There is a global increase in the number of children living with chronic life-threatening illnesses [1,2]. In Singapore, child deaths (age <19 years) due to chronic conditions increased from 204 per year to 245 per year during the 2014 to 2016 period [3]. In fact, congenital anomalies as well as cardiovascular and cerebrovascular diseases account for over half of all deaths in the 0 to 19 years age range in Singapore [4]. Due to technological developments in medical care, children with chronic life-threatening illnesses can live longer, but they simultaneously face the challenge of being dependent on their caregivers (usually, their parents) and disability for a prolonged period [1]. However, not all seriously ill children and their parents receive adequate palliative support services [3].

Being a parent caregiver of a child with a chronic life-threatening illness is a highly stressful experience [5]. In addition to the typical challenges of parenting, parent caregivers must navigate a complex web of stressors, including the practical and financial burden of caregiving [6], strained marital and social relationships [7], and neglect of other healthy children and family members [8]. Furthermore, parent caregivers of children with chronic life-threatening illnesses need to frequently communicate and engage with medical professionals, but such interactions can increase the anxiety and distress of the parents if they are not adequately involved in making decisions regarding their child’s treatment [9,10]. Thus, the stressors associated with caregiving and the elevated demand on resources put parent caregivers at increased risk of depressive symptoms, fatigue, and overall poor quality of life [11].

Our research team recently conducted a qualitative systematic review of 25 high-quality research papers published between 2000 and 2017, exploring the lived experience of parental bereavement due to a child’s chronic life-threatening illness. A 4-phase parental bereavement trajectory of child loss was developed, highlighting appropriate interventions that help parents identify care needs, elicit caregiving strengths, enhance death preparedness, and foster meaning-making throughout the trajectory of their child’s illness to reduce psychoemotional distress during the period of end-of-life caregiving and into bereavement [12]. Our research team conducted a second study to examine the Asian experience of parental bereavement via meaning-oriented strength-focused interviews with 6 couples, 13 mothers, 4 fathers, and 2 primary parental figures (N=25 parental units) who lost their child to a chronic life-threatening illness in Singapore [13,14]. Grounded theory analysis revealed 7 themes and 25 subthemes that were organized into a trauma-to-transformation model of parental bereavement (Figure 1). This culture-specific model shows the milestones of how Asian bereaved parents journeyed through their child’s life-threatening illness and eventual death, describing the ritualistic actions as well as family communication and transactional patterns that aided them to better cope with their loss, to regain control over their lives, to sustain a continuing bond with their deceased child, and to move forward with and ultimately transcend their grief through meaning reconstruction [13,14].

These findings echo previous literature [15-18] positing that parents facing potential child loss could benefit from psychosocial and therapeutic interventions as soon as prognosis is known and throughout the trajectory of the illness, which could ease the transition from caregiving through mortality and bereavement, thus mitigating adverse grief outcomes (Table 1). However, most supportive interventions for parents caring for children with chronic life-threatening illnesses only occur after bereavement [19-21], and a recent systematic review found negligible evidence to support their effectiveness [22]. As such, there is a need to develop a preloss intervention to augment pediatric palliative care and parental bereavement support service—one that empowers parents to reflect on their caregiving experiences, explore and identify resources that could help them better cope with the challenges of caregiving, and support their child to live a meaningful life despite a chronic life-threatening illness.

In developing a preloss intervention that could meaningfully impact families throughout their child’s trajectory of illness and leading to the final days of their child’s life, a number of important therapeutic elements need to be considered and incorporated. First, anticipatory grief, defined as the process of mourning the loss of a loved one before actual loss that enables caregivers to experience and adjust to various grief responses, must be central to such an intervention [17,23]. Anticipatory grief can smoothen the process of coping with death as the individual has the scope to come to terms with the loss in advance [24]. Studies have found that strength-based end-of-life interventions with elements that address anticipatory grief can improve adult patients’ quality of life and mitigate poor bereavement outcomes among family caregivers [25]. It is, therefore, possible that an anticipatory grief-based psychotherapeutic intervention for parents of children with chronic life-threatening illnesses could help parents to understand and regulate their emotions, enhance death preparedness, and thereby build resilience.

