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BACKGROUND: Osteonecrosis (ON) is a potentially disabling skeletal complication of cancer treatment. Although symptomatic osteonecrosis (sON) is well-known in acute lymphoblastic leukemia (ALL), with an incidence around 6%, studies on sON in pediatric Hodgkin lymphoma (HL) are scarce. The aim of this study was to examine the incidence, risk factors, and outcome of sON in children treated for HL. PROCEDURE: A total of 490 children under 18, diagnosed with HL between 2005 and 2019 in Sweden, Finland, and Denmark were eligible for the study. Data on patient characteristics, HL treatment, and development of sON were collected from patients' medical records. Magnetic resonance imaging scans were used to establish ON diagnosis and grade ON according to the Niinimäki grading system. RESULTS: Cumulative 2-year incidence of sON among the 489 included patients was 5.5% (n = 30). The risk for developing sON was higher for those with older age (odds ratio [OR] 1.25, 95% confidence interval [CI]: 1.05-1.49, p < .010), female sex (OR 4.45, CI 1.87-10.58, p < .001), high total cumulative glucocorticoid (GC) doses (OR 1.76, 95% CI: 1.21-2.56, p = 0.003), and advanced HL (OR 2.19, 95% CI: 1.03-4.65, p = .042). Four (13.3%) patients underwent major surgical procedures and 13 (43.3%) had persistent symptoms due to ON at follow-up. CONCLUSIONS: This study shows that sON is as common in pediatric HL as in pediatric ALL, with risk factors such as older age, female sex, high cumulative GC doses, and advanced HL. Future HL protocol development should aim to reduce the burden of ON by modifying GC treatment.
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OBJECTIVE: Central venous saturation (ScvO2) can guide resuscitation of children with septic shock. The normal range of ScvO2 is typically considered as 0.70-0.80, but has not been established in children with cancer. Children with cancer are particularly prone to develop sepsis due to their immunosuppressive therapy, and usually have a permanent central venous catheter, making ScvO2 readily available. We aimed to investigate normal values of ScvO2 in clinically stable children with cancer, and the association between ScvO2, hemoglobin, and lactate. METHODS: We conducted a prospective clinical study at the outpatient clinic of a tertiary pediatric hematology/oncology unit. Blood samples were collected from stable children aged 0-17.9 years who were treated for cancer between January 1 and November 30, 2019, during their routine outpatient clinic visits. RESULTS: A total of 183 blood samples were collected from 68 patients (24 girls and 44 boys). The predicted mean level of ScvO2 with a 95% confidence interval was 0.67 (0.56-0.78). The ScvO2 value was below the expected lower normal limit of 0.70 in 126 (69%) of the samples and in 48 patients (71%) at least once. ScvO2 was significantly associated with hemoglobin (ß1 = 0.012 per g/L hemoglobin, P < 0.001), but not with age, sex, underlying diagnosis, or lactate. CONCLUSIONS: The study revealed that a substantial portion of clinically stable childhood cancer patients exhibited ScvO2 levels below the typical reference value of 0.70, suggesting that these children may have inherently lower baseline ScvO2 levels. This should be kept in mind when evaluating children with cancer for septic shock, emphasizing the importance of tailored assessments in this population. Further understanding of baseline ScvO2 abnormalities may be helpful if ScvO2 is used to guide resuscitation.
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Osteonecrosis (ON) is a common complication of glucocorticoid-based Hodgkin lymphoma (HL) treatment, but the natural evolution and prognosis of ON lesions remain poorly understood. We describe the radiological evolution of ON lesions identified in a Nordic population-based cohort of paediatric HL patients. Magnetic resonance images of suspected ON lesions were centrally reviewed to confirm ON diagnosis and grade the ON lesions according to the Niinimäki classification. The study included 202 ON lesions in 46 patients, of which 77 were joint lesions. Follow-up images were available for 146/202 lesions, with a mean follow-up time of 28 months. During follow-up, 71% of the lesions remained stable, 26% improved or resolved, and 3% progressed. A higher ON grade at diagnosis was associated with a lower likelihood of spontaneous resolution. The likelihood for resolution of ON decreased by 50% for each year of added patient age, when adjusted for sex, ON location, and symptoms. Hip ON showed less spontaneous improvement compared with other joints, and the risk for surgery was 13-fold in hip ON. Grades 3-4 joint ON has the potential to either progress or resolve, warranting follow-up in patients with severe symptoms. Research on secondary prevention should be directed at grade 3-4 joint ON.
