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More knowledge is needed regarding germline predisposition to Ewing sarcoma to inform biological investigation and clinical practice. Here, we evaluated the enrichment of pathogenic germline variants in Ewing sarcoma relative to other pediatric sarcoma subtypes, as well as patterns of inheritance of these variants. We carried out European-focused and pan-ancestry case-control analyses to screen for enrichment of pathogenic germline variants in 141 established cancer predisposition genes in 1,147 individuals with pediatric sarcoma diagnoses (226 Ewing sarcoma, 438 osteosarcoma, 180 rhabdomyosarcoma, and 303 other sarcoma) relative to identically processed cancer-free control individuals. Findings in Ewing sarcoma were validated with an additional cohort of 430 individuals, and a subset of 301 Ewing sarcoma parent-proband trios was analyzed for inheritance patterns of identified pathogenic variants. A distinct pattern of pathogenic germline variants was seen in Ewing sarcoma relative to other sarcoma subtypes. FANCC was the only gene with an enrichment signal for heterozygous pathogenic variants in the European Ewing sarcoma discovery cohort (three individuals, OR 12.6, 95% CI 3.0-43.2, p = 0.003, FDR = 0.40). This enrichment in FANCC heterozygous pathogenic variants was again observed in the European Ewing sarcoma validation cohort (three individuals, OR 7.0, 95% CI 1.7-23.6, p = 0.014), representing a broader importance of genes involved in DNA damage repair, which were also nominally enriched in individuals with Ewing sarcoma. Pathogenic variants in DNA damage repair genes were acquired through autosomal inheritance. Our study provides new insight into germline risk factors contributing to Ewing sarcoma pathogenesis.
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Sarcoma de Ewing , Sarcoma , Niño , Daño del ADN/genética , Predisposición Genética a la Enfermedad , Células Germinativas , Mutación de Línea Germinal/genética , Humanos , Sarcoma/genética , Sarcoma de Ewing/genéticaRESUMEN
OBJECTIVE: The objective of this study was to assess the clinical impact and outcome of the SARS-CoV-2 infection on children with cancer or those who received a hematopoietic stem cell transplantation. METHODS: AIEOP (Italian Association of Pediatric Hematology and Oncology) performed a nationwide multicenter observational cohort study, including consecutive patients between April 2020 and November 2022. RESULTS: Twenty-five Italian centers participated and 455 patients were enrolled. We reported a significant increasing trend of symptomatic cases over the years, while the number of nonmild infections remained stable. Early infection after oncologic diagnosis (<60 days) and severe neutropenia were identified as independent risk factors for developing moderate, severe, or critical infections. The percentage of patients who were asymptomatic and mildly symptomatic and who stopped chemotherapy reduced over the years of the pandemic. Nine patients died, but no death was attributed to SARS-CoV-2 infection. CONCLUSIONS: SARS-CoV-2 infection presented a self-limiting benign course in the Italian pediatric oncohematology population during the pandemic, and its main consequence has been the discontinuation of cancer-directed therapies. The rate of patients who were asymptomatic and stopped chemotherapy reduced over the years, suggesting that the continuation of chemotherapy is a feasible option.
