Abstract INTRODUCTION: Due to recent advancements in surgical techniques, imaging and adjuvant therapies, survival rates for children with central nervous system (CNS) tumours have improved. Research priorities have now shifted to focus on late-effects and quality of survivorship amongst childhood CNS tumour survivors. Our study aimed to assess levels of cardiorespiratory fitness and health-related quality of life (HRQoL) amongst survivors of childhood CNS tumours. A secondary aim was to investigate potential relationships between cardiorespiratory fitness and HRQoL. METHODS: Participants were recruited from the National Children’s Cancer Service in CHI Crumlin. Inclusion criteria included: diagnosis of a primary CNS tumour, aged between 6-17 years, between 3 months and 5 years post completion of oncology treatment, independent mobility, deemed clinically appropriate to participate by treating oncologist. Cardiorespiratory fitness was assessed using the six-minute walk test. HRQoL was assessed with the PedsQL Generic Core Scales, Version 4.0, both self-report and parent proxy report forms were used. RESULTS: 20 participants (n=9 male) were recruited with a mean age of 12.34 (SD = 3.46) years. Mean time since completion of oncology treatment was 2.31 (SD = 1.38) years. Mean 6-minute walk distance (6MWD) was 482.75 (SD = 50.04) metres, equating to the 5.55th (SD = 6.83th) percentile overall. 6MWD was significantly reduced compared to predicted 6MWD (t = -12.52, p <.001, 95% CI [-163.52, -116.68]). Parent proxy-reported HRQoL was significantly reduced compared to healthy population norms (t = -5.82, p <0.001, 95% CI [-25.76, -12.17]). A strong positive correlation exists between 6MWD and both parent-proxy (Pearson’s r = 0.533, p = 0.015) and child-reported HRQoL (r = 0.580, p = 0.007). CONCLUSION: Survivors of childhood CNS tumours present with impaired levels of cardiorespiratory fitness and HRQoL compared to healthy population norms. Higher levels of cardiorespiratory fitness are associated with higher levels of HRQoL.
Background Child study satisfaction has rarely been assessed in longitudinal pediatric research. This study examined child and maternal study satisfaction among 10-15-year-old participants in The Environmental Determinants of Diabetes in the Young (TEDDY) study, an observational multinational (US, Finland, Germany, and Sweden) study of children at increased genetic risk for type 1 diabetes enrolled at birth. Methods Child and maternal study satisfaction was measured by a 3-item annual questionnaire from child-age 10 years until the last visit at child-age 15. Generalized estimating equations identified sociodemographic and study-related variables associated with study satisfaction. Results Children and mothers exhibited high study satisfaction across all ages although their study satisfaction scores were weakly correlated (r ≥ 0.20, p < 0.001). Factors associated with higher study satisfaction were similar for children and mothers: living in the US compared to Finland (Finnish children: -0.78 95%CI -0.86, -0.69; Finnish mothers: -1.48 95%CI -1.61, -1.35; p < 0.001), older child age (15-year-old vs 10-year-old child: 0.09 95%CI 0.03,0.14; mother: 0.42 95%CI 0.37, 0.47; p < 0.001), and accurate perception about the child's type 1 diabetes risk (child: 0.20 95%CI 0.15,0.25; mothers: 0.20 95%CI 0.13, 0.22; p < 0.001). Additional factors associated with greater child study satisfaction included female sex (-0.29 95%CI -0.35, -0.24; p < 0.001), being multiple islet autoantibody positive (0.21 95%CI 0.11, 0.32; p < 0.001), lower child anxiety about type 1 diabetes risk (-0.01 95%CI -0.02, -0.01; p < 0.001), having a mother with higher study satisfaction (0.13 95%CI 0.12, 0.15; p < 0.001), and having read an age-appropriate book about the study, its purpose, and type 1 diabetes (0.19 95%CI 0.12, 0.25; p < 0.001). Conclusions Study satisfaction was high among 10-15-year-old children at increased genetic risk for type 1 diabetes, and their mothers, who had participated in a longitudinal observational study since the child's birth. Factors associated with satisfaction were similar for children and their mothers. Potentially modifiable factors that could be targeted to improve study satisfaction include mother and child accuracy about the child's type 1 diabetes risk and for child study satisfaction in particular - the child's own anxiety about type 1 diabetes, the child's mother's satisfaction and having the child read high quality age-appropriate materials about the study. Trial registration NCT00279318, 06/09/2004.
