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Abstract
Background Infants with life-limiting conditions are a heterogeneous population. Palliative care for infants is delivered in a diverse range of healthcare settings and by interdisciplinary primary healthcare teams, which may not involve specialist palliative care service consultation.
Objective To synthesise the literature for how palliative care is delivered for infants aged less than 12 months with life-limiting conditions.
Methods An integrative review design. MEDLINE, CINAHL, ProQuest, Cochrane, Joanna Briggs Institute and EMBASE were searched for research published in English language, from 2010 to 2022, and peer reviewed. Critical appraisal was completed for 26 patient case series, 9 qualitative, 5 cross-sectional and 1 quality improvement study. Data analysis involved deductive content analysis and narrative approach to summarise the synthesised results.
Results 37 articles met the eligibility for inclusion. Two models of palliative care delivery were examined, demonstrating differences in care received and experiences of families and health professionals. Health professionals reported lack of palliative care education, challenges for delivering palliative care in intensive care settings and barriers to advance care planning including prognostic uncertainty and transitioning to end-of-life care. Families reported positive experiences with specialist palliative care services and challenges engaging in advance care planning discussions.
Conclusion There are complex issues surrounding the provision of palliative care for infants. Optimal palliative care should encompass a collaborative and coordinated approach between the primary healthcare teams and specialist palliative care services and prioritisation of palliative care education for nurses and physicians involved in providing palliative care to infants.
- Chronic conditions
- Communication
- Education and training
- End of life care
- Paediatrics
- Supportive care
Data availability statement
Data sharing not applicable as no datasets generated and/or analysed for this study.
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WHAT WAS ALREADY KNOWN
Palliative care for children provides a total approach to care and is recommended to begin at diagnosis of a life-threatening or life-limiting condition.
The palliative care approach aims to improve quality of life for children with life-limiting conditions and their families.
WHAT ARE THE NEW FINDINGS
Integration of specialist palliative care services was not a standard practice for infants, this was particularly evident in neonatal intensive care units.Although not all infants with LLCs require input from a specialist palliative care service, a palliative approach to care should be delivered by all health professionals.
WHAT IS THEIR SIGNIFICANCE
Clinical: Prioritisation of palliative care education for nurses and physicians involved in the care of infants at an individual and organisational level may lead to improved knowledge and confidence to provide palliative care across a range of clinical settings and further integrate specialist services to optimise palliative care delivery to infants and their families.
Research: Further research to evaluate the impact of palliative care education for nurses and physicians.
Background
Palliative care for infants and children provides a total approach to care and is recommended to begin at diagnosis of life-threatening or life-limiting conditions (LLCs).1–3 It promotes quality of life and can be provided in parallel to curative treatment.1–3 This approach supports patient-centred and family-centred care to provide treatment that meets the family’s preferences and needs.2 3 A recent rapid review of the literature highlighted key areas to enhance the delivery of palliative care for children were: patient-centred and family-centred care, early integration of palliative care to improve access for families and across a variety of settings, advance care planning with support from specialist palliative care teams, and development of educational resources and skills training for health professionals.4
Perinatal and paediatric palliative care services have become established specialties over the past two decades, comprising interdisciplinary health professionals including physicians, nurses, midwives, social workers and chaplains who have advanced training and/or experience in palliative care.3 5 6 Key features of specialist palliative care services include providing consultation to patients and their families and working in partnership with the patient’s healthcare teams to provide management of symptoms, support with decision making, advance care planning, as well as psychosocial, spiritual and bereavement support for the whole family.1–3 Recent research exploring families’ experience of palliative care described what constitutes ‘good’ palliative care as receiving coordinated care, where palliative care is integrated early with specialist palliative care service professionals, involves families throughout the care of the child and at end-of-life, is individual to the family needs and supports the whole family.7