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COVID-19 , Enfermedades Transmisibles , Trasplante de Células Madre Hematopoyéticas , Neoplasias , Niño , Humanos , SARS-CoV-2 , Neoplasias/complicaciones , Neoplasias/terapia , Neoplasias/epidemiología , Trasplante de Células Madre Hematopoyéticas/efectos adversosRESUMEN
International comparisons of cancer surveillance measures may provide insight into inequalities in registration practices, etiological factors, and treatment strategies. This study aimed to compare incidence, survival, and mortality of cancer in children and young adolescents between Belgium and the Netherlands. All children (0-14 years) and young adolescents (15-17 years) diagnosed with cancer between 2004 and 2015 were selected from the population-based cancer registries of Belgium (N = 4739) and the Netherlands (N = 7322). Differences in incidence and mortality were expressed as standardized rate ratios (SRR; BE/NL). Five-year observed survival was calculated using the Kaplan-Meier method. During 2004-2015, the overall cancer incidence among children and young adolescents was similar in both countries. Incidence of neuroblastoma was significantly higher in Belgian children (2010-2015: SRR = 1.3, 95% CI 1.0-1.6). Five-year survival of all malignant cancers was comparable in 2010-2015, exceeding 80% in both age groups. Remarkable differences in survival existed in children for malignant central nervous system (CNS) tumors in 2004-2009 (BE = 62%, NL = 45%), for acute myeloid leukemia (BE = 68%, NL = 78%) and rhabdomyosarcomas (BE = 60%, NL = 79%) in 2010-2015, and for neuroblastoma in both periods (2004-2009: BE = 76%, NL = 64%; 2010-2015: BE = 82%, NL = 64%). Overall cancer mortality in children decreased by approximately 3 percent-points annually in both countries, but was slightly lower in Belgium in 2004-2009 (SRR = 0.9, 95% CI 0.7-1.0). Despite differences for specific cancer types, overall cancer incidence, survival, and mortality were comparable between Dutch and Belgian children and young adolescents in 2010-2015. Variability in screening, diagnosis, and registration practices probably explains the observed differences in incidence and survival of neuroblastoma and malignant CNS tumors.
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Neoplasias , Sistema de Registros , Humanos , Bélgica/epidemiología , Adolescente , Países Bajos/epidemiología , Niño , Preescolar , Lactante , Masculino , Incidencia , Femenino , Recién Nacido , Neoplasias/epidemiología , Neoplasias/mortalidad , Tasa de Supervivencia , Neuroblastoma/epidemiología , Neuroblastoma/mortalidadRESUMEN
Cancer is the leading cause of death in American children older than 1 year of age. Major developments in drugs such as thiopurines and optimization in clinical trial protocols for treating cancer in children have led to a remarkable improvement in survival, from approximately 30% in the 1960s to more than 80% today. Short-term and long-term adverse effects of chemotherapy still affect most survivors of childhood cancer. Pharmacogenetics plays a major role in predicting the safety of cancer chemotherapy and, in the future, its effectiveness. Treatment failure in childhood cancer-due to either serious adverse effects that limit therapy or the failure of conventional dosing to induce remission-warrants development of new strategies for treatment. Here, we summarize the current knowledge of the pharmacogenomics of cancer drug treatment in children and of statistically and clinically relevant drug-gene associations and the mechanistic understandings that underscore their therapeutic value in the treatment of childhood cancer.
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Antineoplásicos , Efectos Colaterales y Reacciones Adversas Relacionados con Medicamentos , Neoplasias , Antineoplásicos/uso terapéutico , Niño , Preescolar , Humanos , Neoplasias/tratamiento farmacológico , FarmacogenéticaRESUMEN
BACKGROUND: Siblings of children with cancer may experience adverse household economic consequences, but their financial outcomes in adulthood are unknown. METHODS: A total of 880 siblings (aged 18-64 years) of adult-aged childhood cancer survivors were surveyed to estimate the prevalence of financial hardship by three established domains (behavioral, material, and psychological). For individual financial hardship items matching the contemporaneous National Health Interview Survey or Behavioral Risk Factor Surveillance System, siblings were compared with the general population by calculating adjusted prevalence odds ratios (ORs) to sample-weighted responses. Multivariable logistic regression models examined associations between sibling characteristics and each hardship domain and between sibling hardship and survivors' cancer/treatment characteristics. RESULTS: Behavioral, material, and psychological hardship was reported by 24%, 35%, and 28%, respectively. Compared with national survey respondents, siblings were more likely to report worries about medical bills (OR, 1.14; 95% confidence interval [CI], 1.06-1.22), difficulty affording nutritious foods (OR, 1.79; 95% CI, 1.54-2.07), and forgoing needed medical care (OR, 1.38; 95% CI, 1.10-1.73), prescription medications (OR, 2.52; 95% CI, 1.99-3.20), and dental care (OR, 1.34; 95% CI, 1.15-1.57) because of cost. Sibling characteristics associated with reporting financial hardship in one or more domains included female sex, older age, chronic health conditions, lower income, not having health insurance, high out-of-pocket medical expenditures, and nonmedical/nonhome debt. No survivor cancer/treatment characteristics were associated with sibling financial hardship. CONCLUSIONS: Adult siblings of childhood cancer survivors were more likely to experience financial hardship compared with the general population. Childhood cancer may adversely affect entire households, with potentially lasting implications.