Second, it would be useful for a preloss intervention for parents of children with chronic life-threatening illnesses to adopt a meaning reconstruction approach [26-28], with each individual actively constructing a phenomenological world of their experiences in relation to various familial and sociocultural contexts and supporting their sense of loss and grief accordingly. Such a meaning reconstruction approach empowers grievers to choose whether to direct their attention to the loss and process turbulent feelings or to focus on practical adjustments to re-engage with their everyday life. Third, a preloss intervention would benefit from a narrative approach [29,30], which could help individuals connect with emotions that are challenging to accept, generate new meaningful stories about life and loss, and restructure negative emotional appraisal of situations such as end-of-life caregiving into more positive ones, thereby generating a sense of hope.

Two effective examples of applying the meaning reconstruction approach and the narrative approach in supporting holistic end-of-life care are dignity therapy [25,31,32] and family dignity intervention [33]. Both of these are evidence-based psychotherapies that address the physical, psychosocial, and existential issues pertaining to one’s dignity at the end-of-life period. Specifically, a family dignity intervention is designed to support the collective experience of grief and loss for Asian families facing mortality, and it could add great value to a preloss intervention for parents of children with chronic life-threatening illnesses. In practice, family dignity intervention comprises a meaning-focused interview with a patient-and-family caregiver dyad that fosters the expression of appreciation and emotional connection through the retelling of important life narratives. The interview is recorded, transcribed, and edited into a legacy document and returned to the dyad for sharing with their loved ones for healing and remembrance.

Finally, a preloss intervention for parents of children with chronic life-threatening illnesses must be mindful of the caregiving responsibilities and limitations that serve as barriers for parents to engage in sit-and-talk therapy [12]. It is possible that a web-based platform that is cost-effective and time-efficient [34] can deliver psychotherapy to such parents. Such therapist-facilitated internet-based platforms are increasingly used for brief and effective psychotherapy for a range of conditions, including depression, anxiety, and stress [35-40]. Moreover, when web-based platforms use writing as the modality for emotional expression and reflection, efficacy is superior compared with audio or video mediums [41]. Finally, the anonymity of an e-writing channel can encourage greater willingness to self-disclose [42,43].

Globally, pediatric palliative care interventions predominantly emphasize the stages of grief and psychological tasks that grieving parents must accomplish after their child’s death [22], and in Singapore, there is no known empirically tested intervention to provide psychoemotional support and psychoeducational resources to parents of children with chronic life-threatening illnesses. Singapore is a leading nation in digital readiness, smartphone utilization for communication is ingrained into the everyday life of its people [44], and web-based intervention services for improving well-being have been welcomed by the Singaporean community [45,46]. Hence, it is reasonable to propose that web-based solutions are vitally useful for enhancing pediatric palliative care and parental bereavement support services. Furthermore, in view of the fact that Asian family caregivers can be uncomfortable with explicit emotional expression even during times of loss and separation [47], a web-based platform and the relative anonymity it offers to participants [43] would be appropriate for an Asian population.

The research team of this study integrated the aforementioned elements necessary for a preloss intervention for parents of children with chronic life-threatening illnesses and conceived a Narrative e-Writing Intervention (NeW-I) to address the gap in pediatric palliative care delivery and research in the local context. NeW-I is a novel web-based, therapist-facilitated, strength-focused, and meaning-oriented intervention designed to provide direct service to parents of children with chronic life-threatening illnesses. The development and evaluation of NeW-I is guided by the Medical Research Council Framework for the Development and Evaluation of Complex Interventions, which is widely recognized in the design and evaluation of complex interventions to improve health outcomes [48,49]. NeW-I is also inspired by the meaning reconstruction model [50], the narrative approach to anticipatory grief [51], dignity therapy [31,32], family dignity intervention for holistic end-of-life care [33], and the findings of a recent investigation on Asian parental bereavement experience of child loss by our research team [13,14].

