The Perceptions and Experiences of Parents and Families of Preterm Infants Who Participate in Family Integrated Care Programs in Neonatal Intensive Care Units: A Qualitative Systematic Review Protocol

The premature birth of an infant coupled with admission to a Neonatal Intensive Care Unit (NICU) can pose complex psychosocial challenges to parents and the family unit. Measures designed to counteract the effects of restricted visitation and prolonged separation from the infant within the NICU setting have led to the development of the Family Integrated Care model; emphasis in this program is placed on the active participation of the parent alongside the healthcare team in providing direct care to the preterm infant. The aim of this systematic review is to explore the perceptions and experiences of parents and families of preterm infants who have used a Family Integrated Care program during hospitalisation in a NICU. A systematic literature search will be performed on six electronic databases (Medline, CINAHL, Embase, PsycINFO, Web of Science, and Scopus) to identify relevant articles which meet the a priori inclusion criteria. A hand-search of the reference lists of all articles taken to full text review will supplement the search. The search will be limited to peer-reviewed, primary research articles written in English only; nil publication date restrictions will be applied. Two independent reviewers will screen all retrieved articles, perform the full-text review, critically appraise the included articles, and extract the data. Where consensus cannot be reached, a third reviewer will be consulted. To assess the quality of included studies and the risk of bias, the Critical Appraisal Skills Programme checklist will be used. Data will be extracted using a data extraction tool developed by the review team and piloted for use by two independent reviewers. A thematic analysis and narrative synthesis of the review ndings will follow. Family


Abstract Background
The premature birth of an infant coupled with admission to a Neonatal Intensive Care Unit (NICU) can pose complex psychosocial challenges to parents and the family unit. Measures designed to counteract the effects of restricted visitation and prolonged separation from the infant within the NICU setting have led to the development of the Family Integrated Care model; emphasis in this program is placed on the active participation of the parent alongside the healthcare team in providing direct care to the preterm infant. The aim of this systematic review is to explore the perceptions and experiences of parents and families of preterm infants who have used a Family Integrated Care program during hospitalisation in a NICU.

Methods
A systematic literature search will be performed on six electronic databases (Medline, CINAHL, Embase, PsycINFO, Web of Science, and Scopus) to identify relevant articles which meet the a priori inclusion criteria. A hand-search of the reference lists of all articles taken to full text review will supplement the search. The search will be limited to peer-reviewed, primary research articles written in English only; nil publication date restrictions will be applied. Two independent reviewers will screen all retrieved articles, perform the full-text review, critically appraise the included articles, and extract the data. Where consensus cannot be reached, a third reviewer will be consulted. To assess the quality of included studies and the risk of bias, the Critical Appraisal Skills Programme checklist will be used. Data will be extracted using a data extraction tool developed by the review team and piloted for use by two independent reviewers. A thematic analysis and narrative synthesis of the review ndings will follow.

Discussion
This review will importantly describe the experiences of parents and families of preterm infants using a Family Integrated Care program delivered within a NICU; review ndings will be used to inform the associated program protocols in a Perth (Western Australia) based NICU and be disseminated via a peerreviewed publication.

