Background

In 2015, 45% (2.7 million) of all deaths in children aged under five years occurred during the neonatal period.1 Of these 2.7 million deaths, one million occurred on the baby's day of birth and approximately two million occurred within the first week of life.1 In Australia, from 2004 to 2013, 8348 infants died, with an average of 927 deaths per year.2 The neonatal period represents the first 28 days of life and is the most vulnerable period for newborn survival.1,2 Although the primary goal of health professionals is the preservation of life, for neonates born with a life-limiting condition, it is also essential to be able to acknowledge, with the consent of the parents and family, when end-of-life or palliative care should be commenced to manage distressing symptoms and enhance quality of life for these neonates.3,4 In addition, palliative care can be provided as an alternative to termination of pregnancy when an unborn baby is diagnosed with a life-limiting condition.5

Neonatal palliative care is defined as care for a baby with a life-limiting condition that involves an active and holistic approach to care from the point of diagnosis through to the time of death.3 It takes into consideration the emotional, social, spiritual and cultural elements that enhance the quality of life and provide support for the parents and family.3 The term neonatal palliative care is often used interchangeably with neonatal end-of-life care, therefore, throughout this protocol and systematic review, the terms "palliative care" and "end-of-life" care will be used synonymously. Although end-of-life care encompasses palliative care, palliative care has been interpreted as an element of end-of-life care, focusing more upon the management of the end stages of a medical condition while preparing for death. Broadly, there are three categories of newborns that would benefit from a palliative model of care: newborns born at the limits of viability; newborns born with a life-limiting/life-shortening condition including those infants who have sustained birth trauma, such as those with neonatal encephalopathy and who despite treatment will not survive6; and newborns who have received intensive care in the neonatal intensive care unit (NICU) but for whom this model of care has become inappropriate because aggressive treatments are leading to undue pain and suffering without the prospect of improvement.7 These categories of newborns have low rates of survival and high morbidity on the occasions that they do survive, and palliative care may be the best approach.

The effect of neonatal death upon parents has been associated with adverse mental health outcomes, traumatic grief, post-traumatic stress disorder, anxiety and suicide.8 During this time, parents are said to experience a sense of loss much different to other types of bereavement such as the death of a spouse.9 A range of profound emotions such as anger, denial and anxiety that are experienced by the parents, siblings, family and staff often accompanies the awareness of the inevitable death of a neonate.8 The natural bond formed between parents and baby can be displaced by news of a life-limiting condition causing some parents to "not get too close" by way of protecting themselves from the forthcoming pain.10

Parents struggle to comprehend their loss and are forced to deal with complex emotional situations such as making the most of engaging with their baby as a living person while having to prepare mentally and emotionally for his/her imminent death.9 These feelings are also shared by the siblings and wider family. A study by Sandler et al.,11 exploring the effects of neonatal loss on siblings, showed they can experience feelings of anxiety, sadness and shock after the passing of their sibling in a neonatal intensive care ward. Research investigating the long-term impact of perinatal and neonatal bereavement on parents have shown prolonged emotional disturbance, an inability to share their feelings of loss with their spouse, as well as permanent loss of relationships with friends, colleagues and extended family members.12,13

In 2012, an Australian Senate inquiry into palliative care identified neonatal palliative care research as a priority area with a particular emphasis on the need for better support of families and siblings.14 To date, there have been numerous systematic reviews that have explored the psychological and emotional impact on parents and families after a stillbirth15-17 or the death of an infant post-birth.18-21 To identify existing systematic reviews exploring the experiences of palliative or end-of-life neonatal care in parents and families, MEDLINE, ProQuest, PsycInfo, Science Direct, Scopus and the JBI Database of Systematic Reviews and Implementation Reports were searched. Only one systematic review was identified that explored exclusively the experiences of parents and families with their healthcare providers during neonatal end-of-life care.21 This present review will therefore go beyond exploring solely the experiences of parents' and families' interaction with their healthcare providers and will provide a more comprehensive review of the multi-factorial nature of end-of-life care or palliative care in the neonatal setting. This may include, but will not be limited to, their experiences of the emotional, social and physical environment of the wards and with healthcare providers or other parents/support services. This systematic review will aim to identify and collate current evidence regarding the experiences, perceptions, challenges, beliefs, facilitators and barriers to appropriate and effective neonatal palliative care to assist in directing and informing future policies and guidelines on end-of-life care that better align with parental expectations.

