Search Results (766)

Background: Childhood, adolescent, and young adult cancer survivors (CAYACS) face significant long-term health risks, yet adherence to long-term follow-up (LTFU) care remains inconsistent. This study explores the concept of akrasia (i.e., acting against one’s better judgment by engaging in behaviors known to be harmful or counterproductive) to understand the psychological, cognitive, and systemic barriers influencing survivor engagement in LTFU. Method: Using an ethical reflection approach based on a literature review, we discussed survivor experiences, behavioral science insights, and ethical principles to identify solutions that balance patient autonomy with supportive interventions. A narrative approach was used to summarize the key points discussed during the ethics reflection group meetings. Results: Our findings highlight key barriers such as trauma, avoidance behaviors, and cognitive constraints that contribute to non-adherence. Strategies such as shared decision-making, digital health tools, and nudge-based interventions are proposed to enhance survivor engagement. Ethical considerations emphasize the need for personalized and flexible care approaches that respect survivor agency while mitigating obstacles to adherence. Conclusions: Addressing akrasia through ethical and behavioral frameworks could improve LTFU adherence, ultimately enhancing survivorship care and long-term health outcomes. Full article

Background/Objectives: Peripheral neuroblastic tumors (pNT) are a biologically heterogeneous group of embryonal tumors that derive from the neural crest and affect the sympathetic nervous system. So far, little is known about the complex immune landscape in these rare childhood cancers. Methods: We focused on the immune cell infiltrate of treatment-naïve pNT from 24 patients, including high-risk neuroblastoma (HR-NBL), non-high-risk neuroblastoma (NHR-NBL), ganglioneuroblastoma (GNBL), and rare ganglioneuroma (GN). To gain novel insights into the immune architecture of these pNT subtypes, we used multiplex immunohistochemistry, multispectral imaging, and algorithm-based data evaluation to detect and characterize T cells, B cells, neutrophils, macrophages, and tertiary lymphoid structures (TLS). Results: The majority of the investigated tumor-infiltrating immune cells were macrophages and T cells. Their detailed phenotypic characterization revealed high proportions of M2-like macrophages as well as activated GrzB⁺ CD8⁺ and PD-1⁺ T lymphocytes. Proportions of these T cell phenotypes were significantly increased in GN compared to HR-NBL, NHR-NBL, or GNBL. In addition, TLS occurred in 11 of 24 patients, independent of immune cell frequencies in the whole tissues. Interestingly, all GN, most GNBL, but only a few NBL contained TLS. We distinguished between three TLS maturation stages that were present irrespective of the pNT subtype. The majority belonged to mature TLS of the primary follicle state. Mature LAMP3⁺ dendritic cells were also found, predominantly in T cell zones of TLS. Furthermore, TLS presence identified pNT patients with significantly prolonged progression-free survival in contrast to all other analyzed immunological features. Conclusions: We propose TLS to be a potential prognostic marker for pNT to predict patient outcomes. Full article

Background: Next-generation sequencing (NGS) has emerged as a transformative tool in precision medicine, offering insights into actionable genomic alterations and informing clinical decision-making in childhood and adolescent/young adult (AYA) solid tumors. Methods: We conducted a systematic review and meta-analysis to assess the utility of NGS in identifying actionable genomic alterations and its impact on clinical decision-making. Studies involving patients aged 0–40 years with solid tumors were included. Data were extracted using Covidence, and pooled estimates were calculated using a random-effects model. Bias was assessed using Begg–Mazumdar, Egger, and Harbord tests. Results: Out of 13,624 references screened, 24 studies met eligibility criteria, comprising 5278 patients and 5359 samples, of which 5207 provided usable data. The pooled proportion of actionable alterations was 57.9% (95% CI: 49.0–66.5%), with minimal evidence of publication bias. Clinical decision-making outcomes were reported in 21 studies, with a pooled proportion of 22.8% (95% CI: 16.4–29.9%). Germline mutation rates, reported in 11 studies, yielded a pooled proportion of 11.2% (95% CI: 8.4–14.3%), consistent with rates typically observed in childhood cancers. Significant heterogeneity was observed across studies due to differences in sequencing methodologies, tumor types, and sampling strategies. Conclusions: NGS demonstrates considerable potential in identifying actionable genomic targets and guiding clinical decision-making in childhood and AYA solid tumors. However, the variability in methodologies underscores the need for standardized protocols and reporting practices to enhance comparability and generalizability. This meta-analysis highlights the promise of genomic medicine while acknowledging challenges posed by heterogeneity in study designs. Full article

