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OBJECTIVES: Adolescents and young adults (AYAs, 18-39 years) with cancer identifying as racially/ethnically minoritized or 2SLGBTQIA+ (Two-Spirit, lesbian, gay, bisexual, transgender, queer, intersex, asexual and "+" referring to other queer identities) have been underrepresented in cancer research. This study explores the aspects of identity that hold significance for these minoritized AYAs and how these facets impact their healthcare experiences. METHODS: Eligible participants comprised English-speaking AYAs who self-identified as racially/ethnically minoritized and/or 2SLGBTQIA+, were diagnosed with cancer between the ages of 15-39, currently aged > 18, and had received or were receiving cancer care within Canadian healthcare system. Additionally, four patient partners meeting the same criteria were recruited as research collaborators. Semi-structured one-on-one virtual interviews guided by an interview script were conducted, and qualitative analysis employed a framework approach. RESULTS: We recruited 23 participants from 4 Canadian provinces (mean age: 28, Range: 20-44); 17 identified as racially/ethnically minoritized, one as sexual/gender minoritized, and five as racially/ethnically and sexually/gender minoritized. Participants emphasized that their culture/ethnicity, religion/spirituality, sexuality, gender, family, career, and being an immigrant are important aspects of their identity, with only one participant recognizing their identity as a "person with cancer". A cancer diagnosis altered the aspects of identity deemed most significant by participants. Both visible and invisible aspects of identity shaped participants' experiences and influenced their level of trust in the healthcare system. CONCLUSION: Racially, ethnically, gender, or sexually minoritized AYAs with cancer place considerable importance on aspects of their identity that are shaped by their respective communities. Recognizing and respecting these identities are paramount for healthcare professionals to deliver safe and inclusive care.
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Neoplasias , Minorías Sexuales y de Género , Adolescente , Adulto , Femenino , Humanos , Masculino , Adulto Joven , Canadá , Minorías Étnicas y Raciales/psicología , Neoplasias/psicología , Neoplasias/etnología , Investigación Cualitativa , Minorías Sexuales y de Género/psicologíaRESUMEN
Importance: Depression is prevalent among patients with cancer, affecting their quality of life and survival. Aerobic physical activity (APA) is an effective strategy for managing depression in the general population, but its effectiveness for reducing depressive symptoms among patients with cancer requires further study. Objective: To evaluate whether APA decreases depression severity in patients with cancer by synthesizing data from published randomized clinical trials (RCTs). Data Sources: Six databases (MEDLINE, Embase, Cochrane Central Register of Controlled Trials, CINAHL, PsycINFO, and Scopus) were systematically searched for relevant citations published between January 1, 1980, and July 5, 2023. Study Selection: This systematic review and meta-analysis included RCTs comparing APA interventions with usual care, waitlist control, or attention control for managing depression in patients with cancer, irrespective of age and cancer type. Data Extraction and Synthesis: Two reviewers independently conducted screening and data extraction. Risk of bias was evaluated using the Cochrane Risk of Bias Tool, version 2. The Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) reporting guideline was followed. Standardized mean differences (SMDs) were calculated using a random-effects model. Data collection and analyses were performed between June 2022 and March 2024. Main Outcomes and Measures: The primary outcome was severity of self-reported depression reported within 1 month of the end of intervention (short term). Secondary outcomes were severity of self-reported depression between 1 and 6 months post intervention (medium term) and between 6 and 12 months post intervention (long term). Results: This meta-analysis included 25 RCTs with a total of 1931 adults with cancer (age range, 18-80 years). Ten RCTs (40%) had low risk of bias. Aerobic physical activity was associated with decreased self-reported depression among adults with cancer across the 25 included trials (n = 1931 participants; SMD, -0.38 [95% CI, -0.59 to -0.18]; P < .001; I2 = 76%). This decrease in depression scores was also significant for the secondary outcomes of long-term depression across 3 trials (n = 299 participants; SMD, -0.32 [95% CI, -0.60 to -0.04]; P = .03; I2 = 31%) but not for medium-term depression across 2 trials (n = 143 participants; SMD, -0.27 [95% CI, -0.60 to 0.06]; P = .10; I2 = 0%). Conclusions and Relevance: In this systematic review and meta-analysis, APA was associated with modest short-term and long-term reductions of depression among adults with cancer. Future studies should discern the effectiveness of APA in combination with other strategies for managing depression across various populations of patients with cancer.