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Supervivientes de Cáncer , Neoplasias , Adulto , Humanos , Niño , Femenino , Hermanos , Neoplasias/epidemiología , Neoplasias/terapia , Estrés Financiero/epidemiología , Costo de Enfermedad , Sobrevivientes , Encuestas y CuestionariosRESUMEN
PURPOSE: Germline testing in pediatric cancer presents opportunities and challenges. Understanding family perspectives, experiences, and preferences will optimize integration into routine care. METHODS: Following Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines, we searched 4 databases for studies exploring perspectives, experiences, and preferences of parents/caregivers and/or patients regarding germline testing of children with cancer. Qualitative and quantitative data were extracted, organized, and summarized by research question and themes. RESULTS: We identified 2286 unique articles, of which 24 were included. Interest in and uptake of testing was high. Families were motivated by altruism and a desire for inheritance/causation information. Testing barriers included psychological concerns, timing of the testing approach if offered at diagnosis or in a high-risk cancer setting and privacy/discrimination. Testing experiences highlighted challenges yet also positive impacts, with results providing psychological relief and informing proactive decision making. Timing preferences varied; however, allowing time to adjust to a new diagnosis was a common theme. Most wanted to receive as many germline sequencing-related results as possible. CONCLUSION: Findings underscore the importance of integrating germline analyses into pediatric cancer care with flexibility and support for families facing challenges. Where possible, consent should be provided at a time that suits each family's situation with access to information aligning with their needs and preferences. PROSPERO: CRD42023444890.
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OBJECTIVE: To determine which groups of children with cancer for whom to apply the newly developed quality measures (QMs) for end-of-life (EOL) care. STUDY DESIGN: In a series of nominal groups, panelists answered the question: "Which children, diagnoses, conditions, or prognoses should be included when examining the quality of EOL care for children with cancer?" In each group, individual panelists proposed answers to the question. After collating individual responses, each panelist ranked their 5 top answers and points were assigned (5 pts for the best answer, 4 pts the second best, etc.). A team of pediatric oncology and palliative care clinician-scientists developed and applied a coding structure for responses and associated themes and subthemes for responses. RESULTS: We conducted 5 nominal groups with a total of 44 participants. Most participants identified as female (88%) and non-Hispanic White (86%). Seventy-nine percent were clinicians, mainly in pediatric palliative care, pediatric oncology, or hospice; 40% were researchers and 12% were bereaved parents. Responses fell into 5 themes: (1) poor prognosis cancer; (2) specific treatment scenarios; (3) certain populations; (4) certain symptoms; and (5) specific utilization scenarios. Poor prognosis cancer and specific treatment scenarios received the most points (320 pts [49%] and 147 pts [23%], respectively). CONCLUSIONS: Participants developed a framework to identify which children should be included in EOL QMs for children with cancer. The deliberate identification of the denominator for pediatric QMs serves as a potent tool for enhancing quality, conducting research, and developing clinical programs.
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Neoplasias , Cuidados Paliativos , Cuidado Terminal , Humanos , Neoplasias/terapia , Cuidado Terminal/normas , Femenino , Masculino , Niño , Cuidados Paliativos/normas , Calidad de la Atención de SaludRESUMEN
BACKGROUND AND AIMS: High-dose chemotherapy (HDC) followed by autologous stem cell transplantation (ASCT) improves the prognosis in pediatric patients with several solid tumors and lymphomas. Little is known about the reconstitution of the immune system after ASCT and the influence of CD34+ cell selection on the reconstitution in pediatric patients. METHODS: Between 1990 and 2001, 94 pediatric patients with solid tumors and lymphomas received autologous CD34+ selected or unmanipulated peripheral stem cells after HDC. CD34+ selection was carried out with magnetic microbeads. The absolute numbers of T cells, B cells and natural killer (NK) cells were measured and compared in both groups at various time points post-transplant. RESULTS: Recovery of T cells was significantly faster in the unmanipulated group at day 30, with no significant difference later on. Reconstitution of B and NK cells was similar in both groups without significant differences at any time. The CD34+-selected group was divided into patients receiving less or more than 5.385 × 106/kg CD34+ cells. Patients in the CD34+ high-dose group displayed significantly faster reconstitutions of neutrophiles and lymphocyte subsets than the CD34+ low-dose group. CONCLUSIONS: Engraftment and reconstitution of leukocytes, B cells and NK cells after transplantation of CD34+ selected stem cells were comparable to that in patients receiving unmanipulated grafts. T-cell recovery was faster in the unmanipulated group only within the first month. However, this delay could be compensated by transplantation of >5.385 × 106 CD34+ cells/kg. Especially for patients receiving immunotherapy after HDC large numbers of immune effector cells such as NK and T cells are necessary to mediate antibody-dependent cellular cytotoxicity. Therefore, in patients receiving autologous CD34+-selected grafts, our data emphasize the need to administer high stem cell counts.