Infants aged less than 12 months comprise the highest number of deaths in childhood.8 9 The leading causes of infant deaths are congenital malformations and perinatal conditions related to low birth weight, preterm births, birth trauma and viral infections.9–11 Many infants’ deaths occur in intensive care settings.12–16 Delivery of palliative care for infants has been reported to be impacted by specific barriers such as uncertain prognosis, use of invasive life-saving interventions including mechanical ventilation and cardiopulmonary resuscitation at end-of-life, complexity of symptom management and lack of continuity of care due to rotational staffing in intensive care units.17–19 A recent study examining the care management trajectories of infants with LLCs who had died found that even for infants born preterm or with congenital conditions, deaths were not anticipated or planned for until close to end-of-life.16 Additional challenges that have confronted families included prolonged hospital stays, prognostic uncertainty while at the same time facing the risk of sudden deterioration and possibility of death of their infant.16 20
Infants with LLCs may be cared for in a variety of hospital and community settings, including intensive care, lower acuity wards and community settings such as hospice and home.3 16 21 Availability of specialist palliative care services may vary across these settings. This means that palliative care for infants is often delivered by the primary or treating healthcare teams such as intensive care physicians and nurses. An Australian study found less than a quarter of infants (11/45) with LLCs who died received input from a specialist palliative care service.16 Given the evolving role of specialist palliative care services, the heterogeneous population of infants with LLCs and their unique needs in a diverse range of healthcare settings, it is unknown if palliative care is being optimally provided for these infants and their families. The aim of this integrative review was to synthesise the literature to better understand how palliative care is delivered for infants aged less than 12 months with LCC. The objectives were: (1) to explore how palliative care is delivered for infants with LLCs; (2) to understand the role of specialist palliative care services for infants with LLCs; (3) to identify factors impacting palliative care delivery to infants.
Methods
The integrative review method was selected to provide a broad understanding of the diverse palliative care literature. The review was guided by the approach used by Whittemore and Knafl22 and followed five steps: problem identification, literature search, data evaluation, data analysis and presentation of results. A protocol was developed and published before the review was started in the Open Science Framework.23 The PRISMA Statement reporting checklist for Systematic Reviews was followed.24
Search strategy
An electronic search of six databases was conducted from a broad variety of health disciplines: MEDLINE, CINAHL, ProQuest, Cochrane, Joanna Briggs Institute and EMBASE. The search strategy included a combination of key words in the title or abstract relating to (palliative or terminal or ‘end of life*’) ADJ3 (care or therap* or treatment*) adjacent to (care or therap* or treatment*) and (“life-limiting*” or chronic* or complex*) adjacent to (condition* or disease* or illness*) and (“congenital abnormalit*” or “congenital anomal*” or prematur*) and (infant* or baby or babies or pediatric* or paediatric* or newborn* or neonat*). Search terms were adapted and modified for each database. See box 1 for Medline search strategy. Hand searching of reference lists of retrieved papers was also undertaken.
Medline search strategy
Medline search strategy
(‘congenital abnormalit*’ or ‘congenital anomal*’ or prematur*).ti,ab. OR
Chronic Disease/OR
((‘life-limiting*’ or chronic* or complex*) ADJ3 (condition* or disease* or illness*)).ti,ab.
AND
((palliative or terminal or ‘end of life*’) ADJ3 (care or therap* or treatment*)).ti,ab. OR
Terminal Care/or Palliative Care/
AND
(infant* or baby or babies or pediatricpaediatric* or paediatric* or newborn* or neonat*).ti,ab. OR
Exp Infant/
Limit to English language and year 2010 to 31 July 2022.
Study selection
Included articles comprised diverse methodologies, theoretical and empirical research about how palliative care is delivered for infants. Specifically, articles focused on palliative care for perinatal, neonatal (the term neonate is defined from birth until 28 days of life)25 and paediatric populations. Studies were included if there was a substantial proportion that included infants. Studies focused on perinatal care were included only if they also included the neonatal period. The starting year 2010 was chosen for inclusion of articles to capture the period of specialist paediatric palliative care emerging as a medical specialty. Inclusions: English language, published online from 31 July 2010 to 31 July 2022, full text available online and peer reviewed. Exclusions: study results obtained more than 2 years prior to 2010, and where results did not specifically relate to infants.