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Depresión , Ejercicio Físico , Neoplasias , Adulto , Femenino , Humanos , Masculino , Persona de Mediana Edad , Depresión/psicología , Depresión/terapia , Ejercicio Físico/psicología , Neoplasias/complicaciones , Neoplasias/psicología , Neoplasias/rehabilitación , Calidad de Vida/psicología , Ensayos Clínicos Controlados Aleatorios como Asunto , Adulto Joven , Anciano , Anciano de 80 o más AñosRESUMEN
Clinical trials conducted by the Intergroup Rhabdomyosarcoma (RMS) Study Group and the Children's Oncology Group have been pivotal to establishing current standards for diagnosis and therapy for RMS. Recent advancements in understanding the biology and clinical behavior of RMS have led to more nuanced approaches to diagnosis, risk stratification, and treatment. The complexities introduced by these advancements, coupled with the rarity of RMS, pose challenges to conducting large-scale phase 3 clinical trials to evaluate new treatment strategies for RMS. Given these challenges, systematic planning of future clinical trials in RMS is paramount to address pertinent questions regarding the therapeutic efficacy of drugs, biomarkers of response, treatment-related toxicity, and patient quality of life. Herein, the authors outline the proposed strategic approach of the Children's Oncology Group Soft Tissue Sarcoma Committee to the next generation of RMS clinical trials, focusing on five themes: improved novel agent identification and preclinical to clinical translation, more efficient trial development and implementation, expanded opportunities for knowledge generation during trials, therapeutic toxicity reduction and quality of life, and patient engagement.
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Ensayos Clínicos como Asunto , Calidad de Vida , Rabdomiosarcoma , Humanos , Rabdomiosarcoma/terapia , Rabdomiosarcoma/tratamiento farmacológico , NiñoRESUMEN
The Adolescent and Young Adult (AYA) Program at CancerCare Manitoba (CCMB) has experienced tremendous growth since its inception. This report provides an overview of how the AYA program at CCMB was established and the crucial factors that led to its early accomplishments and continued expansion. These factors included actions and decisions made at the individual and organizational level that helped lay a strong foundation for the program's sustained success. We hope that some of these lessons learned can be adapted and implemented by other oncology agencies to improve the care outcomes and experiences of AYAs living with cancer.
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Neoplasias , Humanos , Adolescente , Adulto Joven , Neoplasias/terapia , Oncología Médica/métodos , Canadá , Masculino , Femenino , Adulto , ManitobaRESUMEN
Adolescents and young adults (AYAs; 15-39 years) diagnosed with cancer have unique medical and psychosocial needs. These needs could be better addressed through research that is focused on the topics that matter most to them. However, there is currently no patient-oriented research agenda for AYA cancer in Canada. This manuscript describes the early development and project protocol for a priority-setting partnership (PSP) for establishing the top 10 research priorities for AYA cancer in Canada. This project follows the PSP methodology outlined by the James Lind Alliance (JLA) to engage patients, caregivers, and clinicians in research prioritization. The steps of a JLA PSP include establishing a steering group and project partners, gathering uncertainties, data processing and verifying uncertainties, interim priority setting, and a final priority setting workshop. The AYA cancer PSP will result in a top 10 list of research priorities identified by Canadian AYA patients, caregivers, and clinicians that will be published and shared broadly with the research community. The first steering group meeting was held in April 2023, and the project is ongoing. The establishment of a patient-oriented research agenda for AYA cancer will catalyze a long-term and impactful research focus and ultimately improve outcomes for AYA patients with cancer in Canada.
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Neoplasias , Humanos , Adolescente , Canadá , Adulto Joven , Adulto , Investigación , Femenino , Investigación Biomédica , Prioridades en Salud , MasculinoRESUMEN
[This corrects the article DOI: 10.3389/fonc.2024.1376652.].
