Heart disease remains a leading cause of death in infants and children in the United States despite medical and surgical advances.Reference Mathews, Minino, Osterman, Strobino and Guyer 1 Most of these deaths occur in ICUs after prolonged hospitalisations, with over half occurring after withdrawal of life-sustaining treatments including mechanical ventilation and circulatory support.Reference Morell, Wolfe and Scheurer 2
Data from other pediatric populations suggest that the quality of physician–family communication influences the decisions that are made, including decisions about end-of-life care. Data from paediatric oncology suggest that parental understanding that their child is dying despite medical interventions lags behind physicians’; importantly, those parents who had an earlier understanding of the inevitability of their child’s death were more satisfied with their child’s end-of-life care.Reference Wolfe, Klar and Grier 3 Much remains to be explored regarding gaps in communication between physician and parents of seriously ill children. It has been shown, for instance, that physicians and parents often walk away from end-of-life discussions with different perceptions of what was discussed: physicians may believe they offered the possibility of limiting interventions, but parents say they did not hear this option.Reference Boss, Hutton, Sulpar, West and Donohue 4 Studies show that physicians may not explore parents’ understanding often enough, which can perpetuate diverging perceptions of a child’s prognosis and treatment options.Reference Boss, Donohue, Larson, Arnold and Roter 5 , Reference Boss, Lemmon, Arnold and Donohue 6
Despite the prevalence of, and mortality associated with, paediatric heart disease, we lack an evidence base to guide communication and decision-making with these families. There has been research into the importance of teamwork and shared decision-making within paediatric cardiology and cardiac surgery to optimise the care of these children,Reference Jacobs, Wernovsky, Cooper and Karl 7 and it is important to include families as members of the team. Notably, a recent systematic review confirmed that as many as 80% of parents of children with critical heart disease have symptoms of trauma, whereas another 25–50% have symptoms of anxiety, depression, and/or post-traumatic stress disorder.Reference Woolf-King, Anger, Arnold, Weiss and Teitel 8 Given data from a variety of patient groups suggesting that good communication with physicians can moderate parents’ long-term outcome,Reference Meyer 9 , Reference Snaman, Kaye, Torres, Gibson and Baker 10 there is a need for evidence-based, effective, family-centred communication techniques relevant to paediatric heart disease.
This systematic review summarises what is known regarding parent preferences for communication and decision-making for children with critical heart disease.
Methods
Using the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines,Reference Moher, Liberati, Tetzlaff and Altman 11 literature review was completed in February, 2018. Relevant electronic databases, including Pubmed (biomedical and life sciences literature), EMBASE (Excerpta Medica dataBASE), PsycINFO (psychology/psychiatry literature), and CINAHL (The Cumulative Index to Nursing and Allied Health Literature), were searched for English-language studies and dissertations with data about parents’ experience with communication and decision-making for children with high-risk/life-limiting congenital and acquired cardiac disease. Because research in this area is limited, there were no eligibility restrictions on study design, country of origin, single versus multi-institutional recruitment, or inpatient versus outpatient setting. We included studies of prenatal and postnatal communication because critical decisions about heart disease treatment are made during each of these times. Search terms were key words related to communication and decision-making in critical heart disease listed in Table 1. We also hand-searched the reference lists of all included articles to identify additional studies.
Table 1 Search terms.
Three word phrases were used, one from each column in all possible arrangements; “A” AND “B” AND “C”
The initial search yielded 10,247 papers. In a pre-selection process, one author (K.N.) screened titles and abstracts for content relevant to physician–family communication and decision-making for children with critical cardiac disease. In all, fifty-four articles and dissertations remained, and two authors – K.N. and E.P.W. – then performed full-text article review. A third author, P.K.D., adjudicated disputes regarding inclusion of individual articles. Figure 1 summarises study selection and articles excluded from full-text screening.
Figure 1 Summary of search strategy results.
Data extraction from included articles was performed by two authors – K.N. and E.P.W.. Information about study design, sample size, outcomes, and specific findings regarding physician–family communication and decision-making was recorded.
Results
A total of 10 studies – nine articles and one dissertation – met the inclusion criteria. Among them, six were conducted in the United States, three in Scandinavia, and one in Taiwan; all were published between 1996 and 2017. Table 2 summarises study characteristics. In all, four studies recruited families of hospitalised children, two recruited from outpatient cardiology clinics, two from fetal cardiology clinics, and two recruited both from inpatient and outpatient settings. Data collection for nine studies was through qualitative parent interviews; one study utilised parent surveys. The number of parents interviewed or surveyed ranged from 11 to 88 for a total of 255 parents. The majority of parents were mothers.
Table 2 Study characteristics.