Background: Oncological care of adolescent patients is often inconsistent, as they frequently fall between pediatric and adult services. The Childhood Cancer Registry of Campania (CCRC) is the Italian largest population-based registry specializing in children 0–19 years old, with a target population of approximately 1.1 million inhabitants. Material and Methods: This report presents epidemiological indicators and clinical pathways on primary brain tumors in adolescents (15–19 years) from the Campania region. Results: Over the study period (2008–2020), the cohort included 219 adolescents with newly diagnosed central nervous system (CNS) tumors with an annual average incidence rate (IR) of 48.9 cases per million/year. The 5-year observed survival rate after diagnosis of CNS tumor was 84.8%. Overall, the most common tumor site was the pituitary gland and craniopharyngeal duct, representing 22.4% of all tumors. The most frequently occurring malignant primary CNS tumor was germinoma, while the most common non-malignant tumor was pituitary adenoma. Most patients were referred to adult services and nearly half migrated outside the region to receive cancer care. Conclusions: Challenges in the care of adolescent oncology patients include limited access to specialized care, difficulties in transitioning from pediatric to adult institutions, distinct tumor biology, and the underrepresentation of adolescents in clinical trials. The care of adolescents with CNS tumors is fragmented across institutions and significant variations in practice exist between adult and pediatric practitioners. Full article

This study aims to explore the link between exposure to tobacco smoke among nonsmokers and the risk of lung cancer in the United States. We searched six databases for studies on second-hand smoke (SHS) and lung cancer following PRISMA guidelines. Following the random effects model and specific statistical methods, our meta-analysis analyzed studies based on SHS exposure type. A total of 19 eligible studies were included in the review and 15 in the meta-analysis. We covered exposure from parents (childhood), spouses and partners (household), and work-related exposure (colleagues), with higher risk among non-smoking children and domestic partners. Findings reveal a consistent link between SHS exposure and increased lung cancer risk for this population (exposure effect sizes: 1.05–3.11). Analysis of childhood SHS exposure reveals a distinct increased risk associated with parental exposure. For nonsmokers living with smoking spouses, there is a marked 41% increase in risk. Higher risk was associated with more and more prolonged SHS exposure. Exposure to SHS in the workplace shows a correlation with lung cancer risk. Our findings highlight increased SHS-related lung cancer risk, particularly among non-smoking children and domestic partners, intensifying with the amount and duration of exposure, indicating the significant impact of SHS within domestic environments. Full article

Retinoblastoma was one of the first malignant tumors to be described as a genetic disease and its development occurs from the loss of function of the retinoblastoma gene (RB1). The difficulty in accessing the tumor during diagnosis highlights the need for non-invasive diagnostic methods. Studies have shown that liquid biopsy, obtained from any fluid material in the body, for example blood, contains free tumor cells and free and circulating DNA or RNA, making it a convenient tool for diagnosis and prognosis during cancer treatment without the need for invasive procedures. Taking advantage of these events, given this situation, we investigated molecular alterations in samples from retinoblastoma cases, using the NGS strategy as a powerful tool for characterization and aid in diagnosis and prognosis. Genomic data from 76 patients diagnosed with retinoblastoma, comprising 162 samples, tumor (TU), aqueous humor (AH), and peripheral blood (PB), were analyzed using the Oncomine Childhood Cancer Research Panel (OCCRA^®). A total of 22 altered genes were detected, and 54 variants. Of the 76 cases, 29 included paired tumor (TU), aqueous humor (AH), and peripheral blood (PB) samples from the same patient. Alterations in the RB1 gene were detected in 16 of these 29 cases, with concordant alterations identified across all three sample types in three patients. In 12 out of 29 patients, the same genetic alteration was found in both TU and AH. In conclusion, the OCCRA panel enabled the detection, in different samples, of molecular alterations in the RB1 gene, as well as CNAs in the MYCN, ABL2, and MDM4 genes. Limitations of AH were observed, primarily due to the small volume of material available and the consequently low concentration of cell-free DNA (cfDNA). However, as AH provides a viable alternative for analyzing tumors, inaccessible to traditional biopsy methods, liquid biopsy holds significant potential to improve diagnostic accuracy and guide treatment strategies in retinoblastoma cases. Full article

