Participants will be randomized to either the "Storytelling Through Music" (STM) experimental arm or the waitlist control arm. STM is a six-week intervention that utilizes storytelling, reflective writing, self-care skills (i.e., breathing exercises, meditation, self-compassion, body scans), and songwriting. The intervention is delivered online and in a group setting. Online delivery provides convenience to participants, and the group setting provides an environment of people with shared experiences, which has been shown to decrease social isolation and feelings of being alone in their emotional experience. By the end of the intervention, participants will have written a short story and a song based on their story.

Participants will receive the same "Storytelling Through Music" intervention once data collection from the experimental arm is complete.

"Storytelling Through Music" is a six-week expressive arts intervention.

Storytelling Through Music

Carolyn Phillips

Carolyn Phillips, PhD

The death of a child, at any age, is considered one of the most stressful life events a person can experience. In 2020, 11,050 children (under 15 years), 5,000 adolescents (15-19 years) and 60,000 young adults (20-39 years) were estimated to be diagnosed with cancer in the US. While the five-year survival is better for children than adults, over 10,000 children, adolescents, and young adults die from cancer in the US each year.1 Bereaved parents often experience intense and lasting psychological distress resulting in significantly higher morbidity and mortality compared to non-bereaved parents. Twenty-five percent of bereaved parents report new diagnoses of illnesses including prediabetes, anxiety and sleep disorders. Bereaved parents also experience psychological distress such as anxiety, post-traumatic stress disorder, and grief-related depressive symptoms that continue to be significant for years after a child's death. A recent study showed that nearly 33% of bereaved parents suffered from prolonged grief five years after their loss.6 Physiologically, studies show increased cortisol, immune, endocrine, and cardio biomarkers in people with prolonged grief. The death of a child can also affect family and social relationships resulting in decreased communication, feelings of isolation, absence of close social relationships and increased marital strain and divorce.

The purpose of this randomized controlled pilot study is to evaluate the feasibility of implementing a six-week multi-dimensional intervention, Storytelling Through Music (STM), with parents of children who have died from cancer. STM combines multiple modalities of expression (storytelling, writing, and music) to facilitate loss- and restoration-oriented coping by creating a legacy piece (self-written story paired with song) to facilitate continuing bonds with the deceased and find meaning.

The death of a child, at any age, is considered one of the most stressful life events a person can experience. In 2020, 11,050 children (under 15 years), 5,000 adolescents (15-19 years), and 60,000 young adults (20-39 years) were estimated to be diagnosed with cancer in the US. While the five-year survival is better for children than adults, over 10,000 children, adolescents, and young adults die from cancer in the US each year. Bereaved parents often experience intense and lasting psychological distress resulting in significantly higher morbidity and mortality than non-bereaved parents. Twenty-five percent of bereaved parents report new diagnoses of illnesses, including prediabetes, anxiety, and sleep disorders. Bereaved parents also experience psychological distress, such as anxiety, post-traumatic stress disorder, and grief-related depressive symptoms that continue to be significant for years after a child's death. A recent study showed that nearly 33% of bereaved parents suffered from prolonged grief five years after their loss. Physiologically, studies show increased cortisol, immune, endocrine, and cardio biomarkers in people with prolonged grief. The death of a child can also affect family and social relationships resulting in decreased communication, feelings of isolation, absence of close social relationships, and increased marital strain and divorce.

The World Health Organization and the National Coalition for Hospice and Palliative Care (NCHPC) advocate that palliative care should not only improve the quality of life of patients but also extend into bereavement for families. The NCHPC bereavement guideline (7.5.1.c) states that bereavement interventions should include rituals that acknowledge loss and transition, provide opportunities for remembrance, and establish a sense of community. Researchers have examined the use of life review, dignity therapy, and remembrance with pediatric and adolescent patients, as well as bereaved family caregivers of spouses. However, to our knowledge, none have been conducted with bereaved parents. Despite the high risk of negative outcomes and national guidelines recommending bereavement care, the resources for bereaved parents are scarce. In a recent systematic review of intervention studies for bereaved parents, only fifteen interventions were identified. Of those studies, most lacked empirical evidence of effectiveness or alignment with key theoretical concepts. To increase the number of effective resources for this vulnerable and underserved population, interventions need to be developed and tested in order to promote health and disease prevention in this high-risk population.

