'It's Powerful' The impact of involving children and young people in developing paediatric research agendas: A qualitative interview study.

INTRODUCTION: There is a growing consensus that children and young people (CYP) should be involved in matters that concern them. Progress is made in involving CYP in developing pediatric research agendas (PRAs), although the impact of their involvement remains unknown. We aimed to evaluate the impact of involving CYP in developing PRAs and assess the extent to which postpatient and public involvement (post-PPI) activities were planned. METHODS: We conducted a qualitative study using in-depth interviews to identify and gain an in-depth understanding of the impact of involving CYP in developing PRAs. The transcripts were uploaded to Atlas.ti to be coded and organised. Dutch-language interviews were analysed and interpreted together with vocational education and training (VET) students. These students were aged between 14 and 18 years and were training to become nurses. RESULTS: Three CYP and 15 researchers decided to participate. We focused on three categories of impact: agenda-setting impact, individual impact and academic impact. Involving CYP creates a more enriched and clarified agenda. It ensured that both CYP and researchers underwent personal or professional growth and development, it created a connection between the people involved, awareness about the importance of involving CYP and it ensured that the people involved had a positive experience. The participants were unable to indicate the academic impact of their PRAs, but they did understand the key factors for creating it. In addition, the need to measure impact was highlighted, with a particular focus on assessing individual impact. DISCUSSION: Our study outlines the diverse subthemes of impact that arise from involving CYP in developing PRAs. Despite the potential of research agendas to amplify CYP voices, only a minority of researchers strategized post-PPI activities ensuring impactful outcomes, prompting the need for thorough evaluation of various impact forms and consistent alignment with the overarching goal of transforming the research field. PATIENT OR PUBLIC CONTRIBUTION: We involved VET students in the data analysis and interpretation phase by forming a young person advisory group. The data analysis of the interviews analysed by the VET students revealed four distinct themes: 1. Learnt new knowledge. 2. Learnt to collaborate. 3. Learnt to listen. 4. Assessment of the individual impact.

A Dutch paediatric palliative care guideline: a systematic review and evidence-based recommendations for symptom treatment.

BACKGROUND: Children with life-threatening and life-limiting conditions can experience high levels of suffering due to multiple distressing symptoms that result in poor quality of life and increase risk of long-term distress in their family members. High quality symptom treatment is needed for all these children and their families, even more so at the end-of-life. In this paper, we provide evidence-based recommendations for symptom treatment in paediatric palliative patients to optimize care. METHODS: A multidisciplinary panel of 56 experts in paediatric palliative care and nine (bereaved) parents was established to develop recommendations on symptom treatment in paediatric palliative care including anxiety and depression, delirium, dyspnoea, haematological symptoms, coughing, skin complaints, nausea and vomiting, neurological symptoms, pain, death rattle, fatigue, paediatric palliative sedation and forgoing hydration and nutrition. Recommendations were based on evidence from a systematic literature search, additional literature sources (such as guidelines), clinical expertise, and patient and family values. We used the GRADE methodology for appraisal of evidence. Parents were included in the guideline panel to ensure the representation of patient and family values. RESULTS: We included a total of 18 studies that reported on the effects of specific (non) pharmacological interventions to treat symptoms in paediatric palliative care. A few of these interventions showed significant improvement in symptom relief. This evidence could only (partly) answer eight out of 27 clinical questions. We included 29 guidelines and two textbooks as additional literature to deal with lack of evidence. In total, we formulated 221 recommendations on symptom treatment in paediatric palliative care based on evidence, additional literature, clinical expertise, and patient and family values. CONCLUSION: Even though available evidence on symptom-related paediatric palliative care interventions has increased, there still is a paucity of evidence in paediatric palliative care. We urge for international multidisciplinary multi-institutional collaboration to perform high-quality research and contribute to the optimization of symptom relief in palliative care for all children worldwide.

[Active life termination in children aged 1-12 years]. / Actieve levensbeëindiging bij kinderen van 1-12 jaar.