CICU=cardiac ICU; HLHS=hypoplastic left heart syndrome
Outcomes from the 10 studies regarding physician–parent communication and decision-making in the context of paediatric heart disease can be combined into three themes: (1) amount, timing, and content of information provided to parents;Reference Arya, Glickstein, Levasseur and Williams 12 – Reference Lopez, Hanson and Yorke 15 (2) helpful physician characteristics and communication styles;Reference Carlsson, Bergman, Marttala, Wadensten and Mattsson 14 – Reference Wei, Roscigno and Swanson 19 and (3) reinforcing the support circle for families.Reference Bratt, Järvholm, Ekman-Joelsson, Mattson and Mellander 13 , Reference Lan, Mu and Hsieh 17 , Reference Higgins and Kayser-Jones 20
Amount, timing, and content of information that parents want
The most common theme that emerged from the 10 studies was that parents often wanted more information than they felt they received from physicians about their child’s disease and about the larger picture of what the prognosis meant for their child and family, including potential quality of life and physical and developmental limitations.Reference Arya, Glickstein, Levasseur and Williams 12 – Reference Carlsson, Bergman, Marttala, Wadensten and Mattsson 14 , Reference Kupst, Dresser, Schulman and Paul 21 More information, both verbal and written, almost always helped parents make decisions.Reference Arya, Glickstein, Levasseur and Williams 12 – Reference Carlsson, Bergman, Marttala, Wadensten and Mattsson 14 , Reference Kupst, Dresser, Schulman and Paul 21 Parents valued information that was timely and honest, including physician candidness about severity of disease and possibility of a shortened life or significant morbidities.Reference Arya, Glickstein, Levasseur and Williams 12 – Reference Carlsson, Bergman, Marttala, Wadensten and Mattsson 14 This was especially important for parents facing decisions regarding pregnancy continuation or during their child’s acute complications.Reference Bratt, Järvholm, Ekman-Joelsson, Mattson and Mellander 13 , Reference Carlsson, Bergman, Marttala, Wadensten and Mattsson 14 Information about what to expect before cardiac surgery, when surgery is planned, and helping the family visit the ICU to meet staff and ask questions helped parents prepare for future communication with new physicians.Reference Arya, Glickstein, Levasseur and Williams 12 , Reference Lopez, Hanson and Yorke 15 Updates during a surgical procedure helped reduce family anxiety and stress.Reference Lopez, Hanson and Yorke 15
For some parents, disclosure of all potential outcomes, including death, was overwhelming and made it difficult to hear additional information, if not permitted time to process.Reference Lopez, Hanson and Yorke 15 Given this, parents also appreciated repeated information over time.
Physician characteristics and communication styles
Parents described physician characteristics and communication styles that helped them through difficult times and decisions. The most common theme was that parents valued support and encouragement from physicians throughout the process of making serious decisions. Types of support that were meaningful included the opportunity to get to know treating physicians and to be able to contact physicians for follow-up questions;Reference Bratt, Järvholm, Ekman-Joelsson, Mattson and Mellander 13 , Reference Lan, Mu and Hsieh 17 both helped build a trusting relationship.Reference Lopez, Hanson and Yorke 15 – Reference Lan, Mu and Hsieh 17 Parents also trusted physicians who were present and mindful with families during difficult times and were relatable on a human level.Reference Wei, Roscigno and Swanson 19 , Reference Kupst, Dresser, Schulman and Paul 21 Parents appreciated when physicians cared not just for their child but for the whole family;Reference Brown 16 , Reference Wei, Roscigno and Swanson 19 in one study, home visitation by physicians promoted a trusting relationship, demonstrating to parents individualised care.Reference Lopez, Hanson and Yorke 15 Parents valued physicians who spent time exploring the parents’ experience and who tried to anticipate and validate parents’ emotions during difficult times.Reference Wei, Roscigno and Swanson 19 Families appreciated encouragement and guidance from physicians that their family could get through difficult situations.Reference Brown 16 , Reference Wei, Roscigno and Swanson 19
Parents also appreciated when physicians respected parents’ role, acknowledging that parents have their child’s best interest in mind and know them the best.Reference Wei, Roscigno and Swanson 19 Including parents in daily decisions and allowing them to participate in daily care in the hospital allows parents a feeling of control and recognises them as experts in their child’s care.Reference Lopez, Hanson and Yorke 15 They also valued when physicians recognised parents’ need to balance caring for the whole family when making decisions for their child.
Parent desire for physician recommendations was common in situations where treatment options included withholding/withdrawing of life-sustaining therapies.Reference Brown 16 , Reference Vandvik and Førde 18 Parents reported feeling shocked, exhausted, and overwhelmed by these complicated decisions; they were anxious for physician guidance in the face of such important choices.Reference Brown 16 , Reference Vandvik and Førde 18 Some parents felt they could not make decisions about limiting life-sustaining therapies without physician support, believing that such decisions should not be made by parents alone.Reference Brown 16 , Reference Vandvik and Førde 18
Reinforcing family support
The final theme was the need for physicians to reinforce parents’ support outside of the medical setting.Reference Bratt, Järvholm, Ekman-Joelsson, Mattson and Mellander 13 , Reference Lan, Mu and Hsieh 17 , Reference Higgins and Kayser-Jones 20 Parents often prized help from other family members when making serious decisions for a child; this support was strengthened when physicians facilitated inclusion of those family members in important discussions.Reference Bratt, Järvholm, Ekman-Joelsson, Mattson and Mellander 13 , Reference Higgins and Kayser-Jones 20 In multiple studies, parents appreciated when physicians arranged for parents to meet other families who have experienced similar situations, especially families of children with comparable diagnoses.Reference Bratt, Järvholm, Ekman-Joelsson, Mattson and Mellander 13 , Reference Lan, Mu and Hsieh 17
Key take-home points for physicians regarding communication and decision-making with families of children with heart disease are shown in Table 3.