The overarching goal of the novel NeW-I intervention model is to provide an accessible platform for parent caregivers to reflect on their experiences of caring for a child with a chronic life-threatening illness from a new perspective (ie, new eye) as well as to create a renewed sense of self-understanding and empowered identity in the context of their experience (ie, new I). The specific research objectives are three-fold: (1) to develop a standardized protocol for a cultural-specific and meaning-oriented NeW-I for anticipatory grief and bereavement support for parent caregivers of children with chronic life-threatening illnesses and eventual death; (2) to evaluate the efficacy of NeW-I in enhancing quality of life, spiritual well-being, hope, and social support as well as decreasing caregiver burden, depressive symptoms, and risk of complicated grief among parent caregivers; and (3) to examine the challenges and pitfalls in the design and implementation of NeW-I through an integrated feasibility and acceptability study for informing large-scale implementation of the intervention.

This study utilizes an open-label randomized controlled trial design comprising 2 groups: (1) an intervention group (structured NeW-I protocol) and (2) a control group (journaling activity unrelated to their child’s illness). It is hypothesized that intervention participants who successfully complete NeW-I will experience an enhanced quality of life, spiritual well-being, sense of hope, and perceived social support and decreased depressive symptoms, subjective caregiver burden, and risk of complicated grief as compared with control participants. It is also hypothesized that NeW-I is deemed as an accessible and user-friendly service by participants.

The sample comprises 72 individual parents of varying ethnicity in Singapore (N=72). Where both parents of a child participate in the study, their group allocation is randomly determined, and assessments are completed by both parents independently. To be eligible for participation in this study, the individual must be a parent whose child has been diagnosed with a chronic life-threatening illness and has a prognosis of more than 3 months at the time of enrollment. For the purpose of this study, a child has been defined as children and young persons between the ages of 0 and 19 years [52]. The individual must be able to speak, read, and write in English and provide informed consent. Individuals are excluded from this study if they are suffering from severe depressive symptoms and psychological distress, as identified by 2 screening tools. Specifically, to protect participants’ well-being during the pilot testing of NeW-I, those who do not meet the stated cutoff scores of the Patient Health Questionnaire – 9 (PHQ-9; >19) and Kessler psychological distress scale (K-10; >29) are excluded, as formal treatment and therapy would be more beneficial [53,54]. Additionally, if participants cease to meet the inclusion criteria during the study (eg, due to their child’s untimely death), they are excluded from the study and provided with alternative resources for psychosocial support. However, the data that have been collected until the time of their participation will be kept and analyzed so that a complete and comprehensive evaluation of the study will be possible.

For a main trial designed with 90% power and two-sided significance level of at least .05, a pilot sample size of 25 per arm is needed to detect a small effect size of 0.2 in the primary outcome measure [55]. A meta-analysis suggested that many high-quality psychotherapy studies (high quality studies defined by a proficiently trained therapist, treatment integrity, N>50) on the treatment of depression have a mean effect size of 0.22 [56]. Allowing for an attrition rate of 30% at follow-up (a larger estimate due to end-of-life context), the sample size must be inflated by a factor of 1/(1−0.3)=1.43. Therefore, the minimum sample size required for this study was 72 or 36 in each group.

Purposive sampling was adopted to achieve a target sample size of 72. Potential participants are identified and contacted by the collaborating organization (leading pediatric palliative care providers in Singapore, including KK Women’s and Children’s Hospital, Club Rainbow Singapore, Muscular Dystrophy Association Singapore, and Rare Disorders Society Singapore) to introduce the study to their beneficiaries. This sampling strategy allows the recruitment of participants whose children suffer from a wide range of chronic life-threatening conditions, thus allowing for maximum variation in the sampling. If verbal consent is obtained from potential participants, their contact details are passed to the research team at Nanyang Technological University, who subsequently establish telephone contact, explain study procedures, and introduce the NeW-I web-based platform. All personal information pertaining to potential participants are kept confidential, and only the responsible researchers have access to such information.

