Systematic Review of Instruments Assessing Psychosocial Adaptation and Outcomes Among Families of Children With Congenital Heart Disease.

OBJECTIVE: This systematic review identified instruments quantitatively assessing psychosocial adaptation and outcomes in families of children with congenital heart disease (CHD) and evaluated instrument psychometrics. METHODS: Following Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines and a prospectively registered protocol, electronic databases (CINAHL, Embase, PubMed/MEDLINE, PsycINFO, and SCOPUS) were searched from inception until June 20, 2021 for peer-reviewed articles published in English, reporting quantitative data on psychosocial outcomes among parents/caregivers, siblings, or family system. Instrument characteristics and psychometrics were extracted, and adapted COnsensus-based Standards for the selection of health Measurement INstruments (COSMIN) criteria were applied to assess instrument quality. Descriptive statistics and narrative synthesis were used for analysis. RESULTS: Overall, 108 articles reporting on 107 distinct samples across 26 countries met inclusion. Across those articles, 40 instruments assessed psychological functioning or distress, 12 assessed coping, 11 assessed quality of life constructs, 10 assessed parenting stress/caregiver burden, 10 assessed family functioning/impact, 10 assessed stress appraisal, 5 assessed sibling psychosocial outcomes, and 2 assessed couple relationship satisfaction/strain. Applying COSMIN criteria to available data on original instrument development articles/manuals for English language instruments (n = 54), 67% scored a positive property evidence rating for content validity, 39% for internal consistency, 4% for test-retest reliability, and 9% for responsiveness (longitudinal validity). CONCLUSIONS: Studies vary widely in instruments used to assess psychosocial adaptation and outcomes among families of children with CHD. Instrument selection informed by robust key psychometrics, increased psychometric reporting, development of both a "toolkit" approach and a comprehensive CHD-specific family instrument are among key recommendations.

Neurodevelopmental and health-related quality-of-life outcomes in adolescence after surgery for congenital heart disease in infancy.

AIM: To assess outcomes in adolescence after surgery for congenital heart disease (CHD) in infancy. Domains analysed included cognition and executive function, social and emotional well-being, adaptive behaviour, academic achievement, and health-related quality of life (HRQoL). METHOD: Twenty-one participants (10 males, 11 females) ranged in age from 14 to 17 years (mean 15y 4.8mo, SD 8.4mo). Twenty had biventricular repairs. All were classified as New York Heart Association class I. Measures included: Wechsler Intelligence and Achievement scales; Wide Range Assessment of Memory and Learning, Second Edition; California Verbal Learning Test - Children's Version; Behaviour Rating Inventory of Executive Function; Conners, Third Edition; Adaptive Behavior Assessment System, Second Edition; Behavior Assessment System for Children, Second Edition; Rey-Osterrieth Complex Figure; and Pediatric Quality of Life Inventory. RESULTS: Outcomes were significantly lower (p≤0.01) than population norms for processing speed, mathematical achievement, attention, and visual-spatial ability. Participants reported more frequent learning problems but more positive family relations. HRQoL was significantly lower across most domains by self- and parent-proxy report. INTERPRETATION: Individuals with CHD may experience difficulties across a range of domains. These findings emphasize the importance of comprehensive screening, early intervention, and long-term follow-up, as deficits may extend into young adulthood. WHAT THIS PAPER ADDS: Identified cognitive, learning, and attentional impairments in adolescents after congenital heart disease surgery in infancy. Combined self-report, caregiver report, and laboratory tasks in a comprehensive neurodevelopmental assessment protocol. Health-related quality of life was lower across most domains.

"It's more than just a conversation about the heart": exploring barriers, enablers, and opportunities for improving the delivery and uptake of cardiac neurodevelopmental follow-up care.

Introduction: Surveillance, screening, and evaluation for neurodevelopmental delays is a pivotal component of post-surgical care for children with congenital heart disease (CHD). However, challenges exist in implementing such neurodevelopmental follow-up care in international practice. This study aimed to characterise key barriers, enablers, and opportunities for implementing and delivering outpatient cardiac neurodevelopmental follow-up care in Australia. Methods: an exploratory descriptive qualitative study was conducted with healthcare professionals across Australia who had lived experience of designing, implementing, or delivering neurodevelopmental care for children with CHD. Online semi-structured interviews were conducted using a guide informed by the Consolidated Framework for Implementation Research to explore contextual influences. Interview transcripts were analysed using a rapid qualitative approach including templated summaries and hybrid deductive-inductive matrix analysis. Results: fifty-two participants were interviewed. Perceived barriers and enablers were organised into six higher-order themes: factors in the broader environmental, economic, and political context; healthcare system factors; organisational-level factors; provider factors; patient and family factors; and care model factors. The largest number of barriers occurred at the healthcare system level (service accessibility, fragmentation, funding, workforce), while service providers demonstrated the most enabling factors (interprofessional relationships, skilled teams, personal characteristics). Strategies to improve practice included building partnerships; generating evidence; increasing funding; adapting for family-centred care; and integrating systems and data. Discussion: Australia shares many similar barriers and enablers to cardiac neurodevelopmental care with other international contexts. However, due to unique geographical and health-system factors, care models and implementation strategies will require adaption to the local context to improve service provision.

Health-related quality of life and congenital heart disease in Australia.

AIM: To determine whether, in children with congenital heart disease (CHD), disease severity is associated with health-related quality of life (HRQOL) and impact on the family. METHODS: Cross-sectional, single-centre study comparing HRQOL outcomes of age and sex matched children with hypoplasia of the left ventricle (HLV) (n = 31) and tetralogy of Fallot (n = 29) was performed in Queensland, Australia. HRQOL was assessed using generic and disease-specific components of the Paediatric Quality of Life Inventory Measurement Model (PedsQL). Intra-diagnostic age group comparisons of HRQOL were examined. Impact of CHD on families and parental HRQOL was assessed using the PedsQL Family Impact Scale. RESULTS: Child and parent-proxy reporting indicate children with HLV have significantly lower overall HRQOL than children with tetralogy of Fallot across generic domains of HRQOL (P < 0.0001), with significantly lower scores in physical (P < 0.0001) and psychosocial (P < 0.0001) health domains. No significant difference in child reporting across domains of the Cardiac Module is evident. Parent-proxy reporting indicates significantly lower scores on the symptom scales for children with HLV (P < 0.001), with greater cognitive problems (P < 0.02) and perceived treatment anxiety (P < 0.01). No significant differences in HRQOL were identified between age groups. HLV has a greater overall family impact, with significantly lower parental HRQOL (P = 0.0001) and family functioning (P < 0.0001) summary scores. CONCLUSIONS: The more severe condition of HLV is associated with poorer HRQOL in some domains and has greater impact on parental HRQOL and family functioning.