PURPOSE:Ewing sarcoma is the second most common bone cancer in children, accounting for 2% of pediatric cancer diagnoses. Patients who present with metastatic disease at the time of diagnosis have a dismal prognosis compared with the >70% 5-year survival of those with localized disease. Novel therapeutic approaches that can impact metastatic disease are desperately needed, as well as a deeper understanding of the heterogeneity of Ewing sarcoma tumors. EXPERIMENTAL DESIGN:In this study, we utilized single-cell RNA sequencing to characterize the transcriptional landscape of primary Ewing sarcoma tumors and the surrounding tumor microenvironment in a cohort of seven untreated patients with Ewing sarcoma, as well as in circulating tumor cells (CTC). A potential CTC therapeutic target was evaluated through immunofluorescence of fixed CTCs from a separate cohort. RESULTS:Primary tumor samples demonstrate a heterogeneous transcriptional landscape with several conserved gene expression programs, including those composed of genes related to proliferation and Ewing sarcoma gene targets, which were found to correlate with overall survival. Copy-number analysis identified subclonal evolution within patients prior to treatment. Analyses of the immune microenvironment reveal an immunosuppressive microenvironment with complex intercellular communication among the tumor and immune cells. Single-cell RNA sequencing and immunofluorescence of CTCs at the time of diagnosis identified TSPAN8 as a potential therapeutic target. CONCLUSIONS:Ewing sarcoma tumors demonstrate significant transcriptional heterogeneity as well as a complex immunosuppressive microenvironment. This work evaluates several proposed targets that warrant further exploration as novel therapeutic strategies.

BACKGROUND:Cisplatin-induced hearing loss (CIHL) remains a significant complication of pediatric cancer treatment. We evaluated the feasibility, safety, and trends of the efficacy of intratympanic injections of sustained-exposure dexamethasone thermosensitive gel (OTO-104) for otoprotection. PROCEDURE:In this multisite randomized phase 2 trial (NCT02997189), patients aged 0.5-21 years with newly diagnosed cancer treated with cisplatin were eligible. Participants' ears were randomized to receive up to three intratympanic injections of 0.2 mL OTO-104 in one ear and no treatment contralaterally. OTO-104 was administered by an otolaryngologist within 72 hours preceding each cisplatin cycle. Feasibility was determined by the successful administration of intended doses. Treatment-emergent adverse events (TEAEs) and outcomes were monitored by physical examination, concurrent medications, otoscopy, tympanometry, and audiometry. RESULTS:From January 6 to September 26, 2017, 18 doses of OTO-104 were administered to 11 evaluable participants across 5 centers (range, 1-14 years; 9 neuroblastoma and 2 osteosarcoma) via intratympanic injection (9) or tympanostomy tube (2). Sixteen injections were paired with other procedural sedations and two were performed awake; all injections were successfully delivered. The median interval between OTO-104 and cisplatin was 14 hours (range, 7-64). Clinically insignificant tympanic scabs were noted in five participants; no middle ear changes were observed. There were three otologic TEAEs (two transient mild-moderate otalgia [related] and one hypoacusis [unrelated]) and no related non-otologic TEAEs. CIHL developed similarly in treated versus untreated ears; the trial was terminated early. CONCLUSIONS:Although dexamethasone at this dose was not otoprotective, these results suggest that intratympanic injection may be safe, feasible, and a viable delivery platform for testing other otoprotectants.