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The Frontline and Relapsed Rhabdomyosarcoma (FaR-RMS) clinical trial is an overarching, multinational study for children and adults with rhabdomyosarcoma (RMS). The trial, developed by the European Soft Tissue Sarcoma Study Group (EpSSG), incorporates multiple different research questions within a multistage design with a focus on (i) novel regimens for poor prognostic subgroups, (ii) optimal duration of maintenance chemotherapy, and (iii) optimal use of radiotherapy for local control and widespread metastatic disease. Additional sub-studies focusing on biological risk stratification, use of imaging modalities, including [18F]FDG PET-CT and diffusion-weighted MRI imaging (DWI) as prognostic markers, and impact of therapy on quality of life are described. This paper forms part of a Special Issue on rhabdomyosarcoma and outlines the study background, rationale for randomisations and sub-studies, design, and plans for utilisation and dissemination of results.
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BACKGROUND: Implementation of daily cone-beam CT (CBCT) into clinical practice in paediatric image-guided radiotherapy (IGRT) lags behind compared to adults. Surveys report wide variation in practice for paediatric IGRT and technical information remains unreported. In this study we report on technical settings from applied paediatric CBCT protocols and review the literature for paediatric CBCT protocols. METHODS: From September to October 2022, a survey was conducted among 246 SIOPE-affiliated centres across 35 countries. The survey consisted of 3 parts: 1) baseline information; technical CBCT exposure settings and patient set-up procedure for 2) brain/head, and 3) abdomen. Descriptive statistics was used to summarise current practice. The literature was reviewed systematically with two reviewers obtaining consensus RESULTS: The literature search revealed 22 papers concerning paediatric CBCT protocols. Seven papers focused on dose-optimisation. Responses from 50/246 centres in 25/35 countries were collected: 44/50 treated with photons and 10/50 with protons. In total, 48 brain/head and 53 abdominal protocols were reported. 42/50 centres used kV-CBCT for brain/head and 35/50 for abdomen; daily CBCT was used for brain/head = 28/48 (58%) and abdomen = 33/53 62%. Greater consistency was seen in brain/head protocols (dose range 0.32 - 67.7 mGy) compared to abdominal (dose range 0.27 - 119.7 mGy). CONCLUSION: Although daily CBCT is now widely used in paediatric IGRT, our survey demonstrates a wide range of technical settings, suggesting an unmet need to optimise paediatric IGRT protocols. This is in accordance with the literature. However, there are only few paediatric optimisation studies suggesting that dose reduction is possible while maintaining image quality.
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Radioterapia Guiada por Imagem , Tomografia Computadorizada de Feixe Cônico Espiral , Adulto , Humanos , Criança , Radioterapia Guiada por Imagem/métodos , Planejamento da Radioterapia Assistida por Computador/métodos , Abdome , Tomografia Computadorizada de Feixe Cônico/métodos , Europa (Continente) , Imagens de Fantasmas , Dosagem Radioterapêutica , Literatura de Revisão como AssuntoRESUMO
Maintenance chemotherapy (MC) defines the administration of prolonged relatively low-intensity chemotherapy with the aim of "maintaining" tumor complete remission. This paper aims to report an update of the RMS2005 trial, which demonstrated better survival for patients with high-risk localized rhabdomyosarcoma (RMS) when MC with vinorelbine and low-dose cyclophosphamide was added to standard chemotherapy, and to discuss the published experience on MC in RMS. In the RMS2005 study, the outcome for patients receiving MC vs. those who stopped the treatment remains superior, with a 5-year disease-free survival of 78.1% vs. 70.1% (p = 0.056) and overall survival of 85.0% vs. 72.4% (p = 0.008), respectively. We found seven papers describing MC in RMS, but only one randomized trial that did not demonstrate any advantage when MC with eight courses of trofosfamide/idarubicine alternating with trofosfamide/etoposide has been employed in high-risk RMS. The use of MC showed better results in comparison to high-dose chemotherapy in non-randomized studies, including metastatic patients, and demonstrated feasibility and tolerability in relapsed RMS. Many aspects of MC in RMS need to be investigated, including the best drug combination and the optimal duration. The ongoing EpSSG trial will try to answer some of these questions.