Open recruitment is also carried out so that all parents of children with chronic life-threatening illnesses have equal opportunity to participate in a potentially beneficial study and to examine the feasibility of implementing this free and easily accessible intervention in the community. Posters have been placed in strategic locations across Singapore (such as offices of leading pediatric palliative care providers) that provide information regarding the study. When interested participants contact the research team, the study procedures are explained, the NeW-I platform is introduced, and registration information is provided.

The NeW-I therapist-facilitated web-based platform comprises a mobile app and a website (both of which have the same content and functionality). Participants can download the app free of cost from the Apple App Store and the Google Play Store by keying in the relevant keywords or scanning the quick response code provided on the NeW-I study advertisement posters. The NeW-I website is under construction. As shown in Multimedia Appendix 1, when participants initially log in to the app, they are directed to a study participation and informed consent page that provides details about study procedures, institutional affiliations of the research team, rights of research participants, and protection of confidentiality. After participants endorse this web-based informed consent form on the NeW-I platform, they are directed to a demographic information page. Participants are also requested to provide their contact details and last few digits of their national identification number to ensure that only 1 user account is created by each individual.

Following this, participants are directed to a screening page where participants complete the PHQ-9 and the K-10. Those who pass the screening assessments receive confirmation of study participation and are requested to wait for a phone call from the research team. Those who do not pass the screening assessments are thanked for their time and provided with resources for psychosocial support. Individuals who pass the screening assessments receive a phone call from the NeW-I team as a means of identity checking. Following this, participants complete baseline measures (time point 1 assessment). This is followed by a random allocation of participants to either the intervention group or the control group, which is performed via the NeW-I platform using computer-generated random numbers. Participants are then directed to the first writing session. Participants have the option to choose to begin the first writing session immediately or delay for a maximum of 3 days. The day on which participants begin their first writing session is day 1 of week 1. Participants who do not begin their first writing session within 3 days are excluded from the study. However, a concession period of 7 days is given to participants who have a genuine reason for their inability to adhere to the protocol (such as unexpected changes in their child’s health condition) and have expressed keen interest in participating in the study. A similar concession period of 7 days is also maintained for participants who are unable to adhere to the study protocol in weeks 2, 3, and 4 (such that each participant can only be given 1 concession during the entire duration of their study participation). Detailed study procedures are described in Figure 2.

NeW-I is delivered by trained therapists in the research team who are experts in death education and grief counseling and have the clinical competence to work with family caregivers in pediatric palliative settings. All team members have successfully completed research integrity modules under the provisions of Nanyang Technological University’s institutional review board and adhere to the board’s guidelines for safeguarding participants’ identity and confidentiality.

Both intervention and control group participants follow the procedures described in Figure 2. There are 4 weekly sessions of writing. A template has been provided to ensure that the participants’ writings tie in with the session objectives. To improve participants’ adherence to the study protocol, they receive an automated notification on their phone app and email each time a fresh writing session becomes available to them. Participants were assured of anonymity and confidentiality of their writing to encourage open and honest self-expression. The structured writing for each session requires 15 to 30 min, as exposure and time to process ideas through written disclosure over at least three sessions of 15 min each can produce effective outcomes [57].

For the intervention group participants, each weekly session has a unique objective (Table 2). Briefly, in week 1, participants reflect on the demands of caring for a child with a chronic life-threatening illness and the means to cope with these challenges. In week 2, participants consider avenues where they could seek more information about their child’s illness and resources for caregiving. In week 3, participants examine the sources of support that they have within their network of family and friends. In week 4, participants explore how they (and their children) could rise above illness-related challenges and live their lives as fully as possible.

For control group participants, the objective is consistent across the 4 weeks, that is, participants engage in a weekly writing session that is unrelated to their child’s illness and allows them to experience the therapeutic benefits of narrative writing (Table 3).

Table 3

Content and questions for reflective writing for control group.

