Developing a national implementation strategy to accelerate uptake of evidence-based family caregiver support in U.S. cancer centers.

OBJECTIVE: Characterize key factors and training needs of U.S. cancer centers in implementing family caregiver support services. METHODS: Sequential explanatory mixed methods design consisting of: (1) a national survey of clinicians and administrators from Commission-on-Cancer-accredited cancer centers (N = 238) on factors and training needed for establishing new caregiver programs and (2) qualitative interviews with a subsample of survey respondents (N = 30) to elicit feedback on survey findings and the outline of an implementation strategy to facilitate implementation of evidence-based family caregiver support (the Caregiver Support Accelerator). Survey data was tabulated using descriptive statistics and transcribed interviews were analyzed using thematic analysis. RESULTS: Top factors for developing new caregiver programs were that the program be: consistent with the cancer center's mission and strategic plan (87%), supported by clinic leadership (86.5%) and providers and staff (85.7%), and low cost or cost effective (84.9%). Top training needs were how to: train staff to implement programs (72.3%), obtain program materials (63.0%), and evaluate program outcomes (62.6%). Only 3.8% reported that no training was needed. Qualitative interviews yielded four main themes: (1) gaining leadership, clinician, and staff buy-in and support is essential; (2) cost and clinician burden are major factors to program implementation; (3) training should help with adapting and marketing programs to local context and culture; and (4) the Accelerator strategy is comprehensive and would benefit from key organizational partnerships and policy standards. CONCLUSION: Findings will be used to inform and refine the Accelerator implementation strategy to facilitate the adoption and growth of evidence-based cancer caregiver support in U.S. cancer centers.

Availability of Family Caregiver Programs in US Cancer Centers.

Importance: Family caregivers provide the majority of health care to the 18 million patients with cancer in the US. Yet despite providing complex medical and nursing care, a large proportion of caregivers report no formal support or training. In recognition of this gap, many interventions to support cancer caregivers have been developed and tested over the past 2 decades. However, there are few system-level data on whether US cancer centers have adopted and implemented these interventions. Objective: To describe and characterize the availability of family caregiver support programs in US cancer centers. Design, Setting, and Participants: This cross-sectional national survey study was conducted between September 1, 2021, and April 30, 2023. Participants comprised clinical and administrative staff of Commission on Cancer-accredited US cancer centers. Data analysis was performed in May and June 2023. Main Outcomes and Measures: Survey questions about the availability of 11 types of family caregiver programs (eg, peer mentoring, education classes, and psychosocial programs) were developed after literature review, assessment of similar program evaluation surveys, and discussions among a 13-member national expert advisory committee. Family caregiver programs were defined as structured, planned, and coordinated groups of activities and procedures aimed at specifically supporting family caregivers as part of usual care. Survey responses were tabulated using standard descriptive statistics, including means, proportions, and frequencies. Results: Of the surveys sent to potential respondents at 971 adult cancer centers, 238 were completed (response rate, 24.5%). After nonresponse weight adjustment, most cancer centers (75.4%) had at least 1 family caregiver program; 24.6% had none. The most common program type was information and referral services (53.6%). Cancer centers with no programs were more likely to have smaller annual outpatient volumes (χ2 = 11.10; P = .011). Few centers had caregiver programs on training in medical and/or nursing tasks (21.7%), caregiver self-care (20.2%), caregiver-specific distress screening (19.3%), peer mentoring (18.9%), and children caregiving for parents (8.3%). Very few programs were developed from published evidence in a journal (8.1%). The top reason why cancer centers selected their programs was community members requesting the program (26.3%); only 12.3% of centers selected their programs based on scientific evidence. Most programs were funded by the cancer center or hospital (58.6%) or by philanthropy (42.4%). Conclusions and Relevance: In this survey study, most cancer centers had family caregiver programs; however, a quarter had none. Furthermore, the scope of programming was limited and rarely evidence based, with few centers offering caregiving education and training. These findings suggest that implementation strategies are critically needed to foster uptake of evidence-based caregiver interventions.

Patterns of Pediatric Palliative and End-of-Life Care in Neonatal Intensive Care Patients in the Southern U.S.