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BACKGROUND: Families of children and adolescents with cancer strive to maintain routines and normalcy during the child's treatment trajectory that requires frequent hospital visits. Intravenous chemotherapy at home can reduce time spent on the frequent hospital visits and mitigate disruption in daily life. Studies on home chemotherapy for children and adolescents with cancer are limited, as is knowledge of family and health care professionals' needs, and knowledge required to inform adaptation or replication of interventions in other settings. The aim of this study was to develop and describe an evidence-based home chemotherapy intervention that is feasible and safe for children and adolescents and suitable for future feasibility testing. METHODS: The Medical Research Council's guidance for developing complex interventions in health care and the framework of action developed by O'Cathain et al. was used as theoretical frameworks to structure the development process. A literature search, an ethnographic study, and interviews with clinical nurse specialists from adult cancer departments formed the evidence base. Educational learning theory to support and understand the intervention was identified. Stakeholder perspectives were explored in workshops with health care professionals and parent-adolescent interviews. Reporting was qualified using the GUIDED checklist. RESULTS: A stepwise educational program to teach parents how to administer low-dose chemotherapy (Ara-C) to their child at home and a simple and safe administration procedure were developed. Key uncertainties were identified, including barriers and facilitators impacting future testing, evaluation, and implementation. Causal assumptions and reasoning for how the intervention leads to short-term outcomes and long-term impact were clarified in a logic model. CONCLUSIONS: The iterative and flexible framework allowed for integration of existing evidence and new data and was successfully applied to the development process. The detailed report on the development process of the home chemotherapy intervention can enhance adaptation or replication of the intervention to other settings and thereby mitigate family disruption and stress of frequent hospital visits for these treatments. The study has informed the next phase of the research project that aims to test the home chemotherapy intervention in a prospective single-arm feasibility study. TRIAL REGISTRATION: ClinicalTrials.gov ID: NCT05372536.
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Neoplasias , Pais , Adulto , Criança , Humanos , Adolescente , Estudos Prospectivos , Pessoal de Saúde/educação , Aprendizagem , Neoplasias/tratamento farmacológicoRESUMO
BACKGROUND: Detection of circulating tumor-derived material (cTM) in the peripheral blood (PB) of cancer patients has been shown to be useful in early diagnosis, prediction of prognosis, and disease monitoring. However, it has not yet been thoroughly evaluated for pediatric sarcoma patients. METHODS: We searched the PubMed and EMBASE databases for studies reporting the detection of circulating tumor cells, circulating tumor DNA, and circulating RNA in PB of pediatric sarcoma patients. Data on performance in identifying cTM and its applicability in diagnosis, and evaluation of tumor characteristics, prognostic factors, and treatment response was extracted from publications. RESULTS: A total of 79 studies were assigned for the present systematic review, including detection of circulating tumor cells (116 patients), circulating tumor DNA (716 patients), and circulating RNA (2887 patients). Circulating tumor cells were detected in 76% of patients. Circulating DNA was detected in 63% by targeted NGS, 66% by shallow WGS, and 79% by digital droplet PCR. Circulating RNA was detected in 37% of patients. CONCLUSION: Of the cTM from Ewing's sarcoma and rhabdomyosarcoma ctDNA proved to be the best target for clinical application including diagnosis, tumor characterization, prognosis, and monitoring of disease progression and treatment response. For osteosarcoma the most promising targets are copy number alterations or patient specific micro RNAs, however, further investigations are needed to obtain consensus on clinical utility.
