46783429\/jsaa089.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,10,7]],"date-time":"2023-10-07T07:46:42Z","timestamp":1696664802000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/jpepsy\/article\/46\/1\/69\/6032923"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2020,12,14]]},"references-count":47,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2020,12,14]]},"published-print":{"date-parts":[[2021,1,20]]}},"URL":"https:\/\/doi.org\/10.1093\/jpepsy\/jsaa089","relation":{},"ISSN":["0146-8693","1465-735X"],"issn-type":[{"value":"0146-8693","type":"print"},{"value":"1465-735X","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2021,1,1]]},"published":{"date-parts":[[2020,12,14]]}}}