In this article we discuss active termination of life in children aged 1-12 years. It is expected that in 2024 active termination of life will be regulated for children between the ages of 1 and 12 who suffer unbearably, when other options to relieve their suffering are not enough. Using a case, we describe how a request for active termination of life in a child can arise and how physicians can respond to it.

How to move forward in shared decision-making in pediatric palliative care.

Pediatric palliative care has grown immensely in recent years in the world. However, shared decision-making remains a complex process, especially in pediatric palliative care. In particular, a number of issues are priorities to improve the shared decision-making process and ensure high-quality pediatric palliative care for every child. Working on these priorities will improve shared decision-making and thereby enhance high-quality pediatric palliative care around the globe.

Shared decision-making in pediatric palliative care in the Netherlands.

Shared decision-making (SDM) is a process in which health care professionals (HCPs) involve parents and children - when appropriate- to decide together on future treatment. These decisions are based on values that are important for the family, goals of care and preferences for future care and treatment. Elucidation of these values and preferences is preferably done early in the disease trajectory via so-called Advance Care Planning (ACP) conversations. In the Netherlands, ACP and SDM are being adopted by most health care professionals. This has happened only recently. Ten years ago, ACP and SDM were unknown concepts for the vast majority of Dutch HCPs. Today, interest in these conversational approaches is booming in both daily practice and in research. This rise has been reinforced by two recent major advancements in Dutch pediatric palliative care: the Individual Care Plan (ICP) and the Dutch Evidence-Based Guideline on Pediatric Palliative Care (DGPPC). Despite this positive evolution, a lot of work is still ahead. ACP and SDM demand a change in mindset from the traditional paternalistic approach by which the HCP 'knows what is best for this child' to a more humble and open approach in which (non-medical) factors that are important to the child and family and may influence the final treatment decision. Such changes in mindset don't happen overnight. In this article we describe the situation of pediatric palliative care in the Netherlands, with focus on the recent evolution of ACP and SDM.

Pediatric palliative care across continents: Communication and shared-decision-making.

Despite the significant growth and development of pediatric palliative care worldwide, significant challenges remain. One of those challenges is shared decision-making, by which parents, families and professionals all work together to develop a plan of care that reflects both the medical facts and the patient's family's values. Shared decision-making about palliative care and about death and dying may mean different things in different cultures and countries. It is therefore important to learn and compare practices around the world.

Palliative care for children: methodology for the development of a national clinical practice guideline.

BACKGROUND: Provision of paediatric palliative care for children with life-threatening or life-limiting conditions and their families is often complex. Guidelines can support professionals to deliver high quality care. Stakeholders expressed the need to update the first Dutch paediatric palliative care guideline with new scientific literature and new topics. This paper provides an overview of the methodology that is used for the revision of the Dutch paediatric palliative care guideline and a brief presentation of the identified evidence. METHODS: The revised paediatric palliative care guideline was developed with a multidisciplinary guideline panel of 72 experts in paediatric palliative care and nine (bereaved) parents of children with life-threatening or life-limiting conditions. The guideline covered multiple topics related to (refractory) symptom treatment, advance care planning and shared-decision making, organisation of care, psychosocial care, and loss and bereavement. We established six main working groups that formulated 38 clinical questions for which we identified evidence by updating two existing systematic literature searches. The GRADE (CERQual) methodology was used for appraisal of evidence. Furthermore, we searched for additional literature such as existing guidelines and textbooks to deal with lack of evidence. RESULTS: The two systematic literature searches yielded a total of 29 RCTs or systematic reviews of RCTs on paediatric palliative care interventions and 22 qualitative studies on barriers and facilitators of advance care planning and shared decision-making. We identified evidence for 14 out of 38 clinical questions. Furthermore, we were able to select additional literature (29 guidelines, two textbooks, and 10 systematic reviews) to deal with lack of evidence. CONCLUSIONS: The revised Dutch paediatric palliative care guideline addresses many topics. However, there is limited evidence to base recommendations upon. Our methodology will combine the existing evidence in scientific literature, additional literature, expert knowledge, and perspectives of patients and their families to provide recommendations.

Adults born prematurely prefer a periviability guideline that considers multiple prognostic factors beyond gestational age.

