The number of AYAs living with serious illnesses such as cancer is growing globally (Viner et al., 2011; Cohen and Patel, 2014; GBD 2019 Adolescent Young Adult Cancer Collaborators, 2022). In 2020, an estimated 90,000 adolescents and young adults (AYA) were diagnosed with cancer in the United States (Haines et al., 2021). For many, death is an inevitable outcome of their disease, making advance care planning (ACP) a critical component of care (Brown and Sourkes, 2006; DeCourcey et al., 2021). It is difficult for AYAs, their families, and providers to think about or talk about death and dying. The presence of a life-threatening illness adds a multitude of challenges to what is already a difficult period of life, when emerging adults strive to define themself outside of the context of their family and envision their own future (Brown and Sourkes, 2006). Further, disease burden at this age can negatively impact financial security, body image, educational and work trajectories, relationships with spouse/significant other, and plans for having children (Maslow et al., 2011; Warner et al., 2016; Jin et al., 2017). In addition, young adults with advanced cancer have reported significant psychological distress in the form of grief (Jacobsen et al., 2010) and suicidal ideation (Walker et al., 2008).

Effective end-of-life (EoL) discussions are critical for AYAs, especially in the event of disease progression or a poor prognosis, given both the medical challenges and psychosocial risk factors involved (Sansom-Daly et al., 2020). ACP documents and advance directives provide patients with the opportunity to express their preferences for care. These directives can help families and health care agents make informed decisions, alleviate distress (Mack et al., 2005), avoid decisional regret (DeCourcey et al., 2019; Lichtenthal et al., 2020), and potentially improve the patient’s quality of life by respecting their religious, cultural, and familial values and beliefs (Jankovic et al., 2008; Barfield et al., 2010; Kane et al., 2011; Wiener et al., 2012). Families have expressed significant interest in ACP, with parents indicating that the opportunity for these discussions has been a poorly met need (Durall et al., 2012; Lotz et al., 2013; DeCourcey et al., 2019; Hein et al., 2020; Orkin et al., 2020). Research has shown that parents of seriously ill children desire earlier, ongoing opportunities to address ACP with their child’s providers (DeCourcey et al., 2019; Orkin et al., 2020). However, many pediatric providers report a lack of ACP communication training (Dellon et al., 2010; Durall et al., 2012; Lotz et al., 2013; Heckford and Beringer, 2014). Unfortunately, when ACP discussions do take place, they often occur too late and typically during an acute clinical crisis, when there is insufficient time to consider individual goals and values (Davidson et al., 2007; Brudney, 2009; Durall et al., 2012; Snaman et al., 2020; Pennarola et al., 2021).

The literature has clearly demonstrated the need and desire for ACP intervention in this population (Weaver et al., 2015; Kirch et al., 2016), as well as the barriers to initiating ACP (Wolfe et al., 2008; Smith et al., 2012; Kassam et al., 2013; Pinkerton et al., 2018). Studies have shown that less than 3% of AYAs participate in EoL planning conversations without clinician prompting (Lyon et al., 2004, 2014; Liberman et al., 2014; Carr et al., 2021). Data exploring the experience of the family members, friends, and health care providers who are involved in ACP conversations with AYA are limited. Adolescents and families who participated in family-centered ACP found the conversations to be worthwhile (Dallas et al., 2016), notably with a greater understanding of EoL wishes (Madrigal et al., 2017). In a robust multi-site, assessor-blinded, parallel-group, randomized control trial (FACE pACP), ACP surrogates were eight times more likely than controls to have an excellent understanding of adolescent patients’ treatment preferences (Lyon et al., 2018). In another ACP trial, families had more positive appraisals of their caregiving, than families who did not have these conversations (Baker et al., 2020).

Novel tools and interventions are needed to facilitate ACP discussions between AYA and their family members, friends and HCPs (Snaman et al., 2020). Voicing My CHOiCES (VMC), a research informed ACP guide (Wiener et al., 2012; Zadeh et al., 2015), has been shown to both decrease anxiety around EoL planning and enhance communication with both family members and friends (Wiener et al., 2021). This study adds to the literature by providing the perspectives or outcomes on behalf of the family member, friend, or HCP post completion of an ACP document by an AYA. In this study we aimed to gain understanding of the experience of the family member, friend, or HCP pertaining to the ACP discussion, changes in their relationship, conversation quality, and whether the discussion prompted changes in care.

One month after completing the baseline measure and reviewing the VMC tool, 129 participants answered the follow up questions about talking with family members, 124 about talking with friends, and 124 answered the question about talking with HCP. Overall, 84 (65.1%) of participants had shared what they wrote in VMC with a family member and 11 (8.9%) shared with an HCP (Wiener et al., 2021). Twenty-eight participants (22.6%) shared what they wrote in VMC with a friend. Of those with whom document completion was shared, we interviewed 40 (47.6%) family members, 6 (21.4%) friends, and 5 (45.5%) providers about their experience with this conversation. Of note, three interviews (two family members and one provider) were discontinued when the participant indicated the AYA had not shared what they had written in VMC. The remaining analyses are based on interviews with the 48 participants who engaged in such a conversation, according to both the AYA and the study participant.