Ewing sarcoma (ES) is the second most common bone cancer in children, accounting for 2% of pediatric cancer diagnoses. Patients who present with metastatic disease at the time of diagnosis have a dismal prognosis, compared to the >70% 5-year survival of those with localized disease. Here, we utilized single-cell RNA sequencing (scRNA-seq) to characterize the transcriptional landscape of primary ES tumors, and to identify circulating tumor cells (CTCs) in peripheral blood at the time of diagnosis in order to further understand ES transcriptional heterogeneity and factors that drive metastasis. Methods: Viably frozen primary tumor and peripheral blood samples were obtained from 7 ES patients at the time of diagnosis and prior to the initiation of treatment. Tumors were dissociated into a single cell suspension and sorted for viability using FACS (fluorescence activated cell sorting) whereas peripheral blood samples were subjected to a size-based selection with the CellSieve microfiltration system. ScRNA-seq was performed using the 10xChromium platform and count matrices were generated using 10x Genomics Cell Ranger software. Quality control, integration, and cluster analysis was performed with Seurat. Results: Cluster analysis of integrated, primary tumor samples demonstrated that candidate ES cell clusters express FLI1 and ES marker NKX2-2 and cluster separately from immune cell populations. Additionally, using inferCNV, ES clusters were demonstrated to harbor chromosomal copy number alterations known to be associated with ES and that were previously identified clinically on preliminary pathology reports for each patient; further GSEA analysis showed significant overlap between published ES gene sets and genes upregulated in ES clusters. Further analysis of ES cells demonstrated that cell-cycle phase determined a cluster enriched in pro-proliferation gene signatures, and overall heterogeneity of expression of previously known therapeutic targets. Candidate ES CTCs were identified among the peripheral blood samples among clusters which corresponded with distinct immune cell populations. The candidate ES CTCs expressed NKX2-2 and demonstrated enrichment in oncogenic gene signatures. Conclusion: ScRNA-seq of primary ES tumors is feasible and demonstrates that ES tumor cells are largely homogeneous in nature; and candidate ES CTCs can be identified in peripheral blood at the time of diagnosis in ES patient and warrant further investigation as to their utility as a biomarker of metastatic disease. Citation Format: Sarah K. Nelson-Taylor, Avery Bodlack, Andrew Goodspeed, Amy Treece, Nathan Donaldson, Carrye Cost, Tim Garrington, Brian Greffe, Sandra Luna-Fineman, Jenna Sopfe, Masanori Hayashi. Single cell RNA sequencing of primary Ewing sarcoma tumors and identification of circulating tumor cells in patient-matched peripheral blood samples [abstract]. In: Proceedings of the American Association for Cancer Research Annual Meeting 2022; 2022 Apr 8-13. Philadelphia (PA): AACR; Cancer Res 2022;82(12_Suppl):Abstract nr 1681.

Management of unresectable pediatric hepatoblastoma (HB) and hepatocellular carcinoma (HCC) remains challenging. The Society of Pediatric Liver Transplantation (SPLIT) database was used to study survival predictors in pediatric liver transplantation (LT) for HB and HCC. Event-free survival (EFS), associated risk factors, and postoperative complications were studied in children requiring LT for HB/HCC at 16 SPLIT centers. Three-year EFS was 81% for HB (n = 157) and 62% for HCC (n = 18) transplants. Of HB transplants, 6.9% were PRETEXT II and 15.3% were POST-TEXT I/II. Tumor extent did not impact survival (p = NS). Salvage (n = 13) and primary HB transplants had similar 3-year EFS (62% versus 78%, p = NS). Among HCC transplants, 3-year EFS was poorer in older patients (38% in ≥8-year-olds vs 86% <8-year-olds) and those with larger tumors (48% for those beyond versus 83% within Milan criteria, p = NS). Risk of infection (HR 1.5, 95% CI 1.1-2.2, p = .02) and renal injury (HR 2.4, 95% CI 1.7-3.3, p < .001) were higher in malignant versus nonmalignant LT. Survival is favorable for pediatric HB and HCC LT, including outcomes after salvage transplant. Unexpected numbers of LTs occurred in PRE/POST-TEXT I/II tumors. Judicious patient selection is critical to distinguish tumors that are potentially resectable; simultaneously, we must advocate for patients with unresectable malignancies to receive organs.

The detection of tumor-specific nucleic acids from blood increasingly is being used as a method of liquid biopsy and minimal residual disease detection. However, achieving high sensitivity and high specificity remains a challenge. Here, we perform a direct comparison of two droplet digital PCR (ddPCR)-based detection methods, circulating plasma tumor RNA and circulating plasma tumor DNA (ptDNA), in blood samples from newly diagnosed Ewing sarcoma patients. First, we developed three specific ddPCR-based assays to detect EWS-FLI1 or EWS-ERG fusion transcripts, which naturally showed superior sensitivity to DNA detection on in vitro control samples. Next, we identified the patient-specific EWS-FLI1 or EWS-ERG breakpoint from five patient tumor samples and designed ddPCR-based, patient-specific ptDNA assays for each patient. These patient-specific assays show that although plasma tumor RNA can be detected in select newly diagnosed patients, positive results are low and statistically unreliable compared with ptDNA assays, which reproducibly detect robust positive results across most patients. Furthermore, the unique disease biology of Ewing sarcoma enabled us to show that most cell-free RNA is not tumor-derived, although cell-free-DNA burden is affected strongly by tumor-derived DNA burden. Here, we conclude that, even with optimized highly sensitive and specific assays, tumor DNA detection is superior to RNA detection in Ewing sarcoma patients.