An electronic research screening programme was used to screen publications by titles and abstracts.26 A selection of 11 key publications were uploaded to research screener to improve eligibility screening.26 One researcher (RI) independently screened each article in the research screener programme. All three authors (RI, FG, MOC) screened and cross-checked one-third of the articles. Articles that met the inclusion criteria were read in full by one author (RI). Eligible articles were discussed by all three authors to cross-check and address discrepancies.
Data extraction
Articles were grouped according to the research focus: patient case series and perspectives of either health professionals or families. Data extracted from each article were summarised and tabulated; citation (author), year, country, design and method, sample characteristics, specialist palliative care service established, single or multi-site study, aim of study and key findings.
Data evaluation
Critical appraisal of the articles was completed using one of the four checklists according to study designs: patient case series, qualitative, cross-sectional and quality improvement. For quality purposes, a sample of 25% articles from each checklist was completed together by the authors (RI, FG, MOC). One author (RI) independently completed the remaining appraisals and were cross-checked with FG and MOC. Case series and qualitative articles were appraised using checklists from Joanna Briggs Institute Quality Appraisal Framework,27 28 both checklists consisted of 10 items. Cross-sectional articles were appraised using a 20-item checklist.29 The quality improvement study was appraised using a 16-item checklist.30 The quality of the articles was categorised using an approach by Gill et al 31 where the total score from the checklist were converted to a percentage. The following percentages were indicative as: strong (80–100%), good (70–80%), average (50–70%) or poor (less than 50%).
Data analysis
A deductive content analysis5 32 approach was applied using pre-established themes from key palliative care international guidelines.5 6 These included population characteristics, specialist palliative care service, advance care planning, characteristics of end-of-life care and bereavement support. Content analysis steps involved categorising, quantifying, contrasting and comparing the data to synthesise the themes and evidence.22 A narrative approach was used to summarise the synthesised results.
Results
The search generated 3156 articles. Articles were exported to a reference management software programme and, guided by key articles, a research screener system screened and organised each article by title, abstract and relevance.26 Research screener removed 1486 articles that were duplicates or missing titles and abstracts. A total of 1674 articles were screened at title and abstract level. This resulted in 116 articles that met eligibility criteria and were independently read in full by one author (RI), followed by discussion among the research team to cross-check and address discrepancies. Figure 1 displays the PRISMA flowchart. The final number of articles included prior to critical appraisal was 41, including four articles identified through hand searching.
PRISMA flowchart.
Critical appraisal
Twenty-six articles reported a patient case series method (retrospective review), of these, 23 articles were rated as strong, 3 rated as average and were excluded. For nine qualitative research studies (interviews and focus groups), five articles were rated as strong, three were good and one rated as poor and was excluded. For five cross-sectional designs (surveys), three rated strong, two were rated as good. One quality improvement study was rated as strong. In total, four articles were excluded due to poor quality and/or a score of less than 50%. See online supplemental file 1 for critical appraisal of articles. Following critical appraisal, the final number included in the review was 37. See online supplemental file 2 summary of articles.
Supplemental material
Supplemental material
Characteristics of articles
Studies were conducted from the USA 13 (35%), UK 5 (13%), Australia 3 (8%), Brazil 3 (8%), Switzerland 3 (8%), Canada 2 (5%), with the remaining 8 (22%) articles from Belgium, Iran, New Zealand, Malaysia, Italy, China and Turkey. Case series studies included populations of newborns to age 18 years; 13 (35%) articles focused on infants aged less than 12 months and children older than 12 months and 11 (30%) focused on newborns less than 30 days old and infants aged less than 12 months. Five (13%) articles examined the experience of bereaved parents of infants, and eight (22%) focused on health professionals’ experience involving nurses and physicians from areas such as paediatric or neonatal and intensive care units. Most studies 23 (62%) were conducted at a single hospital site and 14 (38%) were multi-site studies. Articles reported on key elements, including the team who delivered palliative care, characteristics of infants who died, advance care planning, barriers and facilitators to provision of palliative care and experience of families. See table 1 for article characteristics.
