Children with complex congenital heart disease (CHD) have known neurological risks and developmental challenges. Reference Marino, Lipkin and Newburger1 The Cardiac Neurodevelopmental Outcome Collaborative (CNOC) is a non-profit organisation founded in 2016 (www.cardiacneuro.org) to “optimise neurodevelopmental and psychosocial outcomes for all individuals with CHD across the lifespan through clinical, quality improvement, and research initiatives.” Reference Marino, Sood and Cassidy2 Cardiac Neurodevelopmental Outcome Collaborative member sites include medical institutions that provide care for children with CHD. Amongst the Cardiac Neurodevelopmental Outcome Collaborative’s accomplishments are the establishment of a clinical data registry and publication of clinical test protocols for neurodevelopmental evaluation in an effort to improve quality of care and research collaborations across member sites and internationally. Reference Ware, Butcher and Latal3,Reference Ilardi, Sanz and Cassidy4
Prior to the novel coronavirus disease 2019 (COVID-19) pandemic, multiple centres had identified barriers to recommended neurodevelopmental care for children with CHD resulting in significant health disparities, especially in terms of insurance status, family geographical location (e.g., urban, suburban, or rural), financial resources, and race and ethnicity. Reference Loccoh, Yu and Donohue5,Reference Glotzbach, Ward and Marietta6 More recently, the COVID-19 pandemic and associated restrictions regarding in-person clinical services have markedly impacted the ability to provide timely neurodevelopmental assessment and care in keeping with best practices for this vulnerable patient population. Reference Marino, Lipkin and Newburger1 Initiating and integrating telehealth services within neurodevelopmental programmes may offer opportunities to reduce delays and disparities in access to neurodevelopmental evaluation and care, as well as broaden service reach to a larger percentage of the CHD population. Use of telehealth may also mitigate the risks associated with lack of timely access to neurodevelopmental evaluation (e.g., child behavioural, emotional and learning difficulties, academic delays, family psychological stress) and facilitate nimble adaptation to events that limit travel and in-person evaluations including, but not limited to, COVID-19. There is, however, a need to balance the clinical information gained via telehealth against the information lost by not having an in-person neurodevelopmental assessment.
Currently, there is limited understanding of how cardiac neurodevelopmental programmes have responded to the COVID-19 pandemic and the extent to which telehealth may have been adopted into practice. Moreover, there is a striking lack of evidence to guide telehealth-based neurodevelopmental services. To address this knowledge gap, and rapidly respond to questions and concerns about telehealth-based neurodevelopmental care, the Cardiac Neurodevelopmental Outcome Collaborative Steering Committee approved the formation of a Telehealth Task Force in June 2020. The multidisciplinary Task Force included representation from neuropsychology, paediatric psychology, cardiology and patient advocacy, and members were charged with collating available evidence and relevant guidelines and identifying current practices across institutional member sites. The purpose of this survey of Cardiac Neurodevelopmental Outcome Collaborative member sites was to assess the telehealth practices of cardiac neurodevelopmental programmes during the COVID-19 pandemic, including adaptation of clinical services, test protocols and interventions, as well as perceived obstacles, disparities, and successes. A secondary goal was to disseminate findings to the cardiac community, to encourage research and collaboration to ensure timely access to high-quality neurodevelopmental services during COVID-19 and into the future.
Materials and methods
Study design and population
Participants for this cross-sectional survey were recruited via 42 Cardiac Neurodevelopmental Outcome Collaborative member sites across North America within a 3-week timeframe, from 22 July to 11 August 2020. During this period, many states across the United States, as well as many other countries, were experiencing increasing COVID-19 cases and implementing closures and restrictions on in-person clinical services. In addition, many hospitals had implemented policies limiting in-person patient care. There was also a high level of uncertainty regarding whether children would return to the classroom for in-person schooling in the Fall.