Mechanisms of Coping with Parental Grief

Parental bereavement is complex because many personal, relational, and end-of-life circumstances affect bereavement, and individuals cope differently. Several factors are associated with prolonged grief and poorer psychosocial outcomes, including intra-personal (i.e., attachment style, sex, religious beliefs, age, history of mental health problems), inter-personal (i.e., social support, family, culture, religious practice, resources), and the unexpectedness of the loss. However, none of these factors are easily changed by interventions. Focusing on modifiable processes that mediate or moderate the adaptation trajectory in bereavement may be more beneficial. In bereaved adults, processes that mediate the relationship between risk factors and mental health outcomes include rumination, deliberate grief avoidance, emotional expression, cognitive appraisals, and meaning-making.

Meta-Affective and Meta-Cognitive Effects of Grief

A growing body of research suggests that self-compassion is positively associated with well-being and negatively associated with depression, anxiety, and post-traumatic stress. Self-compassion recognizes suffering as a universally shared human experience and teaches people to face their suffering non-judgmentally with a kind and mindful approach. Only one study has examined the influence of self-compassion on grief processing, showing a significant relationship between low self-compassion and the severity of complicated grief. Self-compassion may be beneficial in coping with grief because it is associated with engagement in, rather than avoidance of, painful thoughts, memories, and feelings. Furthermore, research on meta-cognition has shown that maladaptive coping strategies such as rumination are driven by metacognitive appraisals of an internal or external event. Meta-cognitive beliefs may keep bereaved people focused on loss issues, preventing them from integrating the loss into their lives and planning for the future.

Affective and Cognitive Effects of Expressive Arts

Expressive arts have been used to improve psychosocial well-being in people with cancer, adolescents with grief, veterans with post-traumatic stress disorder, and to aid bereavement among family caregivers. Yet, many of these studies lack methodological rigor. Interventions aimed at meaning-making are good for individuals at high risk for prolonged grief. Music has been used across cultures, and there is growing evidence that music is often more powerful than language alone in eliciting emotion, is processed throughout spinal, subcortical, and cortical regions, and thus has meaningful impacts on complex cognitive and affective processes. While music and language utilize similar features in the brain, music is more rooted in the primitive brain structures involved in motivation, reward, and emotion. Within the brain, emotional, language, and memory centers are connected during music processing.

Theoretical/Conceptual Framework

Two complementary models guide this study: The Dual-Process Model of Coping with Bereavement and the Meaning Reconstruction Model. Both models view grief as a life-long process of renegotiating continuing bonds with the deceased and finding meaning in life after the loss. The dual-process model posits that grieving a loved one entails oscillating between orientation to the loss (i.e., continuing bonds with the deceased by expressing emotion related to the death and reconnecting with the memory of the loved one) and restoration of contact with a changed world (i.e., re-engaging relationships and experimenting with new life roles). The meaning reconstruction model of grief views grieving as a process of reaffirming or reforming a world of meaning that has been challenged by loss. Research on these models demonstrates signs of efficacy, particularly regarding how continuing bonds with the deceased and meaning-making are important mechanisms of successful adaption to bereavement.

Preliminary Work

This team has implemented two pilot studies examining the in-person and online delivery of STM to professional caregivers. In both studies, the intervention delivery method was feasible and significant improvements were seen in coping (self-compassion (F(3, 105) = 2.88, p\<.05), self-awareness (F(3, 120) = 2.42, p\<.10), psychosocial (loneliness (F(3, 98) = 7.46, p\<.001), and functional (insomnia (F(3, 120) = 5.77, p\<.001) well-being. Qualitatively, participants reported feeling less emotional loneliness, and the stories and songs provided reflection and meaning-making. An unexpected finding from this study was that 60% of participants in the intervention arm had experienced a significant family loss (mostly to cancer) that inspired their oncology nursing careers. During the intervention, this primary family loss, with the grief they needed to examine. This finding informed our team of the need for bereavement interventions for family caregivers.