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INTRODUCTION: The aim of this research is to investigate young cancer patients' cognitive functioning and the underlying neurobiological mechanisms when cognitive functions are impaired. The MyBrain protocol is a multidisciplinary study that investigates cancer-related cognitive impairment in children, adolescents and young adults, combining neuropsychology, cognitive neuroscience and cellular neuroscience. The study is exploratory with a wide focus on trajectories of cognitive functions from diagnosis to the end of treatment and into survivorship. METHODS AND ANALYSIS: Prospective longitudinal study including patients diagnosed with non-brain cancers at age 7-29 years. Each patient is paired with a control matched on age and social circle. PRIMARY OBJECTIVE: Evaluation of neurocognitive function over time. SECONDARY OBJECTIVES: Evaluation of self-perceived quality of life and fatigue, P300 in an electroencephalography (EEG) oddball paradigm, power spectrum in resting state EEG, serum and cerebrospinal fluid levels of biomarkers of neuronal damage, neuroplasticity, proinflammatory and anti-inflammatory markers and their association with cognitive function. ETHICS AND DISSEMINATION: The study is approved by the Regional Ethics Committee for the Capital Region of Denmark (no. H-21028495), and the Danish Data Protection Agency (no. P-2021-473). Results are expected to guide future interventions to prevent brain damage and support patients with cognitive difficulties. TRIAL REGISTRATION NUMBER: The article is registered at clinicaltrials.gov NCT05840575 (https://clinicaltrials.gov/ct2/show/NCT05840575).
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Disfunção Cognitiva , Neoplasias , Adolescente , Adulto Jovem , Criança , Humanos , Adulto , Estudos Longitudinais , Estudos Prospectivos , Qualidade de Vida , Disfunção Cognitiva/etiologia , Disfunção Cognitiva/psicologiaRESUMO
BACKGROUND: This study describes the clinical findings of a consecutive series of pediatric and adolescent patients with a diagnosis of intra-abdominal desmoplastic small round cell tumor (DSRCT) prospectively enrolled in European pediatric Soft tissue sarcoma Study Group (EpSSG) protocols: the BERNIE study, the EpSSG MTS 2008 study, and the EpSSG NRSTS 2005 study. METHODS: Patients aged less than 21 years with a diagnosis of DSRCT arising in the abdomen were included. All trials recommended a multimodal approach including intensive multidrug chemotherapy and loco-regional treatment with surgery and/or radiotherapy whenever possible. RESULTS: The analysis included 32 cases (median age 13.7 years, male:female ratio 1.5:1). Three patients had localized tumors, seven had regionally disseminated disease, and 22 extraperitoneal metastases. All but one patient received multidrug chemotherapy and 11 had maintenance chemotherapy. Loco-regional treatment consisted of surgery only in seven cases, surgery plus adjuvant radiotherapy in 10, and radiotherapy only in six. Among the 17 cases who had radiotherapy, six had irradiation of the primary site, 10 had whole abdominopelvic radiotherapy plus boost to macroscopic residual disease, and one had irradiation to lung metastases only. With a median follow-up of 76 months (range: 18-124 months), 5-year event-free and overall survivals were 19.7% and 21.0%, respectively. Event-free survival was significantly worse for patients who did not receive loco-regional treatment (p-value .007). CONCLUSIONS: The study confirmed that the outcome of patients with DSRCT remains dismal and did not improve over recent years despite an intensive multimodal treatment approach.
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BACKGROUND: Limited data exist on the clinical behavior of pediatric non-rhabdomyosarcoma soft tissue sarcomas (NRSTS) with distant metastases at onset, and a clear standard of care has not yet been defined. METHODS: This cohort study reports on pediatric adult-type metastatic NRSTS enrolled in two concurrent prospective European studies, i.e., the randomized BERNIE study and the single-arm MTS 2008 study developed by the European paediatric Soft tissue sarcoma Study Group. Treatment programs were originally designed for patients with metastatic rhabdomyosarcoma, i.e., nine courses of multidrug chemotherapy (with or without bevacizumab in the BERNIE study), followed by 12 cycles of maintenance therapy, whereas radiotherapy and/or surgery (on primary tumor and/or metastases) were delayed until after seven courses of chemotherapy had been administered. RESULTS: The study included 61 patients <21 years old treated from July 2008 to December 2016. The lung was the site of metastases in 75% of the cases. All patients received multi-agent chemotherapy, 44% had local therapy to primary tumor, and 18% had treatment of metastases. Median time to progression/relapse was 6 months. A high rate of tumor progression was observed during the initial part of the chemotherapy program. With a median follow-up of 41.5 months (range, 2-111 months), 3-year event-free survival and overall survival were 15.4% (95% confidence interval [CI], 7.6-25.7) and 34.9% (95% CI, 22.7-47.5), respectively. There were no statistically significant differences in outcome depending on the type of treatment administered. CONCLUSIONS: The study confirmed the overall poor outcome for patients with metastatic NRSTS, whose treatment remains a challenge. PLAIN LANGUAGE SUMMARY: Pediatric non-rhabdomyosarcoma soft tissue sarcomas form a heterogeneous group of rare tumors. Although recent international studies have defined the standard of care for patients with localized disease, limited data are available on the clinical behavior of patients with distant metastases. This study on 61 metastatic cases treated on two prospective European protocols confirms that the chances of survival of such patients are often dismal and a standard treatment is still lacking.
