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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.

To synthesize and critically evaluate evidence on the effectiveness of integrated psychological care models for children with complex chronic illness within pediatric hospital settings and provide recommendations for successful implementation.Six electronic databases (Medline, Cochrane, Embase, PsycINFO, Scopus, CINAHL) were systematically searched for English language studies including families of children aged 0-17 years with complex chronic illness. Eligible studies reported on psychology or neuropsychology screening, assessment, intervention, or services provided within a pediatric hospital setting.Fifteen studies were identified for review; nine assessed a psychological service, five examined psychosocial screening, and one examined a neuropsychology service. Three studies demonstrated the effectiveness of integrated psychological services in improving child or parent physical, psychological, or behavioral health outcomes. Uptake of psychosocial screening was high (84-96%), but only 25-37% of children or families identified as 'at-risk' engaged with on-site psychology services. Integrated psychological services offering consultations at the same time and location as the child's medical visit reported the highest rates of uptake (77-100%).The available evidence supports co-location of child medical and psychological services. A more consistent and comprehensive approach to the assessment of patient- and caregiver-reported outcomes and implementation effectiveness is recommended.

Abstract Literacy is a major social determinant of health, rooted in skills that develop during early childhood. Children arriving at kindergarten unprepared to learn to read are more likely to have low reading proficiency thereafter. General and health literacy are highly correlated, affecting understanding of health conditions, treatment adherence, and transition to self-care and adult healthcare services. The American Academy of Pediatrics (AAP) recommends literacy and school readiness promotion during well-visits and neurodevelopmental surveillance is emphasized across primary and subspecialty care. While genetic and environmental risk factors for reading difficulties are well-established, risks related to complex and chronic medical conditions are less appreciated and under-researched. This review applies an eco-bio-developmental framework to explore literacy across five complex chronic conditions affecting millions of children worldwide: asthma, cancer, congenital heart disease, epilepsy, and sickle cell disease. In each, integration of an efficient reading brain network may be impacted by direct factors, such as ischemia, anesthesia, and/or medications, and also indirect factors, such as altered parent–child routines, hospital stays, and missed school. By integrating literacy into care management plans for affected children, pediatric primary care and specialty providers are poised to identify risks early, target guidance and interventions, and improve academic and health outcomes. Impact While genetic and environmental risk factors for reading difficulties are well-established, risks related to complex and/or chronic medical conditions such as asthma, cancer, congenital heart disease, epilepsy, and sickle cell disease are substantial, less appreciated, and under-researched. General and health literacy are highly correlated, with implications for the understanding one’s health condition, treatment adherence, and transitioning to self-care, which is especially important for children with complex and/or chronic illness. Pediatric primary care and specialty providers are poised to integrate reading and literacy into care management plans for children with complex and/or chronic illness, including early screening, guidance, support, and interventions.

Objective:COVID-19 has markedly impacted the provision of neurodevelopmental care. In response, the Cardiac Neurodevelopmental Outcome Collaborative established a Task Force to assess the telehealth practices of cardiac neurodevelopmental programmes during COVID-19, including adaptation of services, test protocols and interventions, and perceived obstacles, disparities, successes, and training needs.Study Design:A 47-item online survey was sent to 42 Cardiac Neurodevelopmental Outcome Collaborative member sites across North America within a 3-week timeframe (22 July to 11 August 2020) to collect cross-sectional data on practices.Results:Of the 30 participating sites (71.4% response rate), all were providing at least some clinical services at the time of the survey and 24 sites (80%) reported using telehealth. All but one of these sites were offering new telehealth services in response to COVID-19, with the most striking change being the capacity to offer new intervention services for children and their caregivers. Only a third of sites were able to carry out standardised, performance-based, neurodevelopmental testing with children and adolescents using telehealth, and none had completed comparable testing with infants and toddlers. Barriers associated with language, child ability, and access to technology were identified as contributing to disparities in telehealth access.Conclusions:Telehealth has enabled continuation of at least some cardiac neurodevelopmental services during COVID-19, despite the challenges experienced by providers, children, families, and health systems. The Cardiac Neurodevelopmental Outcome Collaborative provides a unique platform for sharing challenges and successes across sites, as we continue to shape an evidence-based, efficient, and consistent approach to the care of individuals with CHD.

