{"doi":"10.1101/2025.05.07.25327165","title":"Parental stress and family quality of life in families of individuals living with Angelman syndrome","abstract":"Background: Angelman syndrome (AS) is a developmental disorder caused by one of four molecular etiologies. Affected individuals have intellectual disability (ID), limited speech, seizures, and sleep problems. Parents of individuals with AS exhibit elevated stress compared to parents of individuals with other IDs. We examined parental stress and family quality of life (FQOL) over time in families of individuals living with AS. Methods: Data were collected in a natural history study of AS. The Parenting Stress Index, Third Edition (PSI) and the Beach Center FQOL scale assessed parent stress and FQOL. Stress and FQOL were examined across AS molecular subtypes, and predictors were analyzed using a generalised linear model. Relationships between parental stress and FQOL were examined using Pearson correlations and a stepwise mixed-linear model approach. Results: mutations, while FQOL did not differ across subtype in most domains. Increasing age predicted a decrease in parental stress but did not predict FQOL. Elevated parental stress was additionally predicted by maladaptive behaviours and child male sex, while lower FQOL was predicted by child male sex, parent marital status, and family income. Parental stress had a small negative impact on FQOL. Conclusions: Stress is elevated in parents of individuals with AS across subtypes and has a small negative impact on family quality of life. Interventions to reduce stress have potential to improve individual and family well-being.","journal":"medRxiv","year":2025,"id":565534,"datarank":0.0,"base_score":0.0,"endowment":0.0,"self_citation_contribution":0.0,"citation_network_contribution":0.0,"self_endowment_contribution":0.0,"citer_contribution":0.0,"corpus_percentile":null,"corpus_rank":null,"citation_count":0,"citer_count":0,"citers_with_citation_signal":0,"citers_with_endowment":0,"datacite_reuse_total":0,"is_dataset":false,"is_dataset_confidence":0.9545,"is_data_producer":false,"deposit_databanks":null,"is_oa":true,"file_count":0,"downloads":0,"has_version_chain":false,"published_date":"2025-01-01","fair_score":null,"fair_percentile":null,"algorithm_id":"datarank_citation_only_1hop_v6","ranking_scope":"data_only","authors":[{"id":655554,"name":"Angela Gwaltney","orcid":"0000-0002-2305-7187","position":1,"is_corresponding":false},{"id":852640,"name":"Anna Booman","orcid":"0000-0002-8112-6929","position":2,"is_corresponding":false},{"id":1025321,"name":"Sarah Nelson Potter","orcid":"0000-0002-8640-8992","position":3,"is_corresponding":false},{"id":535531,"name":"Anne Wheeler","orcid":"0000-0003-2693-5616","position":4,"is_corresponding":false},{"id":656235,"name":"Rene Barbieri‐Welge","orcid":null,"position":5,"is_corresponding":false},{"id":656236,"name":"Lucia T. Horowitz","orcid":null,"position":6,"is_corresponding":false},{"id":655555,"name":"Rachel J. Hundley","orcid":"0000-0001-5934-7832","position":7,"is_corresponding":false},{"id":249069,"name":"Lynne M. Bird","orcid":"0000-0003-4833-3747","position":8,"is_corresponding":false},{"id":289990,"name":"Wen‐Hann Tan","orcid":"0000-0002-1593-6149","position":9,"is_corresponding":false},{"id":289987,"name":"Anjali Sadhwani","orcid":"0000-0003-0938-0591","position":10,"is_corresponding":false},{"id":1468880,"name":"Catherine Merton","orcid":"0000-0002-1702-3132","position":0,"is_corresponding":true}],"reference_count":51,"raw_metadata":null,"created_at":"2026-07-19T02:56:28.709936Z","pmid":"40385450","pmcid":null,"fwci":null,"citation_percentile":null,"influential_citations":0,"oa_status":null,"license":null,"views":0,"total_file_size_bytes":0,"version_count":0,"fair_f":null,"fair_a":null,"fair_i":null,"fair_r":null,"fair_zscore":null,"fair_rationale":null,"fair_model":null,"fair_agent_version":null,"fair_fulltext_source":null,"fair_has_llm":null,"fair_computed_at":null,"clinical_trials":[],"software_tools":[],"db_accessions":[],"linked_datasets":[],"topics":[]}