Feasibility Study of the Storytelling Through Music Intervention With Bereaved Parents

A Feasibility Study Evaluating Storytelling Through Music Intervention With Bereaved Parents of Children With Cancer

Feasibility will be evaluated by tracking the percentage of participants screened and enrolled. The study will be deemed feasible with a 60% enrollment rate.

Feasibility will be evaluated by tracking the average number of sessions the participants complete. This intervention will be deemed feasible if 85% of the intervention is completed.

An additional primary endpoint is acceptability. Acceptability will be evaluated with semi-structured interview questions to understand the participant's perception of intervention delivery and content, as well as the perceived impact.

The Patient-Reported Outcomes Measurement Information System (PROMIS) Emotional Distress-Anxiety Short-Form 8a will be used. The minimum score is 8, and the maximum score is 40. Higher scores indicate more anxiety.

The Patient-Reported Outcomes Measurement Information System (PROMIS) Depression Short-Form 8a will be used.The minimum score is 8, and the maximum score is 40. Higher scores indicate higher levels of depressive symptoms.

The Patient-Reported Outcomes Measurement Information System (PROMIS) Quality of Life scale (10 items) will be used. The minimum score is 5, and the maximum score is 50. Higher scores indicate more sleep disturbance.

The Patient-Reported Outcomes Measurement Information System (PROMIS) Sleep Disturbance Short-Form 6a will be used. The minimum score is 6, and the maximum score is 30. Higher scores indicate more sleep disturbance.

UCLA Loneliness Scale; range is 0-60; lower score is better outcome.

Self-Compassion Scale; range is 26-130; higher score is better outcome.

The Metacognition Questionnaire-30; range from 30 - 120; higher score is better outcome.

The Continuing Bonds Scale; ranges from 11-55.

The Prolonged Grief-13-Revised; range 10-50; higher score means greater prolonged grief.

University of Texas at Austin

Assistant Professor

Neoplasms

All Conditions

Digestive System Diseases

Carcinoma

Carcinoma, Hepatocellular

Liver Neoplasms

Neoplasms, Glandular and Epithelial

Neoplasms by Histologic Type

Adenocarcinoma

Gastrointestinal Neoplasms

Digestive System Neoplasms

Gastrointestinal Diseases

Liver Diseases

Hematinics

All Drugs and Chemicals

Liver Extracts

Wu Ruei-Jhu, Master

Shiow-Ching Shun, Doctor

The aims of this study are to (1) understand the uncertainty , personality and coping strategies in patients with HCC, and identify the significant factors for coping.

A cross-sectional correlated design will be used and (2) patients will be recruited by purposive sampling from wards and outpatient departments at a medical center in Taipei.

The Illness of Uncertainty, Personality and Coping Strategies in Patients With Hepatocellular Carcinoma

National Taiwan University Hospital

Shiow-Ching Shun

Symptoms and General Pathology

Infections

Respiratory Tract (Lung and Bronchial) Diseases

Musculoskeletal Diseases

Diseases and Abnormalities at or Before Birth

Skin and Connective Tissue Diseases

Nutritional and Metabolic Diseases

Rare Diseases

Syndrome

COVID-19

Facies

Virus Diseases

Coronavirus Infections

Severe Acute Respiratory Syndrome

Osteochondroma

Exostoses, Multiple Hereditary

Osteochondromatosis

Osteogenesis Imperfecta

Enchondromatosis

Exostoses

Osteophyte

Osteochondrodysplasias

Mucopolysaccharidosis IV

Bone Diseases

Bone Diseases, Developmental

Genetic Diseases, Inborn

Rheumatic Diseases

Collagen Diseases

Connective Tissue Diseases

Neoplasms, Bone Tissue

Neoplasms, Connective Tissue

Neoplasms, Connective and Soft Tissue

Hyperostosis

Neoplastic Syndromes, Hereditary

Acute Graft Versus Host Disease

Ollier Disease

Maffucci Syndrome

Mucopolysaccharidosis Type IV

Luca Sangiorgi, MD, Phd

In the COVID-19 outbreak context, people living with rare diseases have been highly troubled with anxiety, loneliness, and depression. The project evaluates resilience and coping strategies to address pandemic impact by discussion in a dedicated focus group using a web-based platform. The goal is to improve, in a sustainable manner, the coping skills and psychological well-being of children, adolescents, and young adults affected by rare skeletal diseases.