An e-mail describing the purpose of the survey was sent to site representatives at all Cardiac Neurodevelopmental Outcome Collaborative member sites, accompanied by a link to an online survey. Each site was invited to nominate a provider from within their cardiac neurodevelopmental programme to complete the survey. A maximum of two e-mail reminders were sent to sites to encourage participation.
To understand telehealth-related practices during the COVID-19 pandemic, the Cardiac Neurodevelopmental Outcome Collaborative Telehealth Task Force developed a 47-item online survey, delivered via a secure web-based application (REDCap, https://projectredcap.org). The survey (see Supplementary Material 1) assessed the seven key areas outlined below.
Pre-Pandemic Clinical Practices (1 item): Types of clinical services routinely provided by each cardiac neurodevelopmental programme prior to COVID-19.
Clinical Practices During COVID-19 (16 items): Service changes in response to COVID-19, use of telehealth, types of patient and caregiver assessments and interventions offered, assessment measures used, patient prioritisation and triage processes, technical aspects of telehealth implementation, and the status of in-person services.
Obstacles (7 items): Frequency of telehealth-related difficulties associated with Current Procedural Terminology coding, billing and insurance reimbursement, patient and provider technology use, staffing or administrative support, access to language interpreting services, and provider confidence delivering telehealth services were assessed on a scale from 1 (“Never”) to 5 (“Always”).
Licensure, Billing, and Reimbursement (4 items): Out-of-state licensure, billing, and reimbursement by insurance providers and use of grant or donor funds to support telehealth services.
Perceived Disparities in Care (4 items): Each site was asked to identify any disparities in access to or delivery of telehealth services for people of non-English speaking backgrounds; people with limited access to technology; type of health insurance; and children with behavioural, age, or ability-related challenges.
Perceived Success and Patient Satisfaction (8 items): Perceptions of success regarding the use of telehealth to offer caregiver or child interviews, infant and child neurodevelopmental evaluations, test result feedback sessions, and parent- or child-focused interventions were assessed. Respondents also indicated how often families expressed satisfaction or a positive experience with telehealth services using a 5-point rating scale from 1 (“Never”) to 5 (“Always”).
Provider Training Needs (7 items): Respondents indicated areas their programme could either share and teach successes and lessons learned with other programmes; or could learn from others.
Descriptive statistics, including frequencies, percentages, mean scores, and standard deviations were calculated using Excel for Microsoft 365 (Microsoft Co, Redmond, WA) to explore site characteristics, telehealth practices, and provider views and experiences.
Of the 42 Cardiac Neurodevelopmental Outcome Collaborative sites approached, 30 completed the survey, yielding a response rate of 71.4%. The annual median cardiac surgical volume of responding sites was 426 surgeries (range: 57–1306). Amongst the respondents, 54.5% were psychologists, 27.3% physicians, 15.2% nurses, and 3% developmental specialists. Figure 1 illustrates regional and telehealth-related characteristics of responding sites.
Use of telehealth across cardiac neurodevelopmental programmes
Prior to the COVID-19 pandemic, the 30 participating sites provided a range of in-person clinical services, including neurodevelopmental assessment for infants (90%), pre-school (93%), and school-aged children (90%) with CHD, and interventions for infants or children (50%), caregivers (43%), or caregiver-child dyads (33%; e.g., Parent-Child Interaction Therapy). During COVID-19, at the time of survey completion, all sites were providing at least some clinical services and 80% (24/30) reported using telehealth to provide these services. One site was offering telehealth services exclusively, six had returned to in-person services exclusively, and 23 sites had developed a hybrid model including both telehealth and in-person visits. Change in the use of telehealth for neurodevelopmental care had been initiated in direct response to COVID-19 in all but one of these 24 sites. Twenty-three sites offered telehealth to both new and established patients, and one site offered telehealth to established patients only. The most frequently used platform for telehealth was Zoom (n = 11), followed by AmWell (n = 4), DoxyMe (n = 3), Vidyo (n = 3), and Microsoft Teams (n = 2). Of the sites that had returned to in-person services, the date of re-commencement ranged from March (in conjunction with cardiac appointments only) to July 2020, with most sites (21/27) returning to some in-person services in June or July.