CONTEXT: Despite high rates of mortality among infants in the Southern U.S., little is known about the timing of pediatric palliative care (PPC), the intensity of end-of-life care, and whether there are differences among sociodemographic characteristics. OBJECTIVES: To describe PPC patterns and treatment intensity during the last 48 hours of life among neonatal intensive care unit (NICU) patients in the Southern U.S. who received specialized PPC. METHODS: Medical record abstraction of infant decedents who received PPC consultation in two NICUs (in Alabama and Mississippi) from 2009 to 2017 (n = 195) including clinical characteristics, palliative and end-of-life care characteristics, patterns of PPC, and intensive medical treatments in the last 48 hours of life. RESULTS: The sample was racially (48.2% Black) and geographically (35.4% rural) diverse. Most infants died after withdrawal of life-sustaining interventions (58%) and had do not attempt resuscitation orders documented (75.9%); very few infants enrolled in hospice (6.2%). Initial PPC consult occurred a median of 13 days after admission and a median of 17 days before death. Infants with a primary diagnosis of genetic or congenital anomaly received earlier PPC consultation (P = 0.02) compared to other diagnoses. In the last 48 hours of life, NICU patients received intensive interventions including mechanical ventilation (81.5%), CPR (27.7%) and surgeries or invasive procedures (25.1%). Black infants were more likely to receive CPR compared to White infants (P = 0.04). CONCLUSION: Overall, PPC consultation occurred late in NICU hospitalizations, infants received high-intensity medical interventions in the last 48 hours of life, and there are disparities in intensity of treatment interventions at end of life. Further research is needed to explore if these patterns of care reflect parent preferences and goal concordance.

Care Intensity and Palliative Care in Chronically Critically Ill Infants.

CONTEXT: Increasingly, chronically critically ill (CCI) infants survive to discharge from Neonatal Intensive Care Units (NICUs). Little is known about their care intensity and the primary and specialty palliative care families receive at and following discharge. OBJECTIVES: To describe care intensity and primary and specialty palliative care received by NICU CCI infants at discharge and one year. METHODS: Chart abstraction of CCI infants at three academic centers discharged at ≥42 weeks corrected gestational age with medical technology between 2016 and 2019, including demographics, care intensity, and primary and specialty palliative care received at discharge and one year. RESULTS: Among 273 infants, NICU median stays were 45 [IQR 23-92] days. Primary diagnoses included congenital and/or genetic conditions (68.5%), prematurity (28.2%), and birth events (3.3%). At discharge, surgical feeding tubes (75.1%) and tracheostomies (24.5%) were the most common technologies. Infants received a median of 6 [IQR 4-9] medications and were followed by a median of 8 [IQR 7-9] providers. At one year, 91.4% continued with one or more technologies, similar numbers of medications and specialty providers. In the NICU, nearly all families had social work involvement, 78.8% had chaplaincy and 53.8% child life; 19.8% received specialty palliative care consultation. At one year, only 13.2% were followed by palliative care. CONCLUSIONS: CCI infants receive intensive medical care including multiple medical technologies, medications, and specialty follow up at discharge and remain complex at one year of life. Most receive primary interprofessional palliative care in the NICU, however these infants and their families may have limited access to specialty palliative care in the short- and long-term.

Missing Voices: Lessons Learned from Nonparticipating Caregivers in Palliative Care Research.

Background: Our previous study to understand end-of-life care of adolescents and young adults (AYAs) had a suboptimal survey response rate by bereaved caregivers. Objective: To identify sociodemographic factors associated with caregiver nonparticipation. Design/Setting/Subjects:Post hoc analysis of a retrospective multicenter cohort study of caregivers of deceased AYAs from 2013 to 2016. Measurements: Exposures: race, ethnicity, area-, and household-poverty. Primary outcome: survey participation. Secondary outcomes: loss to follow-up at each recruitment step. Results: Thirty-five of 263 eligible caregivers participated in the survey (13.3%). Caregivers of AYAs living in high-poverty zip codes were significantly more likely to have a disconnected or incorrect phone number (odds ratio [OR] 2.12; 95% confidence interval [CI] 1.04-4.58; p = 0.03). Caregivers of nonwhite AYAs were significantly less likely to participate (OR 0.35; 95% CI 0.12-0.87; p = 0.01). Conclusions: Caregivers of patients living in poverty are less likely to be reached by traditional recruitment efforts. Caregivers of racial/ethnic minority patients are less likely to participate overall.

An Early Palliative Care Telehealth Coaching Intervention to Enhance Advanced Cancer Family Caregivers' Decision Support Skills: The CASCADE Pilot Factorial Trial.