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Introduction: From the advancement of treatment of pediatric cancer diagnosis, the five-year survival rate has increased significantly. However, the adverse consequence of improved survival rate is the second malignant neoplasm. Although previous studies provided information on the incidence and risk of SMN in long term survivors of childhood cancer, there is still scarce information known for short term (< 5 years) prognosis. This study aims to assess the incidence, characteristics, management, and outcome of children who develop SMN malignancies within 5 years of diagnosis of their initial cancer. Method: This is a retrospective cohort study of early Second Malignant Neoplasms (SMN) in pediatric oncology patients. The Cancer in Young People - Canada (CYP-C) national pediatric cancer registry was used and reviewed pediatric patients diagnosed with their first cancer from 2000-2015. Results: A total of 20,272 pediatric patients with a diagnosis of a first malignancy were analyzed. Of them, 0.7% were diagnosed with a SMN within the first 5 years following their first cancer diagnosis. Development of a SMN impacted survival, shown by an inferior survival rate in the SMN cohort (79.1%) after three years compared to that of the non-SMN cohort (89.7%). Several possible risk factors have been identified in the study including the use of epipodophyllotoxins, exposure to radiation, and hematopoietic stem cell 169 transplant. Discussion: This is the first national study assessing the incidence, 170 characteristics, risk factors and outcome of early SMN in Canadian children 171 from age 0-15 from 2000-2015.
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BACKGROUND: Novel therapies are needed for relapsed and refractory rhabdomyosarcoma (RRMS). Phase II clinical trials in RRMS have typically utilized radiologic response as the primary activity endpoint, an approach that poses several limitations in RRMS. In this analysis, we aimed to estimate an event-free survival (EFS) endpoint for RRMS that could be used as a benchmark for future studies. PROCEDURE: We performed a retrospective study of patients with RRMS enrolling on 13 single-agent phase II Children's Oncology Group and legacy group trials from 1997 to 2016. All included trials used radiographic response as their primary activity endpoint. Six-month EFS was estimated from time of trial enrollment with 95% confidence intervals. Clinical characteristics, including trial of enrollment, sex, age, race, histology, number of prior chemotherapies, and radiographic response were evaluated for their impact on 6-month EFS. RESULTS: We identified 175 patients across 13 trials. The 6-month EFS was 16.8% (11.6%-22.8%). No differences were seen in 6-month EFS based on age, sex, race, or histology. There were nonsignificant trends toward improved 6-month EFS for patients with less than or equal to two prior lines of therapy versus higher than two, for patients enrolled on trials that achieved their primary radiographic response endpoint versus trials that did not, and for patients who achieved complete or partial response compared to those achieving stable disease. CONCLUSIONS: The prognosis of RRMS enrolled on single-agent phase II trials is poor. This pooled 6-month EFS of RRMS on single-agent trials may be used as a RRMS-specific benchmark for future single-agent phase II trials.
