Partnerships for safe care: A meta‐narrative of the experience for the parent of a child with Intellectual Disability in hospital

Abstract Objective To systematically identify and synthesize peer‐reviewed qualitative evidence of the parental experience of hospitalization with a child with Intellectual Disability. Search strategy Key words, synonyms and MeSH subject headings that related to the three key concepts of parental experience, children with Intellectual Disability and hospital settings were applied to six electronic databases: Medline, CINAHL, Embase, PsycINFO, Scopus and Web of Science. Titles and abstracts of publications between January 2000 and February 2019 were screened for relevance. Inclusion criteria Empirical qualitative research involved participants aged 0‐18 years, involved children with Intellectual Disability, involved participants hospitalized as an in‐patient and involved participants focused on parent perspective. Data extraction and synthesis Data were extracted and synthesized using a meta‐narrative approach. Results Eleven publications met the inclusion criteria. Data synthesis revealed three research traditions contributing to this meta‐narrative: Paediatric Nursing Practice, Intellectual Disability Healthcare and Patient Experience. A total of five themes were identified: (a) being more than a parent, (b) importance of role negotiation, (c) building trust and relationships, (d) the cumulative effect of previous experiences of hospitalization and (e) knowing the child as an individual. Discussion and conclusion This review presents a working model for professional‐parent partnership for the safe care of children with Intellectual Disability in hospital. Shifting paediatric healthcare to whole of hospital/multidisciplinary models of care that centre on the child will necessitate partnerships with the parent to identify and manage the needs of the child with Intellectual Disability, in order to achieve safe and equitable care for these children.


| INTRODUC TI ON
Equitable, high-quality and safe healthcare is the aspiration of healthcare systems globally to in order to achieve optimal patient outcomes; yet, despite concerted efforts over the past two decades, rates of avoidable harm have remained largely unchanged. 1 Knowledge of patient experiences is increasingly recognized as critical to inform health systems regarding avoidable harm in healthcare delivery. 2 Children are especially vulnerable to avoidable harms during hospitalization, predominantly those resulting from communication and medication errors. 3 Yet, data regarding patient experiences amongst paediatric populations are challenging to collect and are often interconnected with parental or carer experience. 4,5 In the paediatric healthcare context, parents and carers are routinely used as proxies to obtain patient experience data. [5][6][7] Certain paediatric populations have high healthcare utilization and may be exposed to increased risk. 8 Children with Intellectual Disability (ID) are one such population, 9 with emerging evidence which indicates that children with ID are particularly susceptible to avoidable harm in their care. 10 Vulnerability to avoidable healthcare harm compounds the existing health inequities experienced by these children. 11 Hospital staff rely on the presence of parents and carers to attend to the needs of children with ID. 10,12,13 Being frequent users of healthcare, these parents or carers are therefore often more familiar with the health system and services than the general paediatric population. As such, parents of children with ID are uniquely positioned both in the role that they undertake and to report their observations of hospitalization. Reliable methods to collate patient experience data from children with ID are sparse except through proxy measures such as parents. Thus, parents are a valuable source of healthcare experience data for enhancing the experience of healthcare for children with ID and their parents or carers. 14,15 Parental experience of caring for a child with ID has been explored through the lens of several health disciplines, but exploration of parental experience from the quality and safety perspective is missing. This review aimed to identify evidence to date of the parental experience of hospitalization with a child with ID with regard to care quality and safety, and provide a consolidated narrative evidence synthesis.

| ME THODS
An initial scoping review of the literature identified a small number of key studies from a diverse range of research traditions with comparable findings, which would be ideally synthesized using a meta-narrative approach. The meta-narrative uses an iterative approach to the search strategy and aims to tell a story of the evolution of research into a specific tradition and its disciplines over time. 16 The Realist and Meta-narrative Evidence Syntheses: Evolving Standards (RAMSES) study standards 17 were employed for conducting and reporting this review.

Types of studies
Studies available in English and published since 2000 were eligible for inclusion. The year 2000 is contemporaneous with the study of two seminal healthcare Q&S texts, 18,19 which stimulated growth in the study of patient experience from the healthcare quality and safety perspective.