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INTRODUCTION: The SARS-CoV-2 (COVID-19) pandemic has strained healthcare systems and presented unique challenges for children requiring cancer care, particularly in low- and middle-income countries. This study aimed to assess the impact of the COVID-19 pandemic on access to cancer care for children and adolescents in Northern Tanzania. METHODS: In this cross-sectional study, we assessed the demographic and clinical characteristics of 547 pediatric and adolescent cancer patients (ages 0-19 years old) between 2016 and 2022 using the population-based Kilimanjaro Cancer Registry (KCR). We categorized data into pre-COVID-19 (2016-2019) and COVID-19 (2020-2022) eras, and performed descriptive analyses of diagnostic, treatment, and demographic information. A secondary analysis was conducted on a subset of 167 patients with stage of diagnosis at presentation. RESULTS: Overall admissions nearly doubled during the pandemic (n = 190 versus 357). The variety of diagnoses attended at KCMC increased during the pandemic, with only five groups of diseases reported in 2016 to twelve groups of diseases in 2021. Most patients were diagnosed at a late stage (stage III or IV) across eras, with the proportion of under-five years old patients increasing late-diagnoses from 29.4% (before the pandemic), 52.8% (during the pandemic), when compared to the overall cohort. Around 95% of children in this age category reported late-stage diagnosis during the pandemic. Six out of the twelve cancer site groups also reported an increase in late-stage diagnosis. During the pandemic, the proportion of children receiving surgery increased from 15.8 to 30.8% (p < 0.001). CONCLUSION: Childhood and adolescent cancer care changed in Northern Tanzania during the COVID-19 pandemic, with increased late-stage diagnoses presentations among younger patients and the increased use of surgical therapies in the context of a growing practice. Understanding the impact of the COVID-19 pandemic on pediatric and adolescent cancer care can help us better adapt healthcare systems and interventions to the emerging needs of children and adolescents with cancer in the midst of a health crisis.
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COVID-19 , Neoplasias , Adolescente , Humanos , Niño , Recién Nacido , Lactante , Preescolar , Adulto Joven , Adulto , COVID-19/epidemiología , Estudios Transversales , Pandemias , SARS-CoV-2 , Tanzanía/epidemiología , Neoplasias/diagnóstico , Neoplasias/epidemiología , Neoplasias/terapiaRESUMEN
BACKGROUND: Invasive fungal disease (IFD) is typically aggressive and related with high mortality in children with a hematological malignancy. The association of medical and surgical treatment may ameliorate the outcome. The aim of this study was to analyze the surgical treatment of fungal infections in pediatric oncological populations. METHODS: Retrospective study (2000-2022) of a single-center experience. We reviewed the medical record of all patients with hematologic malignancies and IFD, analyzing the outcome. RESULTS: From the 70 pediatric cases of hematologic malignancies with the diagnosis IFD over 22 years, we included in the present study 44 cases who required surgical approaches for either diagnosis or treatment. Twenty-one patients were males and the mean age was 11 (range 1-23) years. The main indications for surgery were lack of improvement following medical treatment and/or progression of fungal infection (80%) and diagnosis confirmation (20%). Only five patients needed an emergency operation for rapid worsening of symptoms. The most common site of infection was the lung (80%) and the most frequently identified pathogen was Aspergillus (75%). The most common surgical procedures were lobectomy (performed in 17 patients) and atypical lung resection (10). Complications of surgery were mostly treated by medical approach. The mean time of resumption of oncological treatment was 40 (range 0-150) days. CONCLUSIONS: Surgery is an important step in the multimodal treatment of invasive fungal infection with excellent resolution rate. Overall mortality depends on the underlying malignancy.