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Rabdomiossarcoma , Sarcoma , Neoplasias de Tecidos Moles , Adolescente , Criança , Humanos , Adulto Jovem , Protocolos de Quimioterapia Combinada Antineoplásica , Estudos de Coortes , Recidiva Local de Neoplasia/tratamento farmacológico , Estudos Prospectivos , Rabdomiossarcoma/tratamento farmacológico , Sarcoma/tratamento farmacológico , Sarcoma/patologia , Neoplasias de Tecidos Moles/patologia , Ensaios Clínicos Controlados Aleatórios como AssuntoRESUMO
In addition to optimising survival of children with rhabdomyosarcoma (RMS), more attention is now focused on improving their quality of life (QOL) and reducing symptoms during treatment, palliative care or into long-term survivorship. QOL and ongoing symptoms related to the disease and its treatment are outcomes that should ideally be patient-reported (patient-reported outcomes, PROs) and can be assessed using patient-reported outcome measures (PROMS). This commentary aims to encourage PRO and PROM use in RMS by informing professionals in the field of available PROMs for utilisation in paediatric RMS and provide considerations for future use in research and clinical practice. Despite the importance of using PROMs in research and practice, PROMs have been reported scarcely in paediatric RMS literature so far. Available literature suggests lower QOL of children with RMS compared to general populations and occurrence of disease-specific symptoms, but a lack of an RMS-specific PROM. Ongoing developments in the field include the development of PROMs targeted at children with RMS specifically and expansion of PROM evaluation within clinical trials.
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Background: Over the past three decades, complex care and treatment have increasingly become the responsibility of parents as home-based care providers, yet little is known about parents' caregiving experiences when considering the variety of care tasks. It is imperative to gain insight into the challenges parents face when managing treatment and care of their child with cancer to ensure optimal parental support and prior to further expansion of home-based parent caregiving. This study aimed to explore the experiences of children and adolescents with cancer, who had received treatment through a portable infusion pump, and their parents in managing different care tasks. It is the first study and forms the basis of the research project INTACTatHome, that develops and tests interventions of home-based intravenous anti-cancer treatment. Methods: Ethnographic fieldwork comprising participant observation and semi-structured interviewing analyzed using qualitative thematic analysis. Thirteen families participated in the study. Results: Three main themes were identified: (1) being a "mini-nurse"; (2) dividing care; and (3) managing anxiety and fear, each based on separate sub-themes. These themes were bound together by an overarching theme: "Ambiguous expectations of parent caregiving." Discussion: This study contributes to a deeper understanding of the varying experiences of children and adolescents and their parents in managing different care tasks for a child or adolescent with cancer. It underscores the need to establish clear expectations of parents as caregivers throughout the cancer treatment trajectory. This perspective is crucial when developing and implementing future home-based care services.
