The patient pathway after neonatal gastric surgery – how to support the family?

1.0 Background
Rare congenital anomalies and diseases of the gastrointestinal tract include the esophagus, the abdominal wall, bowel, rectum and diaphragm, which almost universally are associated with serious morbidity and for some infants mortality (1). Medical treatment, anesthesia, surgery, and care has developed over the last decades resulting in an increased survival (1). An increasing number of families are therefore caring for infants suffering from these congenital anomalies and diseases and they face many daily challenges. Infants that have had gastrointestinal surgery are at increased risk of morbidity, and have complex care needs after hospital discharge such as; stoma care, tube feeding, wound care, parenteral nutrition and continued observation for risk factors(2). When discharging infants with complex care needs, parents are trained to manage these tasks at home. During the last years, there have been increased focus on the management of infants and children with complex care needs in the community (3). To ensure success in transferring complex medical infants to home, increased collaboration between home health nurses and hospital specialists is essential in the first period after hospital discharge (4). However, the majority of the European countries do not have policies to support the coordination of care between sectors. Further, home health care for complex medical infants as a model of care does not meet the current needs of the families (5). Furthermore, a study has shown that parents of children with congenital malformations are in need of close contact to the hospital after discharge (2). Finally, the experiences and needs from the home health care nurses, managing infants in the home after neonatal gastric surgery, are yet to be studied. A review recommends tailored follow-up post-discharge for this group of infants, but not how follow-up should be carried out (2). Experiences when discharging preterm infants for tube feeding in the home have been widely studied (6–10) and the cross-sectoral collaboration after discharge for this group of infants and their parents is well-organized in many countries (7, 11).
When support does not meet the needs of the parents, it can be a great burden if the responsibility, feels higher than the parents are capable of. Studies have identified posttraumatic stress in parents to children with congenital gastrointestinal malformations with mothers more frequently affected than fathers (12, 13). Parents of children with esophageal atresia report impaired quality of life compared to background populations and again with mothers more affected than fathers (14). This indicates that there is a need for a long-term support to parents of infants receiving gastric surgery as neonates as parental mental challenges may have long-term consequences such as , less sensitive parent-infant interactions and consecutive child attachment- and behavior problems(15–17). A meta-analysis have shown an association between self-compassion and well-being(18), but it is not known if self-compassion protects mental health and well-being in parents with sick infants.
The mentioned studies on parental outcome when having an infant requiring gastric surgery as a neonate in regards to mental health and Quality of Life have not been conducted during their first admission.
On this background, the psychological status of the parents during the infants’ first admission is unknown, as well as the knowledge on the specific needs in the cross-sectoral collaboration in the patient pathway between the hospital and home health care nurses concerning these infants and parents.

2.0 Aim
To investigate experiences and needs from parents and health care professionals at hospital and at home when looking at the patient pathway after infant neonatal gastric surgery.
To answer the overall study aim three sub-studies will be conducted.
2.1 Aim Sub-study one
To conduct an integrative review on the experiences of parents of newborn infants requiring gastrointestinal surgery and their experience of the patient pathway.
2.2 Aim Sub-study two
Identifying risk-factors associated with Post Traumatic Stress Disorder, confidence, well-being and self-compassion in parents of infants receiving neonatal gastric surgery.
2.3 Aim Sub-study three
To identify needs of support and experiences in the patient pathway and the cross-sectional collaboration from the perspectives of hospital clinicians, parents and home health nurses.

3.0 Methods
3.1 Design
A mixed-method approach will be applied to answer the study aims. To answer the aim for sub-study one an integrative review will be conducted. Quantitative methods will be used in sub-study two and qualitative methods in sub-study three.

3.2 Sub-study one
3.2.1 Integrative review – empirical study
The integrative review process of Whittemore and Knafl (19) will be used to guide the study. It is a mixed-method systematic review including qualitative and quantitative studies and is the broadest type of research review providing the opportunity to fully understand a phenomenon of concern. Further, the methodology of an integrative review has the potential to allow findings to be applied to clinical practice (19). The integrative review will be carried out in five stages: 1) problem identification, 2) literature search, 3) data evaluation, 4) data analysis, 5) presentation (19).

3.2.2 Data collection
A structured and comprehensive literature search will be conducted in PubMed (MEDLINE), Embase, CINAHL, PsycInfo, Cochrane, Scopus, Web of Science, SweMed+, and Proquest Dissertation & Thesis Global. The papers from the literature search will be evaluated in a three-step process using Covidence online software for managing and streamlining systematic reviews (20).