AIM: To understand the factors that most influence decision-making in the treatment of infantile spinal muscular atrophy (SMA). METHOD: A discrete choice experiment was conducted among parents of people with SMA (parents), healthcare professionals (HCPs), and members of the Australian general population (GenPop). Respondents were asked to accept/reject treatment for an infant newly diagnosed with SMA in eight hypothetical scenarios, characterized by different combinations of the attributes of the treatment offered. The results were analyzed using probability analysis. RESULTS: Completed responses were provided from 1113 individuals (1024 GenPop, 21 parents, 68 HCPs). Respondents were more likely to accept treatments that improved functioning and mobility. Treatments with higher costs, invasive delivery, and risks of adverse events were accepted less often. Cost most affected treatment choices by HCPs and GenPop, while change in mobility and mode of administration were most influential for parents. INTERPRETATION: These results highlight the importance of understanding value for money and clinical impact in affecting treatment choice, which are crucial for effective planning of healthcare and the successful implementation of treatment programmes for SMA. What this paper adds Spinal muscular atrophy (SMA) treatments with a higher chance of improving functioning and mobility are preferred by the general population, parents, and healthcare professionals. Treatments with higher costs, invasive delivery, and risk of adverse events are less preferred. Willingness to pay for SMA treatments increases with impact on functioning.

Although resilience and high quality of life are demonstrated by many individuals with congenital heart disease, a range of significant psychological challenges exists across the life span for this growing patient population. Psychiatric disorders represent the most common comorbidity among people with congenital heart disease. Clinicians are becoming increasingly aware of the magnitude of this problem and its interplay with patients' physical health, and many seek guidance and resources to improve emotional, behavioral' and social outcomes. This American Heart Association scientific statement summarizes the psychological outcomes of patients with congenital heart disease across the life span and reviews age-appropriate mental health interventions, including psychotherapy and pharmacotherapy. Data from studies on psychotherapeutic, educational' and pharmacological interventions for this population are scarce but promising. Models for the integration of mental health professionals within both pediatric and adult congenital heart disease care teams exist and have shown benefit. Despite strong advocacy by patients, families' and health care professionals, however, initiatives have been slow to move forward in the clinical setting. It is the goal of this scientific statement to serve as a catalyst to spur efforts for large-scale research studies examining psychological experiences, outcomes, and interventions tailored to this population and for integrating mental health professionals within congenital heart disease interdisciplinary teams to implement a care model that offers patients the best possible quality of life.

Diagnosis and treatment of congenital heart disease (CHD) can present challenges to the developing parent-child relationship due to periods of infant hospitalization and intensive medical care, parent-infant separations, child neurodevelopmental delay and feeding problems, and significant parent and child distress and trauma. Yet, the ways in which CHD may affect the parent-child relationship are not well-understood. We systematically reviewed the evidence on parental bonding, parent-child interaction, and child attachment following CHD diagnosis, according to a pre-registered protocol (CRD42019135687). Six electronic databases were searched for English-language studies comparing a cardiac sample (i.e., expectant parents or parents and their child aged 0-5 years with CHD) with a healthy comparison group on relational outcomes. Of 22 unique studies, most used parent-report measures (73%) and yielded mixed results for parental bonding and parent-child interaction quality. Observational results also varied, although most studies (4 of 6) found difficulties in parent-child interaction on one or more affective or behavioural domains (e.g., lower maternal sensitivity, lower infant responsiveness). Research on parental-fetal bonding, father-child relationships, and child attachment behaviour was lacking. Stronger evidence is needed to determine the nature, prevalence, and predictors of relational disruptions following CHD diagnosis, and to inform targeted screening, prevention, and early intervention programs for at-risk dyads.

Growing evidence links mindful awareness to the regulation of emotion, yet little is known about the temporal features of this association. This preliminary prospective study aimed to test the bidi...

Understanding the potential effects of the COVID-19 pandemic on the developing parent-infant relationship is a priority, especially for medically-fragile infants and their caregivers who face distinct challenges and stressors. Observational assessments can provide important insights into parent-child behaviors and relational risk; however, stay-at-home directives and physical distancing measures associated with COVID-19 have significantly limited opportunities for in-person observational parent-infant assessment. To maintain momentum in our research program during the pandemic, we rapidly pivoted to remote, technology-assisted parent-infant observational assessments. In this commentary, we offer a series of strategies and recommendations to assist researchers in adapting observational parent-infant paradigms. We also discuss the benefits, challenges, and limitations of distance-delivered assessments, and offer considerations for clinical service provision and future research during and post the COVID-19 pandemic.