Neurodevelopmental evaluation services during COVID-19
Of the sites offering telehealth-based services (n = 24), 22 sites required in-person visits for infant neurodevelopmental evaluation at the time of the survey, one was offering infant assessment via telehealth, and one did not respond (Fig 2). In terms of neurodevelopmental evaluation for children or adolescents with CHD, one site reported providing this service before COVID-19, eight had initiated child or adolescent evaluation via telehealth during the pandemic, and 15 required in-person visits for this service. When implementing telehealth, just over half the sites (n = 13/24) reported transitioning from comprehensive, standardised, performance-based testing to the use of parent rating scales or semi-structured observational methods (Fig 3). Telehealth was used “often” or “always” to prioritise, triage, or determine the need for an in-person visit by 46% of the sites. For all but one site using telehealth, virtual appointments were “sometimes” or “often” followed by an in-person visit to complete the neurodevelopmental evaluation (n = 23). When collecting background and demographic information from caregivers, nine sites used paper-based surveys, four used web-based surveys, and nine used a combination (two sites did not respond).
Qualitatively, several sites raised concerns regarding the validity of neurodevelopmental evaluation, given the adjustments to test batteries required to accommodate the telehealth setting. No site, for example, was administering the Bayley Scales of Infant and Toddler Development, Fourth Edition (BSID-4) via telehealth and most sites (67%) “never” used the recommended Cardiac Neurodevelopmental Outcome Collaborative test battery Reference Ware, Butcher and Latal3,Reference Ilardi, Sanz and Cassidy4 for children and adolescents during telehealth appointments. Alternative measures used for children under age 6 years included the Ages and Stages Questionnaire (ASQ, n = 7) and the Developmental Assessment of Young Children, Second Edition (DAYC-2, n = 3). Of the sites offering neurodevelopmental assessment for children or adolescents via telehealth, only selected subtests from the Cardiac Neurodevelopmental Outcome Collaborative test battery (assessing domains such as intellectual functioning, academics, memory, executive skills) were administered (Fig 3). Two-thirds of sites (65%) were using parent-proxy rating scales from the Cardiac Neurodevelopmental Outcome Collaborative test battery regularly to assess aspects of child behavioural, socio-emotional, and executive functioning. When administering standardised assessments via telehealth, a few sites implemented a range of strategies to accommodate distance-delivery, including the use of two cameras in the child’s home to increase visibility of the child’s responses during assessments (n = 1), mailing test materials (e.g., response booklets) to the home prior to the appointment (n = 3), use of an online testing platform (n = 1), and requiring number responses for visual items which usually require a pointing response (n = 1).
Psychological interventions with children and caregivers
A limited number of sites were offering in-person intervention services for parents (n = 13) or children (n = 15) prior to COVID-19, and of those sites providing interventions for children, 60% were able to successfully transition to telehealth. At the time of this survey, 15 sites were offering parent-focused interventions via telehealth and 12 were providing child-focused interventions, including 13 sites that were offering intervention services for the first time (Fig 2).
Billing and licensure: At the time of this survey, many payors in the United States expanded billing permissions for telehealth assessment and intervention services in response to the pandemic. Although all surveyed Cardiac Neurodevelopmental Outcome Collaborative sites were able to bill for telehealth services, 46% were unaware of service reimbursement rates. For those with knowledge of reimbursement, rates were the same (34%) or lower (7%) than in-person services or varied based on insurance type (13%). Only two sites reported use of grant or donor funds when families could not pay for telehealth services. Out-of-state psychology licensing requirements were waived in some states;21% of sites provided out-of-state telehealth services, 29% could “sometimes” offer out-of-state services, 21% could not provide out-of-state services, and 29% were unsure of their capacity to provide out-of-state services (Fig 1).