Table 3 Communication and decision-making take-home points.
Discussion
As medical and surgical interventions continue to improve outcomes for paediatric heart disease, there will always remain a group of patients whose conditions will lead to chronic disability and/or early death. The tension between the risks and benefits of escalating interventions for those children means that there may be no consensus about “best” treatment options. Our professional obligation in these situations is to help families understand what they most want to achieve, and avoid, in the course of their child’s care. To do this well, physicians need more than ad hoc counselling skills – we need a robust evidence base regarding family’s needs to inform our approach to communication and decision-making. We performed this review to summarise what is known about family preferences regarding communication and decision-making in the context of critical paediatric cardiac disease. Our most important finding was that the data regarding parent experience are sparse, a concerning deficit in light of disease prevalence and severity. The existing data do offer several indications of what approaches physicians can take when counselling families and joining with them in serious decisions.
First, many parents want more, and timelier, information in order to understand and plan for the future. Because none of the existing studies included recorded family–physician conversations, we cannot assess what families were told; we only know what they feel they heard. Multiple studies show that intense emotions reduce a person’s capacity to hear and process cognitive information;Reference Kralik, Visentin and Van Loon 22 , Reference Jones and Carter 23 physicians should expect and plan for this likelihood when counselling families of very sick children. Parents in the studies we reviewed made it clear that sharing information about their child’s illness, potential treatments, and prognosis should be repeated, in different formats, to reinforce and clarify the essential information. Studies in other populations demonstrate the value of a coordinated, interdisciplinary approach to sharing information – families may hear information more clearly from nurses who spend more time with the family and seem less intimidating than physicians. Importantly, parents’ desire for more information extends well beyond short-term details of expected cardiac interventions: parents want doctors to talk about what to expect from the future, to help them prepare for the “big picture” of their child’s prognosis for quality and length of life. This reflects data from parents of children with other critical illnesses, who report that this “big picture” information is very valuable to families but is often missing from physician counselling.Reference Lemmon, Donohue, Parkinson, Northington and Boss 24
Second, parents were clear that they need more than just information from physicians. Parents whose fetus or child has a life-threatening diagnosis are profoundly stressed and distressed. Physicians can add to this distress if they limit their interactions with families to simply relaying medical information without acknowledging and processing how this information is life-changing for the whole family. Other studies of parents of very ill children suggest that physicians can be supportive in different ways – by physically or verbally sharing their own emotions, by specifically articulating how hard this is for the family, by pausing to allow space for intense emotion – all approaches that take little physician time and build family trust.Reference Boss, Hutton, Sulpar, West and Donohue 4 Third, parents in several of the studies we reviewed wanted treatment recommendations, especially during high-stakes decisions about life-sustaining therapies. Although collaborative decision-making is the ethical gold standard in paediatrics, physicians may instead relegate all decision-making authority to parents, for instance by simply providing information and asking parents to make the decision. Data from other paediatric populations suggest that a minority of families want to be in complete charge of high-stakes medical decisions for their child; most want physicians to share or even direct the decisions.Reference Meert, Eggly and Pollack 25 , Reference Brinchmann, Forde and Nortvedt 26 Therefore, while there is often a physician reluctance to make treatment recommendations,Reference Bastek 27 the fact that many parents find them helpful suggests that physicians should consider including these more commonly in their counselling of families.
Finally, parents in the reviewed papers appreciated physician guidance in accessing sources of support. Families were often discussing their child’s illness and care with extended family and friends, and relied on those people to help them shape their values related to their child’s care. This process was strengthened when physicians included important support people in medical discussions when possible. Parents also found it extremely helpful when physicians facilitated meetings with other parents who had been through similar situations with their child. Most departments have ongoing relationships with parents who have made variable treatment choices; a process for connecting willing families with other families could be an important support.
The findings of this systematic review also highlight that the research on communication and decision-making in the paediatric cardiology population is very limited, and the field is in need of rigorous studies to answer the question of how best to facilitate communication and decision-making between the medical team and families of children with critical heart disease. We are currently conducting a multi-centre study to evaluate how parents of children with cardiac conditions experience their child’s illness and medical treatment and how clinicians can help meet their needs, from diagnosis to end-of-life care. We hope this research will help define how clinicians and families can work as a team to improve the care of children with critical heart disease.
Financial Support
This systematic review received no specific grant from any funding agency or from commercial or not-for-profit sectors.
Conflicts of Interest
None.
Acknowledgements
The authors acknowledge the assistance of Sandra DiBrito, Blair Anton, and Kelly Goles.