Participants
Parents or carers of children (<18 years of age,aligning with the United Nations definition of child. 20 ) with ID in hospital as inpatients. This could include either a specific condition known to include ID, such as Down syndrome, or terms that are synonymous with ID such as cognitive impairment, learning disability or developmental disability. 21

Study design
Qualitative study designs and data are used to understand complex phenomena involving human interactions such as experiences of healthcare delivery, meaning they are ideal for capturing data on healthcare experience. 22,23 Outcomes Parent/carer-reported experiences of hospitalization, or any other terms referring to subjective measures of inpatient healthcare. The parental experience in hospital with a child with ID could be described using any of the following terms: satisfaction, experience or reporting quality of care.

| Exclusion criteria
Studies focussed on children with Autism only were excluded where the participants did not also have ID. 24 Studies of short stay contexts manage the needs of the child with Intellectual Disability, in order to achieve safe and equitable care for these children.

| Study selection and data extraction
Title and abstract screening was conducted by the author, and a copy of the full text was obtained for those studies potentially eligible for inclusion. Inclusion criteria were applied to these studies and discrepancies resolved through discussion with research supervisor. Data extracted included author(s), study year, country, health service setting, participants, study design, main focus and key findings.

| Assessment of study quality
The Critical Appraisal Skills Programme (CASP) qualitative research checklist was used for the assessment of study quality. 27 Each study was assessed for qualitative rigour against the ten CASP criteria and scored on a 3-point, 0-2 scale (No-0, can't tell-1, Yes-2) for a maximum score of 20 indicating a very high-quality study. One author (LM) assessed all the studies, and uncertainties were resolved through discussion with another author (RH).

| Data synthesis
Initial scoping searches of key electronic databases found small pockets of research in this area scattered amongst the broader research fields of nursing practice, disability healthcare and patient experience. With a small yet heterogeneous group of studies emerging, it was determined that data synthesis using a meta-narrative was the best approach. A meta-narrative is suited for sense-making of phenomena as studied through different research perspectives and is presented as an evolutionary story of the topic. 16 Each of the six phases of a meta-narrative (planning, search, mapping, appraisal, synthesis and recommendations) is guided by six principles: pragmatism, pluralism, historicity, contestation, reflexivity and peer review. 16 Applying these principles during each phase, the included studies were reviewed and research traditions and academic disciplines identified by one reviewer (LM). Summaries of F I G U R E 1 Example of search strategy in MEDLINE 11. limit 10 to (english language and yr = "2000 -Current" and "all child (0 to 18 years)") how each research tradition was conceptualized across the included studies were completed. Each study was appraised individually before framing the data through narrative synthesis.

| RE SULTS
A total of 1005 titles were identified from database searches, 932 after duplicates removed. From relevant journal searches, 289 titles were identified. After title screening, 22 were retained from the database searches and eight retained from journal searches for abstract or full-text review. Of these, five studies from the database search and four studies from journal searches were included. Hand searches of the reference lists of these studies identified a further two studies for inclusion. A total of eleven studies were included in this review (see Figure 2 for PRISMA flow diagram of study selection 28 ).

| Characteristics of included studies
A summary of the studies included in this review is provided in Table 1. The 11 included studies reported findings from 10 unique data sets. Three studies were from the United Kingdom, two each from Canada and Sweden and one from each of the following: Australia, Norway, Switzerland and the United States. Two studies each discussed themes derived from a single data set. 29,30 Two studies used mixed methods 15,31 with only findings from qualitative analysis included in this review.
Of the 11 included studies, nine used semi-structured or in-depth interviews for qualitative data collection, one used parent focus group interviews, 32 and one used open-ended questions embedded in a questionnaire. 15 Methods for the analysis of interview data included discourse analysis, 29,30 thematic analysis, 4,12,33 content analysis 15,31,32,34 and hermeneutic analysis. 13 One study did not specify the analytic strategy. 14 The studies identified were published in the last 10 years, with one study from 2008, 33 two published in 2009 12,14 and the subsequent eight studies published from 2013 onwards.
Of the studies reviewed, eight recruited participants from a single site, one recruited from two sites, 4 one identified participants through relevant user organizations, 32 and one recruited from the Australian Rett Syndrome Database. 15 Participant selection and recruitment was based on a recent admission to hospital with their child. All studies included parent/carers as study participants. A total of eight studies specified the participants as mothers, fathers or both; in three of these studies, participants were mothers only, 29,30,32 three studies included either parent, 14,31,34 and two studies included either or both parents. 4,13 Participants were primarily mothers, but four studies included data collected from children with ID and/or healthcare staff. 4,12,29,30 These data were not included in this review.
Diagnosis was included in only five studies 14 Records excluded (n = 12) Full-text articles assessed for eligibility (n = 18) Full-text articles excluded, with reasons (n = 9) Studies included in qualitative synthesis (n = 9) Studies included in synthesis (meta-narrative) (n = 11) Studies from reference lists included (n = 2) sclerosis, spastic quadriplegia with mental retardation, hydrocephalus, myelomeningocele, epilepsy, spinal muscular atrophy and multiple congenital anomalies. A further two studies described the participants' children as having communicative disabilities. 4,32 Quality of the included studies varied, ranging from moderate (11/20) to very high quality (20/20); six studies scored 16 or above (see Table 1). Discussion of researcher reflexivity was inconsistent across the studies, and responder validation was lacking. Due to the low number of studies identified, study quality was not an inclusion criterion.