DICER1 syndrome is a rare hereditary cancer predisposition syndrome that has relevance to pediatric urology providers due to its association with many various pediatric genitourinary malignancies. We describe the case of a pediatric patient who was eventually diagnosed with a pathogenic DICER1 germline variant after undergoing resection of a cystic nephroma and pleuropulmonary blastoma.

NUT midline carcinoma, characterized by the rearrangement of the nuclear protein in testis (NUTM1) gene, is a rare and aggressive subtype of squamous cell carcinoma. This disease is rarely cured and there have been no reports of cure in patients with distant metastatic disease. In fact, patients typically succumb to NUT midline carcinoma within 6 to 12 months from diagnosis. The authors report on a single patient who presented widely metastatic disease who has now been in remission for 37 months after multimodal therapy with compressed cycles of vincristine, cyclophosphamide, and doxorubicin alternating with ifosfamide and etoposide, high-dose radiation, and postchemotherapy resection.

NUT midline carcinoma, characterized by the rearrangement of the nuclear protein in testis (NUTM1) gene, is a rare and aggressive subtype of squamous cell carcinoma. This disease is rarely cured and there have been no reports of cure in patients with distant metastatic disease. In fact, patients typically succumb to NUT midline carcinoma within 6 to 12 months from diagnosis. The authors report on a single patient who presented widely metastatic disease who has now been in remission for 37 months after multimodal therapy with compressed cycles of vincristine, cyclophosphamide, and doxorubicin alternating with ifosfamide and etoposide, high-dose radiation, and postchemotherapy resection.

Purpose: To determine, using the National Cancer Database (NCDB), the impact of the surgery to radiation therapy interval (SRI) on survival in contemporary patients with Wilms tumor (WT). Methods and Materials: The NCDB was queried for patients aged <= 25 years diagnosed from 2004 to 2013 with unilateral WT who underwent definitive surgery and radiation therapy. The SRI was calculated for each patient. A stratified analysis was performed based on presence of metastasis using logistic regression to calculate risk factors for prolonged SRI, with a focus on the recommended SRI according to recent Children's Oncology Group trials (by day 14) and National Wilms Tumor Study-5 (by day 9). Cox regression was performed to assess the association of SRI with overall survival. Results: A total of 1488 patients were included; 32.1% had metastasis at diagnosis. Among both metastatic and nonmetastatic groups, older patients were more likely to have prolonged SRI. For those without metastasis, SRI > 14 days was associated with increased risk of mortality (hazard ratio 2.13, P=.013). Analyzing SRI as a continuous variable also demonstrated an increased risk of death with longer SRI (hazard ratio 1.04 per day, P=006) in this group. In contrast, among patients with metastasis, no significant association between SRI and mortality was found. Conclusion: Early initiation of radiation therapy remains a critical component of multimodal treatment for patients with nonmetastatic WT. For nonmetastatic patients, SRI <= 14 days correlates with improved overall survival. However, no such association was noted for patients with metastases. These results may inform the development of future WT trials. (C) 2018 Elsevier Inc. All rights reserved.

The incidence of central nervous system (CNS) involvement in patients with rhabdomyosarcoma (RMS) is low, and the outcome is dismal. We present a single institution analysis of CNS involvement of pediatric RMS. In 59 patients, the prevalence of CNS involvement was 11.9% (7 patients), higher than prior reports. Of the 6 deaths from disease, all had rapid progression, with a median survival of 14 days. The higher incidence could be secondary to treatment modifications or more sensitive detection. These findings are useful for decisions at the time of CNS involvement and could lead to modifications for future RMS clinical trials.