Contains fulltext : 288352.pdf (Publisher’s version ) (Open Access) OBJECTIVE: Care for fear of cancer recurrence (FCR) is considered the most common unmet need among cancer survivors. Yet the prevalence of FCR and predisposing factors remain inconclusive. To support targeted care, we provide a comprehensive overview of the prevalence and severity of FCR among cancer survivors and patients, as measured using the short form of the validated Fear of Cancer Recurrence Inventory (FCRI-SF). We also report on associations between FCR and clinical and demographic characteristics. METHODS: This is a systematic review and individual participant data (IPD) meta-analysis on the prevalence of FCR. In the review, we included all studies that used the FCRI-SF with adult (≥18 years) cancer survivors and patients. Date of search: 7 February 2020. Risk of bias was assessed using the Joanna Briggs Institute critical appraisal tool. RESULTS: IPD were requested from 87 unique studies and provided for 46 studies comprising 11,226 participants from 13 countries. 9311 respondents were included for the main analyses. On the FCRI-SF (range 0-36), 58.8% of respondents scored ≥13, 45.1% scored ≥16 and 19.2% scored ≥22. FCR decreased with age and women reported more FCR than men. FCR was found across cancer types and continents and for all time periods since cancer diagnosis. CONCLUSIONS: FCR affects a considerable number of cancer survivors and patients. It is therefore important that healthcare providers discuss this issue with their patients and provide treatment when needed. Further research is needed to investigate how best to prevent and treat FCR and to identify other factors associated with FCR. The protocol was prospectively registered (PROSPERO CRD42020142185).

The provision of effective care models for children with neurodevelopmental delay or disability can be challenging in resource constrained healthcare systems. Economic evaluations have an important role in informing resource allocation decisions. This review systematically examined the scope and methods of economic models evaluating interventions for supporting neurodevelopment among children with common neurodevelopmental disorders and identified methods of economic models and presented policy implications. This scoping review employed the Arksey and O'Malley framework and aligned with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses extension for Scoping Reviews (PRISMA-ScR). Four electronic databases were systematically searched to identify eligible model-based economic evaluations of neurodevelopmental care models published since 2000. The Consolidated Health Economic Evaluation Reporting Standards (CHEERS) checklist was used to assess quality of reporting. Data were systematically extracted, tabulated, and qualitatively synthesised across diagnostic categories. Searches identified 1431 unique articles. Twelve studies used a decision analytic model to evaluate care for neurodevelopmental disorders and were included in the review. Included studies focused on attention-deficit/hyperactivity disorder (ADHD, n=6), autism spectrum disorder (ASD, n=3), cerebral palsy (n=2), and dyslexia (n=1). The most used decision analytic modelling approach was a Markov model (n=6), followed by a decision tree (n=3), and a combination of decision tree and Markov model (n=3). Most studies (n=7) adopted a societal perspective for reporting costs. None of the reviewed studies modelled impact on families and caregivers. Four studies reported cost-savings, three identified greater quality of life, and three identified cost increases.

Clinical practice guidelines for paediatric neuromuscular disorders (NMDs) recommend timely provision of wheelchair equipment. The aim of this qualitative study was to understand healthcare professionals' clinical perspectives and practices when recommending wheelchair equipment for the first time, as well as perceived barriers and enablers to timely wheelchair provision.Twenty-one healthcare professionals (HCPs) from Australia and the United States participated in an interview (response rate: 88%, 16/21 women). Participants were from diverse disciplines, based in hospital or community health settings, and had at least one year of experience working in paediatric neuromuscular care.Child fatigue, falls and engagement in age-appropriate activity were common reasons for HCP's wheelchair recommendation. HCPs were acutely aware of parents' experiences of grief and loss throughout the wheelchair prescription process, and over half acknowledged the lack of psychological care available to families affected by NMDs. Multi-disciplinary collaboration, psychologically-informed care, and shared decision-making with stakeholders were perceived enablers of wheelchair transition. Barriers included limited access to equipment, lengthy funding processes and lack of funding for home and vehicle modifications.Integrated psychosocial care is needed to support families throughout their child's disease progression, including wheelchair transition. Implementation of readiness for change tools, and development of tailored informational resources is recommended. Improved access to equipment options and trials, and more efficient funding processes are highly likely to improve parental engagement throughout the wheelchair prescription process.Implications for rehabilitationHealthcare professionals express a strong demand for integrated psychosocial care within paediatric neuromuscular clinics to support families throughout transitions, including wheelchair introduction.Identifying parents' readiness for change can inform their information and support needs, strengthen their decision-making capacity and facilitate timely wheelchair introduction.Stronger collaboration between hospital- and community-based health professionals working in paediatric neuromuscular care is recommended to facilitate knowledge exchange and support families' transition to wheelchair use.Access to equipment options for extended loan or trial in the community can support timely wheelchair introduction.