| Findings
A total of three research traditions contributed to this meta-narrative: Paediatric Nursing Practice, Intellectual Disability Healthcare and Patient Experience. Though these traditions are different, each reflects the broader perspective and principles of family-centred care (FCC).
The research traditions and their conceptualizations of the hospital experience for parents of a child with ID are summarized in Table 2.
All studies were conducted by healthcare professionals with the nursing perspective leading or contributing to nine of the 11 studies. Of the studies reviewed, four were conducted from a nursing perspective only 29,30,33,34 with the remaining seven studies including researchers from other health disciplines such as medical, psychology, physiotherapy, speech pathology and occupational therapy. 4,[12][13][14][15]31,32 Through the review process, findings from the eleven studies were consolidated into five themes; being more than a parent, importance of role negotiation to reduce ambiguity about the role of the parent, building trust and relationships through effective communication, the cumulative effect of previous experiences of hospitalization, and healthcare staff (HCS) taking time to know the child as an individual. The themes crossover, they interrelate to tell the story of the parental experience over time, and how the interactions between the themes highlight the importance of partnerships in care to deliver safe care for children with ID. The five themes are detailed below:

| Being more than a parent
Parents consistently reported that HCS relied on their constant presence, assuming parents would take on multiple roles and provide the necessary care for the child with ID. Coupled with the stress of hospitalization, this impacted on the burden of responsibility for parents in several ways.
They relied on us like another member of staff. Parents consistently reported the burden of responsibility for making decisions on behalf of their child; parents felt guilty for consenting for treatment that subjected their child to both pain and discomfort 13,15,31 or that their decision could be wrong. 13,14,34 One parent expressed torment about the legitimacy of their decision: Did I take the right decision?
Two studies identified this burden of responsibility and ensuing guilt created a tension for parents. 13,15 HCS reliance on parental presence created a sense of helplessness and vulnerability for the parent, 13,31,34 or for their child. 31 For some parents, the burden was overwhelming and contributed to feelings of guilt 15,30 and chronic sorrow. 33 Parents wanted HCS to recognize that the parent was in need of care and support as well. [12][13][14]31,33,34 In two studies, parents reported that the perceived expectation to be constantly available was a significant burden 13,34 :

| Importance of role negotiation for shared care in the context of ambiguity
It is tough to be given the responsibility; we have to stay awake all the time.   4(p748) and this may contribute to parents of children with ID feeling stereotyped as difficult by HCS. 30 Past experiences increased their anxiety about having negative experiences during the next admission, 12-14 and in one study, this contributed to a sense of 'chronic sorrow' for these parents 33(p12) .

| Building trust and relationships through
Past experiences in which there was an apparent lack of continuity of care during a hospital admission also impacted on parental perceptions and expectations of care provided for their child during hospitalization. Parents reported feeling anxious about their child's safety during hospitalization when the nurse did not know their child, 12,32 or HCS were not prepared for their child's hospitalization. 12 Parents reported that inconsistency of HCS led to poor pain management and a lack of information sharing during their child's hospitalization, 4,13,15,32 lessened their confidence in the HCS 15 and impeded partnerships in care. 4,14 Conversely, continuity of HCS, especially those already known to the parent and/or child from previous hospitalizations, had a positive impact on the hospital experience 4,12,14,15,29,32,34 : …The times we've come in and it's been great that's when we see nurses that we know, or with whom everything works well, and who understands the child.
And when the doctor who knows the child best is on duty and everything works.
Parents identified sharing of expertise and knowledge was important for trusting HCS with their child. 4,14,[29][30][31]34 Being included in the care of their child during hospitalization was an opportunity for shared learning with HCS, 4,14,29,30,32,34 and for continuity in the care, they would provide at home. 14

| Healthcare staff taking time to know the child as an individual
The importance of HCS and organizations recognizing the child with ID as an individual with unique needs during hospitalization was identified across most studies. 4,12,14,29,30,32,34 When HCS did not take the time to get to know their child, the parent perceived their child was marginalized 29,30 or unnoticed by HCS. 4