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Ensayos Clínicos Fase II como Asunto , Recurrencia Local de Neoplasia , Rabdomiosarcoma , Humanos , Femenino , Masculino , Niño , Rabdomiosarcoma/mortalidad , Rabdomiosarcoma/tratamiento farmacológico , Rabdomiosarcoma/terapia , Rabdomiosarcoma/patología , Estudios Retrospectivos , Preescolar , Adolescente , Lactante , Recurrencia Local de Neoplasia/patología , Recurrencia Local de Neoplasia/mortalidad , Recurrencia Local de Neoplasia/tratamiento farmacológico , Tasa de Supervivencia , Pronóstico , Estudios de SeguimientoRESUMEN
IMPORTANCE: Data on the middle school outcomes of preterm children are limited and have methodologic issues. OBJECTIVE: To study the association between preterm birth and grade 7 school performance. METHODS: A retrospective population-based cohort study of children born in Manitoba, Canada between 1994 and 2006 using their grade 7 school performance data. A secondary sibling cohort was created comprising children born preterm and their full-term siblings. Primary exposure was preterm birth categorized as <28, 28-33 and 34-36 weeks gestation. The two co-primary grade 7 outcome measures were: not meeting the mathematics competencies, and not meeting the student engagement competencies. Multivariable logistic regression models tested the association between preterm birth and both co-primary outcomes; adjusted odds ratios (aORs) and 95% confidence intervals (CIs) were calculated. RESULTS: 7653 preterm (gestational age median [IQR]: 35 weeks [34,36]) and 110,313 term (40 [39,40]) were included. 43% of < 28 weeks, 18% of 28-33 weeks and 17% of 34-36 weeks had the mathematics co-primary outcome compared to 13% of term children. The corresponding % for the student engagement outcome were 42%, 24%, 24% and 24% respectively. Preterm birth was associated with the mathematics (<28 weeks: 5.48, 3.89-7.70; 28-33 weeks: 1.47, 1.27-1.70; 34-36 weeks: 1.26, 1.16-1.35) and student engagement outcomes (<28 weeks: 2.49, 1.76-3.51; 28-33 weeks: 1.21, 1.06-1.39; 34-36 weeks: 1.09, 1.01-1.16). However, there was no difference in outcomes among the sibling cohort. CONCLUSIONS AND RELEVANCE: Children born preterm had lower grade 7 performance compared to children born term in this population-based cohort. Screening and supports for them in their middle school years are warranted.
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Edad Gestacional , Recien Nacido Prematuro , Humanos , Femenino , Estudios Retrospectivos , Masculino , Niño , Recién Nacido , Modelos Logísticos , Manitoba , Matemática , Nacimiento Prematuro/epidemiología , Rendimiento Académico/estadística & datos numéricos , Instituciones Académicas , Canadá , Análisis MultivarianteRESUMEN
BACKGROUND: The aim of this study was to estimate the event-free survival (EFS) of children and young adults with relapsed or refractory nonrhabdomyosarcoma soft tissue sarcoma (NRSTS) treated in nonrandomized phase 2 studies conducted by the Children's Oncology Group (COG) and predecessor groups to establish a benchmark EFS for future phase 2 NRSTS trials evaluating the activity of novel agents. METHODS: A retrospective analysis of patients with recurrent or refractory NRSTS prospectively enrolled in nonrandomized phase 2 COG and predecessor group trials between 1994 and 2015 was conducted. EFS was defined as disease progression/relapse or death and calculated via the Kaplan-Meier method. The log-rank test and relative risk regression were used to compare EFS distribution by age at enrollment, sex, race, NRSTS histology, prior lines of therapy, calendar year of trial, and type of radiographic response. RESULTS: In total, 137 patients were enrolled in 13 phase 2 trials. All trials used radiographic response rate as a primary outcome, and none of the agents used were considered active on the basis of trial-specified thresholds. The estimated median EFS and 6-month EFS of the entire study cohort was 1.5 months (95% confidence interval [CI], 1.3-1.8 months) and 19.4% (95% CI, 12.7%-26%), respectively. No difference in EFS was observed by age at enrollment, sex, race, NRSTS histology subtype, prior lines of therapies, and trial initiation year. EFS significantly differed by radiographic response. CONCLUSIONS: The EFS for children and young adults with relapsed or refractory NRSTS remains suboptimal. Established EFS can be referenced as a benchmark for future single-agent phase 2 trials incorporating potentially active novel agents in this population.