CONTEXT: Patients with advanced cancer often involve family caregivers in health-related decision-making from diagnosis to end-of-life; however, few interventions have been developed to enhance caregiver decision support skills. OBJECTIVES: Assess the feasibility, acceptability, and potential efficacy of individual intervention components of CASCADE (CAre Supporters Coached to be Adept DEcision Partners), an early telehealth, palliative care coach-led decision support training intervention for caregivers. METHODS: Pilot factorial trial using the multiphase optimization strategy (October 2019-October 2020). Family caregivers and their care recipients with newly-diagnosed advanced cancer (n = 46 dyads) were randomized to1 of 8 experimental conditions that included a combination of one of the following three CASCADE components: 1) effective decision support psychoeducation; 2) decision support communication training; and 3) Ottawa Decision Guide training. Feasibility was assessed by completion of sessions and questionnaires (predefined as ≥80%). Acceptability was determined through postintervention interviews and participants' ratings of their likelihood to recommend. Measures of effective decision support and caregiver and patient distress were collected at Twelve and Twenty four weeks. RESULTS: Caregiver participants completed 78% of intervention sessions and 81% of questionnaires; patients completed 80% of questionnaires. Across conditions, average caregiver ratings for recommending the program to others was 9.9 on a scale from 1-Not at all likely to 10-Extremely likely. Individual CASCADE components were observed to have potential benefit for effective decision support and caregiver distress. CONCLUSION: We successfully piloted a factorial trial design to examine components of a novel intervention to enhance the decision support skills of advanced cancer family caregivers. A fully-powered factorial trial is warranted. KEY MESSAGE: We pilot tested components of CASCADE, an early palliative care decision support training intervention for family caregivers of patients with advanced cancer. CASCADE components were acceptable and the trial design feasible, providing promising future directions for palliative care intervention development and testing. Pilot results will inform a fully-powered trial.

Bereaved Caregivers Perspectives of Negative Communication Experiences Near the End of Life for Adolescents and Young Adults with Cancer.

Purpose: High-quality communication is a standard of palliative care for adolescents and young adults (AYAs) with cancer. Yet, few studies have characterized the negative communication experiences of AYAs near the end of life (EOL). Methods: We performed a secondary analysis of 27 qualitative interviews with bereaved caregivers of AYAs with cancer who died between 2013 and 2016 at 1 of 3 sites. Interviews focused on barriers to optimal EOL care for AYAs. We used thematic analysis using iterative consensus coding to analyze transcripts. Results: Participants were predominantly white (85%), non-Hispanic (93%), and female (74%). Half of the participants were bereaved parents, and 37% were bereaved partners or spouses. Overall, 23/27 (85%) caregivers described at least one negative communication experience related to one of three themes: (1) Insensitivity to patients' needs, preferences, and values; (2) Insufficient discussions of prognosis and/or EOL; and (3) Loss of support from the clinical team near EOL. Both clinician- and patient-related factors contributed to limited EOL discussions. Lack of care continuity related to both clinician factors and systems of care that required new or changing clinical care teams near the EOL. Conclusions: Caregivers report a desire for clinician sensitivity to their needs and values, information about the future, and longitudinal connections with individual clinicians. Clinicians might improve caregivers' EOL experiences by eliciting patient preferences, engaging in EOL discussions, adapting to the AYA's developmental and emotional needs, and demonstrating a commitment to AYAs and caregivers as they approach the EOL.

A lay navigator-led, early palliative care intervention for African American and rural family caregivers of individuals with advanced cancer (Project Cornerstone): Results of a pilot randomized trial.