| Partnerships in care
The value of partnerships between parents and HCS to help care for and make decisions about their child's care needs during hospitalization permeated each research tradition. [13][14][15]29,32,34 Parents wanted to work in partnership with HCS when making decisions about their child's care, as they cannot be objective. 13,34 Parents expressed value in partnerships with HCS to help care for and make decisions about their child's care needs during hospitalization. [13][14][15]29,34 Two studies highlighted that parents viewed participation in the study as an opportunity to be heard, talk about their opinions and share their experiences of hospitalization with their child with ID. 13,33 Based on the findings of this review, we propose a conceptual model of how these five themes may interact in practice to support the development of partnerships between HCS and parents to deliver safe care for children with ID in hospital. This proposed conceptual model is presented below in Figure 3.  Reliance on parents compounded their existing burden and contributed to an overwhelming responsibility of caring for a child with ID, and similar findings have been described by others. 35,37 The parental need for support, role negotiation and partnerships in care have been consistently reported in the paediatric healthcare literature across a variety of populations and settings. 7,37-40 Espezel and Canam (2003) note that it may be that current healthcare environments do not facilitate the parent-nurse rapport and subsequent relationship development that precedes a perception of a partnership. 41 It is essential for healthcare staff to demonstrate empathy, compassion and kindness to engage children and their parents in true partnerships that recognize personhood. 39 Where healthcare delivery is not person-centred, the child is not viewed as an individual while receiving healthcare. This may lead to unnecessary suffering and dehumanization of the child. 42 Furthermore, a recent review found that people with ID, irrespective of their degree of self-awareness, do not consider their ID as a critical component of their self-identity. 43 Keeping the child and their individual needs at the centre of the care experience acknowledges the child has intrinsic value, a humanness and personhood, aligning with principles of person-centred care. 44

| Implications
Models of paediatric healthcare that centre on the child and their healthcare needs will inherently include partnerships with parents, while maintaining focus on the personhood of the child. Such a change will necessitate a systems-wide approach to improvements such as health policy 31 and enhanced undergraduate education for healthcare professionals. 30 Yet to shift values and beliefs around the personhood of the child with ID would necessitate broader social and health system changes. 30 While governing health bodies around the world promote inclusion and reasonable adjustments for people with disabilities in hospital, HCS may lack the necessary understanding, capabilities and resources to implement changes. 45,46 With our conceptual model, we argue for achieving safe and equitable healthcare for children with ID is the goal, realized through partnerships in care and founded on HCS reducing the parental burden through role negotiation, using effective communication to build trust and relationships, recognition of previous poor experiences and getting to know the individual needs of the child with ID. This model will be tested in future qualitative studies.

| Limitations
We have identified several limitations of this meta-narrative, which may impact the generalizability of our findings. Firstly, the literature on this topic, while seeming to come from different research traditions, was inclined towards the paediatric nursing discipline. Most studies spoke to the parental experience with nursing staff, though for this review the term HCS encompasses any clinical disciplines providing acute care within the inpatient hospital setting. Patient experience studies of inpatient care will unavoidably overreport aspects of nursing care as nurses are the key contact for patients.
However, this means these findings cannot be generalized to encompass the healthcare experience outside the inpatient setting.
Another limitation is that participants were chiefly the child's mother, meaning fathers and other family members or caregivers are underrepresented in the research. While an overrepresentation from mothers is to be expected, this has been previously identified by others as a potential bias. 40 Recommendations to researchers include making conscious effort in study design and recruitment strategies to minimize this potential bias by using participant enrolment methods that are unlikely to favour mothers and may capture a broader range of perspectives from all carers involved. 40 Where stated, the diagnoses of some participants, such as myelomeningocele and spinal muscular atrophy, were not specified to include ID. As this was a small number of children, the majority of children in each study had ID, and results were similar across the included studies to those of other parents, this is unlikely to have confounded the findings.
Finally, by limiting the included studies to English, some excluded non-English studies identified during searches of the reference list may have been relevant. With a small number of studies included in this review, it is possible that this has impacted the generalizability of these findings in non-English speaking settings and future studies would benefit from including this perspective.

| Conclusion
This meta-narrative describes a clear need for healthcare staff to develop partnerships in care with parents for there to be safe care for children with ID in hospitals. This starts by negotiating care and shared learning to lessen reliance on parental presence, building trust and relationships to identify the needs of the child with ID and their parent, understanding the impact of previous negative experiences of hospitalization and using rapport to get to know the child as a person.
Models that centre on the child and their healthcare need to include negotiating care roles and partnerships with parents, while maintaining focus on the child. Shifting beliefs about the optimal models of paediatric healthcare will necessitate a systems-wide approach to change the broader social and cultural perceptions of the value of people with ID.