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Ensayos Clínicos Fase II como Asunto , Recurrencia Local de Neoplasia , Sarcoma , Humanos , Femenino , Masculino , Niño , Adolescente , Sarcoma/patología , Sarcoma/mortalidad , Sarcoma/tratamiento farmacológico , Sarcoma/terapia , Preescolar , Recurrencia Local de Neoplasia/patología , Adulto Joven , Estudios Retrospectivos , Adulto , Lactante , Resultado del Tratamiento , Supervivencia sin Progresión , Estimación de Kaplan-MeierRESUMEN
PURPOSE: To compare the cumulative incidence of mental disorders among adolescents and young adults (AYAs) diagnosed with cancer with the general population and their unaffected siblings. METHODS: A retrospective, population-based, matched cohort design was used to investigate the impact of cancer diagnosis on mental disorders among individuals age 15-39 diagnosed between 1989 and 2019. Two cancer-free cohorts were identified: matched population-based and sibling cohorts. Outcomes included incidence of mood and anxiety disorders, substance use disorders, suicide outcomes, psychotic disorders, and any of the preceding four categories within 5 years of cancer diagnosis. Competing risk regression was used to estimate adjusted subhazard ratios (aSHR) and 95% CIs. RESULTS: Among 3,818 AYAs with cancer matched to the population-based cancer-free cohort, individuals with cancer were more likely to be diagnosed with incident mental disorders than those without cancer; the risk was highest immediately after a cancer diagnosis and decreased over time with aSHR [95% CI] for mood and anxiety disorders at 0-6 months (11.27 [95% CI, 6.69 to 18.97]), 6-12 months (2.35 [95% CI, 1.54 to 3.58]), and 12-24 months (2.06 [95% CI, 1.55 to 2.75]); for substance use disorders at 0-6 months (2.73 [95% CI, 1.90 to 3.92]); for psychotic disorders at 0-6 months (4.69 [95% CI, 2.07 to 10.65]); and for any mental disorder at 0-6 months (4.46 [95% CI, 3.41 to 5.85]), 6-12 months (1.56 [95% CI, 1.14 to 2.14]), and 12-24 months (1.7 [95% CI, 1.36 to 2.13]) postcancer diagnosis. In sibling comparison, cancer diagnosis was associated with a higher incidence of mood and anxiety and any mental disorder during first 6 months of cancer diagnosis. CONCLUSION: AYAs with cancer experience a greater incidence of mental disorders after cancer diagnosis relative to population-based and sibling cohorts without cancer, primarily within first 2 years, underscoring the need to address mental health concerns during this period.
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Trastornos Mentales , Neoplasias , Hermanos , Humanos , Neoplasias/psicología , Neoplasias/epidemiología , Adolescente , Masculino , Femenino , Adulto Joven , Hermanos/psicología , Adulto , Trastornos Mentales/epidemiología , Estudios Retrospectivos , Canadá/epidemiología , Incidencia , Estudios de CohortesRESUMEN
OBJECTIVES: We conducted a retrospective multi-centre study to assess the real-world outcome of regorafenib (REGO) and cabozantinib (CABO) in recurrent/refractory bone tumours (BTs) including osteosarcoma (OST), Ewing sarcoma (EWS) and chondrosarcoma (CS)/extra-skeletal mesenchymal CS (ESMC). METHODS: After regulatory approval, data from patients with recurrent BT (11 institutions) were extracted from CanSaRCC (Canadian Sarcoma Research and Clinical Collaboration) database. Patient characteristics, treatment and outcomes were collected. Progression-free survival (PFS) and overall survival (OS) were estimated using the Kaplan-Meier method. RESULTS: From July 2018 to May 2022, 66 patients received REGO or CABO; 39 OST, 18 EWS, 4 CS and 5 ESMC. Median age was 27.8 years (range 12-76); median starting dose was 60 mg for CABO (n = 37, range 40-60) and 120 mg for REGO (n = 29, range 40-160). Twenty-eight (42.4%) patients required dose reduction: hand-foot syndrome 7 (10.6%), nausea/vomiting 1 (1.5%), diarrhoea 1 (1.5%), 2 elevated LFTs (3%), elevated bilirubin 1 (1.5%) and mucositis 1 (1.5%). The median OS for patients with OST, EWS, CS and ESMC was 8.5 months (n = 39, 95% CI 7-13.1); 13.4 months (n = 18, 95% CI 3.4-27.2), 8.1 (n = 4, 95% CI 4.1-9.3) and 18.2 (n = 5, 95% CI (10.4-na), respectively. Median PFS for OST, EWS, CS and ECMS was 3.5 (n = 39, 95% CI 2.8-5), 3.9 (n = 18, 95% CI 2.1-5.9), 5.53 (n = 4. 95% CI 2.13-NA) and 11.4 (n = 5, 95% CI 1.83-14.7), respectively. Age, line of therapy, REGO versus CABO, or time from diagnosis to initiation of TKI were not associated with PFS on univariable analysis. CONCLUSION: Our real-world data show that TKIs have meaningful activity in recurrent BT with acceptable toxicities when started at modified dosing. Inclusion of TKIs in earlier lines of treatment and/or maintenance therapy could be questions for future research.
