The life experience of parents of Congenital Heart Disease adolescents: A meta-synthesis

Introduction: Congenital Heart Diseases (CHD) do not preclude the possibility to become adult due to the different innovations in medical and surgical treatments. The transition from childhood to adulthood is a complex process in the life of CHD adolescents, considering the consequences of their diseases and the need to be adherent with their follow-up indications. In this process, parents play an important role, being a landmark for their children, but the experiences of CHD adolescents’ parents are little studied and the literature about their life experiences appears fragmented. Knowledge of life experience of CHD adolescents’ parents is important to address a tailored and effi cient health-care delivery for the whole family. Therefore, the aim of this study is to synthesize qualitative papers of life experience of CHD adolescents’ parents. Methods: A meta-synthesis study using Noblit and Hare’s interpretative meta-ethnography approach was conducted. Databases searched included PubMed, CINAHL, PsycINFO and Google Schoolar and keywords used are “Congenital heart disease”, “Parents”, “Adolescents” and “life experience”. The search process was performed in accordance with the PRISMA guidelines and only the qualitative papers in the last 20 years were included. Studies resulted by search process were critically appraised using the Critical Appraisal Skills Programme qualitative research appraisal tool. Results: The search yielded 386 potentially relevant studies for screening, and only six articles met all the inclusion criteria. In accordance with the seven phases suggested by Noblit and Hare, the papers were analyzed, discussed and a qualitative meta-synthesis was performed. The meta-synthesis results showed four main themes, exploring also four main contradictions that characterize the life experiences of CHD adolescents’ parents: “fear and uncertainty of the future versus positive coping strategies”; “parents hyperresponsibility and overprotection versus adolescents’ independence desire”; “desire to give support, but not to be supported”; “normality desire versus awareness to live with particular conditions”. Conclusions: The role of the CHD adolescents’ parents is diffi cult and they experience some contradictions. This study explore their life experiences in a preliminary way, but further analysis and studies are needed. Research Article The life experience of parents of Congenital Heart Disease adolescents: A meta-synthesis Federica Dellafi ore1*, Roberta Domanico2, Serena Francesca Flocco3, Francesco Pittella4, Gianluca Conte1, Arianna Magon1, Massimo Chessa3 and Rosario Caruso1 1Health Professions Research and Development Unit, IRCCS Policlinico San Donato, San Donato Milanese (MI), Italy 2Renal medicine, Southend University Hospital NHS Foundation Trust, Southend-on-Sea, England, UK 3Pediatric and Adult Congenital Heart Disease Center, IRCCS Policlinico San Donato, San Donato Milanese (MI), Italy 4Nursing Degree Course, University of Milan, section of IRCCS Policlinico San Donato, San Donato Milanese (MI), Italy Dates: Received: 08 March, 2017; Accepted: 16 May, 2017; Published: 17 May, 2017 *Corresponding author: Federica Dellafi ore, Health Professions Research and Development Unit, IRCCS Policlinico San Donato Via Agadir, 20-24, 20097 San Donato Milanese (MI), Italy, Tel: (+39) 02 52774940; E-mail:


Background
Congenital heart diseases (CHD) are one of the most prevalent and serious birth defects, representing a major global health problem. With a prevalence of 9.1 for 1000 live births [1], the CHD are the leading causes of birth defectsassociated morbidity, mortality, and medical expenditures [2].
Currently, 1.3 million children live with a CHD worldwide [3] and approximately 90% of them can survive into adulthood [4], due to the advances in diagnosis, medical technology, surgical interventions and treatments. It is estimated that about 2800/ million adults are currently living with CHD [5], which need a lifelong follow-up [6].
The transition from childhood to adulthood is a complex process for every young person, being particularly important for CHD adolescents. In fact, the adolescence is a crucial phase for the formation of the personality [7], during which the CHD adolescents have to face with the consequences of their disease and the need to be adherent with their follow-up indications [10]. Moreover, during the transition into adulthood, CHD adolescents could be exposed to many psychological issues related to their development of self-identity, self-esteem and self-image [8] and they should learn about their disease, overcoming frustration and anxiety and developing self-care strategies [9]. For this reason, the recent clinical research focused to explore the peculiarity of the transition period synthesis. Arch Nurs Pract Care 3(1): 031-037. DOI: http://dx.doi.org/10.17352/anpc.000022 from childhood to adulthood in patients with CHD, in order to develop the purposeful transition care models, which are aimed to provide self-management and to satisfy the medical, psychosocial, educational, and employment needs of CHD adolescents [1,8].
In this process, parents play a key role, being a landmark for CHD adolescents [10]. In fact, the CHD adolescents' parents are extensively involved in care activities, such as accompanying their child to visits, always staying with them for the entire time and administering their medications every day [11]. Moreover, they could be considered to be an integral part of team members for their children [11], supporting them to develop their independence and to promote their responsibility [12].
Currently, there is a lack of information about the experiences and dilemmas of parents during the CHD transition process from childhood to adulthood. Recent fi ndings seem to highlight that it is not easy to be a CHD adolescents' parents, because they experience anxiety, stress, and depression [13], often feeling uncertain about their roles during transition [14]. However, knowing the experiences of CHD adolescents' parents is important to address tailored and effi cient healthcare delivery for the whole family. Therefore, the aim of this study is to synthesize qualitative papers of life experience of CHD adolescents' parents.

