The NeW-I study protocol is guided by the SPIRIT 2013 checklist for reporting of clinical trial protocols [41] (see Figure 1 for the corresponding Standard Protocol Items: Recommendations for Interventional Trials (SPIRIT) Figure).
[Insert Figure 1 here]
Aims, Objectives and Hypothesis
The development and evaluation of NeW-I is informed by the medical research council framework for the development and evaluation of complex interventions [39, 40], the meaning-reconstruction model [42], the narrative approach to anticipatory grief [43], dignity therapy [27, 28], and family dignity intervention for holistic end-of-life care [29], and guided by the findings of a recent investigation on Asian parental bereavement experience of child loss by our research team [10]. The objectives of this study are:
- to develop a detailed protocol for a culture-specific and meaning-oriented Narrative e-Writing Intervention (NeW-I) for anticipatory grief and bereavement support for parents facing their child’s chronic life-threatening illness and impending death;
- to evaluate the efficacy of NeW-I in enhancing quality of life, spiritual well-being, hope and perceived social support, and decreasing depressive symptoms, caregiver burden and risk of complicated grief among participants; and
- to examine positives and challenges in the design and implementation of NeW-I through a built-in accessibility and feasibility study for establishing a solid foundation towards full-scale implementation.
It is hypothesized that intervention group participants who successfully complete NeW-I 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 to control group participants. It is also hypothesized that NeW-I is deemed an accessible and user-friendly service by participants.
Study Design
This study adopts an open-label randomized controlled trial design comprising two groups: (1) an intervention group (structured NeW-I protocol) and (2) a control group (unstructured journaling activity).
Sample
The sample comprises 66 parents of varying ethnicity in Singapore (N = 66). 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 between the ages of 0–19 years and has a prognosis of more than 3 months at the time of study registration. The individual must be able to speak, read, and write in English and provide informed consent. Individuals are excluded from this study if they cannot provide informed consent and are suffering from severe depressive symptoms and psychological distress as identified by two screening tools. Specifically, to protect participants’ well-being during the pilot testing of NeW-I, those who do not meet the stated cut-off scores of Patient Health Questionnaire–9 (>19) and Kessler Psychological Distress Scale (>29) are excluded as formal treatment and therapy would be more beneficial [44, 45].Additionally, if participants cease to meet the inclusion criteria during the study (such as, due to their child’s untimely death), they are excluded from the study and provided alternative resources for psychosocial support.
Sample Size Calculation
For a main trial designed with 90% power and two-sided 5% significance, a pilot sample size of 25 per arm is needed to detect a small effect size of 0.2 in the primary outcome measure [46]. In fact, many high quality psychotherapy studies (as defined by proficiently trained therapist, treatment integrity, N ≥ 50) for the treatment of depression have a mean effect size of d = 0.22 [47]. Allowing for an attrition rate of 30% at follow-up (a larger estimate due to end-of-life context), the minimal sample size required for this study is 66, or 33 in each group.
Study Sites and Recruitment Procedures
In collaboration with 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, purposive sampling is adopted to achieve the target sample size of 66. Potential participants are identified and contacted by the relevant collaborating organization to introduce the study. If verbal consent is obtained from potential participants, their contact details are passed to the NTU research team who subsequently establish telephone contact, explain study procedures and introduce the NeW-I online platform. All personal information pertaining to potential participants is 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 illness 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 are placed in strategic locations across Singapore providing information regarding the study. When interested participants contact the research team, study procedures are explained, the NeW-I platform is introduced, and registration information is provided.
App and Intervention Procedure
The NeW-I therapist-facilitated online platform comprises a mobile app and a website. When participants initially log on to the app or website, they are directed to a study participation and informed consent page that provides details about study procedures, rights of research participants and protection of confidentiality. After participants endorse this online informed consent form on the NeW-I platform, they are directed to a demographic information page. This is followed by 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 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. This is followed by random allocation of participants to either the intervention or the control group which is done via the NeW-I platform by using computer-generated random numbers. Following this, participants complete baseline measures (T1 assessment) and are then directed to the first writing session. Participants may choose to begin the T1 assessment and first writing session immediately, or delay for a maximum of 3 days. The day on which participants begin the writing session is day 1 of week 1. Study procedures are described in detail in Figure 2.
