Navigating the healthcare system with my child with CHD: parental perspectives on developmental follow-up practices

Background: Parents of children with CHD face several barriers when trying to access the services needed to support their child ’ s development. In fact, current developmental follow-up practices may not identify developmental challenges in a timely manner and important opportunities for interventions may be lost. This study aimed to explore the perspectives of parents of children and adolescents with CHD with respect to developmental follow-up in Canada. Methods: Interpretive description was used as a methodological approach for this qualitative study. Parents of children aged 5 – 15 years with complex CHD were eligible. Semi-structured interviews that aimed to explore their perspectives regarding their child ’ s developmental follow-up were conducted. Results: Fifteen parents of children with CHD were recruited for this study. They expressed that the lack of systematic and responsive developmental follow-up services and limited access to resources to support their child ’ s development placed an undue burden on their families, and as a result, they needed to assume new roles as case managers or advocates to address these limitations. This additional burden resulted in a high level of parental stress, which, in turn, affected the parent – child relationship and siblings. Conclusions: The limitations of the current Canadian developmental follow-up practices put undue pressure on the parents of children with complex CHD. The parents stressed the importance of implementing a universal and systematic approach to developmental follow-up to allow for the timely identification of challenges, enabling the initiation of interventions and supports and promoting more positive parent – child relationships.


Abstract
Background: Parents of children with CHD face several barriers when trying to access the services needed to support their child's development. In fact, current developmental follow-up practices may not identify developmental challenges in a timely manner and important opportunities for interventions may be lost. This study aimed to explore the perspectives of parents of children and adolescents with CHD with respect to developmental follow-up in Canada. Methods: Interpretive description was used as a methodological approach for this qualitative study. Parents of children aged 5-15 years with complex CHD were eligible. Semi-structured interviews that aimed to explore their perspectives regarding their child's developmental followup were conducted. Results: Fifteen parents of children with CHD were recruited for this study. They expressed that the lack of systematic and responsive developmental follow-up services and limited access to resources to support their child's development placed an undue burden on their families, and as a result, they needed to assume new roles as case managers or advocates to address these limitations. This additional burden resulted in a high level of parental stress, which, in turn, affected the parent-child relationship and siblings. Conclusions: The limitations of the current Canadian developmental follow-up practices put undue pressure on the parents of children with complex CHD. The parents stressed the importance of implementing a universal and systematic approach to developmental follow-up to allow for the timely identification of challenges, enabling the initiation of interventions and supports and promoting more positive parent-child relationships.
Children with a complex CHD requiring open heart surgery in infancy are at high risk of developmental delays that may affect multiple developmental domains including motor skills, language, cognition, behaviour, and academic skills, which can arise at different time points during childhood and adolescence. [1][2][3] Although developmental difficulties are often mild to moderate, their frequency remains high, and they are associated with activity limitations at school, in selfcare, and in community participation. 3,4 Thus, in recent years, the improvement of developmental outcomes has become a priority for both the clinical and research communities. Improvement of developmental outcomes is dependent on a system that provides timely identification of developmental delays and subsequent referral for interventions. The effectiveness of these interventions is well supported in the literature on other high-risk infant populations, especially when implemented in a timely manner. 5 There is also growing evidence of the effectiveness of early interventions in the CHD population. 6,7 Moreover, children and families can be offered support, resources, and strategies to functionally adapt to some challenges that cannot be remediated. 8 In an effort to optimise the developmental trajectories of children with CHD, the American Heart Association released a statement emphasising the importance of systematic follow-up services for all children with CHD. 9 This statement indicated that follow-up care for high-risk children with CHD should include surveillance (monitoring of parents' concerns over time), screening (questionnaires), and formal evaluations. The Cardiac Neurodevelopmental Outcomes Collaborative recently proposed to expand the follow-up strategies recommended by the AHA by suggesting new key time points for re-evaluating outcomes at 6, 18, and 36 months, 5, 8-9, 10-11, 13-14, and 18 years as well as additional assessment and screening tools. 10,11 However, studies conducted in various countries have reported on the presence of systemic barriers that limited the implementation of these recommendations. [12][13][14] In Canada, only half of the tertiary care centres that perform open heart surgery have structured developmental follow-up programmes for children with CHD. 12 As a consequence, important opportunities for interventions to enhance outcomes may be lost.
A recent study has shown that parents of children with CHD highly value ongoing medical and developmental monitoring. 15 However, no study to date has specifically explored developmental follow-up experiences, needs, and preferences of families of children with CHD. Therefore, it remains unclear if the services offered meet the needs of families. Hence, the aim of this study was to explore the perspectives of Canadian parents of children and adolescents with CHD with respect to the developmental follow-up of their child.