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Neoplasias Hematológicas , Infecciones Fúngicas Invasoras , Humanos , Niño , Adolescente , Masculino , Femenino , Infecciones Fúngicas Invasoras/diagnóstico , Infecciones Fúngicas Invasoras/cirugía , Infecciones Fúngicas Invasoras/mortalidad , Infecciones Fúngicas Invasoras/etiología , Infecciones Fúngicas Invasoras/tratamiento farmacológico , Preescolar , Estudios Retrospectivos , Lactante , Adulto Joven , Neoplasias Hematológicas/complicaciones , Neoplasias Hematológicas/terapia , Resultado del Tratamiento , Manejo de la EnfermedadRESUMEN
BACKGROUND: The aim of this study was to investigate the applicability of the central line-associated bloodstream infection (CLABSI) criteria of the Centers for Disease Control and Prevention in pediatric oncology patients. METHODS: Bacteremia episodes from 2020 to 2022 from a prospective cohort of pediatric oncology patients with a central venous catheter were included. Episodes were classified by three medical experts following the CLABSI criteria as either a CLABSI or non-CLABSI (i.e., contamination, other infection source, or mucosal barrier injury-laboratory confirmed bloodstream infection (MBI-LCBI)). Subsequently, they were asked if and why they (dis)agreed with this diagnosis following the criteria. The primary outcome was the percentage of episodes where the experts clinically disagreed with the diagnosis given following the CLABSI criteria. RESULTS: Overall, 84 bacteremia episodes in 71 patients were evaluated. Following the CLABSI criteria, 34 (40%) episodes were classified as CLABSIs and 50 (60%) as non-CLABSIs. In 11 (13%) cases the experts clinically disagreed with the diagnosis following the CLABSI criteria. The discrepancy between the CLABSI criteria and clinical diagnosis was significant; McNemar's test p < .01. Disagreement by the experts with the CLABSI criteria mostly occurred when the experts found an MBI-LCBI a more plausible cause of the bacteremia than a CLABSI due to the presence of a gram negative bacteremia (Pseudomonas aeruginosa n = 3) and/or mucositis. CONCLUSIONS: A discrepancy between the CLABSI criteria and the evaluation of the experts was observed. Adding Pseudomonas aeruginosa as an MBI pathogen and incorporating the presence of mucositis in the MBI-LCBI criteria, might increase the applicability.
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Bacteriemia , Infecciones Relacionadas con Catéteres , Cateterismo Venoso Central , Mucositis , Neoplasias , Sepsis , Niño , Humanos , Infecciones Relacionadas con Catéteres/diagnóstico , Infecciones Relacionadas con Catéteres/etiología , Estudios Prospectivos , Bacteriemia/diagnóstico , Bacteriemia/etiología , Neoplasias/complicaciones , Neoplasias/diagnóstico , Estudios RetrospectivosRESUMEN
OBJECTIVE: Parents of children with cancer are at risk for developing psychosocial problems. The present study aims to evaluate the effect of an online group intervention (Op Koers Online, in English: On Track Online) on psychosocial wellbeing and coping skills. METHODS: Parents of a child with cancer (diagnosis <5 years ago) participated in a randomized controlled trial. In six consecutive (and one booster-) protocolled sessions in an online chatroom, trained psychologists and social workers taught coping skills using cognitive behavioral and acceptance and commitment techniques. Questionnaires assessed anxiety, depression, distress, situation-specific emotional reactions and coping skills (Op Koers Questionnaire/Cognitive Coping Strategies Scale Parent Form) and evaluated the intervention. Linear mixed-model analyses were performed to detect differences between the conditions in changes over time; T0-T1 and T0-T2 (6-week and 6-month follow-up), and to detect changes in scores T2-T3 (12-month follow-up) for the intervention group only. RESULTS: 89 parents were included in analyses (mean age 41.9 years, 86% female, 62%/38% post/during treatment of their child). Beneficial intervention effects (p < 0.05) were found at T1 for anxiety, depression, distress, loneliness and relaxation, and at T2 for anxiety, uncertainty and relaxation. In the intervention condition, scores did not change from T2 to T3, except loneliness that decreased and relaxation that improved. All effect sizes were small to medium (ß = -0.21 to 0.46). Parents were generally positive about the intervention. CONCLUSIONS: Op Koers Online for parents of children with cancer has a positive effect on psychosocial wellbeing and the coping skill relaxation. Implementation is recommended to prevent psychosocial problems. CLINICAL TRIAL REGISTRATION: Dutch Trial Register https://onderzoekmetmensen.nl/en NL73763.041.20.