Introduction: Pediatric cancer survivors are at high risk for visual–motor and cognitive deficits that persist throughout life. These domains are related to academic performance. The current study examined (i) whether both visuomotor and cognitive functions and (ii) whether visuomotor functions alone mediate the relationship between age and cognitive functions. Methods: In total, there were 210 participants (7–17 years): 70 posterior fossa tumors (M_age = 12.1  ±  3.2 years, 44% female) and 70 acute lymphoblastic leukemia (M_age = 12.3  ±  3.4 years, 45% female) survivors and 70 (M_age = 12.2 ± 3.3 years, 41% female) healthy controls. Visual motor integration, motor coordination and visual perception were assessed using the Beery VMI test. Working memory, attention and planning were assessed using CANTAB. Results: Impaired motor function is significantly more pronounced than cognitive impairment in both groups of cancer survivors (effect size from 25 to 30% for visual–motor and from 5 to 7% for cognitive functions). A multiple regression model revealed that age and visual motor functions are significant predictors of attention (in the ALL group β = −0.490, t = −4.88, p = 0.000) and working memory (in the PFT group β = 0.264, t = 2.72, p = 0.008; in the ALL group β = 0.215, t = 2.24, p = 0.028). Conclusions: In children who have experienced acute lymphoblastic leukemia and tumors of the posterior cranial fossa, visual–motor dysfunction is more pronounced than cognitive impairment. In addition, there is an association between visual–motor function disorders and working memory. These findings can be used to develop more specific rehabilitation protocols. Full article

Given the understandable patient and caregiver distress that often fluctuates across treatment, especially at diagnosis and with major disease milestones, it is important to prioritize interventions that are known to best support families during these key times of transition. Individualized and time-sensitive psychoeducation and anticipatory guidance are effective interventions in reducing distress and fostering adaptive coping across the cancer trajectory and are a standard for the psychosocial care of children with cancer and their families. Patient education materials have proven to be a flexible, accessible, cost-effective, and easily adaptable tool for delivering these interventions to this population. Moreover, within the field, there has been a call to action for the inclusion of patients and families in the development of research study design and clinical programming, recognizing them as the experts of their own experiences. This commentary aims to explore how patients and families can be actively involved in the creation, assessment, and implementation of patient education materials, fostering a collaborative partnership between families and clinicians. We provide detailed examples of this collaboration in the creation of patient education materials focused on promoting coping at diagnosis, supporting siblings and assisting with social reintegration at the end of treatment, promoting responsive caregiving for young children, and supporting caregiver coping through the creation of a podcast. Ultimately, this commentary seeks to emphasize the importance of partnering with patients and families to ensure their voices remain central to intervention development and implementation. Full article

Background: Effective and timely treatment of vitamin B12 deficiency in childhood is crucial because it can lead to serious issues, including delayed growth and neuromotor development. Available treatment options include oral, intramuscular, and sublingual administration. Aim: This study investigated the efficacy of a new dosing protocol for sublingual methylcobalamin. Materials and Methods: In total, 312 patients with vitamin B12 deficiency (serum level < 250 ng/L) were divided into two groups based on their treatment type: intramuscular cyanocobalamin (Group 1) and sublingual methylcobalamin (Group 2). Group 1 included 29 (9.3%) patients, and Group 2 included 283 (90.7%) patients, with 56 (18%) patients in Group 2 undergoing treatment for childhood cancer. The sublingual methylcobalamin protocol consisted of 1 puff (500 μg) daily for children under 8 years of age and 2 puffs (1000 μg) daily for those 8 years and older, administered for 1.5 months and then three times weekly for an additional 1.5 months. Results: The mean ages in Groups 1 and 2 were 10.07 ± 6.05 years (range, 1–17 years) and 7.43 ± 5.86 years (range, 0.1–17 years), respectively. The female/male ratio was 19/10 in Group 1 and 145/138 in Group 2. The most common diagnoses were anemia (72, 22.9%), cancer (56, 18.0%), and hemangioma (40, 12.7%). The median serum levels of vitamin B12 in Group 1 were 177 ng/L before treatment, 447 ng/L after 1.5 months, and 321.5 ng/L after 3 months. In Group 2, the levels were 172 ng/L before treatment, 438 ng/L after 1.5 months, and 360 ng/L after 3 months. There were no significant between-group differences. Both groups showed a statistically significant increase in levels above 300 ng/L. Discussion: Sublingual methylcobalamin, a noninvasive treatment option, was as effective as intramuscular cyanocobalamin. This study is to compare the standard intramuscular protocol with a new dosing regimen for sublingual methylcobalamin. This study showed that it is also useful for children to hold the spray in their mouths for 1 min and avoid food intake for the next 15 min. Full article