Article characteristics
The availability of specialist paediatric and perinatal palliative care services was reported in 25 (68%) articles. For 10 (27%) articles, it was not reported and two (5%) articles reported that specialist palliative services were not established or limited. In articles where specialist palliative care services were reported, for 16 (43%) it was not routine practice for infants with LLCs and their families to receive specialist palliative care service consultation.14 15 33–39 Three (8%) articles reported that palliative care was delivered by the primary healthcare team in the neonatal intensive care unit (NICU).13 33 40 Congenital abnormalities and complications secondary to extreme prematurity were the most reported cause of deaths for infants.13 15 35 36 38 41–46
Synthesis of results
This review explored how palliative care was delivered for infants with LCCs and their families, and the role of specialist palliative care services. The 24 case series articles were grouped into two categories of (1) palliative care delivery with specialist palliative care services and (2) palliative care delivery by the primary healthcare team without specialist palliative care consultation. Key topics reported included population characteristics, place of care and death, advance care planning, end-of-life characteristics and bereavement support. The two groups were compared to examine key differences in how palliative care was delivered and how these impacted on care. To explore factors that impacted on palliative care delivery, synthesis of the cross-sectional and qualitative articles included family experience of palliative care and health professional perspective.
Palliative care delivery with specialist palliative care service consultation
The population who received specialist palliative care service consultation were infants and children with a high risk of mortality or medical complexity including receiving invasive life-saving interventions such as mechanical ventilation, older infants (aged more than 12 months),34 37 43 47 with congenital conditions diagnosed prenatally or following birth trauma,14 33 42 44 48 and infants with longer NICU stays (more than 30 days).33 However, this was inconsistently reported. For example, one Canadian article reported for infants diagnosed with congenital heart disease, consultation with a specialist palliative care service was less common (16%), and an article from Ireland reported infants with congenital heart disease comprised 25% of specialist palliative care consultations.34 48 Reported reasons for consultation with a specialist palliative care service were to guide goals of care/advance care planning discussions, support decision making, optimise symptom management, end-of-life care planning, and care coordination.33 44 49
Advance care planning discussions occur between the healthcare team and family and involve consideration of how to provide care to the infant during deterioration and at end-of-life that aligns with the families’ preferences and needs.5 Advance care planning discussions were reported in 10 (27%) articles and included antenatal birth planning, family’s preferences for end-of-life care and treatment limitations.15 37 41–43 Infants with advance care planning and treatment limitations were reported to have received fewer invasive procedures close to end-of-life.15 35 37 41 50 Antenatal birth plans focused on comfort rather than invasive treatment and resuscitation measures.42 In contrast, one article from the USA compared infants who received palliative care with those who did not and reported infants in both groups received similar invasive procedures close to end-of-life.44
End-of-life care was delivered in a variety of settings including the labour and delivery ward, neonatal and paediatric intensive care, hospice and home.12 15 42 48 50 A distinguishing feature for families of infants who received specialist palliative care service consultation was that these families were offered options for place of care at end-of-life, resulting in deaths occurring outside of intensive care setting, including inpatient hospital wards, hospice or home.12 15 21 37 41 43 48 50 51 Two articles captured bereavement support . In both studies, families who had received specialist palliative care service consultation received bereavement support.15 50
Palliative care delivery by primary healthcare team without specialist palliative care service consultation
The population who received palliative care delivered by their primary team appeared to be infants in NICUs,15 35–37 39 43 45 46 in particular infants born preterm or with congenital heart disease.14 34
For infants in this group, advance care planning discussions were infrequently reported.15 35 36 41 43 50 Infants received invasive interventions close to end-of-life, such as mechanical ventilation, cardiopulmonary resuscitation, insertion of central venous access devices, administration of vasoactive medications and antibiotics.12 35 37 38 45 52 A Brazilian study at a tertiary NICU reported that infants with prognostic uncertainty received invasive treatments more frequently in comparison to infants with confirmed prognosis.13 One article from the USA compared outcomes for infants between parents who opted for comfort care versus infants who received intensive care treatment and identified no difference in the median age of death.40 Infant death following withdrawal of life-sustaining treatment was reported in six studies.12 35 38 41 45 52 A nation-wide Swiss study including children of all ages, that formed part of a larger research programme to understand current end-of-life practices and parental perspectives, reported 84% of infant deaths were preceded by withdrawal of life-sustaining treatment.38
A high proportion of infant deaths occurred in intensive care settings.36 41 45 46 51 Younger infants (aged under 12 months) appeared to be less likely to be transferred or discharged home for end-of-life care.12 15 21 38 45 Discussions for options for preferred place of death was infrequently reported,15 and hospice deaths were less common.43 Bereavement support was not reported in any of the studies.