The natural history and treatment of spinal muscular atrophy (SMA) is currently being transformed by the development and availability of novel therapies, with significant related changes in practice. This not only has important implications for the health and wellbeing of patients with SMA and their families, as well as improves the quality of care. The present study aimed to investigate the processes and factors that influence treatment and healthcare decisions for children and adults with SMA and their families and healthcare providers. Four focus groups comprising adults, or parents of children and adolescents, with SMA and an expert panel of healthcare providers (N = 25) explored experiences of SMA, its treatment and related decision making and expectations for future care. Group discussions were recorded and transcribed verbatim for thematic analysis using NVivo12.0. People with SMA, their families and healthcare providers described confronting complex healthcare decisions in the context of a rapidly changing SMA treatment environment. Across all groups, five key themes were identified: hope, yearning and searching, patient-centred care and support, community and a sense of connectedness and weighing up potential treatment benefits and costs. Essential to these themes was the notion of what it means to live with SMA and complexities relating to ‘quality of life’. Identifying and more deeply understanding the factors that influence patient, family and healthcare providers’ decision making regarding SMA treatment is an important first step in improving the quality of patient- and family-centred care and in informing clinical practice and future health policy incorporating personalized medicine and optimal supportive and mental health care.

IntroductionFear of cancer recurrence (FCR) is commonly reported by patients diagnosed with early-stage (0–II) melanoma and can have a significant impact on daily functioning. This study will pilot the implementation of the Melanoma Care Program, an evidence-based, psychological intervention to reduce FCR, into routine practice, using a stepped-care model.Methods and analysisIntervention effectiveness and level of implementation will be investigated using a hybrid type I design. Between 4 weeks before and 1 week after their next dermatological appointment, patients with melanoma will be invited to complete the Fear of Cancer Recurrence Inventory Short-Form, measuring self-reported FCR severity. Using a stepped-care model, clinical cut-off points will guide the level of support offered to patients. This includes: (1) usual care, (2) Melanoma: Questions and Answers psychoeducational booklet, and (3) three or five psychotherapeutic telehealth sessions. This longitudinal, mixed-methods pilot implementation study aims to recruit 108 patients previously diagnosed with stage 0–II melanoma. The primary effectiveness outcome is change in FCR severity over time. Secondary effectiveness outcomes include change in anxiety, depression, stress, health-related quality of life and melanoma-related knowledge over time. All outcomes are measured at baseline, within 1 week of the final telehealth session, and 6 and 12 months post-intervention. Implementation stakeholders at each study site and interested patients will provide feedback on intervention acceptability and appropriateness. Implementation stakeholders will also provide feedback on intervention cost, feasibility, fidelity and sustainability. These outcomes will be measured throughout implementation, using questionnaires and semistructured interviews/expert group discussions. Descriptive statistics, linear mixed-effects regression and thematic analysis will be used to analyse study data.Ethics and disseminationEthics approval was granted by the Sydney Local Health District–Royal Prince Alfred Zone (2020/ETH02518), protocol number: X20-0495. Results will be disseminated through peer-reviewed journals, conference presentations, social media and result summaries distributed to interested participants.Trial registration details(ACTRN12621000145808).

Traditional congenital heart disease (CHD) outcomes include mortality (survival to adulthood and life expectancy) as well as cardiac and noncardiac morbidity. Strategies to identify and manage sequelae have primarily focused on objective data obtained though invasive and noninvasive diagnostic approaches. In contrast, patient-reported outcomes (PROs) provide subjective information, using standardized measures, about patients' health and well-being as reported directly by patients, without interpretation, interference, or assumptions made by clinicians or others. Selection of PRO measures entails thoughtful consideration of who the individuals being surveyed are, why assessment is occurring (eg, what are the domains of interest; clinical vs research), and what processes are in place for acquisition, administration, interpretation, and response. In this review, we focus on 3 domains of PROs for pediatric and adult patients with Fontan physiology: physical health status, psychological functioning, and quality of life. Infants, children, adolescents, and adults with CHD face a spectrum of challenges that might influence PROs across the life span. In general, patients with Fontan palliation tend to have lower physical health status, experience more psychological distress, and have equivalent or reduced quality of life compared with healthy peers. Herein, we provide an overview of PROs among people with Fontan circulation as a group, yet simultaneously emphasize that the optimal way to understand the experiences of any individual patient is to ask and listen. We also offer clinical and research initiatives to improve the adoption and utility of PROs in CHD settings, which show commitment to capturing, understanding, and responding to the patient voice.