BACKGROUND: The objective of this study was to assess the feasibility, acceptability, and potential efficacy of ENABLE (Educate, Nurture, Advise, Before Life Ends) Cornerstone-a lay navigator-led, early palliative care telehealth intervention for African American/Black and/or rural-dwelling family caregivers of individuals with advanced cancer (ClinicalTrials.gov identifier NCT03464188). METHODS: This was a pilot randomized trial (November 2019 to March 2021). Family caregivers of patients with newly diagnosed, stage III/IV, solid-tumor cancers were randomized to receive either an intervention or usual care. Intervention caregivers were paired with a specially trained lay navigator who delivered 6 weekly, 20-minute to 60-minute telehealth coaching sessions plus monthly follow-up for 24 weeks, reviewing skills in stress management, self-care, getting help, staying organized, and future planning. Feasibility was assessed according to the completion of sessions and questionnaires (predefined as a completion rate ≥80%). Acceptability was determined through intervention participants' ratings of their likelihood of recommending the intervention. Measures of caregiver distress and quality of life were collected at 8 and 24 weeks. RESULTS: Sixty-three family caregivers were randomized (usual care, n = 32; intervention, n = 31). Caregivers completed 65% of intervention sessions and 87% of questionnaires. Average ratings for recommending the program were 9.4, from 1 (not at all likely) to 10 (extremely likely). Over 24 weeks, the mean ± SE Hospital Anxiety and Depression Scale score improved by 0.30 ± 1.44 points in the intervention group and worsened by 1.99 ± 1.39 points in the usual care group (difference, -2.29; Cohen d, -0.32). The mean between-group difference scores in caregiver quality of life was -1.56 (usual care - intervention; d, -0.07). Similar outcome results were observed for patient participants. CONCLUSIONS: The authors piloted ENABLE Cornerstone, an intervention for African American and rural-dwelling advanced cancer family caregivers. The acceptability of the intervention and data collection rates were high, and the preliminary efficacy for caregiver distress was promising. LAY SUMMARY: To date, very few programs have been developed to support under-resourced cancer family caregivers. To address this need, the authors successfully pilot tested an early palliative care program, called Educate, Nurture, Advise, Before Life Ends (ENABLE) Cornerstone, for African American and rural family caregivers of individuals with advanced cancer. Cornerstone is led by specially trained lay people and involves a series of weekly phone sessions focused on coaching caregivers to manage stress and provide effective support to patients with cancer. The authors are now testing Cornerstone in a larger trial. If the program demonstrates benefit, it may yield a model of caregiver support that could be widely implemented.

Caregiver Reported Quality of End-of-Life Care of Adolescent and Young Adult Decedents With Cancer.

Background: The quality of palliative and end-of-life (EOL) care for adolescents and young adults (AYAs) with cancer remains largely unknown. Objective: To describe caregivers of AYA cancer decedents perspectives' on EOL care quality related to EOL care communication. Design: Cross-sectional observational study. Setting/Subjects: Caregivers (n = 35) of AYAs who died from a cancer diagnosis from 2013-2016 were recruited from 3 U.S. academic medical centers. Measurements: Caregiver participants completed structured surveys (FAMCARE scale and the Toolkit After-Death Bereaved Family Member Interview) by telephone to gather perceptions of quality of EOL care of their AYA cancer decedents. Results: Caregivers reported unmet needs regarding preparation for the time of death (50%), the dying process (45%) and unmet spiritual/ religious needs (38%). Lowest quality of EOL care scores related to communication and emotional support. Conclusions: Our findings call for special focus on providing information about what to expect during the dying process and adequately addressing spiritual and religious preferences during EOL care for AYAs.

Simulating Telehealth Experiences in the Neonatal Care Environment: Improving Access to Care.

Telehealth in the neonatal environment can improve remote medical care and access to specialized care and training eliminating barriers for effective health care delivery. Clinicians are utilizing telehealth in their practice to provide specialized care and training in areas that have little access. Educating health care clinicians on the basics of telehealth is an essential component of clinical training programs. Use of simulation-based telehealth experiences as part of that training can provide hands-on learning in a safe, realistic environment. Simulation can prepare health care teams in using telehealth technology in managing patient care, postdischarge care, and specialized care programs.

Barriers to Optimal End-of-Life Care for Adolescents and Young Adults With Cancer: Bereaved Caregiver Perspectives.

BACKGROUND: Adolescents and young adults (AYAs; aged 15-39 years) with cancer frequently receive intensive measures at the end of life (EoL), but the perspectives of AYAs and their family members on barriers to optimal EoL care are not well understood. METHODS: We conducted qualitative interviews with 28 bereaved caregivers of AYAs with cancer who died in 2013 through 2016 after receiving treatment at 1 of 3 sites (University of Alabama at Birmingham, University of Iowa, or University of California San Diego). Interviews focused on ways that EoL care could have better met the needs of the AYAs. Content analysis was performed to identify relevant themes. RESULTS: Most participating caregivers were White and female, and nearly half had graduated from college. A total of 46% of AYAs were insured by Medicaid or other public insurance; 61% used hospice, 46% used palliative care, and 43% died at home. Caregivers noted 3 main barriers to optimal EoL care: (1) delayed or absent communication about prognosis, which in turn delayed care focused on comfort and quality of life; (2) inadequate emotional support of AYAs and caregivers, many of whom experienced distress and difficulty accepting the poor prognosis; and (3) a lack of home care models that would allow concurrent life-prolonging and palliative therapies, and consequently suboptimal supported goals of AYAs to live as long and as well as possible. Delayed or absent prognosis communication created lingering regret among some family caregivers, who lost the opportunity to support, comfort, and hold meaningful conversations with their loved ones. CONCLUSIONS: Bereaved family caregivers of AYAs with cancer noted a need for timely prognostic communication, emotional support to enhance acceptance of a poor prognosis, and care delivery models that would support both life-prolonging and palliative goals of care. Work to address these challenges offers the potential to improve the quality of EoL care for young people with cancer.