Family perspective
Five articles reported experiences of infants’ bereaved parents using in-depth interviews20 53 54 or cross-sectional surveys.39 55 Each article focused on a different aspect of the family experience; consultation from a specialist palliative care service, advance care planning, end-of-life care decision making and parental satisfaction. Families reported that the specialist palliative care team provided support with decision making, facilitated communication for advance care planning, provided emotional support and optimised symptom management.53 55 Of note, in one study families initially associated the term palliative care with the healthcare team giving up on providing curative treatment and, as a result, they experienced a loss of hope.
One parent stated:
So when I heard palliative care, I heard hospice, I knew we were teetering on the edge of life. Instead of looking at the positives that it could do, I was just looking at, you know, I’m not ready to transition to that. We’re still fighting, I still want aggressive care.53 p.483
Interestingly, the family later reflected on their experience and stated they found consultation with the specialist palliative care service was beneficial, and they would recommend it for other families.53
Three articles reported families’ experiences of advance care planning and end-of-life care decision making.20 53 55 An article from a paediatric intensive care unit in the UK investigated parental experiences of decision making and reported continuity of care and trusted relationships were fundamental to supporting parents with end-of-life care decision making.20 Advance care planning was reported by parents as being emotionally overwhelming, and they felt they needed to be accepting of the situation before they could engage in these discussions.20 In addition, not all parents were familiar with advance care planning, some parents felt it would have been helpful and others reported they would not be able to engage in the process to plan for or consider their infant or child’s end-of-life.20 Preferences for end-of-life care (including cultural and spiritual needs) appeared to be unique to each family.20 Parents described factors that influenced decision making for their infant or child’s end-of-life care was their knowledge and experience of the condition, recognition of progressive deterioration, their acceptance of end-of-life, honest and clear communication from health professionals and parents ‘goal’ for more time with their child.20 39 53 Another Swiss study, that formed a component of the research programme discussed earlier, involved a survey of 135 bereaved parents from four diagnostic groups (cardiology, neonatology, neurology and oncology) to examine their perceptions of their child’s end-of-life care following six quality domains of care; support for the family unit, communication, shared decision making, symptom management, continuity and coordination of care and bereavement support.39 Parental satisfaction about end-of-life care was reported positively by all diagnostic groups.39 Interestingly, neonatology parents reported lower satisfaction with shared decision making than the other three diagnostic groups.39 Furthermore, 80% of neonatology parents reported discussions regarding withdrawal of life-sustaining treatment occurred, however, 65% reported they were actively involved in decisions for withdrawing life-sustaining treatment.39
Specific challenges for parents in NICUs were reported to be: not being able to comfort and hold their infant, financial burden of having an infant in hospital, communication conflict with health professionals, lack of continuity of care due to changeover of staff and the open environment of the intensive care unit resulting in lack of privacy.53 Families who accessed hospice care described the added value of being in a homely environment, availability of continuous support from health professionals, privacy and the opportunity to care for their infant or child and create memories.54
Factors that impacted on palliative care delivery
Eight articles reported perspectives of health professionals (physicians and nurses involved in the care of infants) using surveys,56–58 in-depth interviews47 59–61 and focus group.62 Several facilitators and barriers were identified for the provision of palliative care, including referrals/involvement of specialist palliative care service and advance care planning.