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Neoplasias Óseas , Condrosarcoma , Osteosarcoma , Sarcoma de Ewing , Sarcoma , Neoplasias de los Tejidos Blandos , Humanos , Adulto , Niño , Adolescente , Adulto Joven , Persona de Mediana Edad , Anciano , Recurrencia Local de Neoplasia/tratamiento farmacológico , Canadá , Neoplasias Óseas/tratamiento farmacológico , Neoplasias Óseas/patología , Sarcoma/tratamiento farmacológico , Sarcoma de Ewing/tratamiento farmacológico , Sarcoma de Ewing/patología , Osteosarcoma/patología , Estudios RetrospectivosRESUMEN
Rhabdomyosarcoma (RMS) is the most common soft tissue sarcoma among children and adolescents. The management of RMS involves risk stratification of the patients based on various clinicopathological characteristics. The multimodality treatment approach requires chemotherapy, surgery, and/or radiation. The treatment of RMS necessitates the involvement of multiple disciplines, such as pathology, pediatric oncology, surgery, and radiation oncology. The disease heterogeneity, molecular testing, evolving treatment regimens, and limited resources are some of the challenges faced by clinicians while treating a patient with RMS in low- and middle-income countries (LMICs). In this review, we endeavor to bring experts from varying fields to address clinicians' common questions while managing a child or adolescent with RMS in LMICs. This review is most applicable to level 2 centers in LMICs as per the levels of services described by the Adapted Treatment Regimens Working Group of the Pediatric Oncology in Developing Countries committee of the International Society of Pediatric Oncology.
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Rabdomiosarcoma , Sarcoma , Niño , Adolescente , Humanos , Países en Desarrollo , Rabdomiosarcoma/patología , Terapia Combinada , América del NorteRESUMEN
Objective was to update a clinical practice guideline (CPG) for the management of fatigue in children and adolescents with cancer or pediatric hematopoietic cell transplant recipients. We reconvened a multi-disciplinary and multi-national panel. While the previous 2018 CPG evaluated adult and pediatric randomized controlled trials (RCTs) to manage fatigue, this 2023 update revised previous recommendations based only on pediatric RCTs. Twenty RCTs were included in the updated systematic review. Physical activity significantly reduced fatigue (standardized mean difference -0.44, 95% confidence interval -0.64 to -0.24; n = 8 RCTs). Using the 2018 recommendations as a basis, the panel continued to make strong recommendations to use physical activity, and to offer relaxation, mindfulness or both, to manage fatigue in pediatric patients. Cognitive or cognitive behavioral therapies may be offered. Pharmacological approaches should not be routinely used. The panel made a new good practice statement to routinely assess for fatigue, ideally using a validated scale.
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BACKGROUND: Despite the widespread use of medical cannabis, little is known regarding the safety, efficacy, and dosing of cannabis products in children with cancer. The objective of this study was to systematically appraise the existing published literature for the use of cannabis products in children with cancer. METHODS: This systematic review, registered with the International Prospective Register of Systematic Reviews (CRD42020187433), searched four databases: MEDLINE, Embase, PsycINFO, and the Cochrane Library. Abstracts and full texts were screened in duplicate. Data on types of cannabis products, doses, formulations, frequencies, routes of administration, indications, and clinical and demographic details as well as reported efficacy outcomes were extracted. Data on cannabinoid-related adverse events were also summarized. RESULTS: Out of 34,611 identified citations, 19 unique studies with a total of 1927 participants with cancer were included: eight retrospective chart reviews, seven randomized controlled trials, two open-label studies, and two case reports. The included studies reported the use of various cannabis products for the management of symptoms. Cannabinoids were commonly used for the management of chemotherapy-induced nausea and vomiting (11 of 19 [58%]). In controlled studies, somnolence, dizziness, dry mouth, and withdrawal due to adverse events were more commonly associated with the use of cannabinoids. Across all included studies, no serious cannabis-related adverse events were reported. CONCLUSIONS: Although there is evidence to support the use of cannabis for symptom management, in children with cancer, there is a lack of rigorous evidence to inform the dosing, safety, and efficacy of cannabinoids. Because of the increasing interest in using cannabis, there is an urgent need for more research on medical cannabis in children with cancer.