Introduction Testicular germ cell tumors (GCTs) are themost common solid tumor among adolescent and young adult (AYA) males. AYA patients with GCTs most typically have non-seminoma compared with seminoma, and accordingly there are fewer data reported on the AYA experience with testicular seminoma. Objective To evaluate national trends in postoperative treatment and overall survival (OS) outcomes in testicular seminoma by age group, specifically comparing AYAs with older adults. Study design The National Cancer Data Base (NCDB) was queried for patients with testicular seminoma diagnosed between 2004 and 2012, who underwent orchiectomy followed by observation or adjuvant therapy (chemotherapy, radiation (RT), or both). Patients were grouped by age: AYA (15-39 years), adults between 40 and 55 years, and adults > 55 years. Overall survival (OS) was presented using KaplaneMeier curves and groups compared via a log-rank test. Univariate (UVA) and multivariate (MVA) analyses were performed using Cox proportional hazards regression models. Binary multiple logistic regression identified differences in variables by age category. Results Of the total 22,361 patients the majority were AYAs (12,880, 57.6%), followed by adults 40-55 years (8,022, 35.9%), and > 55 years (1,459, 6.5%). Unadjusted 5-year OS was significantly better for AYAs versus adults 40-55 years and > 55 years (98.0%, 96.4%, 87.7%; p < 0.001), as was 10-year OS (96.1%, 91.8%, 71.3% respectively; p < 0.001). The Table shows that on a MVA, OS was significantly better for AYAs versus adults 40-55 years and adults > 55 years. AYA patients were also more commonly treated at centers with greater clinical volume. Additionally, AYA patients were less likely to present with metastatic disease. Accordingly, AYA patients were less likely to undergo retroperitoneal lymph node dissection (OR 0.81; p = 0.001) and were less often managed with adjuvant therapy including chemotherapy (OR 0.91; p = 0.027), RT (OR 0.93; p = 0.025), or both (OR 0.68; p = 0.020). Discussion AYA patients with testicular seminoma present with earlier stage disease and in the clinical Stage I setting are more often are managed with active surveillance following orchiectomy when compared with older adults in this population-based analysis. Among AYA patients, OS was modestly better when compared with adults 40-55 years and significantly better when compared with adults > 55 years. Conclusion Our objective to describe the patterns of care and survival outcomes for AYA patients with testicular seminoma in the USA was met by reviewing this large national dataset. These results may inform future guidelines for management of AYA seminoma.

BackgroundThe ability of intraoperative frozen section (IFS) to reliably diagnose renal tumors in children and adolescents is largely unknown. The objective of our study is to evaluate the ability of IFS to establish a histologic diagnosis for renal tumors in this population.MethodsWe reviewed our experience with patients who underwent IFS at the time of surgery for a renal tumor suspicious for malignancy from 2005 to 2015. The IFS was compared to the final pathology (FP). Data on concordance and reliability were analyzed.ResultsOne hundred thirty patients underwent surgical interventions for a renal tumor suspicious for malignancy, and 32 (25%) patients underwent IFS. Median turnaround time for IFS was 20 min (range 13-44). The histologic IFS diagnosis correlated with FP in 26 (81.2%) cases was discrepant in three (9.4%) cases, and IFS was deferred to FP in three (9.4%) cases (kappa 0.71, 95% confidence interval [CI]: 0.52-0.899, P < 0.001). The IFS correctly distinguished between Wilms tumor and non-Wilms tumor in 30 (94%) cases (kappa 0.874, 95% CI: 0.705-1, P < 0.001). A total of 17 of 19 (89.5%) Wilms tumors were correctly diagnosed by IFS, yielding a sensitivity of 0.89 (95% CI: 0.67-0.99) and a specificity of 1 (95% CI: 0.75-1).ConclusionIFS is a reliable tool to establish a histologic diagnosis and to differentiate between Wilms and non-Wilms tumors in children and adolescents with renal tumors. The use of IFS should be encouraged in cases in which obtaining a diagnosis will provide guidance for important real-time medical decision making, specifically additional adjunctive surgical procedures.

PURPOSE:The purpose of this study was to evaluate the risk of cardiac death in pediatric Hodgkin's lymphoma (HL) survivors and identify high-risk groups that may need additional surveillance.METHODS:The Surveillance, Epidemiology and End Results program database was queried to analyze the rates of radiation therapy (RT) use and cardiac-specific mortality (CSM) in HL patients, aged 0-21 years, treated from 1973 to 2007. Primary endpoint was cardiac mortality.RESULTS:A total of 6552 patients were included. Median follow-up was 12 years (range, 0-40). Median age at diagnosis was 17 years (range, 0-21). The majority were white (85.5%), from western states (41.2%), had nodular sclerosis HL (73.2%), presented with stage I or II disease (51.5%), and received RT (56.1%). Death from cardiac disease occurred in 114 patients (9.2% of all deaths). CSM for the entire cohort at 10-, 20-, and 30-year time points was 0.3%, 1.6%, and 5.0%, respectively. Median age at the time of cardiac death was 39 years (range, 18-58 years). Under multivariate analysis (MVA), adolescent patients (ages 13-21) had higher rates of CSM (hazard ratio [HR], 3.05; p = 0.005). Female gender (HR, 0.43; p < 0.001), patients treated from 1998 to 2007 (HR, 0.19; p = 0.018), and those with lymphocyte-rich histology (HR, 0.14; p = 0.047) had significantly lower rates of CSM. Use of RT was not associated with CSM under MVA (HR, 1.18, p = 0.452).CONCLUSION:The cumulative incidence of CSM in this population analysis of pediatric HL was 9.2%, with a steady decline over the past several decades. Adolescent patients at diagnosis and males were more likely to die of cardiac-related causes.