This study has been approved by the institutional review board of Nanyang Technological University, Singapore (IRB-2018-07-009). Web-based, endorsed informed consent was obtained from all participants before study participation. Participants’ confidentiality, safety from unintended outcomes, and right to withdraw without any adverse consequences are safeguarded under the ethical provisions of Human Biomedical Research Act studies reviewed by the institutional review board of Nanyang Technological University, Singapore.

As such, there is minimal risk of engaging in a web-based narrative writing activity. The experienced NeW-I therapist will be available to participants to offer web-based support, in the event that some aspects of the intervention cause them distress or discomfort. If participants need further support, a referral system has been set up such that participants who are recruited via purposive sampling would be referred to their health and social care provider for follow-up assistance. Finally, any deviations from or changes to the study protocol, unexpected breaches in privacy, or major technical difficulties will be promptly reported to the institutional review board of Nanyang Technological University, Singapore, and further steps will be taken after seeking advice from the board.

NeW-I is a first-of-its-kind, web-based, therapist-facilitated, strength-focused, and meaning-oriented intervention for parents of children with chronic life-threatening illnesses, thereby filling in a critical service gap in local pediatric palliative care. Through an open-label randomized control trial, the efficacy of NeW-I for improving such parents’ quality of life, spiritual well-being, hope, and social support and decreasing depressive symptoms, caregiver burden, and risk of complicated grief is investigated across 5 time points. It is expected that parents who successfully complete the structured NeW-I intervention protocol will experience enhanced quality of life, spiritual well-being, sense of hope, and perceived social support and decreased depressive symptoms, subjective caregiver burden, and risk of complicated grief as compared with parents who engage in a journaling activity unrelated to their child’s illness (control group). It is also expected that parents will find NeW-I to be an accessible and user-friendly service.

In addition to improving the psychosociospiritual well-being of parent caregivers, this study creates opportunities for (1) a longitudinal assessment of the mental states of 36 parents (ie, control group) to obtain a naturalistic trajectory of anticipatory grief, (2) an evaluation of the extent to which the intervention is successful in improving the mental well-being of 36 parents (ie, intervention group), and (3) an evaluation of the extent to which these potential positive effects are sustained over time. The findings will inform the development of a full-scale NeW-I protocol that will be disseminated via research papers and presentations. This will form the foundation for further empirical research to test the effectiveness, acceptability, and feasibility of NeW-I in Singapore and in other Asian communities around the world.

The web-based narrative writing model of NeW-I and the relative anonymity it offers to participants [43] supports the unique needs of Asian family caregivers who are uncomfortable with emotional expression even during times of loss and separation [47]. It is hoped that NeW-I is perceived by parents to be a safe platform for engaging in intimate dialogue regarding their child’s caregiving, thereby enhancing parents’ experience of their child’s illness trajectory, empowering them to harness available resources to provide the best possible care to their child, while simultaneously reducing psychosocial distress and caregiver burden. Evidence from a recent systematic review [71] suggests that the guided web-based intervention protocol of NeW-I is likely to be cost-effective. Additionally, the web-based intervention protocol is convenient to access, allows expression of disenfranchised emotions, and promotes meaning ascription to traumatic experiences. Finally, the current format of NeW-I is tailored for parents of children with chronic life-threatening illnesses. However, after a detailed examination of the structural and implementation strengths and challenges of NeW-I, its effectiveness in enhancing mental health as well as feasibility and accessibility, the web-based therapeutic protocol can be adapted to deliver psychotherapy to diverse populations including young adults who are diagnosed with a life-limiting condition, siblings of terminally ill young persons, and caregivers of patients with dementia, to name a few.

Presently, NeW-I can only be implemented with participants who speak, read, and write English. Singapore is a multicultural and multilinguistic nation [72], and future research should expand the delivery language of NeW-I and assess its acceptability and effectiveness among different linguistic and ethnic groups in Singapore and globally. Despite this limitation, NeW-I could enhance participants’ wellness by drawing attention away from their illness narrative and instead emphasize areas that research has demonstrated to be most meaningful at the end of life. Expected study outcomes can generate new knowledge to inform research and practice in pediatric palliative care and parental bereavement support locally and globally.