AIM: The aim of the study was to explore the perspectives of adults born prematurely on guidelines for management at extreme premature birth and personalisation at the limit of viability. METHODS: We conducted four 2-h online focus group interviews in the Netherlands. RESULTS: Twenty-three participants born prematurely were included in this study, ranging in age from 19 to 56 years and representing a variety of health outcomes. Participants shared their perspectives on different types of guidelines for managing extremely premature birth. They agreed that a guideline was necessary to prevent arbitrary treatment decisions and to avoid physician bias. All participants favoured a guideline that is based upon multiple prognostic factors beyond gestational age. They emphasised the importance of discretion, regardless of the type of guideline used. Discussions centred mainly on the heterogeneity of value judgements about outcomes after extreme premature birth. Participants defined personalisation as 'not just looking at numbers and statistics'. They associated personalisation mainly with information provision and decision-making. Participants stressed the importance of involving families in decision-making and taking their care needs seriously. CONCLUSION: Adults born prematurely prefer a periviability guideline that considers multiple prognostic factors and allows for discretion.

Neonatal euthanasia in the context of palliative and EoL care.

Neonatal deaths can be categorized in 5 modes along the dimension of intervention and physiology. This classification can be helpful to analyze the choices that can be made in end-of-life care in the NICU. In the Netherlands, neonatal euthanasia became an optional 6th mode of death since publication and legalization of the Groningen Protocol. This paper summarizes the history, legal status and ethical justification of the Groningen Protocol, and describes end-of-life practice in the subsequent years. Since the implementation of the Groningen Protocol, the practice of neonatal euthanasia has almost disappeared. Simultaneously, there has been spectacular growth in neonatal palliative care programs in the Netherlands. Is there still a need for this last-resort option?

Dutch professionals' discussion preferences with the parents of extremely premature infants varied, but the trend was towards shared decision-making.

AIM: We explored professionals' views on sharing decision-making with parents before and after an extremely preterm birth and what healthcare professionals considered severe outcomes. METHODS: A nationwide, multi-centre online survey was carried out among a wide range of perinatal healthcare professionals in the Netherlands from 4 November 2020 to 10 January 2021. The medical chairs of all nine Dutch Level III and IV perinatal centres helped to disseminate the survey link. RESULTS: We received 769 survey responses. Most respondents (53%) preferred to place equal emphasis on two treatment options during shared prenatal decision-making: early intensive care or palliative comfort care. The majority (61%) wanted to include a conditional intensive care trial as a third treatment option, but 25% disagreed. Most (78%) felt that healthcare professionals were responsible for initiating postnatal conversations to justify continuing or withdrawing neonatal intensive care if complications were associated with poor outcomes. Finally, 43% were satisfied with the current definitions of severe long-term outcomes, 41% were unsure and there were numerous for a broader definition. CONCLUSION: Although Dutch professionals expressed diverse preferences on how to reach decisions about extremely premature infants, we observed a trend towards shared decision-making with parents. These results could inform future guidelines.

Improvement and implementation of a national individual care plan in paediatric palliative care: a study protocol.

INTRODUCTION: Paediatric palliative care (PPC) is care for children with life-threatening or life-limiting conditions, and can involve complex high-tech care, which can last for months or years. In 2015, the National Individual Care Plan (ICP) for PPC was developed and has shown to be successful. The ICP can be seen as an instrument to facilitate coordination, quality and continuity of PPC. However, in practice, an ICP is often completed too late and for too few children. We aim to improve the coordination, quality and continuity of care for every child with a life-threatening or life-limiting condition and his/her family by further developing and implementing the ICP in the Netherlands. METHODS AND ANALYSIS: To evaluate the original ICP, ICP 1.0, interviews and questionnaires will be held among parents of children who have or have had an ICP 1.0 and healthcare professionals (HCPs) who used ICP 1.0. Based on the results, ICP 1.0 will be further developed. An implementation strategy will be written and the renewed ICP, ICP 2.0, will be nationally tested in an implementation period of approximately 7 months. During the implementation period, ICP 2.0 will be used for all children who are registered with Children's Palliative Care teams. After the implementation period, ICP 2.0 will be evaluated using interviews and questionnaires among parents of children who received ICP 2.0 and HPCs who worked with ICP 2.0. Based on these results, ICP 2.0 will be further optimised into the final version: ICP 3.0. ETHICS AND DISSEMINATION: This study received ethical approval. The ICP 3.0 will be disseminated through the Dutch Centre of Expertise in Children's Palliative Care, to ensure wide availability for the general public and HCPs within PPC. Additionally, we aim to publish study results in open-access, peer-reviewed journals and to present results at national and international scientific meetings.