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Serviços de Assistência Domiciliar , Neoplasias , Humanos , Criança , Adolescente , Motivação , Pais , Cuidadores , Hospitais , Neoplasias/terapiaRESUMO
BACKGROUND: Investigations of migraine among childhood cancer survivors have predominantly relied on self-reported information and hospital discharge diagnoses. Alone, both approaches are liable to bias. We used Danish nationwide registers to obtain data on both prescriptions of acute migraine medications (antimigraines) and hospital discharge diagnoses of migraine to assess the relative risk of migraine across a wider spectrum of migraine presentations than previously studied. METHODS: We followed a Danish population-based cohort of 7771 individuals with childhood cancer diagnosed in the period from Jan 1st, 1978 to Dec 31st, 2017, for risk of prescription antimigraine initiation and for risk of hospitalization due to migraine. Rates of hospitalization were assessed for the entire follow-up period whereas rates of antimigraine initiations were assessed in the period from Jan 1st, 1997, to Dec 31st, 2017. Relative to the general population without childhood cancer, standardized incidence ratios (SIRs) were calculated for each outcome. RESULTS: Individuals exposed to childhood cancer were at increased risk of antimigraine initiation (SIR of 1.24, 95% CI: 1.11-1.38) and of migraine hospitalization (SIR of 2.44, 95% CI: 1.87-3.12) from the day of their cancer diagnosis and up to 40 years after. CONCLUSIONS: Individuals diagnosed with childhood cancer have a higher risk of migraine of varying presentations, in addition to migraine resulting in hospitalization as previously reported. This potentially preventable problem warrants clinical attention.
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The European pediatric Soft tissue sarcoma Study Group analyzed all children with epithelioid hemangioendothelioma prospectively registered in the NRSTS-05 (EUDRACT 2005-001139-31) and in MTS-2008 (NCT00379457) studies: 10 patients with localized and one with metastatic disease. Median age was 14.3 years (range, 9.0-18.8). Local therapy was initial primary surgery in seven cases, and five patients received systemic therapy. No patients received radiotherapy. After a median follow-up of 50 months (range, 6-176) for living patients, nine patients remain alive off therapy and two died. Five-year progression free and overall survivals are, respectively, 77.1% (95% confidence interval [CI]: 34.5-93.9) and 74.1% (95% CI: 28.1-93.0).
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Hemangioendotelioma Epitelioide , Sarcoma , Neoplasias de Tecidos Moles , Adolescente , Criança , Estudos Clínicos como Assunto , Hemangioendotelioma Epitelioide/terapia , Humanos , Sarcoma/patologia , Sarcoma/terapia , Neoplasias de Tecidos Moles/patologia , Neoplasias de Tecidos Moles/terapiaRESUMO
OBJECTIVES: In many countries, there is a lack of structured psychosocial health interventions to support young people with cancer. Thus, we developed the nurse-led intervention the Youth-Check Program with the aim of supporting young people with cancer. The aim of the study was to evaluate young patients with cancer and their parents' experiences of the program. DATA SOURCES: In total, 23 semi-structured interviews were conducted. Participants were young patients with cancer, aged 12-18 years (nâ¯=â¯10) and parents aged 41-53 years (nâ¯=â¯13). Data were analyzed thematically using Malterud's systematic text condensation. CONCLUSION: Three themes were derived: "The Youth-Check Program offers a safe space that led to openness among the young people," "participation in the Youth-Check Program met parents' needs for support for their teenager," and "the Youth-Check Program provided new insights that were not always taken into account." IMPLICATIONS FOR NURSING PRACTICE: The Youth-Check Program is a feasible and useful nurse-led intervention, which can be implemented for the benefit of young people with cancer. However, to strengthen the Youth-Check Program, it is important to define young people's individual needs to make sure they are met according to their specific preferences. Most young people took on more treatment responsibility, and they were empowered to set their own agenda in terms of what kind of support they needed in relation to topics that preoccupied them. The parents were given much needed support for their teenagers.