You have accessJournal of UrologySexual Function/Dysfunction: Penis/Testis/Urethra: Benign Disease & Malignant Disease I1 Apr 2016PD34-01 SURVIVAL OUTCOMES OF ADOLESCENT PATIENTS WITH NON-SEMINOMA TESTICULAR GERM-CELL TUMORS: A POPULATION-BASED STUDY Arya Amini, Timothy Waxweiler, Paul Maroni, Elizabeth Kessler, Carrye Cost, Brian Greffe, Timothy Garrington, Arthur Liu, and Nicholas Cost Arya AminiArya Amini More articles by this author , Timothy WaxweilerTimothy Waxweiler More articles by this author , Paul MaroniPaul Maroni More articles by this author , Elizabeth KesslerElizabeth Kessler More articles by this author , Carrye CostCarrye Cost More articles by this author , Brian GreffeBrian Greffe More articles by this author , Timothy GarringtonTimothy Garrington More articles by this author , Arthur LiuArthur Liu More articles by this author , and Nicholas CostNicholas Cost More articles by this author View All Author Informationhttps://doi.org/10.1016/j.juro.2016.02.986AboutPDF ToolsAdd to favoritesDownload CitationsTrack CitationsPermissionsReprints ShareFacebookTwitterLinked InEmail INTRODUCTION AND OBJECTIVES Testicular germ cell tumors (TGCT) are the most common solid malignancy in adolescent and young adult men. In adolescents, approximately 90% of TGCTs are non-seminomas (NS). Few studies have evaluated the impact of age, specifically adolescence, on outcomes of NS TGCTs. METHODS The SEER database was queried for individuals ≥13yr diagnosed with NS TGCTs from 1995-2012. Patients were categorized into adolescent (13-19yr) and adult (≥ 20yr) cohorts. A Cox proportional hazards model was used for multivariate analysis (MVA). RESULTS 13,964 patients (1,496 adolescents, 12,467 adults) were included. Median follow up was 71mo (range, 1-215mo). 5yr OS for adolescent and adult patients was 94% and 92% respectively (log-rank p=0.007); 5yr CSS was 95% and 94% respectively (log-rank p=0.139), Fig 1. Under MVA, adolescent patients had both improved OS (HR, 0.61; 95% CI, 0.50-0.75; p<0.001) and CSS (HR, 0.65; 95% CI, 0.51-0.82; p<0.001), when compared to adults. Additional predictors for improved OS included: Pure embryonal histology (HR 0.55; p<0.001), being married (HR 0.80; p=0.001), and additional tumor excision (HR 0.44; p<0.001). Factors associated with decreased OS included: African American race (HR 1.73; p<0.001), Hispanic descent (HR 1.17; p=0.019), pure choriocarcinoma histology (HR 1.59; p<0.001), residence in neighborhoods with >10% poverty (HR 1.19; p=0.016) or >10% unemployment (HR 1.16; p=0.047), presenting with regional or distant metastatic disease (HR 6.09; p<0.001), and receiving radiation (HR 2.48; p<0.001). In a logistic regression analysis adjusting for demographics, compared to adults, adolescent patients more commonly presented with regional or distant metastatic disease (OR 1.16; 95% CI, 1.01-1.35), were more likely to undergo an additional tumor excision (OR 2.43; 95% CI, 1.57-3.77) and other adjuvant surgery (OR 5.87; 95% CI 2.25-15.3). Additionally, adolescents were less likely to undergo radiation (OR 0.61; 95% CI, 0.39-0.97). CONCLUSIONS Adolescent patients with NS TGCTs had slightly improved survival compared to adults, despite more advanced disease. This may imply that while adolescent patients present at more advanced stage, they achieve excellent survival outcomes but possibly at the cost of greater therapeutic burden. © 2016FiguresReferencesRelatedDetails Volume 195Issue 4SApril 2016Page: e838-e839 Advertisement Copyright & Permissions© 2016MetricsAuthor Information Arya Amini More articles by this author Timothy Waxweiler More articles by this author Paul Maroni More articles by this author Elizabeth Kessler More articles by this author Carrye Cost More articles by this author Brian Greffe More articles by this author Timothy Garrington More articles by this author Arthur Liu More articles by this author Nicholas Cost More articles by this author Expand All Advertisement Advertisement PDF downloadLoading ...