Facilitators
A key facilitator for consulting with specialist palliative care service was physicians’ and nurses’ appreciation of palliative care teams’ knowledge and experience navigating uncertainty, expertise in providing emotional and spiritual care for families and assisting with communication,47 and health professionals’ recognition that palliative care shares parallel importance as curative care.58 Other facilitators to consult with specialist palliative care service were: provision of support for patients with medical complexity and those with lengthy hospital stays.47 Nurses valued specialist palliative care consultation services’ capacity to enable families to have options for preferred place of care and death, provide bereavement support, and care coordination with community teams including hospice care.59 An important feature reported in six articles was health professionals’ recognition that palliative care education and training, including understanding when to refer to specialist palliative care services, is essential for health professionals to deliver optimal palliative care.47 57–61
Barriers
A key challenge to offering specialist palliative care service consultation in the NICU appeared to be that a high proportion of infants died within 24 hours of admission.37 This short time period reduced opportunity for specialist palliative care consultation.37 Further, health professionals reported how intensive care environments were suboptimal settings for supporting infants at end-of-life and for delivering a palliative approach.57 58 62 These factors were compounded by time constraints and frequent staff change overs that prohibited opportunity to develop rapport with families.47 57 58 62 In addition, insufficient knowledge about palliative care and advance care planning due to a lack of formal training and education was reported by nurses and physicians in NICU and paediatric intensive care units.59–61 Barriers to discussing advance care planning and transitioning to end-of-life care were reported to be due to: prognostic uncertainty, lack of medical consensus about a prognosis, availability of newer technologies and procedures to prolong life, parental desire for and expectations of cure and not ‘giving up’, and ethical challenges balancing the burdens and benefits of invasive treatments.56 58 59 62 Neonatology physicians’ decision to cease curative treatment was mostly influenced by prognosis—f it was predictable, and their assessment of infants’ quality of life.56
Discussion
This review identified 37 articles from 14 countries, predominately originating from developed countries. The articles reported a heterogeneous population of infant characteristics, study settings, aims and methods. There was clearly great variation in the care infants received and complex issues surrounding the provision of palliative care and factors impacting consultation with specialist palliative care services. The infant population in the case series articles included a range of disease trajectories, with complex medical needs, and a high proportion of infants was managed in intensive care units. Infants and their families also faced the ever-present possibility of sudden deterioration and death.
While the principles and practice of palliative care for children and adults have many similarities, infants and their families experience unique challenges and considerations.63 Issues for infants include the rarity of diseases, difficulty to diagnose a condition, prognostic uncertainty, possibility of sudden deterioration, the role of decision makers which may involve extended family members and impact on the whole family.63 A common theme reported by health professionals and in two of the patient case series studies was prognostic uncertainty, which contributed to the challenges of transitioning from cure-oriented care to palliative care and was a factor that influenced the care infants received. This mirrors a recent study in Australia categorising the trajectories of infants with LLCs who died, which found infants with prognostic uncertainty received curative treatment and were associated with infrequent referral to specialist palliative care services.16 However, it is important to acknowledge the inherent challenges in the neonatal population where infant prognosis is unknown, and intensive care treatment needs to be initiated before a diagnosis and prognosis can be confirmed. Findings suggest that this is a phenomenon experienced across several continents.
In this review, two distinct models of palliative care were examined: (1) palliative care delivery for the infant and family who received specialist palliative care service consultation and (2) palliative care delivered by the primary healthcare team. Specialist palliative care services were reported as being available in the majority of the case series articles. However, less than a quarter of the articles reported specialist palliative care service involvement in infants’ care. For the remaining articles, palliative care was delivered by the primary healthcare team, a feature particularly evident in NICUs. This reflects previous empirical findings and suggests that despite recommendations as best practice, integration of specialist palliative care services is not yet a standard practice for infants with LLCs.4 19 This finding is not totally surprising given specialist palliative care services are considered relatively new in some countries, so understandably integration and service availability may vary. Specifically, studies conducted in Iran, Brazil, Malaysia and China did not report whether specialist palliative care services were available, perhaps indicating specialist palliative care consultation may not be a uniform approach in all countries and it is unknown the impact cultural beliefs may have on palliative care services.