Pediatric weight management is often hampered by poor engagement and adherence. Incentives based on loss have been shown to be more effective than gain-based incentives in improving outcomes among children with health conditions other than obesity. In preparation for a clinical trial comparing loss-framed to gain-framed incentives, a survey of youth and caregiver attitudes on weight management incentives, reasons for program attendance, and an economic evaluation of a theoretical trial were conducted. Ninety of 835 (11%) surveys were completed by caregiver and child. The economic evaluation showed that loss-framed incentives had a preferable incremental cost-effectiveness ratio (a lower value is considered preferable) than gain-based incentives. Most youth and caregivers felt a gain incentive would be superior, agreed that the full incentive should go to the youth (vs. the caregiver), and identified “improving health” as a top reason for pursuing weight management.

Objective In the wake of the COVID-19 pandemic, psychologists were pushed to look beyond traditional in-person models of neurodevelopmental assessment to maintain continuity of care. A wealth of data demonstrates that telehealth is efficacious for pediatric behavioral intervention; however, best practices for incorporating telehealth into neurodevelopmental assessment are yet to be developed. In this topical review, we propose a conceptual model to demonstrate how telehealth can be incorporated into various components of neurodevelopmental assessment. Methods Harnessing existing literature and expertise from a multidisciplinary task force comprised of clinicians, researchers, and patient/parent representatives from the subspecialty of cardiac neurodevelopmental care, a conceptual framework for telehealth neurodevelopmental assessment was developed. Considerations for health equity and access to care are discussed, as well as general guidelines for clinical implementation and gaps in existing literature. Results There are opportunities to integrate telehealth within each stage of neurodevelopmental assessment, from intake to testing, through to follow-up care. Further research is needed to determine whether telehealth mitigates or exacerbates disparities in access to care for vulnerable populations as well as to provide evidence of validity for a wider range of neurodevelopmental measures to be administered via telehealth. Conclusions While many practices are returning to traditional, face-to-face neurodevelopmental assessment services, psychologists have a unique opportunity to harness the momentum for telehealth care initiated during the pandemic to optimize the use of clinical resources, broaden service delivery, and increase access to care for pediatric neurodevelopmental assessment.

Background People with melanoma want and need effective interventions for living with fear of cancer recurrence (FCR). Objectives This study reports the 12-month outcomes of a brief, psychological intervention designed to reduce FCR in people at high risk of developing another primary melanoma compared with usual care. Methods In this two-arm randomized controlled trial, adults previously diagnosed with stage 0, I or II melanoma were randomly allocated to the intervention (n = 80) or control (usual care) arm (n = 84). The trial was registered with the Australian and New Zealand Clinical Trials Registry on 19 March 2013 (registration: ACTRN12613000304730). The intervention comprised a 76-page psychoeducational resource and three individually tailored, telephone-based sessions with a psychologist, scheduled at specific time points around participants' dermatological appointments. The primary outcome was the level of self-reported fear of new or recurrent melanoma assessed at 12 months postintervention using the severity subscale of the Fear of Cancer Recurrence Inventory. Results Compared with the control arm, the intervention group reported significantly lower FCR at 12 months postintervention; the between-group mean difference was -1·41 for FCR severity [95% confidence interval (CI) -2·6 to -0·2; P = 0·02] and -1·32 for FCR triggers (95% CI -2·6 to -0·02; P = 0·04). The odds ratio for FCR severity scores ≥13 (54% intervention, 63% control) was 0·59 (95% CI 0·30-1·14, P = 0·12). There were no differences between groups in secondary outcomes, such as anxiety, depression or health-related quality of life. Conclusions The previously reported 6-month benefits of this brief, patient-centred psychological intervention in reducing FCR were found to continue 12 months postintervention, with no known adverse effects, supporting implementation as part of routine melanoma care.

This study aimed to evaluate the cost effectiveness of a newly developed psycho-educational intervention to reduce fear of cancer recurrence (FCR) in early-stage melanoma patients. A within-trial cost-effectiveness and cost-utility analysis was conducted from the Australian health system perspective using data from linked Medicare records. Outcomes included FCR, measured with the severity subscale of the FCR Inventory; quality-adjusted life years (QALYs) measured using the preference-based instrument, Assessment of Quality of Life-8 Dimensions (AQoL-8D) and 12-month survival. An incremental cost-effectiveness ratio (ICER) was calculated for two economic outcomes: (1) cost per additional case of ‘high’ FCR avoided and (2) cost per QALY gained. Means and 95% CIs around the ICER were generated from non-parametric bootstrapping with 1000 replications. A total of 151 trial participants were included in the economic evaluation. The mean cost of the psycho-educational intervention was AU$1614 per participant, including intervention development costs. The ICER per case of high FCR avoided was AU$12,903. The cost-effectiveness acceptability curve demonstrated a 78% probability of the intervention being cost effective relative to the control at a threshold of AU$50,000 per extra person avoiding FCR. The ICER per QALY gained was AU$116,126 and the probability of the intervention being cost effective for this outcome was 36% at a willingness to pay of AU$50,000 per QALY. The psycho-educational intervention reduced FCR at 12 months for people at high risk of developing another melanoma and may represent good value for money. For the QALY outcome, the psycho-educational intervention is unlikely to be cost effective at standard government willingness-to-pay levels. The trial was prospectively registered in the Australian and New Zealand Clinical Trials Registry (CTRN12613000304730).