[Insert Figure 2 here]
Intervention and Control Group
Both intervention and control group participants follow the procedures described in Table 2. There are 4 weekly sessions of writing. A template is provided to ensure that participants’ writings are structured and 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 are 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 minutes, since exposure and time to process ideas through written disclosure over at least 3 sessions of 15 minutes each can produce effective outcomes [48].
[Insert Table 2 here]
For intervention group participants, each weekly session has a unique objective (see Table 3). Briefly, in week 1, participants reflect on the demands of caring for a child with chronic life-threatening illness, and the means to cope with these challenges. In week 2, participants consider avenues where they can seek more information about their child’s illness and resources for caregiving. In week 3, participants examine the sources of support which they have within their network of family and friends. In week 4, participants explore how they (and their children) can rise above illness-related challenges and live their lives as fully as possible.
[Insert Table 3 here]
For control group participants, the objective is consistent across the 4 weeks, that is, participants engage in a weekly unstructured writing session which allows them to experience the therapeutic benefits of unstructured narrative writing (see Table 4).
[Insert Table 4 here]
Evaluation of Outcomes
Quantitative assessments. Via the NeW-I platform, both intervention and control group participants fill out a socio-demographic form at baseline, and are then assessed on a battery of standardized and validated measures across 5 time-points. This includes: (i) Kemp Quality of Life Scale [49], (ii) a modified version of the Functional Assessment of Chronic Illness Therapy- Spiritual Well-Being [51], (iii) Herth Hope Index [50], (iv) Patient Health Questionnaire–9 [44], (v) Burden Scale for Family Caregivers-Short [51], (vi) Inventory of Social Support [52] and (vi) a modified version of the Brief Grief Questionnaire [53] (see Table 5).
[Insert Table 5 here]
Assessment for both intervention and control group takes place at five time-points: baseline (T1), immediately after completion of the intervention or control protocol (T2), one month after completion of the intervention or control protocol (T3), three months after completion of the intervention or control protocol (T4), and a final follow-up six months after completion of the intervention or control protocol (T5) (described in both Figure 1 and Figure 2). Participants receive an automated notification on their phone app and email each time an assessment set becomes available to them. For each completed assessment, participants receive a 30 SGD voucher.
Accessibility study. To evaluate the accessibility and effectiveness of NeW-I, all intervention group participants are invited to participate in a written interview at the completion of all intervention components at T2, which explores the impact of the intervention, aspects of the intervention found to be helpful, aspects of the intervention found to be unhelpful and how they could be improved, challenges encountered in completing the intervention, and scope for enhancing intervention usability.
Feasibility study. To maintain a detailed audit trail and to assess the implementation and delivery of NeW-I, the research team records the time needed to provide feedback to participants and restructure their narrative writing, deviations from the intervention protocol (if any), uncompleted interventions and their reasons, and NeW-I therapists’ perceptions of competence, intervention delivery, observations of participants’ experiences and responses, and difficult or deviant cases. All feedback provided to participants is vetted by at least two members of the research team for data monitoring, quality and safety assurance.
Data Analysis
Quantitative data. All quantitative data are stored and analyzed using the SPSS computer software. Reliability tests are reported for all scales with the sample population. Between-and within-participant comparison of outcomes are conducted, and appropriate effect size estimates are reported. Further, comparisons are done at T3, T4 and T5 follow-up with baseline for both groups to assess longitudinal outcomes and potential maintenance effects. To characterize and predict changes in outcome variables, multilevel analyses are conducted. The intervention and control group are also compared on demographic characteristics and baseline measures. If necessary, these are controlled in the analyses. Deviant cases which could impact intervention outcomes, recruitment rates, comparison of drop-out rates and missing data in the two groups are reported.
Qualitative data. All qualitative data are stored and analyzed using the QSR NVivo computer software. Phenomenological analysis is used to obtain an in-depth and comprehensive view of the dataset from an insider perspective [56]. Unique or minority voices are elicited to illuminate counterpoints to the stated views. Throughout the data analysis process, strategies to maximize research rigor and trustworthiness are prioritized. The use of such a method of data analysis has been demonstrated in previous research involving grief therapy with bereaved parents [54].