Neonatal physicians and nurses have a comprehensive role in providing cure-oriented care and expertise in quality end-of-life care to infants. These healthcare teams may not consider or prioritise referral to specialist palliative care services for infants where the care management is focused on curative intent, or in the context of infants’ rapid deterioration, and short time periods of care. Interestingly, this was not a factor reported by health professionals and may require further research.
The review highlighted key differences in practice between the two models of palliative care. Infants who received specialist palliative care service consultation were more likely to have had advance care planning discussions and end-of-life planning, including preference for place of death. Without these elements of care, families may not be afforded the opportunity to plan and prepare for their infant’s end-of-life care that aligns with their goals and values.59 Advance care planning discussions were recognised by parents as emotionally overwhelming, however were also acknowledged as important and valuable. Parents’ willingness to engage in advance care planning discussions were also variable. This may be influenced by the approach taken by health professionals to frame advance care planning. This involves explaining to parents that advance care planning guides health professionals to deliver care that aligns with the family wishes and can also be achieved alongside infants receiving curative treatment. Using this approach, parents may better appreciate the importance of advance care planning and be able to engage in discussions.
It has long been recognised that parents with a critically ill infant requiring intensive care support experience depression, anxiety and loss of control and hope.64 At the same time, parents are required to discuss the future care of their infant, a discussion which may occur when the infant’s condition is rapidly evolving or has an uncertain prognosis. For some, therapeutic relationships may not yet be established between the family and healthcare team recognised as important factors enabling parents to engage in advance care planning discussions.20 The challenges experienced by families with advance care planning for infants have also been described in paediatric and adult palliative care settings. For example, studies involving parents of children aged up to 17 years and in the adult patient setting similarly reported family reluctance and difficulty facing reality to have end-of-life discussions.7 65 66 The review findings support timely referrals to specialist palliative care teams to establish therapeutic relationships, provide continuity of care, assist with decision making and provide psychosocial and emotional support for the whole family.7 67 Involvement of specialist palliative care teams may play a key role in mitigating some of the challenges experienced by families to support families to engage in advance care planning.
Current guidelines recommend that preferred place of care or death is discussed or considered with the family and can be achieved with support from specialist palliative care services to assist in the transfer of patients for end-of-life care where feasible.3 5 63 A key finding from this review was that a high proportion of infant deaths occurred in neonatal intensive care settings following withdrawal of life-sustaining treatment . It was not reported what the preferred place of death was for these families. Only a small number of deaths occurred at home or in hospice settings. Those infants who died at home or in hospice care were those who had received specialist palliative care service. While providing choices to families where they wish to be cared for is an important aspect of palliative care, transferring medically complex patients from intensive care units to other settings for end-of-life care may not be practical or feasible.48 Significant preparation and collaboration of hospital and community providers are required, which can be resource intensive and logistically difficult to arrange.15 68 Despite these challenges, with timely planning and collaboration, care of the infant at home may provide many benefits, including a homely environment, increased privacy, involving family members such as siblings in care and enhanced ability to meet cultural or spiritual needs.3 Although a systematic review found inadequate evidence to support home as the preferred place of death for children and young people,69 the majority of the studies reported perspectives of parents of children with cancer and the results may not be generalisable to other populations, in particular for infants.69
Challenges to providing optimal palliative care for infants and their families included primary healthcare professionals’ reported lack of knowledge and experience with advance care planning and introducing palliative care resulting in lack of confidence, skills and knowledge to talk with families about palliative care and support decision making. These findings add to existing evidence in both paediatric and adult settings where the importance is recognised of building capacity of health professionals to deliver quality palliative care through education.70 71 Given the diverse healthcare settings and health professionals involved in providing palliative care to infants, education that addresses specific challenges and complex issues for infants is essential. Targeted paediatric palliative care education through workshops, mentoring and delivering patient-specific education have been found to be useful in developing confidence and improving clinical practice for health professionals.70 72 Key focus areas for education are recommended to align with current palliative care guidelines, including communication skills, assessment and management of symptoms, and psychosocial care of the patient and family.3 71 73