3.3 Setting and participants in the clinical studies (sub-study two and three)
Odense University Hospital (OUH) is one of two Danish hospitals with the highest level of medical care for patients with neonatal and pediatric surgery as defined by the Danish Health Authorities. Parents of infants with abdominal gastric malformations and diseases requiring surgery as neonates will be included in sub-study two and three. Further, clinicians involved in the patient pathway will be included in sub-study three.

3.3.1 Recruitment (sub-study two and three)
Parents will be recruited for the study during infant admission. The parents will be approached when survival of the infant is expected and will be informed about the study from a project nurse or the primary investigator (PhD student). Inclusion criteria for participation is being a parent of an infant requiring neonatal gastric surgery due to necrotizing enterocolitis, spontaneous intestinal perforation, meconium ileus, abdominal wall defect, esophagus atresia, atresia in the gut, or diaphragma hernia, and being Danish speaking. Annually approximately 50 infants requiring gastrointestinal surgery receive treatment and care at OUH. Parents will be included consecutively and parents will be included for the study during a two year period (Appendix 1). It is estimated, that 30% of the parents will decline participation in the study. We aim for parents of 50 infants to complete the questionnaires. A power calculation is not suitable for this type of study and has therefore not been performed.

3.4 Sub-study two
3.4.1 Data and collection
The primary outcome is parental Post Traumatic Stress Disorder (PTSD) measured by using International Trauma Questionnaire (ITQ). Secondary outcomes is Parental Confidence measured by Karitane Parenting Confidence Scale (KPCS) (21), well-being measured by The World Health Organisation – Five well-being Index (WHO-5) (22) and self-compassion measured by the self-compassion scale – short form (SCS-SF)(23). Data will be collected through self-reported questionnaires at four time points; 2 weeks after birth, 3 months after birth, 6 months after birth and 12 months after birth. Besides the instruments for measuring parental outcome the parents will be asked to fill out information demographic data. Data will be collected using Research Electronic Data Capture (REDCap) hosted by Open Patient data Explorative Network (OPEN). Health data on infants will be drawn from hospital database of infants requiring gastrointestinal surgery (The gastroped database).

3.4.2 Questionnaires
All the questionnaires used for this study is validated. ITQ includes 18 simply-worded questions focusing on the core features of PTSD and has been developed to be consistent with the organizing principles of the ICD-11 set by the World Health Organization(24). The KPCS instrument is a 15 item scale measuring parental confidence for parents with infants aged 0-12 months(21). Parents must reply to a statement with an answer that comes closest to how he/she feels. The WHO-5 instrument measure current mental well-being on a 5 item likert scale(25) and the SCS is a 14-item scale that measures how a person reacts towards one self in tough times(23).

3.4.3 Analysis
Descriptive statistics will be used to describe the characteristics of the included participants. Categorical data will be analysed using χ2/Fishers exact test. Multivariate regression analysis will be used to study associations for posttraumatic stress, parental confidence, self-compassion, and well-being. P-values ≤ 0.05 will be considered statistical significant and analysis will be performed using STATA (version 17.0, Stata Corporation, College Station, Texas, USA).

3.5 Sub-study three
3.5.1 Qualitative design
The aim of sub-study three is to identify needs of support and experiences in the patient pathway and the cross-sectional collaboration from the perspectives of hospital clinicians, parents and home health nurses.
In accordance with James P. Spradley (26) the qualitative research method of this project will be participant observation with both informal ethnographic interviews(27), and formal semi structured interviews(28). Participant observation allows the researcher to participate in the field being investigated, converse with the informants to get insight into their experience and participate in their daily lives (26).

3.5.2 Participant observation
The participation degree in the project will be moderate participation, which maintains a balance between being an insider and an outsider, and thereby between participation and observation (26).
The observational study is found to be suitable, as it is aimed to investigate the cross-sector collaboration and what needs arise from the perspective of the parents and from the health care professionals, as well as their experiences of the patient pathway and mental health consequences. The families will be observed in the first time after discharge, to see what situations the families are experiencing, what challenges may appear and what needs arise in this connection.
In accordance with Spradley’s research cycle the researcher will be making descriptive observations, to get an overview over the social situation. After recording and analyzing the data, the research will be narrowed and then focused observations can begin. Finally, after more analysis and repeated observations in the field, the researcher will be able to narrow the investigation further and make selective observations (26).
In accordance with Spradley, an ethnographic protocol will be prepared during the fieldwork, which will include field notes, and possibly audio recordings and images (26). The field notes will be transcribed immediately after, and will contain detailed observations and quotes.