ACP discussions have been associated with a range of positive outcomes, including increased congruence between treatment preferences expressed by AYAs and their caregivers and increased likelihood that these preferences will be honored at the EoL. Yet, AYAs and their caregivers find it difficult to engage in these conversations (Jimenez et al., 2018). For the majority of study participants, completing an age-appropriate ACP guide prompted a first conversation regarding EoL preferences with a family member. To a lesser degree, it also prompted a first conversation with a friend. Notably, many of these conversations covered more than just EoL preferences. Participants described having deeply honest discussions about hopes, fears, and relationships. These findings support using VMC to enhance communication about EoL preferences, adding to the existing literature on the myriad benefits of such interventions (Feraco et al., 2016; Lin et al., 2020; Laronne et al., 2021).

In addition to families requiring assistance in navigating these discussions (Kenney et al., 2021), we found few AYAs shared what they wrote in VMC with their HCP. Challenges surrounding ACP conversations with HCPs include provider discomfort and a lack of training and resources (Dellon et al., 2010; Durall et al., 2012; Lotz et al., 2013; Heckford and Beringer, 2014). In fact, most family members and friends, as well as half of the HCP in our study, did not think the AYA would have talked to them about their EoL preferences without having completed VMC. This highlights the need for training on how to introduce ACP more comfortably with AYAs so that goals regarding current and future care can be addressed. While just one of five HCPs noted a treatment change following the discussion, these numbers may reflect the limited number of HCPs who the AYAs engaged in an ACP conversation. This is consistent with current literature suggesting patients and their family members will wait for the topic to be raised by their clinician (Clayton et al., 2005; Brighton and Bristowe, 2016). Research focused on training clinicians and preparing patients and families to engage in high-quality discussions using an age-appropriate ACP guide, like VMC, may help to achieve higher quality EoL care.

The specifics of what the AYA shared following completion of VMC was varied and included their preferences on mechanisms of comfort and support, how aggressively they would like to be treated if there was little to no chance of recovery, planning for how they would like to be remembered, and care/concern for others after death. These conversations were well received and described as being beneficial to both parties, despite being emotionally difficult to initiate. Similar to extant literature (Aldridge et al., 2017; Hein et al., 2018; Weaver et al., 2021), participants in the current study recognized that communicating important EoL care preferences can help prepare for future situations and create a pathway to goal-concordant EoL care. Critical longitudinal data is needed to assess whether communicated preferences were honored after an AYAs death.

Other benefits from these conversations were also reported. Half of family members and a third of their friends reported a change in their relationship with the AYA. Changes were all self-reported to be positive. Additionally, when describing the overall experience of talking about ACP with the AYA, participants again highlighted benefit despite also being seen as burdensome. Many participants spoke to the value of the ACP conversations and the relief in having the discussion despite the stress of thinking about worst-case scenarios. These findings can reassure family members and HCPs of advantages associated with these courageous conversations.

Some limitations are important to note. First, many family members (n = 44, 53.7%), friends (n = 6, 54.5%), and HCP (n = 5, 50%) who AYAs shared their VMC completion with were not interviewed. For some, the AYA wasn’t comfortable with having the researcher reach out to them, and for others the family member, friend, or HCP was unreachable or declined participation. Second, interviews were only conducted with English-speaking individuals. Therefore, we do not know if the themes that were identified would be different from the family members, friends and HCP who were not interviewed. Third, we contacted AYA participants 1 month after they completed VMC. We don’t know if a conversation occurred with a family member, friend, or HCP about what they wrote in VMC past this point. Fourth, demographic data was not collected on the contact participants, so it is unknown whether one demographic was more represented than another. Last, the perspectives obtained might have been affected by recall bias. Despite these limitations, to the best of our knowledge, this is the first multisite study that describes the unique perspectives of family members, friends, and HCP after discussing ACP preferences with AYA post completion of VMC. The concurrent voices captured here poignantly illustrate the shared sense of burden and benefit AYAs and all those who care for them experience trying to communicate during the complex journey at the end of life.

The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.

The studies involving human participants were reviewed and approved by NIH Institutional Review Board: National Institutes of Health. The patients/participants provided their written informed consent to participate in this study.

SB, LW, and MP contributed to the conception and design of the study. SB, LW, JT, PM, CH, and KZ collected the data. SB organized the database and wrote the first draft of the manuscript. MG, SB, and AF performed the statistical analysis. LW wrote sections of the manuscript. All authors contributed to manuscript revision, read, and approved the submitted version.

The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.

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