March 11th, 2020, the World Health Organization (WHO) declared the SARS-CoV-2 pandemic accordingly with the spread and size of the infection.

The restrictions implemented by the governments have substantially changed people's lifestyles, facilitating the emergence of emotions such as fear, anxiety, and a sense of loneliness.

The age groups most involved by these burdens have been childhood, adolescence, and young adults. These groups have experienced dramatic changes in their daily life in order the relational, educational, and social networks. Nonetheless, these links outline the ideal environment for the development of resilience and the promotion of coping strategies to face traumatic events.

These lifestyle challenges and their consequences are even more evident in the context of the rare diseases since the outbreak containment measures have emphasized the troubles with which people affected by these conditions are daily forced to confront, in terms of access to care and treatments, resulting in increased stress and anxiety.

The current phase represents a sensitive step for the resumption of the activities in charge of the health and social service aimed at the citizens and especially to people with special needs, such as people suffering from rare diseases.

The pandemic experience has promoted new forms of meeting and contact between individuals using web-based platforms, playing a fundamental role in preserving the community and "sociality" dimensions.

In this scenario, the rare diseases could be an attractive model since the pandemic impinges over their bio-psycho-social characteristics at different levels. Resilience and coping strategies adopted by rare diseases patients could configure as an exemplum to face the psychological distress resulting from anti-pandemic measures, suggesting new approaches of taking charge by health service.

This study evaluates resilience and coping strategies to address pandemic impact by discussion in a dedicated focus group using a web-based platform. The goal is to improve, in a sustainable manner, the coping skills and psychological well-being of children, adolescents, and young adults affected by rare skeletal diseases.

Resilience and Coping in a Rare Skeletal Disease Population to Face Coronavirus (COVID-19) Outbreak Distress: a Longitudinal Study

Exploring Resilience and Coping Strategies of Young Population in Response to Severe Acute Respiratory Syndrome Corona Virus 2 (SARS-CoV-2) Outbreak: a Longitudinal Study in a Cohort of Patients Affected by Rare Skeletal Disorders.

Ego-Resiliency Scale (ER89); 14 items; 4-point Likert scale (1=does not apply at all; 2=applies slightly, if at all; 3=applies somewhat; and 4=applies very strong); Score Resiliency Trait: 47-56 Very High, 35-46 High, 23-34 Undetermined, 11-22 Low, 0-10 Very Low

Hospital Anxiety and Depression Scale (HADS); 14 items: 7 items relate to anxiety and 7 relate to depression; a 4-point (0-3) Likert-scale; Cutoffs for both HADS-anxiety and HADS-depression as \>= 8, and for HADS-total \>= 15

UCLA Loneliness Scale (Version 3); 20-item scale; 4-point rating scale (1=Never; 2=Rarely; 3=Sometimes; and 4=Often); score ranging from 20 to 80.

UCLA Loneliness Scale (Version 3); UCLA Loneliness Scale (Version 3); 20-item scale; 4-point rating scale (1=Never; 2=Rarely; 3=Sometimes; and 4=Often); score ranging from 20 to 80.

Study title	NCT ID	Conditions	Interventions	Eligibility	Organization
Feasibility Study of the Storytelling Through Music Intervention With Bereaved Parents	NCT06047808	Pediatric Cance... Grief Well-Being, Psy... Coping Strategi... Bereavement	Storytelling Th...	18 Years - 99 Years	University of Texas at Austin
The Illness of Uncertainty, Personality and Coping Strategies in Patients With Hepatocellular Carcinoma	NCT01380392	Liver Carcinoma		20 Years -	National Taiwan University Hospital
The Illness of Uncertainty, Personality and Coping Strategies in Patients With Hepatocellular Carcinoma	NCT01380392	Liver Carcinoma		20 Years -	National Taiwan University Hospital
Resilience and Coping in a Rare Skeletal Disease Population to Face Coronavirus (COVID-19) Outbreak Distress: a Longitudinal Study	NCT04844697	Multiple Osteoc... Osteogenesis Im... Ollier Disease Maffucci Syndro...		8 Years - 24 Years	Istituto Ortopedico Rizzoli

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