Review method
A literature review with meta-synthesis was undertaken to address the following research questions:  What are the experiences of CHD adolescents' parents?
 Which factors infl uence the life of CHD adolescents' parents?
Meta-synthesis is a relatively new technique for examining qualitative research [15]. The literature shows different approaches and techniques types to conduct a meta-synthesis [16]. Noblit and Hare's [17], meta-ethnographic approach was used to guide this meta-synthesis, subsequently adapted for health services research by Britten et al. [18]. This metasynthesis method includes 7 phases (Table 1), that overlapping as the synthesis proceeds: phase 1 -getting started; phase 2deciding what is relevant to the initial interest; phase 3 -reading studies; phase 4 -determining how the studies are related; phase 5 -translating the studies into each other; phase 6 -synthesizing translations; phase 7 -expressing the synthesis [17].

Search strategy
In accordance with phase 2 [17], a systematic search was performed in electronic databases including MEDLINE, PsycINFO, CINAHL, and Google Scholar, in December 2015. Before the begin of the systematic search process, a pilot test was carried out in electronic databases to exclude that a metasynthesis on CHD adolescents parents was already performed and to identify the key words. The relevant key terms used to shape the queries were as follows: 'Congenital Heart Disease', 'Transition' 'adolescents', 'parents', 'Life experience'. Indeed, the search comprised four separate search queries based on thesaurus terms, free-text terms and broad terms relevant to topics searches: (1) population-related terms (Parents, Parenting, Caregivers, Family); (2) transition phase -related terms (Adolescents, Youth, Transition phase, Transition); (3) illness -relation terms (Congenital Heart Disease, Congenital Heart Defects); (4) outcomes-related terms (Life Experiences; Perception; Life Change Events; Quality of life, Health related quality of life, Experience, Life). Afterwards, the search terms were combined using the Boolean terms 'OR' and 'AND' and further combined using free text search. The time limit was set to 1995-2015, because the advanced cardiac surgery was developed and the CHD adolescents there was not before to 20 years ago. Other search limits were the English language and the full text availability.

Inclusion and exclusion criteria
The review included primary qualitative studies about the experiences of CHD adolescents' parents. The studies identifi ed by queries was selected considering the following inclusion criteria: (a) qualitative studies, (b) published between January 2005 and December 2015, (c) written in English, (d) available full text, (e) focus on parents of CHD adolescents, and (f) explore their life experiences. Studies with other outcomes, with theoretical or quantitative design, in languages other than English were excluded.

Search process and quality appraisal
The search process was conducted by two authors (DF and DR) independently. The authors have discussed frequently to reach agreement on the search process and appraisal. The procedure to select the articles during search is summarized in the fl ow diagram, according to the PRISMA fl ow-chart (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) [19], as shown in Table 2. The depth literature search, performed using the key terms, led to identify 386 articles (identifi cation phase). In the screening phase, 246 articles were excluded using the inclusion criteria, such as time limit, Getting started. Identifying a topic of intellectual interest and the background of the research themes that the qualitative research informs. Defi ning the aim.

Phase 2
Deciding what is relevant to the initial interest. Including relevant studies, describing the search strategy and criteria for inclusion and exclusion Phase 3 Reading studies. Repeating reading of the studies noting their interpretative metaphors.

Phase 4
Determining the relationships between the studies. Reading the fi ndings of the primary studies and extracting metaphors, concepts and themes and their potential relationships in the primary studies (fi rst-level analysis), which ends in an assumption of their relationship being reciprocal (fi ndings across studies are comparable), reputational (fi ndings stand in opposition to each other) or as representing a line of arguments (second-level analysis).

Phase 5
Translating the studies into each other (fi rst-level synthesis). Comparing metaphors and their interactions within single studies and across studies and at the same time protecting uniqueness and holism. English language, title and abstract selection and deletion of duplicate. The articles were reduced to 20, and assessed for eligibility (eligibility phase). Those articles were assessed using the Critical Appraisal Skills Programme qualitative research appraisal tool [20]. Thus, after full-texts reading, only six articles were included (included phase), excluding 14 records due to they did not meet CASP assessment (poor methodology) or they did not focused on our research questions (lack of relevance), as shown in Table 2. Also data extraction and critical appraisal were undertaken independently by two researchers (DF and DR) and then discussed until to reach consensus.