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Neoplasias , Intervenção Psicossocial , Adolescente , Humanos , Neoplasias/terapia , Pais/psicologiaRESUMO
Background: Improved survival rates for children and adolescents diagnosed with cancer call for novel strategies for reducing short- and long-term treatment-related side effects. These include the physical and metabolic sequelae that are exacerbated by sedentary behavior and treatment-induced toxicities. We aim to investigate the effect of an integrative neuromuscular training intervention during the first 6 months of anti-cancer treatment primarily on muscle strength, and secondarily on exercise capacity, physical function, markers of metabolic syndrome, dysmetabolism, and health-related quality of life during and after ended treatment. Methods: One hundred and twenty-seven children and adolescents, newly diagnosed with malignant and benign neoplasia, aged 6-17 years, and treated with chemotherapy or radiation will be randomized to either the intervention or the control arm of the study. The intervention group will, in addition to usual care, be offered a combination of 6 months of supervised physical exercise (integrative neuromuscular training) and home-based exercise. The active control group will, in addition to usual care, receive information along an unsupervised written home-based training program. All participants, including parents, will receive information about the importance of physical exercise during the course of cancer treatment, at the start of treatment, and in 5 monthly sessions. The primary outcome is measured in terms of isometric quadriceps muscle strength. Secondary outcomes include muscle strength and endurance, markers of metabolic syndrome and dysmetabolism, exercise capacity, physical function and activity, days of hospitalization, and health-related quality of life. Assessment will be conducted at treatment initiation (baseline), at 3 and 6 months after inclusion, and 1 month and 1 year after ended treatment. The primary endpoint for lower-body muscle strength is at 6 months after treatment initiation. The effects of the intervention will be evaluated through a constrained linear mixed model. Discussion: This national randomized controlled study has the potential to provide new knowledge concerning the short- and long-term effects of a novel, inclusive approach for youth exercise programming (integrative neuromuscular exercise) in children and adolescents during anti-cancer treatment. Using a pragmatic, low-cost, and time-efficient training design, this intervention can be easily adapted to both hospital and home settings. Clinical Trial Registration: ClinicalTrials.gov (NCT04706676), first released January 5, 2021.
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PURPOSE: Childhood cancer is rare, and treatment is frequently associated with long-term morbidity. Disparities in survival and long-term side effects encourage the establishment of networks to increase access to complex organ-conservative strategies, such as brachytherapy. We report our experience of an international cooperation model in childhood cancers. METHODS AND MATERIALS: We examined the outcome of all children referred to our center from national or international networks to be treated according to a multimodal organ-conservative approach, including brachytherapy. RESULTS: We identified 305 patients whose median age at diagnosis was 2.2 years (range, 1.4 months to 17.2 years). Among these patients, 99 (32.4%) were treated between 2015 and 2020; 172 (56.4%) were referred from national centers; and 133 (43.6%) were international patients from 31 countries (mainly Europe). Also, 263 patients were referred for primary treatment and 42 patients were referred for salvage treatment. Genitourinary tumors were the most frequent sites, with 56.4% bladder/prostate rhabdomyosarcoma and 28.5% gynecologic tumors. In addition to brachytherapy, local treatment consisted of partial tumor resection in 207 patients (67.9%), and 39 patients (13%) had additional external radiation therapy. Median follow-up was 58 months (range, 1 month to 48 years), 93 months for national patients, and 37 months for international patients (P < .0001). Five-year local control, disease-free survival, and overall survival rates were 90.8% (95% confidence interval [CI], 87.3%-94.4%), 84.4% (95% CI, 80.1%-89.0%), and 93.3% (95% CI, 90.1%-96.5%), respectively. Patients referred for salvage treatment had poorer disease-free survival (P < .01). Implementation of image guided pulse-dose-rate brachytherapy was associated with better local control among patients with rhabdomyosarcoma referred for primary treatment (hazard ratio, 9.72; 95% CI, 1.24-71.0). At last follow-up, 16.7% patients had long-term severe treatment-related complications, and 2 patients (0.7%) had developed second malignancy. CONCLUSIONS: This retrospective series shows the feasibility of a multinational referral network for brachytherapy allowing high patient numbers in rare pediatric cancers. High local control probability and acceptable late severe complication probability could be achieved despite very challenging situations. This cooperation model could serve as a basis for generating international reference networks for high-tech radiation such as brachytherapy to increase treatment care opportunities and cure probability.