Finally, bereavement support was only reported in two of the case series articles, perhaps indicating a potential gap in the provision of palliative care services and bereavement care. Gaps in bereavement support have been widely recognised across adult and paediatric settings, with many services lacking bereavement guidelines.74 A coordinated approach to bereavement support is recommended, including assessment of individual needs of the family, and may involve providing anticipatory support.2 Bereavement interventions may include telephone contact, information and resources to navigate grief, referral to counselling services, age-appropriate support for siblings and opportunity to meet with the healthcare team to address questions.2 74 75
Strengths and limitations
Strengths of this review were the inclusion of a diversity of palliative care studies from a range of countries, study settings, aims and methods. A rigorous critical appraisal assessing the quality of each of the four study designs was conducted. The qualitative and cross-sectional study designs provided a layer of understanding and insight through exploring family and health professional experiences. Limitations were that only articles published in English and full text available were included. Consequently, there is potential for cultural bias, resulting in stronger perspectives from developed countries and limiting the generalisability of the findings. There is opportunity for researchers to advocate attention to capturing diversity. Additionally, bereavement support was not specifically a search term and was only reported in two of the case series articles. This may have also reflected gaps in documentation in patient records rather than gaps in service.
Conclusion
The review highlighted the complexity surrounding provision of palliative care for infants and their families. The infants comprised of a heterogeneous population, with a range of disease trajectories, many receiving intensive care level support. Complexity was compounded by prognostic uncertainty, possibility of sudden deterioration and death. Two models of palliative care delivery were examined, demonstrating differences in both care received and experiences of families and health professionals. For infants who received specialist palliative care service consultation, their families more frequently had advance care planning discussions and end-of-life care planning including preferred place of death. Health professionals reported challenges in transitioning from cure-oriented care to palliative care approaches for infants and their families. Lack of education and training about palliative care was identified by health professionals as a barrier that may impact on their capacity to deliver palliative care and to consult with specialist palliative care services. Optimal palliative care should encompass a collaborative and coordinated approach between the primary healthcare teams and specialist palliative care services. Prioritisation of palliative care education for nurses and physicians involved in the care of infants at an individual and organisation level may lead to improved knowledge and confidence to provide palliative care across a diverse range of clinical settings and further integrate specialist services to optimise palliative care delivery to infants and their families.
Data availability statement
Data sharing not applicable as no datasets generated and/or analysed for this study.
Ethics statements
Patient consent for publication
Ethics approval
Not applicable.
Acknowledgments
The authors thank Vanessa Varis, Librarian from Curtin University for support and assistance in developing the search terms for the databases.
References
Supplementary materials
Supplementary Data
This web only file has been produced by the BMJ Publishing Group from an electronic file supplied by the author(s) and has not been edited for content.
Footnotes
Contributors RI is responsible for the overall content as guarantor, made substantial contributions to the study design, completed the formal search, analysis, interpretation of data, appraisal of the studies, synthesis of results, manuscript writing and approval of the final version to be published. FG and MOC provided supervision to RI and both made substantial contribution to the study design, analysis, interpretation of data, appraisal of studies, synthesis of results, reviewing and editing of the manuscript, and approval of the final version to be published.
Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.
Competing interests None declared.
Provenance and peer review Not commissioned; externally peer reviewed.
Supplemental material This content has been supplied by the author(s). It has not been vetted by BMJ Publishing Group Limited (BMJ) and may not have been peer-reviewed. Any opinions or recommendations discussed are solely those of the author(s) and are not endorsed by BMJ. BMJ disclaims all liability and responsibility arising from any reliance placed on the content. Where the content includes any translated material, BMJ does not warrant the accuracy and reliability of the translations (including but not limited to local regulations, clinical guidelines, terminology, drug names and drug dosages), and is not responsible for any error and/or omissions arising from translation and adaptation or otherwise.