Background
The clinical care of the preterm infant in Neonatal Intensive Care Units (NICUs) has historically been provided by medical, nursing, and allied healthcare providers (1). Whilst this specialised care has provided a structured approach which aligns with advancements in technology, the intensity of the NICU environment and the acuity of the infant, coupled with restricted visitation, prolonged separation and minimal physical contact, has resulted in a broad range of poor health outcomes for parents. For example, delayed infant attachment and bonding, high parental stress, and an increased risk of postnatal depression and post-traumatic stress disorder (1)(2)(3)(4). From a mother's perspective, the premature delivery and loss of the third trimester can be emotionally traumatic and physically challenging; their role following the preterm birth is often ill-de ned which can potentiate a degree of vulnerability and isolation as they navigate the hospital system. For the preterm infant, the impact can also be signi cant with evidence of behavioural issues, disability, neurodevelopmental delays, inadequate or variable weight gain, higher rates of nosocomial infection and increased rates of hospital re-admission often observed (1,2,5).
In the 1970s, measures designed to advance the clinical outcomes of NICU standard practice provided to preterm infants were rst observed in Tallinn, Estonia (6). In this revolutionary model, all non-technical infant care was performed by mothers. These guiding principles have led to the development of the Family Integrated Care program (FICare) which acknowledges the signi cant and critical contributions of parents in improving the health outcomes of the preterm infant (1,2,7,8). The integration of parents into the direct care of the preterm infant is broadly based on a shared-care philosophy where parents are viewed as active partners with the healthcare team; emphasis is placed on the physical interactions that occur between the parent and infant, and the relationships formed between the healthcare team and parents (8). To support parents and family members throughout the preterm infant hospitalisation and in preparation for hospital discharge, clinical staff provide ongoing education within a safe environment to enable a successful role transition to that of primary caregiver following hospital discharge (2, 3, 8).
Stelwagen et al. (4) described the ability of parents to develop the skills required to care for the preterm infant following discharge, con dence and control in in uencing the health outcomes of the infant and having insight into the infant's condition as critical factors of FICare program success. Conversely, where self-e cacy was challenged due to power struggles between parents and staff, participants felt that this considerably impacted their con dence.
An alternate model often delivered in the NICU is the Family Centred Care (FCC) program. The FCC program (6, 7) has subtle differences to FICare, which are mostly seen in the degree to which parents are actively involved in providing direct care to the infant; the FICare program requiring more direct involvement. FCC programs are also not conceptually well understood in terms of how the parents are embedded into the care protocols (6, 7,9). With this understanding, our review will only consider literature that describes the involvement of parents or family members who have participated in a NICU FICare program only, and not FCC.
A review of the published literature based on NICU FICare programs revealed that most studies were observational or randomised controlled trials (RCTs) that focussed on improving the health outcomes of the infant (1,2,8). Commonly reported outcome measures in these studies included NICU length of stay (LOS), rate of hospital re-admission, infant weight gain and duration of breastfeeding. From a qualitative perspective, there has been a few studies that have narratively described the experiences of parents and families of preterm infants who have participated in FICare programs (1,2).
A preliminary search of PROSPERO registered protocols was conducted on 19 March 2021 to determine the extent of work undertaken in the proposed review area. Broad search terms were used to identify systematic review protocols of similar content. The search revealed six registered review protocols which were examined for similarity. However, none were found to be comparable to our proposal or based on a qualitative interpretation of perceptions and experiences. All review protocols identi ed in PROSPERO reported quantitative measures only to determine and synthesise outcome ndings, and two protocols excluded qualitative studies from the search. The Cochrane library was also examined on 25 March 2021. The search terms used to review work undertaken in this area were, 'Family Integrated Care in NICU'. The search did not retrieve any registered Cochrane reviews, but 31 trials. A screening of the trials via title only, identi ed one article which required further exploration. However, this item was not a published systematic review or protocol. Following a comprehensive review of PROSPERO and the Cochrane Library, the originality of our proposed qualitative systematic review was con rmed.
The overall aim of the proposed review is to explore the experiences of parents and families of preterm infants participating in a FICare program delivered in a NICU. The review has three primary objectives: To explore the perceptions and experiences of parents and families of preterm infants who are participating in a FICare program delivered in a NICU; To describe the facilitators and barriers experienced by parents and families of preterm infants participating in a NICU FICare program; and To appraise and synthesise qualitative ndings to better understand where improvements to program protocols could potentially be made.

Review Question
What are the perceptions and experiences of parents and families of preterm infants who participate in Family Integrated Care Programs in Neonatal Intensive Care Units?

Methods
This systematic review protocol was submitted for registration with the International Prospective Register of Systematic Reviews (PROSPERO submission ID # 243818) and will be reported in accordance with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses Protocols (PRISMA-P) statement (see checklist in Appendix 1) (10). A PRISMA ow diagram will display the total articles retrieved throughout each stage of the review, describe reasons for exclusion and document total articles submitted for nal data extraction.

Study design
The current review will consider studies that feature qualitative evidence only and include, but not limited to, qualitative and mixed methods research, focus groups, phenomenological studies, ethnography, and grounded theory research.

Participants
There will be no restrictions applied to the age, ethnicity, or gender of participants. The participants should meet the a priori inclusion criteria: parents, families, guardians or caregivers of a preterm infant who have used or are participating in a NICU FICare program. Within the description of the families, siblings, grandparents and aunts and uncles will also be included. For the purposes of this review, a preterm infant is classi ed as a being born < 37 weeks of age. As the FICare program is typically offered to all infants in a NICU setting, we will include studies that report that at least 50% of the infants included in the study, are preterm infants.

Phenomena of interest
This review will consider studies that explore the perceptions and experiences of parents and families of preterm infants who are participating in a NICU FICare program. We will include studies where the authors have de ned the NICU program they deliver as FICare. Studies will be excluded if they have investigated the perceptions and experiences of families participating in an FCC program or any other NICU integrated model of care, which is not described as being FICare.