Can you hear me now?: Improving palliative care access through telehealth.

Telehealth has been increasingly used to expand healthcare access over the last two decades. However, this had not been the case for palliative care (PC), because telehealth was considered nontraditional and impractical due to the sensitive nature of conversations and a "high touch" philosophy. Motivated by limited PC access to rural and underserved populations and positive PC telehealth studies, clinical PC telehealth models have been developing. However, nearly overnight, the COVID-19 pandemic accelerated the use and uptake of telehealth across health care and especially in PC. As a result, clinicians, administrators, and others agree that telehealth is "here to stay," and will likely maintain widespread use and refinement beyond rural areas. The purpose of this review is to describe exemplar PC telehealth programs in research and clinical practice, including pros and cons, lessons learned, and future directions for the ongoing development and expansion of PC via telehealth across diseases and the lifespan.

The Challenges of Taking Breastfeeding Infants to Academic Nursing Conferences.

Nursing faculty are predominantly women, and many are of childbearing age. Academic responsibilities for nursing faculty include attendance at scientific meetings and conferences. When nursing faculty members are breastfeeding, situations arise that may require them to make difficult decisions to meet both demands as mothers and academics. The purpose of this article is to describe the experiences of three nursing faculty members who brought their breastfed infants to professional nursing conferences, as well as identify major barriers to an inclusive working environment that cultivates "work-life balance" and propose workable solutions for breastfeeding mothers in academe.

Palliative Care Knowledge and Characteristics in Caregivers of Chronically Ill Children.

There is a growing population of children with complex chronic conditions (CCCs) whose caregivers would benefit from palliative care (PC). However, little is known about caregivers' PC awareness. We aimed to describe PC awareness among caregivers of children with CCCs and identify factors associated with lack of PC awareness. We used the National Cancer Institute's national Health Information National Trends Survey 2018 data to determine the percentage of caregivers of ill children who have PC awareness. After matching, caregiver PC awareness was compared with that of (1) the general survey population, (2) other caregivers, and (3) caregivers not caring for children. We used multivariable regression to determine factors associated with lack of PC awareness. Of 131 caregivers, 60% had "never heard of" PC. Caregivers of children were no more likely to have heard of PC than the general survey population (P = .76), noncaregivers (P = .97), or caregivers of nonchildren (P = .13). Caregivers younger than 40 years and without a college degree were less likely to have PC awareness than their peers. Most caregivers of ill children have no PC awareness, with no more PC awareness than the general population. Nurses caring for children with CCCs can help educate families and other health care team members about PC.

Highlighting Simulation Practices in the Neonatal Environment.

Simulation is an effective teaching methodology to enhance clinical thinking and reasoning skills among nursing students and practicing nurses. The opportunity to practice in a safe environment maintains a structure that promotes learning at all levels. There are various levels of fidelity as well as cost to facilitate simulation in the neonatal setting. This at times hinders the ability to incorporate simulation into educational practices. The purpose of this article is to provide a discussion on simulation practices in the neonatal setting, an overview of low-cost neonatal simulation exemplars, and implications for practice.

Bereaved parents' views on end-of-life care for children with cancer: Quality marker implications.