Should the Dutch Law on Euthanasia Be Expanded to Include Children?

The 2002 Dutch Euthanasia law applies to patients aged 12 years and older. Developments in end-of-life care and decision-making in the last decade have sparked the debate about usefulness and necessity to extend euthanasia to include children under 12 years of age. This paper describes two opposite positions: the affirmative position of a pediatrician and expert in pediatric palliative care and the negative position of a jurist and specialist in health law.

The age limit for euthanasia requests in the Netherlands: a Delphi study among paediatric experts.

BACKGROUND: The Dutch Euthanasia Act applies to patients 12 years and older, which makes euthanasia for minors younger than 12 legally impossible. The issue under discussion specifically regards the capacity of minors to request euthanasia. OBJECTIVE: Gain insight in paediatric experts' views about which criteria are important to assess capacity, from what age minors can meet those criteria, what an assessment procedure should look like and what role parents should have. METHODS: A Delphi study with 16 experts (paediatricians, paediatric nurses and paediatric psychologists) who work in Children Comfort Teams in Dutch academic hospitals. The questionnaire contained statements concerning criteria for capacity and procedural criteria. Consensus was defined as ≥80% agreement. RESULTS: The experts agreed that five criteria for capacity, found in a previous literature study, are all important. They agreed that some children between ages 9 and 11 could meet all the criteria. Consensus was reached for the statements that the entire medical team should be involved in the decision making and that a second independent expert must assess the case. Experts agreed that the parents' opinion is relevant and should always be taken into account, but it need not be decisive. CONCLUSION: This study shows that the age limit of 12 years in the Dutch Euthanasia Act is too strong according to paediatric experts. Letting go of the age limit or lowering the age limit combined with adequate capacity assessment for minors younger than 12 are options that should be discussed further.

Intestinal Oxygenation and Survival After Surgery for Necrotizing Enterocolitis: An Observational Cohort Study.

OBJECTIVE: To assess whether regional intestinal oxygen saturation (rintSO2) and regional cerebral oxygen saturation (rcSO2) measurements aid in estimating survival of preterm infants after surgery for NEC. SUMMARY OF BACKGROUND DATA: Predicting survival after surgery for NEC is difficult yet of the utmost importance for counseling parents. METHODS: We retrospectively studied prospectively collected data of preterm infants with surgical NEC who had available rintSO2 and rcSO2 values measured via near-infrared spectroscopy 0-24 hours preoperatively. We calculated mean rintSO2 and rcSO2 for 60-120 minutes for each infant. We analyzed whether preoperative rintSO2 and rcSO2 differed between survivors and non-survivors, determined cut-off points, and assessed the added value to clinical variables. RESULTS: We included 22 infants, median gestational age 26.9 weeks [interquartile range (IQR): 26.3-28.4], median birth weight 1088 g [IQR: 730-1178]. Eleven infants died postoperatively. Preoperative rintSO2, but not rcSO2, was higher in survivors than in non-survivors [median: 63% (IQR: 42-68) vs 29% (IQR: 21-43), P < 0.01), with odds ratio for survival 4.1 (95% confidence interval, 1.2-13.9, P = 0.02) per 10% higher rintSO2. All infants with rintSO2 values of >53% survived, whereas all infants with rintSO2 <35% died. Median C-reactive protein [138 mg/L (IQR: 83-179) vs 73 mg/L (IQR: 12-98), P < 0.01), lactate [1.1 mmol/L (IQR: 1.0-1.6) vs 4.6 mmol/L (IQR: 2.8-8.0), P < 0.01], and fraction of inspired oxygen [25% (IQR: 21-31) vs 42% (IQR: 30-80), P < 0.01] differed between survivors and non-survivors. Only rintSO2 remained significant in the multiple regression model. CONCLUSIONS: Measuring rintSO2, but not rcSO2, seems of added value to clinical variables in estimating survival of preterm infants after surgery for NEC. This may help clinicians in deciding whether surgery is feasible and to better counsel parents about their infants' chances of survival.