Abstract INTRODUCTION: Diffuse midline glioma (DMG) is a fatal childhood CNS tumor. Magnetic resonance imaging (MRI) is the gold standard for DMG diagnosis and monitoring of response to therapy. Leveraging novel MRI analytical approaches, including volumetric and machine learning based analyses, may aid in the prediction of patient overall survival (OS) and help to identify high-risk cases. METHODS: T1- and T2-weighted MR images were retrospectively collected from children and young adults diagnosed with DMG (n=43). MRI features, including manually determined 3D tumor volume (T2), T1 contrast-enhancing tumor volume, T1 relative to T2 volume (T1/T2), tumor relative to whole brain volume, tumor average intensity, and tumor heterogeneity (i.e., intensity skewness and kurtosis), were evaluated at upfront diagnosis. MRI features were analyzed to identify significant predictors of OS outcome, which was defined as OS shorter, or longer, than one year from diagnosis. A support vector machine was used to predict OS outcomes using combinations of these features. RESULTS: The presence of T1 contrast-enhancing tumor at diagnosis (p=0.01), and a high T1/T2 ratio (>25%, p=0.009), predicted significantly shorter median OS. Moreover, feature selection identified T2 mean intensity (p<0.001), T2 image intensity skew (p=0.006), T1/T2 ratio (p=0.02), and T1 volume relative to whole brain (p=0.03) as significant predictors of OS outcome (short versus long). Combining T2 mean intensity, T2 image skew, T1 segment kurtosis and patient gender resulted in OS outcome prediction accuracy of 83.3% (sensitivity=85%, specificity=81.8%, n=42 cases).CONCLUSION: We have identified MRI volume and imaging features that significantly predict OS outcome in children diagnosed with DMG. Our findings provide a framework for incorporating MRI volumetric and machine learning analyses into the clinical setting, allowing for the customization of treatment based on tumor risk characteristics.
Abstract Diffuse midline glioma with H3 K27 alterations (DMG) is the most aggressive primary malignant brain tumor in the pediatric population. Radiation therapy (RT) is the standard-of-care; however, it has not demonstrated a significant improvement in overall survival or stalled disease progression, and children usually succumb to disease within 12-15 months of diagnosis. Consequently, finding efficient treatments for DMG remains one of the most critical unmet needs in modern neuro-oncology. Interleukin 13 receptor subunit alpha 2 (IL-13Rα2) is a cell-surface receptor upregulated in ~80% of DMG versus normal brain, posing a potentially promising therapeutic target. Immunotherapies harnessing IL-13Rα2 to selectively delivery cytotoxic payloads, such as pseudomonas exotoxin A (PE), to tumor cells have been previously demonstrated to be safe in DMG patients and efficacious in preclinical models of DMG with high IL-13Rα2 expression using convection-enhanced delivery (CED). Furthermore, there is evidence that PE-based therapies can sensitize cells to RT. In this study, we utilized in vitro and in vivo models of DMG to evaluate clinically-relevant combination treatments of RT and the IL-13/PE immunotoxin GB-13 (IL13.E13K-PE4E). GB-13 improved the effectiveness of low-dose RT in multiple DMG cell lines by promoting caspase activation, which boosted apoptotic cell death. Intratumoral administration of GB-13 via chronic CED decreased tumor burden and prolonged survival in both DMG patient-derived orthotopic xenograft and genetically engineered mouse models. When GB-13 and RT were administered concurrently, this impact became even more prominent, informing future preclinical and clinical investigation of a potentially efficacious therapeutic approach in a subset of DMG.
Background Child abuse remains a significant public health challenge. Healthcare workers serve as critical frontline responders, yet gaps persist between legislative frameworks and implementation. Objective The objective of this study is to assess knowledge, attitudes, and practices of healthcare workers in Al Dhahira regarding child abuse identification and reporting, identify barriers to consistent implementation, and examine factors associated with reporting behavior. Methods A descriptive cross-sectional study of 150 healthcare workers at 18 primary healthcare centers in Al Dhahira was conducted in 2023 (sample size calculated as 148 for a population of 600, 95% confidence level). Data were analyzed using IBM SPSS Statistics for Windows, Version 25 (Released 2017; IBM Corp., Armonk, New York, United States) with descriptive statistics and chi-square tests (p<0.05). Results The sample comprised 102 (68.0%) physicians, 38 (25.3%) nurses, and 10 (6.7%) allied health professionals. While 140 (93.3%) were aware of the Omani Child Law and 128 (85.3%) knew a child protection hotline exists, only 110 (73.3%) knew the correct hotline number; 91 of 128 (71.1%) among those aware. Although 138 (92.0%) believed suspected abuse should always be reported, only 110 (73.3%) consistently reported cases, revealing an 18.7-percentage-point attitude-practice gap. Major barriers included fear of legal consequences (44; 29.3%), clinical identification challenges (42; 28.0%), and lack of feedback (39; 26.0%). Previous child protection training was significantly associated with consistent reporting behavior: 73 of 89 (82.0%) trained workers consistently reported versus 37 of 61 (60.7%) untrained workers (p=0.001). Conclusions Healthcare workers demonstrate good theoretical knowledge of child protection frameworks but face significant practical barriers to consistent implementation. Evidence-based interventions including professional development programs, clear legal protection communications, feedback mechanisms, and multi-professional reporting capacity warrant prospective evaluation to bridge the attitude-practice gap and ensure effective child protection implementation.