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Cannabinoides , Marihuana Medicinal , Neoplasias , Niño , Humanos , Cannabinoides/uso terapéutico , Cannabis , Marihuana Medicinal/uso terapéutico , Neoplasias/tratamiento farmacológico , Estudios Retrospectivos , Vómitos/inducido químicamente , Vómitos/tratamiento farmacológico , Antineoplásicos/efectos adversosRESUMEN
In the United States, approximately 850-900 children and adolescents each year are diagnosed with soft tissue sarcomas (STS). STS are divided into rhabdomyosarcoma (RMS) and non-rhabdomyosarcoma STS (NRSTS). RMS and NRSTS are risk stratified into low-, intermediate-, and high-risk categories, with 5-year survival rates of approximately 90%, 50%-70%, and 20%, respectively. Recent key achievements from the Children's Oncology Group (COG) STS Committee include the identification of new molecular prognostic factors for RMS, development and validation of a novel risk stratification system for NRSTS, successful completion of a collaborative NRSTS clinical trial with adult oncology consortia, and collaborative development of the INternational Soft Tissue SaRcoma ConsorTium (INSTRuCT). Current COG trials for RMS are prospectively evaluating a new risk stratification system that incorporates molecular findings, de-intensification of therapy for a very low-risk subgroup, and augmented therapy approaches for intermediate- and high-risk RMS. Trials for NRSTS exploring novel targets and local control modalities are in development.
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Rabdomiosarcoma , Sarcoma , Neoplasias de los Tejidos Blandos , Adulto , Adolescente , Niño , Humanos , Sarcoma/tratamiento farmacológico , Rabdomiosarcoma/terapia , Neoplasias de los Tejidos Blandos/terapia , Neoplasias de los Tejidos Blandos/diagnóstico , Tasa de Supervivencia , Oncología MédicaRESUMEN
BACKGROUND: Temsirolimus has shown in vivo activity against rhabdomyosarcoma (RMS). We aimed to determine the feasibility of incorporating temsirolimus within the standard Children's Oncology Group (COG) chemotherapy backbone of vincristine, actinomycin-D, and cyclophosphamide (VAC) alternating with vincristine and irinotecan (VI) in children with intermediate-risk (IR) RMS. METHODS: The feasibility phase of the COG IR-RMS trial, ARST1431 (NCT02567435), assigned 10 patients to receive 15 mg/m2 /dose (dose level 1) of temsirolimus on days 1, 8, and 15 of each of three weekly VAC and VI cycles for the first 12 weeks of induction chemotherapy. The primary endpoint of the feasibility phase was to establish the safe dose and safety of combining temsirolimus with VAC/VI. The combination regimen was deemed feasible if less than 40% of patients developed a priori defined nonhematological dose-limiting toxicities (DLTs). RESULTS: Ten patients (seven males and three females; median age = 4.5 years [range: 0.2-14.4 years]) with IR-RMS were enrolled and received dose level 1 of temsirolimus. Eight patients had FOXO1-negative disease, while two had FOXO1-positive disease. Two patients had metastatic disease. Of 10 patients, two developed DLTs: grade 3 oral mucositis and pneumonitis. Four patients (40%) had grade 4 neutropenia. No treatment-related mortality occurred. The median duration of the completion of the feasibility phase was 12.1 weeks (range: 11.7-15 weeks). CONCLUSIONS: Weekly temsirolimus at 15 mg/m2 /dose during VAC/VI chemotherapy was feasible and well tolerated. The efficacy of this regimen is currently being tested in a phase III randomized trial against VAC/VI chemotherapy alone in the ARST1431 trial.