Medical informed consent is the process by which a 'competent', non-coerced individual receives sufficient information including risks of a medical procedure and gives permission for it to occur. The capacity to give an informed consent might be impaired during labour. This study aimed to examine women's abilities to understand and remember during labour. Women were prospectively recruited at 36 weeks of gestation and randomised to undertake questionnaires which assessed their ability to understand and remember information. They were randomised to: (1) information given in labour only, written format (2) information in labour, verbal (3) information at 36 weeks plus labour, written (4) information at 36 weeks plus labour, verbal. Immediate comprehension and retention was assessed at 36 weeks, in labour, and 24-72 hours after birth. Forty-nine women completed the questionnaires regarding understanding and retention of information at 36 weeks, six intrapartum, and five postpartum (90% attrition). Women receiving information at 36 weeks and in labour

Diagnosis of CHD substantially affects parent mental health and family functioning, thereby influencing child neurodevelopmental and psychosocial outcomes. Recognition of the need to proactively support parent mental health and family functioning following cardiac diagnosis to promote psychosocial adaptation has increased substantially over recent years. However, significant gaps in knowledge remain and families continue to report critical unmet psychosocial needs. TheParent Mental Health and Family Functioning Working Groupof the Cardiac Neurodevelopmental Outcome Collaborative was formed in 2018 through support from an R13 grant from the National Heart, Lung, and Blood Institute to identify significant knowledge gaps related to parent mental health and family functioning, as well as critical questions that must be answered to further knowledge, policy, care, and outcomes. Conceptually driven investigations are needed to identify parent mental health and family functioning factors with the strongest influence on child outcomes, to obtain a deeper understanding of the biomarkers associated with these factors, and to better understand how parent mental health and family functioning influence child outcomes over time. Investigations are also needed to develop, test, and implement sustainable models of mental health screening and assessment, as well as effective interventions to optimise parent mental health and family functioning to promote psychosocial adaptation. The critical questions and investigations outlined in this paper provide a roadmap for future research to close gaps in knowledge, improve care, and promote positive outcomes for families of children with CHD.

Comprehensive psychological care of people with melanoma spans all phases of the disease trajectory, including risk assessment, diagnosis, treatment, recovery, and survivorship, as well as the transition from curative to palliative care, death, and bereavement. At least one-third of people with melanoma report levels of psychological distress indicative of a need for clinical intervention, and over 70% report high levels of fear of cancer recurrence or progression. Many patients experience the period of diagnostic uncertainty between detection of a suspicious lesion and receipt of biopsy results as particularly stressful. High psychological distress is associated with reduced participation in cancer screening, treatment and risk management recommendations, delays in seeking medical advice, lower satisfaction with clinical care, higher health care costs, poorer social functioning, greater morbidity and mortality, and poorer overall quality of life. Screening for distress in people with melanoma is strongly recommended as part of routine, multidisciplinary care, followed by a stepped approach to psychological support and treatment, based on patient risk and resilience factors, needs, values, and preferences. A range of psychotherapeutic and psychoeducational interventions, delivered via a variety of modalities, are effective in reducing psychological distress in people with melanoma in the short and longer term. Cutting-edge research is focused on harnessing technology to bring effective interventions to scale.

Prenatal diagnosis of congenital heart disease (CHD) can be a life-altering and traumatic event for expectant parents. Parental anxiety, depression, and traumatic stress are common following a prenatal cardiac diagnosis and if untreated, symptoms often persist long-term. During prenatal counseling, parents must try to manage psychological distress, navigate uncertainty, process complex medical information, and make high-stakes medical decisions for their unborn child and their family. Physicians must deliver the diagnosis, describe the expected perinatal management plan, discuss short and long-term prognoses and introduce elements of uncertainty that may exist for the particular diagnosis. Physican training in these important skills is highly variable and many in our field acknowledge the need for improved guidance on best practices for counseling and supporting parents during pregnancy and early parenthood after prenatal diagnosis, while also sustaining physicians' own emotional well-being. We describe these challenges and the opportunities that exist to improve the current state of prenatal counseling in CHD.