BACKGROUND: End-of-life (EOL) quality markers in adult oncology include home death and intensive care unit avoidance. Corresponding markers are lacking in pediatric oncology. This study was aimed at describing bereaved parents' perspectives of high-quality EOL care in pediatric oncology. METHODS: This study enrolled a convenience sample of 28 bereaved parents (English- or Spanish-speaking) whose children (0-21 years old) had died of cancer ≥6 months before. Semistructured interviews were conducted to elicit parental perceptions of medically intense/quality EOL care. Interviews were recorded and transcribed verbatim (30 hours), and study team consensus and content analyses identified themes related to EOL quality markers. Related quotes were scored on a 5-point Likert scale ranging from 1 (supported comfort care) to 5 (supported medically aggressive care). RESULTS: The children died in 1998-2017 at a mean age of 10 years (SD, 5.2 years); 50% had a solid tumor, and 46% were Spanish-speaking. Themes included 1) home death preference (unless home support was inadequate; median score, 1.6), nonaggressive care (median score, 2.4), and continued anticancer therapy (median score, 3.2); 2) programs/policies that could alleviate barriers limiting a family's time with a dying child (visiting restrictions and financial strains); 3) the need to prepare the family for death (eg, what would happen to the child's body), and 4) perceived abandonment. CONCLUSIONS: This is the first qualitative study to identify quality makers for children dying of cancer from bereaved parents' perspectives. Natural death is generally preferred, and quality measures that address barriers to parents' spending time with their children, a lack of preparation for the events surrounding death, and feelings of abandonment are critical. Future studies need to validate these findings and develop targeted interventions.

Correction: Despite mutation acquisition in hematopoietic stem cells, JMML-propagating cells are not always restricted to this compartment.

An amendment to this paper has been published and can be accessed via a link at the top of the paper.

Hospital or Home? Where Should Children Die and How Do We Make That a Reality?

CONTEXT: Most of the 20,000 U.S. children dying of serious illnesses annually die in the hospital. It is unknown if this hospital death predominance reflects family wishes or systemic issues such as lack of hospice access. Hence, we need to better understand location of death preferences for children and their families. OBJECTIVE: To better understand location of death preferences in North America, we reviewed the literature to examine the evidence for and against home death in seriously ill children (0-18 years). METHODS: We searched English articles in PubMed, PsycINFO, and Embase published during 2000-2018 for articles related to parental, child/adolescent, and provider preference for death location and articles that correlated death location with bereavement or quality of life outcomes. RESULTS: The search results (n = 877 articles and n = 58 abstracts of interest) were reviewed, and 34 relevant articles were identified. Parent, child, and provider preferences, bereavement outcomes, and associated factors all point to some preference for home death. These findings should be interpreted with several caveats: 1) many studies are small and prone to selection bias, 2) not all families prefer home death and some that do are not able to achieve home death due to inadequate home support, 3) studies of bereavement outcomes are lacking. CONCLUSION: Adequate resources are needed to ensure children can die in their chosen location-be that home, hospital, or free-standing hospice. This review highlights research areas needed to better understand death location preference and programs and policies that will support home death for those that desire it.

Close quarters: Counseling aboard the Training Ship Golden Bear.

The mental health practice setting of the maritime training cruise provides an uncommon combination of opportunities and challenges from college and maritime military counseling. This article addresses the opportunities available in a clinical role that allows the clinician to be an active member of the community they serve and promote mental health among the next generation of maritime workers. The challenges addressed include promoting mental health in a setting in which emotional expression and mental health treatment are stigmatized, accompanied by disincentives for help-seeking behavior, as well as the potential risks of multiple relationships and limited confidentiality. (PsycInfo Database Record (c) 2020 APA, all rights reserved).

Despite mutation acquisition in hematopoietic stem cells, JMML-propagating cells are not always restricted to this compartment.

Juvenile myelomonocytic leukemia (JMML) is a rare aggressive myelodysplastic/myeloproliferative neoplasm of early childhood, initiated by RAS-activating mutations. Genomic analyses have recently described JMML mutational landscape; however, the nature of JMML-propagating cells (JMML-PCs) and the clonal architecture of the disease remained until now elusive. Combining genomic (exome, RNA-seq), Colony forming assay and xenograft studies, we detect the presence of JMML-PCs that faithfully reproduce JMML features including the complex/nonlinear organization of dominant/minor clones, both at diagnosis and relapse. Further integrated analysis also reveals that although the mutations are acquired in hematopoietic stem cells, JMML-PCs are not always restricted to this compartment, highlighting the heterogeneity of the disease during the initiation steps. We show that the hematopoietic stem/progenitor cell phenotype is globally maintained in JMML despite overexpression of CD90/THY-1 in a subset of patients. This study shed new lights into the ontogeny of JMML, and the identity of JMML-PCs, and provides robust models to monitor the disease and test novel therapeutic approaches.