Paediatric End-of-LIfe CAre Needs in Switzerland (PELICAN) : Current end-of-life care practices and the persepctives of bereaved parents

Despite continued advancements in medical care and improved survival or life expectancy, childhood deaths due to complex chronic conditions (CCC) or prematurity are inevitable. Deaths during the first year of life constitute approximately 50% of disease-related deaths, the causes of which include perinatal complications, prematurity, or congenital anomalies. Beyond the age of one year, the three most common life-limiting CCCs are neurological/neuromuscular and cardiovascular conditions (including genetic disorders), and malignancies. The majority of disease- and prematurity-related deaths occur in hospitals and for children dying at home, hospital use in their terminal stage is high. Symptom burden and reliance on medical technology has been reported to be considerable. Circumstances and characteristics of deaths, however, are known to vary by age and medical conditions.
When facing the death of their child, parents experience an unimaginably painful life event and severe crisis that affects the whole family for life. In this highly stressful time parents are confronted with uncertainty and are required to make difficult decisions, e.g. withdrawal of life-sustaining interventions. Their need for compassionate professional support is high. Paediatric palliative (PPC) and end-of-life (EOL) care emerged as a medical subspecialty aimed at meeting the specific needs of seriously ill children and their families. Meeting these needs requires a comprehensive and integrative approach from a compassionate and skilled multidisciplinary team. Parental needs have been studied in the past and an overview of themes/domains most important to parents can be summarised as: sincere relationships and emotional, spiritual and cultural support; genuine communication; alleviation of suffering; continuity, coordination and accessibility of care; and bereavement support. Deficiencies in meeting parental needs were identified across all themes, e.g. insufficient communication, lack of respect, and lack of emotional support.
Most of the evidence related to the parental perspective of their child’s EOL care originates from qualitative research. To assess and explore the parental perspective effectively, an approach combining quantitative and qualitative information is likely to provide the most comprehensive view of experiences and unmet needs. A few self-administered questionnaires designed to assess the experiences and needs of parents of a dying child exist. None of the instruments available were applicable to the heterogeneous field of paediatric EOL care, including children of different age groups, with different underlying illnesses and in different clinical settings.
The development of PPC in Switzerland lags behind when compared with other developed countries, such as the United Kingdom or the USA. Need for action has been recognised by the Federal Office of Public Health and the call for a thorough assessment of current practices, needs, available resources and existing services as requirement to formulate and plan further measures to promote PPC in Switzerland has been issued.
This dissertation is embedded in the PELICAN study (Paediatric End-of-LIfe CAre Needs in Switzerland, 2012 – 2015). The overarching aims of the nationwide PELICAN study were to provide comprehensive information and to understand the current practice of EOL care (i.e. in this study, the last 4 weeks of life prior to death) in paediatric settings in Switzerland (hospital and community care) and to explore and describe parental perspectives and the perspectives of the healthcare professionals involved. Based on the results, recommendations for best-practice during this highly vulnerable and critical phase of life not only for the affected child but also for the family will be formulated.
The dissertation is organized in 9 chapters:
Chapter 1 introduces in the field of paediatric EOL care and reviews relevant topics. The epidemiology of childhood illness- and prematurity-related death is described together with characteristics and circumstances of death. PPC and EOL are being defined and the he provision of paediatric palliative and EOL care are introduced. Existing evidence about the parental perspective of their child’s EOL care is summarised, and a potential framework for the quantitative assessment of parental experiences and needs is described. The emerging research gaps, and the rational and aims of this dissertation are described in chapter 2 together with the context in which the PELICAN study took place.
Chapter 3 comprises the PELICAN study protocol. It provides an introduction, rational and specific aims of the entire research project including three main studies and two subs-studies combining quantitative and qualitative methodology. Methods for each study arm are detailed. This dissertation comprises PELICAN I and the quantitative part of PELICAN II, including the development and testing of the Parental PELICAN Questionnaire (PaPEQu).
Chapter 4 reports on patterns of care at EOL in neonates and children with complex chronic conditions. Data from 149 paediatric patients who died in the years 2011 or 2012 due to a cardiac, neurological or oncological condition, or during the neonatal period were collected in 13 hospitals, two long-term institutions and 10 community-based healthcare service providers throughout Switzerland. Sixty-two percent of the patients died in intensive care units, 84% of them following the withdrawal of life-sustaining treatment. Reliance on invasive medical interventions was prevalent, and the use of medication was high, with a median count of 12 different drugs during the last week of life. Patients experienced an average number of 6.42 symptoms. The prevalence of various types of symptoms differed significantly among the four diagnostic groups. Only half of the patients who spent days at home received community-based healthcare. The study provides a comprehensive overview of current EOL care practices in a real-life setting of different healthcare providers and the findings provide a knowledge base for paediatric palliative care teams.
Chapter 5 describes the development and initial validation of the Parental PELICAN Questionnaire (PaPEQu) – an instrument to assess parental experiences and needs during their child’s EOL care. The PaPEQu was developed in four phases between August 2012–March 2014: phase 1: item generation; phase 2: validity testing; phase 3: translation; phase 4: pilot testing. Psychometric properties were assessed after applying the PaPEQu in a sample of 224 bereaved parents in April 2014. Validity testing covered the evidence based on tests of content, internal structure and relations to other variables. The PaPEQu consists of approximately 90 items in four slightly different versions accounting for particularities of the four diagnostic groups. The questionnaire’s items were structured according to six quality domains described in the literature. Evidence of initial validity and reliability could be demonstrated with the involvement of healthcare professionals and bereaved parents. The PaPEQu holds promise as a measure to assess parental experiences and needs and is applicable to a broad range of paediatric specialties and settings.
Chapter 6 reports on the results of the survey with the newly developed PaPEQu. Responses regarding parental experiences and perceived satisfaction are described. Differences between the four diagnostic groups are analysed using a generalized estimation equation to account for the dyadic data structure. Of 307 eligible families, 267 could be contacted and 135 (51 %) consented to participate in this questionnaire survey. Our findings show positive parental experiences of their child’s EOL care and high perceived satisfaction with the care their child received. Parents of a child with cancer rated their experiences highest in most of the six quality domains and reported the highest satisfaction with care. The lowest scores were mainly reported by parents from the neurology group, with the exception of the shared decision making domain, where parents of neonates reported significantly less positive experiences. Although positive in general, our study results suggest some areas for improvement. The integration of specialised paediatric palliative care has the potential to minimise lost opportunities to support and assist parents.
In chapter 7 finally, the results of all studies are being synthesized and discussed within the state of science. Strengths and weaknesses of methods are examined and implications on the level of policy, practice and research are derived. This dissertation concludes with the recognition that urgently needed data to advance the development of PPC in Switzerland is now available. It also recognises however, that we are challenged to demonstrate the effectiveness of PPC services in order to grow out of the infancy of this medical subspecialty.