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Neoplasias , Padres , Niño , Humanos , Femenino , Adulto , Masculino , Padres/psicología , Estrés Psicológico/terapia , Estrés Psicológico/psicología , Ansiedad/terapia , Habilidades de Afrontamiento , Neoplasias/terapia , Neoplasias/psicologíaRESUMEN
OBJECTIVE: End of pediatric cancer treatment requires family adjustment. Caregivers who struggle to incorporate the child's condition into family life have poorer family outcomes. To better understand factors that contribute to successful transition off active childhood cancer treatment, we sought to examine caregiver perceived management ability of the youth's condition and family functioning as predictors of caregiver distress, evaluate family functioning as a mediator between perceived ability and distress, and explore race and ethnicity as a moderator between perceived ability and family functioning. METHODS: Caregivers (N = 141) completed measures assessing family management (condition management ability; CMA), family functioning, and distress as part of a clinical education and screening program within 1 year of the end of treatment. Bias-corrected bootstrap regression analyses examined mediation and moderated mediation models with patient race and ethnicity as the moderator. RESULTS: The overall mediation model was statistically significant for CMAâfamily functioningâdistress. Race and ethnicity moderated the relationship between CMA and family functioning, but the full model was not significant. CMA was related to family functioning for caregivers of non-Hispanic white youth, but not caregivers of Hispanic youth. Family functioning was related to distress for all caregivers. CONCLUSIONS: Family functioning serves as an initial intervention target to reduce caregiver distress. Caregiver perceived management ability of their child's condition is a meaningful predictor of family functioning and distress for caregivers of non-Hispanic white youth, yet CMA may be limited as a screener of family management patterns for diverse populations, and other family management dimension may be more applicable.
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Cuidadores , Neoplasias , Adolescente , Humanos , Niño , Etnicidad , Neoplasias/terapia , Relaciones Familiares , Análisis de RegresiónRESUMEN
OBJECTIVE: Cancer predisposition syndromes are being more frequently recognized in the etiology of pediatric oncology and genetic-related technologies are evolving rapidly, leading to an increasing availability of genetic testing for families. This systematic review assessed the psychological impact of genetic testing on children and parents in the context of childhood cancer. METHODS: Searches were performed using three databases (Web of Science, Pubmed and Embase) to identify relevant empirical studies. Following Cochrane guidelines, we screened 3838 articles and identified 18 eligible studies, representing the perspectives of children and/or parents. RESULTS: The included studies described the impact of genetic testing in different contexts (e.g. predictive testing and diagnostic testing) and in different subgroups, (e.g. carriers and non-carriers). Overall, the studies did not identify clinically-relevant long-term increases in negative emotions (depression, anxiety, distress, uncertainty, guilt) as a result of genetic testing. Negative emotions were typically time-limited and generally occurred in families with particular characteristics (e.g. those with a history of multiple cancer diagnoses, families receiving an unfavorable result for one child and a favorable result in siblings, and those with pre-existing mental health difficulties). Positive emotions (hopefulness, relief and peace of mind) were also reported. Knowing their genetic risk status appeared to help to foster empowerment among families, regardless of the result and any associated emotions. CONCLUSIONS: Genetic testing in pediatric oncology does not appear to cause significant additional harm and can lead to positive outcomes. Clinicians need to be especially attentive when counseling families at increased risk of distress.