Background/Objectives: Early-onset cancer is an emerging global health concern, including in the United States. However, data on early-onset liver and intrahepatic bile duct cancer remain limited. This study aims to fill this gap by analyzing trends in early-onset liver and intrahepatic bile duct cancer in the United States over the past two decades. Methods: This study used National Childhood Cancer Registry data to examine temporal trends in early-onset liver and intrahepatic bile duct cancer in the United States. The analysis involved estimating age-adjusted incidence rates of early-onset liver and intrahepatic bile duct cancer, stratified by histological type, ethnicity, and sex. Results: In 2021, the age-adjusted incidence rate of early-onset liver and intrahepatic bile duct cancer was estimated at 0.53 per 100,000 population (95% Confidence Interval [CI]: 0.48–0.59). From 2001 to 2021, the age-adjusted incidence rate showed a significant annual percent change (APC) of 1.35% (95% CI: 0.87–1.83%). When stratified by sex, the age-adjusted incidence rate in females increased significantly (APC: 3.07%, 95% CI: 2.26–3.87%) while remaining stable in males. Among racial and ethnic groups, non-Hispanic American Indian and Alaska Native (AIAN) individuals had the highest age-adjusted incidence rate, recorded at 2.67 per 100,000 population (95% CI: 0.95–5.85). By histological type, hepatic carcinoma had the highest age-adjusted incidence rate, significantly increasing over time (APC: 1.47%, 95% CI: 0.96–1.99%). In contrast, the incidence rates for hepatoblastoma and unspecified hepatic tumors remained stable between 2001 and 2021. Conclusions: Our study identified an increasing incidence of early-onset liver and intrahepatic bile duct cancer in the United States, primarily driven by cases in females and hepatic carcinoma. Full article

Objective: This study was conducted to investigate the effects of exercise on psychological disorders in patients receiving childhood cancer treatment. Methods: The study group consisted of patients with cancer between the ages of 9 and 17 who were treated in the Hematology–Oncology units of Erciyes University. For the sample group, children with cancer were informed about the content of the study, and 40 children with cancer agreed to participate in the study voluntarily. The volunteers were divided into two groups, control and experimental, each consisting of 20 people. For the pre-test, participants completed a socio-demographic information form, as well as the Kovacs Depression scale, Beck Anxiety Inventory, and the pediatric cancer quality of life scale for children. Volunteers in the experimental group engaged in aerobic and strength exercises for an eight-week period. The study was completed with 8 volunteers in the control group and 14 volunteers in the experimental group due to various factors, such as voluntary withdrawal, disease progression, and mortality. After this period, the volunteers were asked to complete the same scales once more as a post-test. The SPSS 22.00 statistical analysis program was used. The independent samples t-test was employed to compare the pre-test and post-test findings of the control and experimental groups, while the paired samples t-test was used for within-group evaluations. Results: In the within-group comparisons, significant differences were observed in favor of the post-test scores in the experimental group for both the anxiety scale (p < 0.05, Mean: 8.14) and the quality-of-life child form (p < 0.05, Mean: 38.14). For intergroup comparisons, significant differences were found in favor of the experimental group in terms of post-test scores of depression (p < 0.05, mean: 10.57) and anxiety scales (p < 0.05, mean: 8.14). Conclusions: It is postulated that this outcome stems from the positive effects of sports activities in helping children undergoing cancer treatment distance themselves from their psychological adversities. Full article

(1) Background: Inadequate physical activity is an ongoing issue for pediatric survivors of childhood cancer and hematopoietic stem cell transplant (HSCT), increasing their risk for chronic health conditions and decreasing quality of life. Team Survivors is a 12-week program in which survivors train as a group to participate in a community triathlon. Preliminary findings from a quality improvement project of Team Survivors were used to assess its feasibility in influencing survivors’ physical activity and quality of life and their family’s perception of the program. (2) Methods: Repeated measures t-tests evaluated changes in caregiver-reported exercise self-efficacy and health-related quality of life (HRQoL). These findings were supplemented by a narrative review of qualitative data from caregivers and survivors on their overall experience and satisfaction with the program. (3) Results: All participants successfully completed the triathlon, and families reported satisfaction with the program’s logistics (i.e., coordination, coaching, and practice). Neither caregiver-reported exercise self-efficacy nor HRQoL significantly differed between pre- and post-intervention, but notable improvements were reported in all domains of HRQoL. Qualitatively, the perceived benefits reported by caregivers and survivors were multifaceted. Physical benefits included improvements in survivors’ physical activity level and endurance. Psychosocially, caregivers and survivors reported greater confidence and motivation for physical activity, improved coping, and increased social engagement and feelings of normalcy. (4) Conclusions: Mixed method results support the feasibility of Team Survivors in positively impacting survivors’ ability to engage in physical activity. The multifaceted structure of the program may more broadly impact psychosocial functioning in addition to physical activity. Future studies require a larger sample size to adequately power the analyses. Full article