3.5.3 Data collection
3.5.3.1 Interview
Informal ethnographic interviews will take place under focused and selective observations, which gives the researcher the opportunity to clarify questions or get insight to the informants’ perspective to situations that arise in the present (26). A formal semi structured interview (28) with the parents will take place after analysis of the field data and after the selective observations. In addition to the families that are included in the observational study, an estimated number of 10 more families will be interviewed in order to gain a broader insight into families’ experience of the discharge process and the families’ first time at home.
Individual semi structured interviews will also be conducted with the family’s home health nurses and other clinicians involved in the patient pathway to get their perspectives.
In Accordance to Kvale and Brinkmann, all interviews will be supported by an interview guide, recorded and verbally transcribed (28).

3.5.3.2 Participant observation
The field studies take place in the families own homes. A number of 10 families will be observed during a year. The observational study will take place from the day after the families' discharge from the hospital, and continue during the first weeks and months. Each family will be observed in the home for five days during this period. The observations will take place during day and evening hours to make sure, that both parents will be present. The observations will include the families meetings with the home health nurse(26).

3.5.4 Analysis
The qualitative data analysis and interpretation will be inspired by Paul Ricoeur and his Phenomenological-hermeneutic thinking (29). In accordance with Ricoeur the analysis and interpretation of the text will take place in three stages; the naïve reading, the structural analysis and the critical interpretation and discussion. The naïve reading will provide the researcher with an overview and an understanding of, what the text is about. The structural analysis identifies units of meaning (“what is said”) and units of significance (“what the text speaks about”) and will reveal themes. The final phase will lead the themes to further interpretation and discussion (29).

Et spædbarn med sygdom eller medfødte misdannelser i mave/tarmkanalen kan efter udskrivelse have et komplekst plejebehov udover almindelig spædbarnspleje som stomipleje, sondeernæring, observation af sårheling efter operation og symptomer på genopstået sygdom.
Der påhviler forældrene et stort ansvar efter udskrivelse, og for mange familier kræver spædbarnsplejen, at de stadig er i tæt kontakt med sundhedsprofessionelle efter udskrivelsen. Yderligere, skal familielivet rumme gentagne akutte genindlæggelser.
Barnets komplekse plejebehov og sygdom kan med tiden slide på en families livskvalitet (1,2) og resultere i blandt andet posttraumatisk stress (3,4), følelser af meningsløshed og skilsmisse (1). Der foreligger ingen systematisk identificering af forældrenes behov og ønsker for omsorg, og hvordan man kan understøtte dette i det tværsektorielle samarbejde mellem hospital og kommune efter barnets udskrivelse.
Projektet har til hensigt at understøtte forældre i at varetage og vedligeholde deres sundhed og trivsel ved at understøtte deres behov i samarbejde med sundhedsprofessionelle. Projektet vil pege på indsatser som kan sætte fokus på- og optimere sektorovergange, hvilket vil gavne familierne og øge følelsen af tryghed og sammenhæng. Optimering af sektorovergange vil endvidere gavne sundhedspersonale i både primær og sekundær sektor, da fokus vil være et forbedret samarbejde på tværs over en længere periode end ved blot overlevering fra hospital til kommune. Dette vil styrke fagligheden omkring familierne og det syge barn, og øge tryghed og kompetencer ved både familier og sundhedspersonale, heriblandt også sundhedsplejersken, som vil klædes bedre på til at kunne vejlede forældrene ved et sygt barn. Kvaliteten ved sygeplejen til familierne i eget hjem vil øges, og projektet har dermed stor relevans for temaet ”Sygepleje til borgere i eget hjem”. Projektet imødekommer endvidere efterspørgslen på differentierede sundhedsydelser, og vil have fokus på en Care and Compassion tilgang, hvor det søges at afdække hvorvidt alle eller nogle familier har behov for særlige sygeplejefaglige tiltag i eget hjem, og anerkender at alle familier måske ikke har behov for de samme tilbud eller andelen heraf.