Results
The included studies in this meta-synthesis are summarized in Table 3. Two studies were conducted in USA, one study in Canada and three studies in Europe. Four included papers were related to the adolescent with heart defects and CHD, while two studies focused on adolescent with chronic illness, where there also described a sub-sample of CHD adolescents. The data collection in all the studies was conducted through semistructured interview or narrative interview, and data analysis were made by content analysis, phenomenological hermeneutic method, thematic analysis or grounded theory procedure.
The six identifi ed papers were analyzed by the two authors (DF and DR) (phase 3 of Noblit and Hare approach) to determine the communalities between studies and to integrate the themes emerging from each one (Phase 4 of Noblit and Hare approach) [21]. Afterwards, the same authors identifi ed new concepts and then four new themes were developed (phase 5 of Noblit and Hare approach). Those four themes were related to the synthesis of the data referred to experiences of CHD adolescents' parents (phase 6 of Noblit and Hare approach), representing the meta-synthesis results.
Thus, each theme explored four main contradictions that characterize the life experiences of CHD adolescents' parents: "fear and uncertainty of the future versus positive coping strategies"; "parents hyper-responsibility and overprotection versus adolescents' independence desire"; "desire to give support, but not to be supported"; "normality desire versus awareness to live with particular conditions". Therefore, the continuous experience of contradictions lived by parents during the transition phase of the adolescents affected by CHD is the main underpinning the meta-synthesis. Nevertheless, parents and adolescents may have different perceptions: parents could typically be anxious, whereas adolescents display a wait-and-see attitude [22,23]. A detailed description of new four themes are presented in the following sections, supported by data from the original studies.

Theme 1: fear and uncertainty of the future versus positive coping strategies
This theme described mainly the parents' fear about their own son's future, in term of working environment and social life, and their will to face at best the congenital heart defect: this disease is a huge part of everyday lives and parents seem to be uncertain about their roles during transition and how to promote their children [11]. "A parent never wants to have a kid die before her and that is what she ( [24].
Although the predictability of their child's future is no more uncertain than for a child without CHD, the parents struggled with the uncertainty of what impact CHD would have on their adolescent's life [25]. During infancy and early childhood, most of the parents had been counseled about their child's limited life span, leaving them to deal with the challenge of uncertainty. Mark's father said of the early years, "You just never know when you're going to wake up and fi nd if he's alive or not. It was terror." John's mother remembered, "For a while I was afraid to get attached, because I sort of felt I might lose him" [25]. This often has led the families to live experiences that they would not live: "It has caused us to do things in our lives at a diff erent point than we would have otherwise. For example, we went to Disney World when Zoe was seven and Rick was three. We said that 'they are well now, and we do not know how long they will be well for, so let's go now.' So there are things we have done in our lives, because we wanted to make sure that they had the opportunity to do those things when there were no health issues" [24].
The parents feel helpless and afraid, because their child cannot fulfi ll his own dreams and ambitions as their healthy peers: they are wondering how hard is to push their child to excel were areas of considerable concern for parents of adolescents.
As their children progressed through adolescence into young adulthood, parents wanted their children to go to college, obtain stable jobs, move from home, and become independent.
Faced with these fears and uncertainties some families have developed some approaches to cope with the future and with the illness, such as some parents have tried to put the disease into background and live as normal as possible. Other parents have instead tried to put aside uncertainties concerning the future, focusing on daily-based fl exible problems scheme. An example given concerned the management of smoking as a

Theme 2: parents' hyper-responsibility and overprotection versus adolescents' independence desire
This theme described the contrast between the parent's inclination in taking charge of adolescent's responsibilities and their wish of getting their own children more independent. Parents expressed ambivalence about whether it was appropriate to encourage their child to prepare for the working world. Jane's mother said: "I'd like to see her graduate from eighth grade and high school. I really would like to see her have the skills she needs to pursue what she would like to do in life... I guess the health I can't really worry about, because! Can't do anything about it…." [25]. The transition phase included the transfer of responsibility from pediatric care to the adult one, and represented the period in which parents start to give more autonomy in decision making to the young adult. Some families consider this situation as a diffi cult and intense period, and they would like that their child will never grow up; but, at the same time, they realize how this transfer is considered as a fundamental step, in which they need to release responsibility and independence to their children [26,27].
Parents' natural desire is to act as a parent: they feel the need to be involved in the care of the adolescents and feel entitled to responsibility for their adolescents' health. They grieve the experiences that their sick children are missing because of their heart defects and mourn the tough school years that they must undergo. They also express concerns that the world would not treat the young adult fairly because of CHD [25]. Parents become aware that their children were no longer children and this could give a sense of sorrow in not being needed anymore. At the same time, this new and unknown condition could represent for parents a sort of a relief to shift responsibility.