Materials and method
Design This qualitative study used interpretive description as a methodological approach. This methodology is designed to develop an understanding of human experiences that recognises each experience as constructed and contextual and is applicable to clinical contexts. 16

Sample and procedure
Parents of children aged 5-15 years with CHD requiring openheart surgery before 2 years of age and who had received health services in Canada were approached to participate in this study. Parents whose children were born prematurely or with genetic conditions were not eligible because these factors presumably affected the extent and type of developmental follow-up received. Participants were recruited through support groups, associations for families of children with CHD and flyers available in the Cardiology Divisions of selected children's hospitals across Canada. Participants were purposively recruited, from the pool of interested participants, to capture users' experiences across childhood, to represent different geographical locations (urban and rural), children from both sexes. Recruitment ended when no new themes were identified in two subsequent interviews.
Questionnaires including the age and sex of the child with CHD, geographical location, socio-economic information, and developmental challenges encountered were emailed to the families. Parents were asked to return the completed questionnaire prior to their interview. Semi-structured interviews were conducted by videoconference or telephone (see Interview Guide, Appendix A). Interviews were conducted in French or English. Informed consent was obtained from each study participant.

Analyses
Interviews were transcribed verbatim and the transcripts were analysed using NVivo 11 software (QSR International). Data were coded using both inductive and deductive coding. The deductive codes were based on the interview guide questions. Comparative analysis was then used to find commonalities and differences in parents' experiences. This method allowed us to discern commonalities between participants' experiences and preferences while acknowledging individual care experiences and their complexity. New conceptualisations of the parents' perspectives on the developmental follow-up of children with CHD were then developed. Finally, quotes that best represented the data collected were selected and the interpretations were developed. French quotes were translated by the first author. Data collection and analysis took place concurrently in an iterative manner. This allowed for tentative interpretations to be discussed in subsequent interviews, to allow participants to expand, clarify, and/or elaborate on the proposed interpretations.