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Pruebas Genéticas , Neoplasias , Niño , Humanos , Neoplasias/psicología , Emociones , Ansiedad , Oncología MédicaRESUMEN
OBJECTIVES: Childhood cancer survivors' social reintegration may be hampered in low and middle-income countries. The nature and extent of social challenges and prejudices that survivors encounter in such settings are largely unknown. This study explores caregivers' perspectives on social reintegration and stigmatization of Kenyan childhood cancer survivors. METHODS: Caretakers of childhood cancer survivors (<18 years) were interviewed using mixed-methods questionnaires during home or clinic visits between 2021 and 2022. Stigma was assessed with an adjusted Social Impact Scale and risk factors were investigated. RESULTS: Caretakers of 54 survivors (median age 11 years) were interviewed. Families' income (93%) decreased since start of treatment. Caretakers (44%) often lost their jobs. Financial struggles (88%) were a burden that provoked conflicts within communities (31%). School fees for siblings became unaffordable (52%). Families received negative responses (26%) and were left or avoided (13%) by community members after cancer disclosure. Survivors and families were discriminated against because the child was perceived fragile, and cancer was considered fatal, contagious, or witchcraft. Survivors repeated school levels (58%) and were excluded from school activities (19%) or bullied (13%). Performance limitations of daily activities (p = 0.019), male sex (p = 0.032), solid tumors (p = 0.056) and a short time since treatment completion (p = 0.047) were associated with increased stigma. Caretakers recommended educational programs in schools and communities to raise awareness about cancer treatment and curability. CONCLUSIONS: Childhood cancer survivors and their families experienced difficulties with re-entry and stigmatization in society. Increasing cancer and survivorship awareness in schools and communities should facilitate social reintegration and prevent stigmatization.
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Supervivientes de Cáncer , Cuidadores , Estigma Social , Humanos , Supervivientes de Cáncer/psicología , Supervivientes de Cáncer/estadística & datos numéricos , Kenia , Masculino , Femenino , Niño , Cuidadores/psicología , Adolescente , Encuestas y Cuestionarios , Adulto , Neoplasias/psicología , Preescolar , Persona de Mediana EdadRESUMEN
BACKGROUND: Effective communication is founded on bidirectional participation from families and healthcare providers. In adult medicine, bidirectional communication promotes treatment adherence and builds the family-provider relationship. However, the relationship between communication styles in pediatrics remains poorly understood, particularly in culturally diverse settings. This study aims to investigate parent-provider communication dynamics and parental involvement during diagnostic cancer communication in Guatemala. PROCEDURE: This qualitative study included 20 families of children with cancer and 10 providers at Unidad Nacional de Oncología Pediátrica in Guatemala. Psychoeducation and diagnostic conversations between parents, psychologists, and oncologists were recorded and thematically analyzed using a priori and novel codes exploring communication behaviors, parental engagement, and interpersonal dynamics. RESULTS: Participating parents had children with various diagnoses. Only 15% of fathers and 5% of mothers reported education beyond primary school. Providers spoke 68% of words during psychoeducation and 85% of words during diagnosis conversations. Providers used supportive communication behaviors providing explanations, demonstrating verbal attentiveness, and soliciting questions and non-supportive behaviors including paternalistic talk. Parental participation was considered active when they asked questions, expressed hopes or concerns, or asserted their opinions, and non-active when participation was limited to brief responses to closed-ended questions. Supportive provider communication often encouraged active participation; non-supportive communication did not. Furthermore, active parental participation prompted supportive communication from providers, while non-active participation did not. CONCLUSIONS: Our findings highlight the bidirectional nature of effective communication, establishing that provider communication styles both influence and are influenced by parental participation, and emphasizing the importance of supportive provider communication for patient-centered care.