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Braquiterapia , Neoplasias da Próstata , Rabdomiossarcoma , Neoplasias da Bexiga Urinária , Braquiterapia/métodos , Criança , Feminino , Humanos , Cooperação Internacional , Masculino , Recidiva Local de Neoplasia/radioterapia , Neoplasias da Próstata/radioterapia , Estudos Retrospectivos , Rabdomiossarcoma/radioterapia , Neoplasias da Bexiga Urinária/radioterapiaRESUMO
INTRODUCTION: The prospective TEDDI protocol investigates the feasibility of radiotherapy delivery in deep inspiration breath-hold (DIBH) for pediatric patients. To secure optimal radiotherapy planning, a diagnostic baseline FDG PET/CT in free breathing (FB) and DIBH was acquired. The anatomical changes in the mediastinum and the effect on PET metrics between the two breathing conditions were assessed for pediatric patients with mediastinal lymphoma. MATERIAL AND METHODS: Ten patients aged 5-17 were included and had a PET/CT in FB and DIBH. Metabolic active lymphoma volumes were manually delineated with a visually based segmentation method and the PET metrics were extracted. The anatomical lymphoma, lung and heart volumes were delineated on CT. RESULTS: The lung volume increased while the heart was displaced caudally and separated from the lymphoma in DIBH compared to FB. Both the anatomical and the metabolically active lymphoma volumes appeared different regarding shape and configuration in the two breathing conditions. The image quality of the DIBH PET was equal to the FB PET regarding interpretation and delineation of lymphoma lesions. All PET metrics increased on the DIBH PET compared to the FB PET with the highest increase observed for the maximum standardized uptake value (33%, range 7-56%). CONCLUSION: Diminished respiratory motion together with anatomical changes within the lymphoma increased all PET metrics in DIBH compared to FB. The anatomical changes observed in DIBH compared to FB are expected to reduce radiation doses to the heart and lungs in pediatric patients with mediastinal lymphoma referred for radiotherapy delivery in DIBH and, thereby, reduce their risk of late effects. TRIAL REGISTRATION: The Danish Ethical Committee (H-16035870, approved November 24th 2016), the Danish Data Protection Agency (2012-58-0004, approved 1 January 2017). Registered retrospectively at clinicaltrials.gov (NCT03315546, 20 October 2017).
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Linfoma , Tomografia por Emissão de Pósitrons combinada à Tomografia Computadorizada , Adolescente , Suspensão da Respiração , Criança , Pré-Escolar , Fluordesoxiglucose F18 , Coração , Humanos , Linfoma/diagnóstico por imagem , Linfoma/radioterapia , Órgãos em Risco/diagnóstico por imagem , Estudos Prospectivos , Dosagem Radioterapêutica , Planejamento da Radioterapia Assistida por Computador , Estudos RetrospectivosRESUMO
BACKGROUND: While underlying mechanisms and pathways of social inequalities in cancer survival have been extensively examined in adults, this is less so for children with cancer. Hypothesized mechanisms include prediagnostic utilization of and navigation through the health care system, which may differ by socioeconomic resources of the families. In this nationwide register-based study we investigated the association between measures of family socioeconomic position in relation to prediagnostic health care contacts and stage of disease at diagnosis in children with cancer in Denmark. METHODS: We identified all children diagnosed with a cancer at ages 0-15 years in 1998-2016 (N = 3043) from the Danish Childhood Cancer Registry. We obtained comprehensive information on measures of socioeconomic position, parental health and prediagnostic contacts to both general practitioners and hospitals 24 months prior to diagnosis from various national registries. We fitted multivariable conditional logistic regression models for the association of family socioeconomic and health-related variables with firstly, frequent health care contacts and secondly, advanced stage. RESULTS: We found higher odds ratios (OR) of frequent both overall and emergency health care contacts in the last 3 months before diagnosis in children from households with short parental education and mixed affiliation to work market, when compared to children with high family socioeconomic position. Further, children of parents with depression or of non-Western origin, respectively, had higher OR for frequent overall and emergency contacts. We found no association between socioeconomic position, parental health and stage of disease. CONCLUSION: Families with socioeconomic disadvantage, non-Western origin or depression more frequently utilize prediagnostic health care services, both generally and in the acute setting, indicating that some disadvantaged families may struggle to navigate the health care system when their child is sick. Reassuringly, this was not reflected in disparities in stage at diagnosis. In order to improve the diagnostic process and potentially reduce health care contacts, attention and support should be given to families with a high number of health care contacts over a short period of time.