Background: Neurocognitive outcomes beyond childhood in people with a Fontan circulation are not well defined. This study aimed to investigate neurocognitive functioning in adolescents and adults with a Fontan circulation and associations with structural brain injury, brain volumetry, and postnatal clinical factors. Methods: In a binational study, participants with a Fontan circulation without a preexisting major neurological disability were prospectively recruited from the Australia and New Zealand Fontan Registry. Neurocognitive function was assessed by using Cogstate software in 107 participants with a Fontan circulation and compared with control groups with transposition of the great arteries (n=50) and a normal circulation (n=41). Brain MRI with volumetric analysis was performed in the participants with a Fontan circulation and compared with healthy control data from the ABIDE I and II (Autism Brain Imaging Data Exchange) and PING (Pediatric Imaging, Neurocognition, and Genetics) data repositories. Clinical data were retrospectively collected. Results: Of the participants with a Fontan circulation who had a neurocognitive assessment, 55% were male and the mean age was 22.6 years (SD 7.8). Participants with a Fontan circulation performed worse in several areas of neurocognitive function compared with those with transposition of the great arteries and healthy controls ( P P P =0.01 and working memory, P =0.02). Structural brain injury was present in the entire Fontan cohort; the presence of white matter injury was associated with worse paired associate learning ( P P P P P ≤0.04). Conclusions: Neurocognitive impairment is common in adolescents and adults with a Fontan circulation and is associated with smaller gray and white matter brain volume. Understanding modifiable factors that contribute to brain injury to optimize neurocognitive function is paramount.

Background People with a Fontan circulation experience a range of physical, psychosocial and neurodevelopmental challenges alongside, or caused by, their cardiac condition, with significant consequences for health‐related quality of life ( HRQOL ). We meta‐analyzed HRQOL outcomes reported by people with a Fontan circulation or their proxies and evaluated predictors of poorer HRQOL . Methods and Results Six electronic databases were searched for peer‐reviewed, English‐language articles published before March 2019. Standardized mean differences (SMD) were calculated using fixed and random‐effects models. Fifty articles reporting on 29 unique studies capturing HRQOL outcomes for 2793 people with a Fontan circulation and 1437 parent‐proxies were analyzed. HRQOL was lower in individuals with a Fontan circulation compared with healthy referents or normative samples (SMD, −0.92; 95% CI , −1.36 to −0.48; P HRQOL domains, with the largest differences found for physical (SMD, −0.90; 95% CI , −1.13 to −0.67; P CI , −0.90 to −0.52; P P =0.004), and diagnosis of hypoplastic left heart was associated with poorer social functioning (β=−0.007; P =0.048). Sensitivity analyses showed use of the PedsQL Core Module was associated with lower HRQOL scores compared with the Short‐Form Health Survey‐36. Conclusions HRQOL outcomes for people with a Fontan circulation are lower than the general population. Optimal care acknowledges the lifelong impact of the Fontan circulation on HRQOL and offers targeted strategies to improve outcomes for this growing population.

Background Spinal muscular atrophy (SMA) is a neurodegenerative disease that has a substantial and multifaceted burden on affected adults. While advances in supportive care and therapies are rapidly reshaping the therapeutic environment, these efforts have largely centered on pediatric populations. Understanding the natural history, care pathways, and patient-reported outcomes associated with SMA in adulthood is critical to advancing health policy, practice and research across the disease spectrum. The aim of this study was to systematically review research investigating the healthcare, well-being and lived experiences of adults with SMA. Methods In accordance with the Preferred Reported Items for Systematic Reviews and Meta-Analysis guidelines, seven electronic databases were systematically searched until January 2020 for studies examining clinical (physical health, natural history, treatment) and patient-reported (symptoms, physical function, mental health, quality of life, lived experiences) outcomes in adults with SMA. Study risk of bias and the level of evidence were assessed using validated tools. Results Ninety-five articles met eligibility criteria with clinical and methodological diversity observed across studies. A heterogeneous clinical spectrum with variability in natural history was evident in adults, yet slow declines in motor function were reported when observational periods extended beyond 2 years. There remains no high quality evidence of an efficacious drug treatment for adults. Limitations in mobility and daily activities associated with deteriorating physical health were commonly reported, alongside emotional difficulties, fatigue and a perceived lack of societal support, however there was no evidence regarding effective interventions. Conclusions This systematic review identifies the many uncertainties regarding best clinical practice, treatment response, and long-term outcomes for adults with SMA. This comprehensive identification of the current gaps in knowledge is essential to guide future clinical research, best practice care, and advance health policy with the ultimate aim of reducing the burden associated with adult SMA.