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Comunicación , Neoplasias , Padres , Relaciones Profesional-Familia , Investigación Cualitativa , Humanos , Masculino , Femenino , Niño , Neoplasias/psicología , Neoplasias/diagnóstico , Neoplasias/terapia , Guatemala , Adulto , Padres/psicología , Adolescente , Preescolar , Lactante , Oncología Médica , Personal de Salud/psicologíaRESUMEN
Inconsistencies in the definition of clinically unsuspected venous thromboembolism (VTE) in pediatric patients recently led to the recommendation of standardizing this terminology. Clinically unsuspected VTE (cuVTE) is defined as the presence of VTE on diagnostic imaging performed for indications unrelated to VTE in a patient without symptoms or clinical history of VTE. The prevalence of cuVTE in pediatric cancer patients is unclear. Therefore, the main objective of our study was to determine the prevalence of cuVTE in pediatric cancer patients. All patients 0-18 years old, treated at the IWK in Halifax, Nova Scotia, from August 2005 through December 2019 with a known cancer diagnosis and at least one imaging study were eligible (n = 743). All radiology reports available for these patients were reviewed (n = 18,120). The VTE event was labeled a priori as cuVTE event for radiology reports that included descriptive texts indicating a diagnosis of thrombosis including thrombus, central venous catheter-related, thrombosed aneurysm, tumor thrombosis, non-occlusive thrombus, intraluminal filling defect, or small fragment clot for patients without documentation of clinical history and or signs of VTE. A total of 18,120 radiology reports were included in the review. The prevalence of cuVTE was 5.5% (41/743). Echocardiography and computed tomography had the highest rate of cuVTE detection, and the most common terminologies used to diagnose cuVTE were thrombus and non-occlusive thrombus. The diagnosis of cuVTE was not associated with age, sex, and type of cancer. Future efforts should focus on streamlining radiology reports to characterize thrombi. The clinical significance of these cuVTE findings and their application to management, post-thrombotic syndrome, and survival compared to cases with symptomatic VTE and patients without VTE should be further studied.
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Neoplasias , Tromboembolia Venosa , Humanos , Tromboembolia Venosa/epidemiología , Tromboembolia Venosa/etiología , Niño , Neoplasias/complicaciones , Neoplasias/epidemiología , Femenino , Masculino , Preescolar , Adolescente , Lactante , Recién Nacido , Prevalencia , Estudios Retrospectivos , Estudios de Seguimiento , Canadá/epidemiología , Pronóstico , Nueva Escocia/epidemiologíaRESUMEN
Researchers use patient-reported outcomes (PRO) to capture the patient experience, and in pediatrics, parent report of symptom and function often accompanies the child's assessment. Children with cancer and their family caregivers frequently evaluate symptom burden and functional status differently. Child-caregiver PRO disagreement often worries pediatric oncology clinicians, who do not know how to best approach sharing it with families. Openly discussing differences in symptom and functional status reporting is a crucial element of supporting families and can improve family harmony throughout the child's illness journey. This paper offers strategies and language for communicating with families about PRO disagreement.
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Cuidadores , Neoplasias , Niño , Humanos , Medición de Resultados Informados por el PacienteRESUMEN
Wilms tumor is the most common pediatric renal cancer, and lungs represent the major site of metastasis and recurrence. Relapse occurs in 15%, months or years after treatment; so due to the small sample, acquiring more data about the pattern of lung relapse remains a challenge. The aim of our study was to evaluate if pulmonary relapse, detected by computed tomography (CT), occurred at the initial site of lung metastases or in a different location. According to our data, the CT pattern of lung relapse showed high probability of recurrence at the same site of initial metastasis.
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Neoplasias Renales , Neoplasias Pulmonares , Tumor de Wilms , Niño , Humanos , Recurrencia Local de Neoplasia , Tumor de Wilms/patología , Neoplasias Renales/patología , Neoplasias Pulmonares/secundario , Pulmón/patologíaRESUMEN
Eligible pediatric hospitals can purchase clinician-administered drugs at discounted rates through the 340B Drug Pricing Program and charge payers prices exceeding drug acquisition costs, but the magnitude of these markups is not known. In a study of newly approved oncology drugs at pediatric 340B hospitals, median negotiated prices ranged from 102% (interquartile range [IQR]: 91%-156%) of average sales price (ASP) at Phoenix Children's Hospital to 630% (IQR: 526%-630%) at Driscoll Children's Hospital. Pediatric hospitals participating in the federal 340B Drug Pricing Program can extract steep payments on new drugs from commercial insurers, though with wide variation between and within hospitals.