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Purpose: Cancer-related cognitive deficits (CRCDs) are common among young adults (YAs) (ages: 18-39) with cancer and can be debilitating. We aimed to determine the feasibility and acceptability of a virtual Coping with Brain Fog intervention among YAs with cancer. Our secondary aims were to explore the intervention's effect on cognitive functioning and psychological distress. Methods: This prospective feasibility study involved eight weekly, 90-minute virtual group sessions. Sessions focused on psychoeducation on CRCD, memory skills, task management, and psychological well-being. The primary outcomes were feasibility and acceptability of the intervention evaluated through attendance (>60% not missing >2 consecutive sessions) and satisfaction (Client Satisfaction Questionnaire [CSQ] score >20). Secondary outcomes included the following: cognitive functioning (Functional Assessment of Cancer Therapy-Cognitive Function [FACT-Cog] Scale) and symptoms of distress (Patient-Reported Outcomes Measurement Information System [PROMIS] Short Form-Anxiety/Depression/Fatigue) and participants' experiences using semistructured interviews. Paired t-tests and summative content analysis were used for quantitative and qualitative data analyses. Results: Twelve participants (five male, mean age = 33 years) were enrolled. All but one participant met feasibility criteria of not missing >2 consecutive sessions (11/12 = 92%). The mean CSQ score was 28.1 (standard deviation 2.5). Significant improvement in cognitive function as measured by FACT-Cog Scale was observed postintervention (p < 0.05). Ten participants adopted strategies from the program to combat CRCD, and eight reported CRCD symptom improvement. Conclusion: A virtual Coping with Brain Fog intervention is feasible and acceptable for the symptoms of CRCD among YAs with cancer. The exploratory data indicate subjective improvement in cognitive function, and will inform the design and implementation of a future clinical trial. ClinicalTrials.gov Registration: NCT05115422.
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RMS most commonly presents in children and adolescents, however a subset of tumors are diagnosed in infants under one year of age. Due to the rarity of infant RMS, utilization of different treatment approaches and goals, and small sample sizes, the published studies of infants with RMS have yielded heterogeneous results. In this review, we discuss the outcomes of infants with RMS treated in various clinical trials and the strategies that various international cooperative groups have employed to reduce the morbidity and mortality related to treatment without compromising the overall survival of this population. This review discusses the unique scenarios of diagnosing and managing congenitals or neonatal RMS, spindle cell RMS and relapsed RMS. This review concludes by exploring novel approaches to diagnosis and management of infants with RMS that are currently being studied by various international cooperative groups.
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Published outcomes for children with cancer with coronavirus disease 2019 (COVID-19) have varied. Outcome data for pediatric oncology patients in Canada, outside of Quebec, have not been reported. This retrospective study captured patient, disease, and COVID-19-related infectious episode characteristics and outcome data for children, 0 to 18 years, diagnosed with a first COVID-19 infection between January 2020 to December 2021 at 12 Canadian pediatric oncology centers. A systematic review of pediatric oncology COVID-19 cases in high-income countries was also undertaken. Eighty-six children were eligible for study inclusion. Thirty-six (41.9%) were hospitalized within 4 weeks of COVID-19; only 10 (11.6%) had hospitalization attributed to the virus, with 8 being for febrile neutropenia. Two patients required intensive care unit admission within 30 days of COVID-19 infection, neither for COVID-19 management. There were no deaths attributed to the virus. Of those scheduled to receive cancer-directed therapy, within 2 weeks of COVID-19, 20 (29.4%) experienced treatment delays. Sixteen studies were included in the systematic review with highly variable outcomes identified. Our findings compared favorably with other high-income country's pediatric oncology studies. No serious outcomes, intensive care unit admissions, or deaths, in our cohort, were directly attributable to COVID-19. These findings support the minimization of chemotherapy interruption after COVID-19 infection.