Background: Development and implementation of effective family-based intervention and treatment strategies during COVID-19, and future public health crises, will require a detailed understanding of the ways that COVID-19 has affected parents, children, and families. Qualitative and mixed-methods research is an important means of gaining such detailed information, particularly in the early phases of the pandemic, when the research community continues to explore the meaning of this experience. Methods: We employed thematic analysis to identify themes in parents’ and children’s reflective writing about COVID-19, and explored how citation of certain themes mapped onto quantitative measures of distress commonly used in past psychosocial research on pandemics. Data were collected from N = 56 parents and N = 43 children between April to May 2020.Findings: Analysis of parent and child written reports revealed positive and negative thematic content, though parents expressed more positive themes than children. The most common themes identified from parent reports surrounded concerns about the impact of COVID-19 on their children, health concerns for others, and the stressful balancing act of parenting, schooling, and working from home. In addition, many parents reported gratitude for what they had, and reflected on the upsides of the pandemic for family relations. For children, the most common themes identified surrounded missing school and social relationships. When children expressed positive themes, they tended to be more egocentric reflections (e.g. learning to ride a bike). Both parent and child thematic content mapped onto parent-proxy and parent-self reported quantitative dimensions of parent and child distress. Finally, we observed that parents who reflected on gratitude in their written reports exhibited a decrease in negative affect pre- to -post-writing.Interpretation: Both parents and children are suffering from reduced wellbeing during the COVID-19 pandemic, particularly with the burdens imposed by the closure of in-person schooling. The psychosocial impact of the pandemic on families would likely be even greater in magnitude within less advantaged populations. Future research and policy should focus on identifying how to fulfill children’s social needs and lessen the caregiving burdens during this time.

As the molecular basis of congenital heart disease (CHD) comes into sharper focus, cardiac genetics services are likely to play an increasingly important role. This study aimed to identify parents’ preferences for, and willingness to participate in, clinical genetics services for CHD. A discrete choice experiment was developed to assess parents’ preferences for pediatric cardiogenetics services based on four attributes: appointment format, health professionals involved, waiting time, and information format. Data were analyzed using a mixed logit model. One hundred parents with a living child diagnosed with CHD requiring surgical intervention between 2000 and 2009 completed the discrete choice experiment. Parents expressed a clear preference for cardiac genetics services featuring (i) a single appointment, (ii) the presence of a clinical geneticist and a genetic counselor, (iii) both verbal (oral) and Web-based information about CHD and genetics, and (iv) availability of an appointment within 2 weeks. If offered such conditions, 93% of respondents indicated that they would attend. The choice of service was most strongly influenced by the presence of both a clinical geneticist and a genetic counselor. Parents of children with CHD favor a single, timely genetics appointment with both a geneticist and a genetic counselor present. If appointments offered match these preferences, uptake is likely to be high.

Purpose: To conduct a scoping review of the published evidence on parents' perceptions of power wheelchair prescription for children with a neuromuscular disorder and clinicians' influence on timely wheelchair implementation.Method: Nine electronic databases and reference lists of all retrieved full-text articles were searched up to March 2017. Eligibility criteria included (1) at least one child participant with a neuromuscular disorder, (2) power wheelchair as an intervention, and (3) qualitative, quantitative or mixed methods parent-reported outcomes related to power wheelchair equipment.Results: None of the 67 eligible studies examined parental perceptions of wheelchair prescription as a primary aim, and only 10 studies included children with a neuromuscular disorder. Parents reported their own emotional responses including grief and loss, emphasis on their child's walking and lack of accessibility as key barriers to power wheelchair prescription. Clinicians' perspectives on walking and powered mobility influenced parental decision-making regarding power wheelchair use for their child.Conclusion: Parents' experiences of initial wheelchair prescription have not been explored in existing literature. Clinicians' understanding of the benefits of power wheelchair equipment, particularly in the context of progression of neuromuscular disorders, is critical to facilitating timely wheelchair prescription with children. Condition-specific evidence is urgently needed to inform and support multidisciplinary management of children and their families.Implications for RehabilitationIt is important that rehabilitation professionals recognize parental barriers to initial power wheelchair prescription, such as strong emotional responses, an emphasis on their child's walking and lack of access.Clinicians' perspectives on walking and powered mobility may influence parental decision-making regarding engagement in power wheelchair prescription and rehabilitation.It is critical that clinicians are aware of and actively educate families regarding the benefits of powered mobility to facilitate timely prescription and support physical and psychological adjustment.