{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"institution":[{"name":"medRxiv"}],"indexed":{"date-parts":[[2026,5,15]],"date-time":"2026-05-15T16:08:09Z","timestamp":1778861289910,"version":"3.51.4"},"posted":{"date-parts":[[2026,5,13]]},"group-title":"Psychiatry and Clinical Psychology","reference-count":48,"publisher":"openRxiv","license":[{"start":{"date-parts":[[2026,5,13]],"date-time":"2026-05-13T00:00:00Z","timestamp":1778630400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"accepted":{"date-parts":[[2026,5,13]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                <jats:sec>\n                  <jats:title>Background<\/jats:title>\n                  <jats:p>A range of rare chromosomal micro-deletions or -duplications (Copy Number Variants - CNVs) are associated with high risk of neurodevelopmental and mental health conditions (ND-CNVs). There is great individual variability in outcomes, but we lack insights into the contributing social factors, including family functioning.<\/jats:p>\n                <\/jats:sec>\n                <jats:sec>\n                  <jats:title>Methods<\/jats:title>\n                  <jats:p>Caregivers of 598 children and young people (CYP) with a range of 16 ND-CNVs and 222 siblings without ND-CNVs (controls) completed questionnaires on overall family climate (cohesion and conflict) as well as caregiver-CYP relationship warmth and hostility and took part in a research diagnostic interview about CYPs\u2019 psychiatric symptoms. CYPs\u2019 intelligence quotient (IQ) was also measured.<\/jats:p>\n                <\/jats:sec>\n                <jats:sec>\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>Comparisons with published data from neurotypical families indicated that families affected by ND-CNVs are characterised by higher family cohesion and conflict as well as lower caregiver-CYP warmth and hostility. Symptoms of oppositional defiant disorder reduced more steeply in CYP with ND-CNVs compared to controls with increasing family cohesion (interaction effect: \u03b2 = -0.14, p = 4.65 \u00d7 10\u207b\u00b2). In contrast, they rose more steeply with increasing family conflict (interaction effect: \u03b2 = 0.18, p = 1.05 \u00d7 10\u207b\u00b2). Furthermore, symptoms of mood disorder increased more steeply with increased caregiver-CYP hostility in CYP with ND-CNVs (interaction effect: \u03b2 = 0.15, p = 4.55 \u00d7 10\u207b\u00b2).<\/jats:p>\n                <\/jats:sec>\n                <jats:sec>\n                  <jats:title>Conclusions<\/jats:title>\n                  <jats:p>Raising a CYP with a rare genetic condition is challenging. Timely access to interventions that support caregivers in fostering a positive family environment may reduce behavioural difficulties in CYP, with subsequent benefits for family functioning.<\/jats:p>\n                <\/jats:sec>","DOI":"10.64898\/2026.05.10.26352561","type":"posted-content","created":{"date-parts":[[2026,5,13]],"date-time":"2026-05-13T15:45:18Z","timestamp":1778687118000},"source":"Crossref","is-referenced-by-count":0,"title":["Family functioning and psychiatric outcomes in children and young people with intellectual and developmental disabilities caused by rare genetic mutations"],"prefix":"10.64898","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-5975-813X","authenticated-orcid":false,"given":"Josephine E","family":"Haddon","sequence":"first","affiliation":[{"name":"Division of Psychological Medicine and Clinical Neurosciences, Centre for Neuropsychiatric Genetics and Genomics, Cardiff University, Cardiff, UK"},{"name":"Neuroscience and Mental Health Innovation Institute, Cardiff University, Cardiff, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3433-4784","authenticated-orcid":false,"given":"Jessica H","family":"Hall","sequence":"additional","affiliation":[{"name":"Division of Psychological Medicine and Clinical Neurosciences, Centre for Neuropsychiatric Genetics and Genomics, Cardiff University, Cardiff, UK"},{"name":"Cardiff University Brain Research Imaging Centre (CUBRIC), School of Psychology, Cardiff University, Cardiff, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"name":"IMAGINE ID","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jeremy","family":"Hall","sequence":"additional","affiliation":[{"name":"Division of Psychological Medicine and Clinical Neurosciences, Centre for Neuropsychiatric Genetics and Genomics, Cardiff University, Cardiff, UK"},{"name":"Neuroscience and Mental Health Innovation Institute, Cardiff University, Cardiff, UK"},{"name":"Department of Psychiatry, Warneford Hospital, University of Oxford, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Michael J","family":"Owen","sequence":"additional","affiliation":[{"name":"Division of Psychological Medicine and Clinical Neurosciences, Centre for Neuropsychiatric Genetics and Genomics, Cardiff University, Cardiff, UK"},{"name":"Neuroscience and Mental Health Innovation Institute, Cardiff University, Cardiff, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4426-3254","authenticated-orcid":false,"given":"Marianne B M","family":"van den Bree","sequence":"additional","affiliation":[{"name":"Division of Psychological Medicine and Clinical Neurosciences, Centre for Neuropsychiatric Genetics and Genomics, Cardiff University, Cardiff, UK"},{"name":"Neuroscience and Mental Health Innovation Institute, Cardiff University, Cardiff, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"54368","reference":[{"key":"2026051508100930000_2026.05.10.26352561v1.1","doi-asserted-by":"publisher","DOI":"10.1111\/jir.12054"},{"key":"2026051508100930000_2026.05.10.26352561v1.2","doi-asserted-by":"publisher","DOI":"10.1097\/00004583-200001000-00015"},{"key":"2026051508100930000_2026.05.10.26352561v1.3","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpeds.2011.02.039"},{"key":"2026051508100930000_2026.05.10.26352561v1.4","doi-asserted-by":"publisher","DOI":"10.1177\/0004867412450076"},{"key":"2026051508100930000_2026.05.10.26352561v1.5","doi-asserted-by":"publisher","DOI":"10.1111\/j.2517-6161.1995.tb02031.x"},{"issue":"4","key":"2026051508100930000_2026.05.10.26352561v1.6","first-page":"1165","article-title":"The Control of the False Discovery Rate in Multiple Testing under Dependency","volume":"29","year":"2001","journal-title":"The Annals of Statistics"},{"key":"2026051508100930000_2026.05.10.26352561v1.7","doi-asserted-by":"publisher","DOI":"10.3389\/fpsyt.2020.00547"},{"key":"2026051508100930000_2026.05.10.26352561v1.8","doi-asserted-by":"publisher","DOI":"10.1186\/s40359-024-01609-9"},{"key":"2026051508100930000_2026.05.10.26352561v1.9","doi-asserted-by":"publisher","DOI":"10.1002\/aur.2297"},{"key":"2026051508100930000_2026.05.10.26352561v1.10","doi-asserted-by":"publisher","DOI":"10.1176\/appi.ajp.2020.20010015"},{"key":"2026051508100930000_2026.05.10.26352561v1.11","doi-asserted-by":"publisher","DOI":"10.1038\/s41398-022-02296-z"},{"key":"2026051508100930000_2026.05.10.26352561v1.12","doi-asserted-by":"publisher","DOI":"10.1016\/s2215-0366(19)30123-3"},{"key":"2026051508100930000_2026.05.10.26352561v1.13","doi-asserted-by":"publisher","DOI":"10.1111\/jomf.12836"},{"key":"2026051508100930000_2026.05.10.26352561v1.14","unstructured":"Conger R (1989) Behavioral affect rating scale (BARS): Young adult perception of parents\u2019 hostility and warmth: Iowa youth and families project.. 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