Perceived obstacles and disparities: Across the 24 sites using telehealth for neurodevelopmental and psychosocial care, the most frequently reported obstacles were patient and family difficulties with technology (e.g., poor internet connection), reduced provider confidence delivering neurodevelopmental services via telehealth, and difficulties integrating language interpreting services during telehealth appointments (Table 1). Factors perceived to contribute to disparities in care included limited access to language interpreting services, limited patient access to adequate technology, and children not being able to participate in telehealth due to age, behavioural, or ability-related challenges (Table 2).
Each potential obstacle was rated by the respondent on a Likert scale from 1 ("Never") to 5 ("Always"), or as "Unknown" or "Resolved". CPT: Current Procedural Terminology.
Response options ranged from 1 ("No disparities identified") to 5 ("Always").
Perceived success, patient satisfaction, and provider training needs: Sixty per cent of sites reported that families “often” or “always” expressed satisfaction with their telehealth experience and the average perceived patient and family satisfaction rating was 3.8 out of 5. Neurodevelopmental care providers also offered spontaneous feedback covering a range of themes related to their subjective experiences with telehealth, including an increased ability to reach at-risk patients, reduced no-show rates, and requests from parents for continued telehealth services after the pandemic due to the convenience afforded. Many respondents expressed an eagerness for opportunities to learn more about and develop or expand skills in telehealth-based neurodevelopmental evaluation and care. A substantial subset of respondents was willing to share information and help train providers at other sites to increase successes and reduce obstacles (see Supplementary Material 2).
We surveyed 30 cardiac neurodevelopmental programmes across North America within a 3-week period during the COVID-19 pandemic (22 July to 11 August 2020) and found that most sites (80%) were providing at least some clinical services via telehealth. All but one of these 24 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 via telehealth, and none had completed comparable testing with infants and toddlers. Moreover, a substantial subset of respondents expressed concerns about the validity and reliability of neurodevelopmental evaluation via telehealth, indicating a need for more data correlating in-person and telehealth evaluation results. In contrast, most sites were administering parent interviews and rating scales and providing feedback on test results using telehealth. While parent interviews and rating scales are a critically important component of neurodevelopmental evaluation for infants, children, and adolescents with CHD, these cannot replace the information gleaned from performance-based neurodevelopmental testing using standardised measures administered by trained professionals.
Concerns regarding various practical challenges associated with telehealth implementation highlight the need for collaborative efforts across congenital heart centres. The wide range of barriers identified in this survey, along with a strong interest in learning more about the use of standardised tests via telehealth, provided the catalyst for the Cardiac Neurodevelopmental Outcome Collaborative Telehealth Task Force to deliver a series of webinars to support its member institutions as they develop and expand telehealth services. These have been well-attended (172 registrations), and additional webinars are planned. Concerns regarding insurance reimbursement rates and the potential inability to serve out-of-state patients are well-founded, given the marked variability in reimbursement laws between states and the durability of limited pandemic-related changes to state laws. 7–9 Ongoing advocacy for adequate reimbursement will require understanding of how telehealth challenges may both contribute to disparities in access to care Reference Hirko, Kerver and Ford10,Reference Chunara, Zhao and Chen11 and facilitate access for some children and families who could not otherwise participate in an in-person visit. Telehealth may expand the footprint and breadth of cardiac neurodevelopmental care by bringing expertise to under-served, under-resourced, and remote communities and efficiently directing children and families to the most appropriate services. Recently, the American Academy of Pediatrics (AAP) Section on Telehealth Care, Committee on Practice and Ambulatory Medicine, and Committee on Pediatric Workforce advocated for telehealth as a means of decreasing disparities in access to care by extending paediatric expertise and best practices to children and adolescents no matter where they are located. Reference Curfman, Hackell and Herendeen12 This is limited, however, by our capacity to address barriers associated with language, digital literacy, special needs, and access to and payment for technology infrastructure. In addition, the AAP argue for payment for telehealth services at parity with the equivalent services provided in-person by private and public insurers. Such efforts are essential to avoid furthering disparities. Future research is needed to examine the specific ways in which telehealth mitigates or exacerbates disparities in access to and outcomes of neurodevelopmental care for vulnerable populations.
