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 aetiologies. 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 parental stress and FQOL. Stress and FQOL were examined across AS molecular subtypes, and predictors were analysed using a generalised linear model. Relationships between parental stress and FQOL were examined using Pearson correlations and a stepwise mixed-linear model approach.

RESULTS: Our sample consisted of 231 families of individuals living with AS. Parental stress was clinically elevated and was highest in families of individuals with UBE3A pathogenic variants, whereas FQOL did not differ across subtypes 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, whereas 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 negative impact on FQOL. Interventions to reduce stress have the potential to improve individual and family well-being.