Theme 3: desire to give support, but not to be supported
This third theme described the parents' need to help their child with CHD and the relationship of the family with the care providers. In order to support the teenager, a parent seeks security and force by the care providers, who understand the clinical and emotional situation that the families and young must face every day. The parents feel safe when the relationship with the clinical staff is based on solid trust: the greatest desire is to feel accepted and understood not only by pediatric care but also adult's one. Especially the latter has to provide assistance to parents, which now fi nd themselves lost and looking for advice to better deal with the transition process and move to the new adult health care setting [14,28]. "It felt good because, in the position I was in, I needed all the information I could get, even though I might not have understood the whole thing at once. It felt good to be involved in it and not be pushed away." [28].
Parents know how much is important the care providers' participation, but at the same time, they underline how much their role is crucial. In fact, they are aware to be needed to their children, which may not be able to face this new reality alone.
In fact, a mother of 18 year-old boy says: "Just because he is a grown up, they can't expect that he knows everything..." [14]. For this reason the parents are willing to accept the help offered by care providers and they start to be condescending towards them in order to be considered and therefore more helped. The parents consider themselves a strong and supportive fi gure to their own children [25] [14].
Parents' desire is to receive the most possible clinical information they can, even if these latter are frightening [28]. When parents have not been informed about the medical condition of their children, they begin to feel confusion, suffering and helplessness: in this situation they need to fi ght for their own children, in order to support them in the best way possible [28].  don't expect her to live a year" [26].
Parents would like to be considered as a normal family: despite copious clinical visits, surgeries, or unexpected health deterioration of health, some parents attempt to adapt themselves to the new life, "normalizing" as much as possible, as suggested by health care providers [26]. Others parents, instead, tend to make a comparison between their own children and healthy peers, because schools and society are the fi rst to defi ne the adolescents affected by CHD as" sick" and then different [24,25] That wouldn't be fair towards the other kids.' So, we got out of that school." [25].
Parents suffer thinking about the experiences their children are losing [28]: they perceive that CHD adolescents feel alienated and treated differently by teachers and coaches [29] and feel that the illness has affected all family life, even in the smallest aspects. In the articles by Gantt (2002) a mother states: "We used to go camping before he was born. We never went again. Not as a family. Not like we did with the other two kids." [26].
Parents also ask themselves how to inform family and friends about their children's disease; others use instead the disclosure of the disease to vent themselves and to be considered, after all, normal [25].  According to the recent guidelines ACC/AHA for the management of patients with CHD [30], the process of transitioning should prepare young patients for successful transfer to an adult healthcare provider at a later time, and it should begin since the age of 12 [31]. Furthermore, the transition care models have to orient and to encourage a sound healthbehavior, considering how those patients could be exposed to many psychological issues related to their development of selfidentity, self-esteem and self-image [8].

Discussion
Moreover, heavy medical expenses [32] and frequent hospitalizations [33], bring CHD adolescent patients and their family members to other types of complications, from the burden of the disease on the whole family [34], to issues related to the unknown future, treatment program or prognosis. For these reasons, the understanding of CHD parents' needs is crucial to face the diversity and complexity of adolescents with CHD, guiding the whole care delivery.
As many studies have already shown, parents of children with CHD showed lower well-being compared to parents of healthy children [35]. These fi ndings are amplifi ed when children with CHD will grow up. For all these reason, the literature shows that the parental role during the transition phase is extremely diffi cult, being the parents a landmark for their children. They live feelings of helplessness, they are unable to relate and understand the needs of their sons, and consequently parents of CHD adolescents have a decreased well-being compared to parents of healthy adolescents.

Limitations
This meta-synthesis was developed based on data collected within original studies on the experiences of CHD adolescents' parents. Therefore, this study's quality is largely a product of the quality of data collected in the original studies chosen for meta-synthesis. A strength of the current study was that the two authors conducted their reviews independently and are compared for confi rming data. While the integration of different individual's perspectives and experiences has not altered the focus of this study, it produced a more complete picture of the study phenomenon. On the contrary, the inclusion of only six qualitative papers is the main limitation of this meta-synthesis, which is not intended to be comprehensive or cumulative. In other words, the results of this meta-synthesis could help to frame the peculiarities of adolescents with CHD and parents, addressing future empirical studies and even the necessity to develop specifi c tools to measure each identifi ed peculiarity.