Limited data exist on the use of telehealth for cardiac neurodevelopmental evaluation and care. Most telehealth assessment studies with children thus far have focused on addressing unmet needs in rural or remote communities, such as in Australia, and have used a satellite clinic with additional personnel. Reference Hodge, Sutherland and Jeng13,Reference Hodge, Sutherland and Jeng14 While promising, preliminary data on the use of telehealth testing are extremely limited and very few studies have explored telehealth for neuropsychological assessment in the child’s home. Reference Harder, Hernandez and Hague15–Reference Peterson, Ludwig and Jashar18 Early motivation for telehealth psychology services was, in large part, associated with attempts to minimise barriers to care imposed by geographical distance. A pre-pandemic survey of parents of children with complex medical conditions identified geographical distance as a major barrier to access to behavioural health services and parents reported a clear preference for telepsychology initiatives over in-person therapy. Reference Williams, McDonald and Roberts19 Evidence supporting the feasibility, acceptability, and efficacy of telehealth-based psychological interventions for children and adolescents predates the pandemic Reference Wade, Oberjohn, Burkhardt and Greenberg20–Reference Comer, Furr and Miguel23 and indicates that telehealth can be as effective as in-person therapies, Reference Gloff, LeNoue, Novins and Myers24,Reference Osenbach, O’Brien, Mishkind and Smolenski25 with documented improvements in child behavioural and emotional outcomes, parent engagement and mental health outcomes, and family retention and satisfaction with treatment. Reference Thongseiratch, Leijten and Melendez-Torres26–Reference Jenkins-Guarnieri, Pruitt, Luxton and Johnson29 Benefits of telehealth also include potentially increased generalisability to the natural environment, with greater clinician access to challenges in the family environment resulting in more effective problem-solving or skills implementation. Reference Antonini, Raj, Oberjohn and Wade30 Elsewhere, our Task Force has proposed a conceptual model to illustrate how telehealth can be integrated within each stage of cardiac neurodevelopmental care, from intake to evaluation through to follow-up care and intervention, both during and post-pandemic. Reference Cox, Butcher and Sadhwani31
Strengths and Limitations
Strengths of this first survey of clinical services in cardiac neurodevelopmental programmes during the COVID-19 pandemic include the relatively high response rate and representation from a diverse group of providers (psychologists, physicians, nurses), sites (various surgical volumes), and locations across North America (Fig 1). The survey captured data on a broad range of clinical practices applicable across paediatric populations, including caregiver interviews, assessments for children of various ages, family feedback sessions, and child and parent intervention services. The survey findings have already been translated into educational practice, including the launch of a series of webinars hosted by the Cardiac Neurodevelopmental Outcome Collaborative for neurodevelopmental care providers and opportunities for ongoing learning and development between sites. Despite these strengths and the insights provided by the survey, several important limitations must also be considered. First, there are limited data on the validity and reliability of paediatric neurodevelopmental evaluation using telehealth. Any recommendations about current practices should be considered in this context and await empirical support from future studies. Second, the findings described may be specific to the period during which the survey was carried out. The COVID-19 pandemic has served as a catalyst for numerous positive changes in psychology practice, including the issuing of emergency licensure to facilitate out-of-state access to care and the relaxing of restrictions around insurance reimbursement for telehealth services. Many survey respondents did, however, voice concerns about the potential for future contraction of these changes and a high degree of uncertainty about the future of telehealth. Third, respondents predominately represented cardiac neurodevelopmental programmes located in the Northeastern region of the United States. Only four of the participating centres were located in the Western region of the United States and only one programme in Canada participated, limiting the capacity to generalise findings to other countries and health systems. Fourth, while the survey response rate was relatively high (71.4%) and robust in the midst of a pandemic, a notable proportion of Cardiac Neurodevelopmental Outcome Collaborative sites did not take part, potentially due to the rapid changes occurring in the clinical setting limiting time and capacity for survey engagement. It is possible, therefore, that the sample may over-represent sites with greater resources or more established cardiac neurodevelopmental follow-up programmes.