Results
A total of 38 parents contacted us to participate in the study. Six were not eligible because they did not meet the inclusion criteria. Purposive sampling, using the criteria described above, was performed amongst the remaining 32 parents. We selected fifteen parents of children with CHD now aged 5-14 years (mean 9.4 years) to participate in the study. All interviews were conducted with parents who self-identified as mothers of children with CHD. In three cases, fathers were also present for part of the interview. Interviews lasted an average length of 42 minutes (range: 22-95 minutes). Participants' children were born with a wide variety of CHD diagnoses requiring open-heart surgery and families lived in different regions and had variable socio-economic characteristics (Table 1). All but one child with CHD experienced challenges in one or more of the following surveyed domains: academic performance (n = 10), behaviour (n = 9), gross motor (n = 9), fine motor (n = 9), cognitive (n = 7), and language (n = 6) skills. Four parents also reported that their child with CHD had sleep or sensory disturbances. In terms of developmental follow-up, 12 of the 15 parents had experienced screening and/or formal evaluation for their child at some point, and three had access to surveillance only.
Although parents of children with CHD expressed gratitude and were very satisfied overall with the care their child had received for their cardiac condition, most of them voiced concerns regarding the developmental follow-up they received. Two main themes provided an overview of the perspectives of parents on their child's developmental care: A. Perspectives on current developmental follow-up care: Limited accessibility from identification to intervention; B. Increased parental burden: Struggling to fill the gaps in developmental follow-up while seeking to establish a sense of normalcy.
Perspectives on current developmental care: Limited accessibility from identification to intervention Lack of systematic and responsive developmental follow-up services across childhood Even if most parents reported having access to a formal evaluation for their child through public services, private resources, or research studies, many parents expressed concerns with the absence of a systematic approach to developmental follow-up for all children with CHD at risk of developmental delays.
"I wish it was more just universal, that all kids get referred into whatever the program is in their community, at least for a primary assessment. In my ideal world, that is just more standard of care as opposed to something that you have to wait for a problem to be discovered and know how to navigate the system to get there : : : ." (Participant 8) Parents also voiced disquiets with regard to the responsiveness of current practices to formally identify the developmental delays or challenges their child was experiencing. They expressed that, sometimes, when they brought up concerns with their healthcare professionals, they preferred to wait and see if the problems their child was facing would resolve by themselves. In these situations, they felt that they had to insist on obtaining a referral for further assessment or intervention. Therefore, most parents, especially first-time parents, stressed the importance of systematic developmental follow-up at key timepoints throughout childhood. Conversely, two parents, including one whose child did not experience challenges, expressed that those formal evaluations can sometimes be stressful for both the child and the parents and that adding additional hospital visits can also be overwhelming, especially in the first years of life. In addition, one parent who was part of a research project found that using multiple questionnaires that often overlapped was both frustrating and time-consuming for them.
Parents were aware and sensitive to the lack of resources in the healthcare system. In response to this barrier, parents suggested a tiered approach where they could start with screening questionnaires and follow up with a formal evaluation only if the questionnaire raised concerns. Nevertheless, they insisted that an in-depth assessment of their child's challenges was essential before school entry. Furthermore, some participants identified the importance of a centralised follow-up process to facilitate the circulation of information between professionals to avoid having to describe difficult moments they had gone through on multiple occasions.
Supporting developmental needs: From community resources to education for parents and educators Parents referred to intervention services were often confronted by the scarcity of resources both in the healthcare and educational systems. Participants reflected that the subtleness of challenges associated with their child's CHD and the absence of formal diagnoses for their developmental challenges may contribute to the difficulties in accessing the resources.
"We squeaked until we were able to get something [resources] because they often overlook kids that [have moderate difficulties], because they aren't as bad as other kids, or they feel that other kids are in more need of, and it's the kids that are in need but just kind of on the borderline that fall through the cracks". (Participant 10) The presence of waitlists for resources was also problematic, especially when referrals were not made in a timely manner. For families who lived further from large urban centres, the limited accessibility to professionals was even more striking.
Some parents had access to resources they appreciated. Parents from two provinces (British Columbia and New Brunswick) mentioned they could self-refer to a developmental follow-up programme or rehabilitation services for the first 3-5 years of life depending on the province. Other parents had access to community services to support their child development. For example, one parent had a weekly inclusive physical literacy class offered by students from various backgrounds such as kinesiology and physiotherapy, through a programme affiliated with their local college. Community services such as swim therapy were also found to be beneficial by parents of children with CHD. Finally, parents suggested that providing them with the tools and strategies to best support their child's development would be valuable.
Increased parental burden: Struggling to fill the gaps in developmental follow-up while seeking to establish a sense of normalcy Parents of children with CHD expressed that they experienced high levels of stress. Initially, their stress was caused by their child's heart condition, the diagnosis, perinatal circumstances, and surgery. However, after this critical period, most parents began feeling anxious about their child's development. Many participants explained that the limitations in current developmental followup practices resulted in increased parental burden and stress as they had to fill these gaps if they wanted their child to receive the care they needed. Since developmental follow-up mostly took the form of surveillance, parents felt that the responsibility of identifying delays rested on their shoulders. Some parents identified that they had lower expectations for their child with CHD. Conversely, other parents felt that they were constantly keeping Parents also expressed that they did not have the same opportunity to spend quality time with their child as a baby due to the stress they were experiencing, a decreased sense of empowerment and their child's critical health. One parent highlighted that the education on the medical management of their child they received completely eclipsed the education on the general infant care first-time parents typically receive.
"My son was our first child, so they forgot the normal things you need to do with a baby and the normal steps. Your child is so sick that at first it is like survival, but they must not forget that you are going home anyway with a baby, you have to take care of this baby. I need the basics. The other parents, they showed them how to wash their babies before they go home. We haven't been shown anything. I knew how to give her injections and gavage, but all the normal things were forgotten. [ : : : ] I find that there is this side there that they should not forget also, the human being." (Participant 5) Given the limited access to resources, parents often became advocates for their child to acquire the services they needed. The lack of systematic and centralised follow-up systems meant that parents had to navigate the healthcare system to find the resources they needed to support their child. Parents also felt they had to take on the roles of case manager to coordinate their child's appointments and ensure information was communicated to all professionals. The interviewees reported an overall sense of being overwhelmed by time and energy required by the multiple roles and the added responsibilities that they must take on. Some mothers even had to stop working given the amount of time required for these additional responsibilities. The parents expressed that these challenges not only have a profound impact on them, on their child with CHD, but also on the overall family dynamics and on other siblings. A number of parents shared how important community resources such as support groups with other parents of children with CHD are during these difficult times.