Search strategy
A systematic search strategy using a three-stage approach will be undertaken using the SPIDER tool framework for qualitative evidence (sample, phenomenon of interest, design, evaluation, research type) (11). Following the identi cation of key concepts and relevant search terms, a review of google scholar and the use of an online thesaurus will be performed to identify commonly used terms and synonyms.
Boolean operators, speci c lters (English only and peer-reviewed articles) and truncation for terms and phrases will then applied to enhance the search strategy. Speci c Medical Subject Headings (MeSH), index terms and controlled vocabulary will be applied to six individual bibliographic databases: Medline (Ovid), CINAHL (EBSCOhost), Embase (Ovid), PsychInfo (EBSCOhost), Web of Science, and Scopus (Elsevier). No publication date restrictions will be applied, and articles written in English only will be accepted. To supplement the review, a manual hand-search will also be performed on the reference lists of all articles included in the full text review (Stage 3). A draft search strategy for Medline is provided in Screening of studies Following the electronic database search, all retrieved articles will be imported into the online data manager system Covidence, and duplicates removed (12). The titles and abstracts will be screened by two independent reviewers. Articles will be excluded if they do not meet the a priori study design criteria, do not include preterm infants in a NICU setting or if the study intervention is not described as FICare. If there is uncertainty about the intervention used, a full-text review will be conducted. Where resolution cannot be reached concerning article inclusion following title/abstract review, consultation with a third more senior reviewer will occur.
All articles meeting the inclusion criteria will be submitted for full-text review. These will be screened by two independent reviewers and reasons for exclusion of full-text studies presented in a PRISMA ow diagram. For studies where disagreement occurs, discussions will be conducted between the two reviewers until consensus is reached. Where consensus cannot be reached, consultation with a more senior third reviewer will take place to adjudicate a nal decision.

Data extraction
A standardised data extraction tool will be developed by the review team and piloted for use by two independent reviewers for all eligible studies. The data to be extracted from each of the included studies will include: study title, rst author, year of publication, country of origin, study design, sample size, sample demographics, setting, data collection methods, documentation of the FICare program used, and key ndings including experiences, perceptions, barriers and facilitators.
If additional content data is available, we will consider qualitative ndings based on groups of people who experience higher than average preterm births. These may be classi ed by socioeconomic factors, Indigenous status, low-and middle-income countries, and mothers who have increased risk factors (e.g. high alcohol or other drug use) or mental health concerns.

Quality appraisal
To assess the quality of study ndings, assess the risk of bias and critically appraise each eligible study article, the Critical Appraisal Skills Programme (CASP) checklist (13) will be used by two independent reviewers. Where disagreement occurs in the appraisal, discussions will be conducted between the two reviewers; a third more experienced reviewer will be used to reach consensus for cases where resolution cannot be achieved. The ndings of the critical appraisal will be presented in table format.

Data analysis and synthesis
The research articles taken to full text review will be imported into NVivo for data management and to provide structure during the analysis (14). A six-stage thematic analysis of all data will then be performed by two reviewers (15,16). Thematic analysis is a useful iterative method for contextual research questions to provide interpretation of explicit and discreet data identi ed in primary healthcare research (15,16). The development of key concepts and themes which arise from the analysis will be mapped to graphically display the interactions and associations of each theme; a narrative synthesis of data to present study ndings will then follow.
The synthesis of qualitative data will be supported by extracted quotes to provide a descriptive summary of the included studies. A summary of the qualitative ndings will also be presented in table format.
Although the thematic analysis will be performed by two reviewers, discussions between all members of the review team will occur to reach consensus about the preliminary coding of data and the development of the overarching key themes.

Discussion
For parents, families and main caregivers of infants who are born prematurely, the NICU environment can be a source of high stress and anxiety (1,2,5,8). Prolonged separation, ill-de ned roles, and limited direct contact between infant and parent serving to compound a range of health outcomes observed in this setting. This review of qualitative studies will aim to identify the facilitators and barriers associated with FICare programs and importantly attempt to address FICare program utility from the experiences of parents and family members. Review ndings will serve to inform and in uence current and future NICU FICare programs in the review location (Perth, Western Australia) and importantly contribute to hospital quality improvement. Ethical approval for this systematic review has been waived by Edith Cowan University (ECU) Human Research Ethics Committee (HREC).

Not applicable
Availability of data and materials The data set supporting the results of this review will be included within the paper and its additional les.

Competing interests
The reviewers advise that they have no con ict of interest in conducting this systematic review Funding Funding for this work has been provided by the Women's and Infant Research Foundation. The funder has no role in the development of this protocol.