Future directions and recommendations
Across cardiac neurodevelopmental programmes, interest in and questions regarding telehealth are currently ubiquitous and the speed at which practices are adapting outstrips the ability to make scientifically supported recommendations. Reference Taddei and Bulgheroni32–Reference Koterba, Baum and Hamner35 Collaborative learning, opportunities for research and quality improvement projects, and knowledge dissemination in real-time are critical to ensuring we can rapidly respond to changing circumstances in the future. Research is needed to guide evidence-based recommendations regarding the practical administration of standardised psychological and neurodevelopmental assessments with children via telehealth. Studies are also needed to assess the validity and reliability of neurodevelopmental evaluation in the home via telehealth, including how results compare across settings (i.e., telehealth vs. in-person evaluation). Such studies need to take into consideration the social and psychological effects of the pandemic, changes to the school environment and potential effects on academic and cognitive development, patient and family risk and resilience factors, and variations associated with neurodevelopmental programme characteristics. Collection of data on neurodevelopmental outcomes, and on the validity and reliability of telehealth-based neurodevelopmental services could bolster advocacy efforts to increase insurance authorisation and reimbursement, and expand waivers for out-of-state licensing requirements for telehealth psychology services. While offering neurodevelopmental services via telehealth could improve access to care and broaden services in resource-limited settings, investigation into the balance between risks and benefits and reimbursement rates of telehealth for families and providers are needed. Mixed methods studies, combining qualitative and quantitative methodologies, should explore patient, family, and provider satisfaction with different approaches to cardiac neurodevelopmental care. In addition, multi-centre longitudinal studies should examine how the implementation of telehealth for neurodevelopmental screening and evaluation influences the total number of patients who access services, the percentage of at-risk individuals reached, the number of referrals for additional services or testing, as well as disparities in care and health outcomes.
The COVID-19 pandemic challenged institutions to rapidly transition to telehealth, and it is clear there is still much to learn to overcome challenges and leverage the progress made. Telehealth has the potential to augment the delivery of neurodevelopmental care for children and young people with CHD and other medical conditions; however, further study is required to broaden the evidence base and strengthen practice. Our survey showed that the implementation of telehealth-based neurodevelopmental services has presented novel challenges for providers and institutions in the areas of clinical practice, technology requirements, patient and family access to care, service reimbursement, and outcomes measurement. Despite these challenges, we found that most cardiac neurodevelopmental programmes we surveyed had pivoted to delivering at least some neurodevelopmental services via telehealth in response to COVID-19 and many of these sites expressed a strong interest in receiving additional training and resources or sharing their successes with others. We envisage that the Cardiac Neurodevelopmental Outcome Collaborative will play a role in guiding providers and institutions as they plan and adapt to new telehealth-based neurodevelopmental services. This coordinated effort across institutions will allow for an evidence-based, efficient, and consistent approach to the care of individuals with CHD and other medical conditions.
To view supplementary material for this article, please visit https://doi.org/10.1017/S1047951122000579
We gratefully acknowledge our colleagues and fellow providers from across North America, for their thoughtful responses to the survey, as well as the research assistance provided by Meghan Ford, Jennifer Hembree, Nicola Porter, Diya Sanghavi, and Jessica Cowin. We also thank the Cardiac Neurodevelopmental Outcome Collaborative Publications Committee, for insightful feedback on an earlier version of this manuscript.
Nadine Kasparian is the recipient of a National Heart Foundation of Australia Future Leader Fellowship (101229) and support from the Heart Institute Research Core at Cincinnati Children’s Hospital.
Conflicts of interest
The authors have no potential, perceived, or real conflicts of interest to declare. The first draft of the manuscript was written by Nadine Kasparian, Dawn Ilardi, Anjali Sadhwani and Renee Sananes, and all authors contributed to manuscript review and revision. No honorarium, grant, or other form of payment was given to any author to produce this manuscript.