Dutch guidelines on care for extremely premature infants: Navigating between personalisation and standardization.

OBJECTIVE: There is no international consensus on what type of guideline is preferred for care at the limit of viability. We aimed to conceptualize what type of guideline is preferred by Dutch healthcare professionals: 1) none; 2) gestational-age-based; 3) gestational-age-based-plus; or 4) prognosis-based via a survey instrument. Additional questions were asked to explore the grey zone and attitudes towards treatment variation. FINDING: 769 surveys were received. Most of the respondents (72.8%) preferred a gestational-age-based-plus guideline. Around 50% preferred 24+0/7 weeks gestational age as the lower limit of the grey zone, whereas 26+0/7 weeks was the most preferred upper limit. Professionals considered treatment variation acceptable when it is based upon parental values, but unacceptable when it is based upon the hospital's policy or the physician's opinion. CONCLUSION: In contrast to the current Dutch guideline, our results suggest that there is a preference to take into account individual factors besides gestational age.

Involving children and young people in paediatric research priority setting: a narrative review.

OBJECTIVE: The objective of this study is twofold: first, to describe the methods used when involving children and young people (CYP) in developing a paediatric research agenda and, second, to evaluate how the existing literature describes the impact of involving CYP. We distinguish three forms of impact: impact on the research agenda (focused impact), impact on researchers and CYP (diffuse impact) and impact on future research (research impact). DESIGN: A narrative review of MEDLINE, PsycINFO, Web of Science and Google Scholar was conducted from October 2016 to January 2022. The included studies involved at least one CYP in developing a research agenda and were published in English. RESULTS: 22 studies were included; the CYP involved were aged between 6 years and 25 years. Little variation was found in the methods used to involve them. The methods used were James Lind Alliance (JLA) approach (n=16), focus groups (n=2), workshop (n=2), research prioritisation by affected communities (n=1) and combined methods (n=1). Impact was rarely described: focused impact in nine studies, diffuse impact in zero studies and research impact in three studies. CONCLUSION: This study concludes that the JLA approach is most frequently used to involve CYP and that all methods used to involve them are rarely evaluated. It also concludes that the reported impact of involving CYPs is incomplete. This study implies that to convince sceptical researchers of the benefits of involving CYPs and to justify the costs, more attention should be paid to reporting these impacts.

Development of the Dutch Structure for Integrated Children's Palliative Care.

Children's palliative care (CPC) is gaining attention worldwide, facilitated by the exchange of knowledge during regular specialised congresses. This article describes the developments in the Netherlands over the past 15 years. The Foundation for Children's Palliative Expertise (PAL) was established as a nationwide initiative committed to improving palliative care for children countrywide. This led to the development of the first hospital-based CPC team in 2012, which expanded to a total of seven teams adjacent to children's university hospitals. Regional networks for CPC were developed in parallel to these teams from 2014 onwards. The networks are a collaboration of professionals from different disciplines and organisations, from hospital to homecare, and have covered the aspects of CPC nationally from 2019 onwards. They are connected through the Dutch Knowledge Centre for CPC. This centre was established in 2018 by the PAL Foundation in collaboration with the Dutch Association for Pediatrics. In 2013, the first evidence-based guideline, 'palliative care for children', provided access to knowledge for parents and healthcare providers, and in 2017, a format for an individual palliative care plan was established. Within the Knowledge Centre for CPC, a physician's support centre for dilemma's regarding the end of life of children was set up. The efforts to have children's palliative care embedded in the regular Dutch health care insurance are ongoing.