Background/Objectives: Dental caries is believed to be one of the most frequent dental long-term adverse effects of anticancer therapy. It may occur due to numerous chemotherapy-dependent oral symptoms or due to the possible neglection of oral care because of parental involvement in the monitoring of other long-term effects of the therapy. This study aimed to determine the incidence of dental caries and the impact of other risk factors in cancer survivors and age-matched controls. Methods: This cross-sectional study was conducted on 40 cancer survivors and 80 peers divided into three age groups. Indices such as dmft/s, DMFT/S, ft/s, FT/S, Plaque Index (PI), and Gingival Index (GI) were calculated to compare the study participants. The sum of dmft and DMFT was used to assess the possible impact of socioeconomic, oral hygiene, and dietary factors. Results: Caries frequency was found to be lower in cancer survivors (92.50%) than in controls (97.50%). No statistically significant differences were found between the study groups within all the caries indices examined. Strong positive correlations with PI and cariogenic diet in the youngest survivors and with PI in middle-aged survivors were observed. Strong negative correlations in middle-aged children were noticed in terms of father’s education in survivors and mother’s education in controls. Conclusions: The study findings suggest that there is no relationship between chemotherapy and dental caries in long-term cancer survivors. Careful dental care still remains a major contributor to maintaining oral health. Full article

Background: Exposure to pesticides has been associated with an increased risk of developing childhood leukemia. However, the impact of pesticides on childhood leukemia survival has not been examined. We investigated the associations between residential pesticide use during key developmental periods and 5-year survival in children treated for acute lymphoblastic leukemia (ALL). Methods: Residential use of insecticides, herbicides, rodenticides, and flea control products from preconception up to 12 months prior to diagnosis and sociodemographic characteristics were collected via parental interview among 837 children diagnosed with ALL between 1995 and 2008 in California, USA. Data on clinical features were abstracted from medical records. Vital status was obtained through linkage to the National Death Index (NDI) up to 2020. Cox proportional hazards regression models were used to estimate hazard ratios (HRs), adjusting for sociodemographic factors and clinical risk group. Results: A total of 108 children with ALL (~13%) died within 5 years of diagnosis. Exposure to any pesticides pre- and/or postnatally was slightly higher among deceased compared to alive children (95.4% vs. 91.5%; p = 0.23), while use of rodenticides was significantly higher in children who died (25.0%) vs. those who survived (15.5%; p = 0.02). In fully adjusted models, exposure to rodenticides was associated with an increased risk of mortality (HR 1.70; 95% confidence interval (CI) 1.08–2.64; p = 0.02), especially when the child was exposed during pregnancy (HR 1.90; 95% CI 1.15–3.16; p = 0.01) and possibly 12 months before diagnosis (HR 1.60; 95% CI 0.98–2.61; p = 0.06). Increased hazards of death were also observed with other types of pesticides during pregnancy, but those associations were not statistically significant. Conclusions: This study is the first to report reduced survival among children with ALL previously exposed to rodenticides, particularly during pregnancy, underscoring the need to further evaluate mechanisms by which environmental exposures during key developmental stages may later impact cancer outcomes. Full article

The WHO Global Initiative for Childhood Cancer will likely increase the number of childhood cancer survivors in resource-poor countries. This study explored survivorship care in Kenya through parental reports on late effects and the follow-up needs of childhood cancer survivors. Parents of Kenyan childhood cancer survivors (under 18 years old) who completed treatment for at least one year were interviewed using semi-structured questionnaires from 2021 to 2022. Parents of 54 survivors were interviewed. Survivors had solid tumors (52%) and hematological tumors (48%). Most (52%) received chemotherapy combined with either surgery or radiotherapy. Many survivors (72%) experienced symptoms according to their parents. The most prevalent symptoms were pain (37%), fatigue (26%), and ocular problems (26%). Eleven percent of parents observed limitations in the daily activities of the survivors. Parents of survivors with two or more symptoms were more likely to rate symptoms as moderate to severe (p = 0.016). Parents expressed concern about late effects (48%). Only 28% were informed about late effects at the hospital, despite 87% indicating they would have welcomed this information. Follow-up care was deemed important by 98%. Recommendations included providing education about late effects and organizing survivor meetings. Survivorship clinics should be established to ensure that follow-up information and care are accessible. Full article