BACKGROUND:In adolescents, approximately 90% of testicular germ cell tumors (T-GCTs) are non-seminomas (NS T-GCTs). Few studies have evaluated the impact of age, specifically in adolescence, on outcomes of NS T-GCTs. OBJECTIVE:The purpose of this study was to review all patients diagnosed with NS T-GCTs in the Surveillance, Epidemiology, and End Results (SEER) database to evaluate the association between age (adolescents vs. adults) and survival outcomes. METHOD:The SEER database was queried for individuals ≥13 years old diagnosed with NS T-GCTs from 1995 to 2012. Patients were categorized into adolescent (13-19 years) and adult (≥20 years) cohorts. A Cox proportional hazards model was used for multivariate analysis (MVA). RESULTS:A total of 13,963 patients (1496 adolescents, 12,467 adults) was included. Median follow-up was 71 months (range 1-215). Five-year overall survival (OS) for adolescent and adult patients was 94% and 92%, respectively (p = 0.007); 5-year cancer-specific survival (CSS) was 95% and 94%, respectively (p = 0.139). Under MVA, adolescent patients had improved OS (HR 0.61; 95% CI 0.50-0.75; p < 0.001) and CSS (HR 0.65; 95% CI 0.51-0.82; p < 0.001), when compared with adults (Table). In a logistic regression analysis adjusting for demographics, adolescent patients were more likely to present with regional or distant metastatic disease (OR 1.16; 95% CI 1.01-1.35; p = 0.039), undergo an orchiectomy (OR 2.44; 95% CI 1.50-4.00; p < 0.001) or tumor excision (OR 2.43; 95% CI 1.57-3.77; p < 0.001), and receive other adjuvant surgery (OR 5.87; 95% CI 2.25-15.30; p < 0.001). CONCLUSIONS:To our knowledge, this is the largest population-based comparative analysis in NS T-GCTs comparing outcomes between these two age groups. Adolescent patients with NS T-GCTs had slightly improved survival compared with adults, despite presenting with more advanced disease. While adolescent patients present at more advanced stage, they achieve excellent survival outcomes possibly at the cost of a greater therapeutic burden.

Describe the development and evolution of a primary-care-based, multidisciplinary clinic to support the ongoing care of adult survivors of childhood cancer.

1.To understand the successful partnership model in developing a pediatric palliative care program.2.To understand the partnership model approach in providing pediatric palliative care. As pediatric palliative care (PPC) evolves, delivery of PPC has been approached using a variety of models. Our approach over the past 13 years has been to tap into the expertise of a local adult oriented hospice organization to provide emotional and nursing support (pediatric RNs only) to the outpatient population while also using the medical and nursing expertise of a pediatric focused organization (a children's hospital) for pain and symptom management and nursing issues and to oversee the inpatient palliative care service. We present here demographic data and patient satisfaction outcomes related to our pediatric palliative care program. Demographic data and patient satisfaction outcomes have been tracked since the inception of the program in 1999. This model of care has been successful in educating both community and hospital-based providers about PPC issues. The Butterfly Program has served to date over 400 patients and families. The average length of stay in the program reflects the true palliative nature of the program and has ranged up to over 700 days. Because the program seeks to enroll patients earlier in the trajectory of their illnesses, there has been no reimbursement for the emotional care services and these costs have been covered by the two partner organizations. The Butterfly Program has been instrumental in working with the state Medicaid organization to successfully implement a pediatric palliative and hospice care waiver allowing palliative care services to start from the time of diagnosis of a life-limiting illness with Medicaid reimbursement for emotional support services. Successful partnership of a children's hospital and local healthcare agency/hospice to provide PPC services can be accomplished.