Discussion
Participants in this study stressed the importance of implementing systematic, accessible, and responsive developmental follow-up services, where information on the child and available resources is centralised and accessible across Canada. Without such a system, the burden of identifying delays and adequately supporting the needs of children with CHD falls on the shoulders of the parents, increasing their stress and ultimately impacting the entire family.
As the healthcare focus for children with CHD expands beyond survival to improving developmental outcomes, it is important to ensure that family-centered care is authentically used across institutions. To our knowledge, this is the first study to report on the impact of the current gaps in services for children with CHD on the families and to describe parental perspectives of optimal follow-up for children with CHD in Canada. Systematic developmental follow-up is required to identify challenges in a timely manner so that the resources can be put in place to support development, thus avoiding gaps in continuity of care. Although current guidelines 9-11 recommend formal evaluation at various time points during childhood, some parents suggested a tiered approach in which formal evaluation would be used only when screening results indicate suspicion of delays rather than systematic formal evaluations at all key time points. This approach has been successfully implemented in other countries 13 and could offer a cost-effective alternative to ensure that challenges are identified early before they have long-term consequences on the child. This process may also be less stressful and burdensome for the parents of children who experience fewer challenges and for youth with CHD. Hence, a thorough analysis of current barriers to implementation of systematic developmental follow-up approaches and a reflection on how the recommendations could be adapted to different contexts is warranted.
Parents were also concerned with the limited availability of resources to stimulate their child's development or interventions for identified difficulties both in the healthcare and school systems. This need for enhanced support throughout childhood and adolescence has also been reported by youth with CHD. 17 Enhancing the accessibility of resources within the school system needs to be formally examined in future studies. Nevertheless, the parents have identified various strategies that rely on education, community resources, telehealth, and other technologies that could be put in place to support their child's needs.
The participants in this study reported a heavy burden related to managing the care of their child with CHD. Parents expressed that they had to assume new roles such as case managers, administrators, or advocates, as a direct consequence of the current gaps in developmental follow-up practices. This is line with a previous Swiss study in which parents reported feeling exhausted from additional responsibilities with regard to the neuromotor development of their infant with CHD. 18 The increased burden resulted in increased levels of stress for participants in our study. This could explain, in part, the increased level of parental stress, anxiety, and depression reported in previous studies. 19,20 Studies have shown that parental stress is associated with the child's cognitive ability and behaviour. 21,22 Hence, this study further supports the need for screening for parental mental health and access to psychological and social supports for their child to be included as part of family-centered care. Those supports should include education on parental self-care and personal resilience as well as the promotion of healthy child development and supports for successful social participation. 23 Furthermore, the lack of quality time with their child and a decreased sense empowerment and self-efficacy was described as having an impact on the parent-child relationship by study participants. This could result in changes in parenting styles and decreased attachment between the mother and child. 24 Finally, welcoming a child with CHD may be associated with changes in family functioning. 25,26 A recent systematic review reported psychosocial well-being to be negatively impacted in 40% of siblings of children with CHD. 27 Thus, assessment and support for siblings also need to be considered. The paediatrics post-intensive care syndrome model can provide a framework for comprehensive family-centered care. 28 This study presents some limitations. Indeed, our sample consisted almost exclusively of mothers and may not represent the views of fathers of children with CHD. Nevertheless, the mothers often described the experience they had as parents or as a family and some fathers were present during a portion of the interviews. In addition, despite our far-reaching recruitment strategy, we could not enroll participants from all 10 Canadian provinces; therefore, potential gaps and strategies that exist in provinces from which we did not have participants may have been missed.

Conclusion
The limitations of current developmental follow-up practices put undue stress and burden on Canadian parents of children with complex CHD. The parents stressed the importance of implementing a universal and systematic approach to developmental followup to allow for timely identification of challenges, enable initiation of interventions and supports, and promote more positive parentchild relationships. It is now essential that we identify strategies to facilitate the implementation of systematic approaches to developmental follow-up across the Canadian provincial healthcare systems.