Inclusion criteria

Types of participants

The current review will consider studies that include parents and families (e.g. grandparents and siblings) of neonates (infants aged 28 days or less)1,2 with a life-limiting condition. A life-limiting condition is an incurable illness where it is expected that loss of life will be a direct consequence of the illness.14,22

Phenomena of interest

The phenomenon of interest for this systematic review is the experiences of parents and families of babies in palliative and end-of-life care in the hospital, community or home settings. These experiences may include barriers and facilitators to appropriate and effective neonatal palliative and end-of-life care identified by parents and family members, experiences of communication with and care provided by the entire multidisciplinary team, their experience of the emotional, social and physical environment of the wards (if in a hospital setting) and the general perceptions of parents whose infant has received palliative or end-of-life care.

Context

Any neonatal setting will include, but not limited to, hospitals of any size, within the community or home setting and in any country. A neonatal setting is a place where care is provided to sick neonates such as the special care nursery or NICU.

Types of studies

The current systematic review will consider studies that focus on qualitative data including, but not limited to, designs such as phenomenology, grounded theory, ethnography and qualitative components of studies using mixed method designs.

Search strategy

The search strategy aims to find both published and unpublished studies. Studies published in English will be included in this review. Studies published from 2000 to present will be considered for inclusion in this review. The time range was selected to capture a wide range of data to yield results relevant to current contemporary practice. A three-stage search strategy will be utilized in this review.

Stage 1: An initial search of MEDLINE (OVID-SP) and CINAHL will be undertaken using a limited set of keywords. Initial keywords to be used will be experiences, palliative care, end-of-life, neonate, infant, death, bereavement, parent, family, siblings, hospital care, support, barriers, facilitators, resources, perinatal, neonatal intensive care and special care. The titles of studies in the initial search will be reviewed to expand the keywords, and the index terms used to describe the studies will be identified.

Stage 2: Individual search strategies will be developed for each database using identified keywords and index terms. A second search will then be undertaken using the individual search strategies developed for each database.

The databases that will be searched for published literature include:

CINAHL

MEDLINE via OvidSP

ProQuest

Scopus

PsycInfo

Science Direct

Web of Knowledge.

The database that will be searched for unpublished literature or gray literature will be ProQuest Dissertations and Theses.

Stage 3: The reference lists of all identified studies will be searched for additional studies.

Assessment of methodological quality

Papers selected for retrieval will be assessed by two independent reviewers for methodological quality prior to inclusion in the review using standardized critical appraisal instruments from the Joanna Briggs Institute Qualitative Assessment and Review Instrument (JBI-QARI) (Appendix I). Any disagreements that arise between the reviewers will be resolved through discussion or with a third reviewer.

Data extraction

Data will be extracted from papers included in the review using the standardized data extraction tool from JBI-QARI (Appendix II). The data extracted will include specific details such as the phenomena of interest, participants and study methods.

Data synthesis

Qualitative research findings will, where possible, be pooled using JBI-QARI. This will involve the aggregation or synthesis of findings to generate a set of statements that represent that aggregation, through assembling the findings (Level 1 findings) rated according to their quality, and categorizing these findings on the basis of similarity in meaning (Level 2 findings). These categories will then be subjected to a meta-synthesis to produce a single comprehensive set of synthesized findings (Level 3 findings) that can be used as a basis for evidence-based practice. Where textual pooling is not possible, the findings will be presented in narrative form.

Appendix I: QARI appraisal checklist

Appendix II: Data extraction instrument

Appendix III: QARI appraisal instrument

References