Background: Post-concussion sequelae in adolescents include physical, cognitive, and psychosocial effects that may result in reduced quality of life (QOL) and academic challenges when returning to school. The COVID-19 pandemic resulted in altered teaching and learning models outside the traditional in-person classroom, and elevated psychosocial stress and isolation among adolescents. It is unknown whether these changes affected QOL or academic challenges post-concussion. Hypothesis/Purpose: To compare self-reported QOL and academic challenges among concussed adolescents prior to and during the COVID-19 pandemic. Methods: Concussed adolescents were enrolled from secondary schools and children’s medical centers across seven sites into the Sport Concussion Outcomes in Pediatrics (SCOPE) study (PRiSM Concussion RIG). Participants completed demographics, medical history, symptom report, the Concussion Learning Assessment and School Survey (CLASS) and QOL via PROMIS Pediatric Global 25 during their first evaluation visit. Participants were grouped by date ranges: September 2018-January 2020 (Pre-COVID-19) and September 2020-May 2021 (COVID-19). No participants were enrolled between February-August 2020 (cessation of clinical research activities during this time.). Results: A total of 141 patients were enrolled. Mean age=14.9±2.4 years, 61% self-reported as male, 82% white and 85% not Hispanic or Latino, 5.2+3.1 days post-injury (range=1-14 days). Seventy-four patients were enrolled prior to COVID, and 67 were enrolled during COVID (Table 1). No significant differences were found between the ‘Pre-COVID-19’ group versus ‘During COVID-19’ group in symptom resolution, symptom severity score, QOL, or academic concerns (Table 2). The only difference was the ‘person at school who is best to coordinate support.’ A significantly lower proportion of participants who sustained their concussion during COVID-19 reported their principal/assistant principal would be the best person to coordinate support compared to those who sustained their concussion before the COVID-19 pandemic. In contrast, a significantly higher proportion of those who sustained their concussion during the COVID-19 pandemic reported their coach would be the best person to coordinate support. Conclusion: In this convenience yet nationally representative sample, students who sustained a concussion during the pandemic did not report increased academic concerns or reduced QOL compared to those whose injury was prior to the pandemic. Median symptom severity was lower during the pandemic, though not statistically significant. These findings could represent shifted priorities of the principal/assistant principal but still adequate support from medical and school-based staff; less academic stress and greater flexibility during the COVID-19 pandemic; or may represent limitations in the CLASS instrument to detect differences that occurred during the pandemic.Table 1 Participant characteristics by group. Data are presented as mean (standard deviation) or n (% within group).Table 2 Clinical and Quality of Life characteristics (via PROMIS Pediatric Global 25) between groups, assessed at the initial post-concussion assessment. Data are presented as median [interquartile range] and compared using Mann Whitney U tests.
Objective This study examined the relationship between adolescent sports participation and school adjustment and analyzed the serial mediating effects of peer relationships and depressive symptoms. Methods The study sample comprised 8,096 adolescent participants. Assessments were conducted using the Physical Activity Questionnaire for Children (PAQ-C), the School Social Behavior Scale (SSBS), the Quality of Life Scale for Children and Adolescents (QLSCA), and the Symptom Checklist-90 (SCL-90). The hypothesized mediation effects were tested using the SPSS 26.0 macro PROCESS 4.5 with bias-corrected bootstrapping (5,000 resamples) to assess the significance of the indirect effects. Results Group comparisons showed that boys scored higher than girls on sports participation, school adjustment and reported lower depressive symptoms scores (p < 0.001), while no significant gender difference emerged for peer relationships. No significant age differences were observed for sports participation, school adjustment, or peer relationships (all p > 0.05), but depressive symptoms increased significantly with age (p < 0.01), with the highest levels at age 19. Only children scored higher on sports participation and school adjustment and lower on depressive symptoms, but lower on peer relationships, than non-only children (p < 0.001). Students in remote areas showed lower school adjustment and higher depressive symptoms scores than those in several non-remote groups (p < 0.001). Sports participation was positively correlated with school adjustment (r = 0.260, p < 0.001) and peer relationships (r = 0.201, p < 0.001) and negatively correlated with depressive symptoms (r = -0.166, p < 0.001). Peer relationships were positively correlated with school adjustment (r = 0.344, p < 0.001) and negatively correlated with depressive symptoms (r = -0.362, p < 0.001). Depressive symptoms were negatively correlated with school adjustment (r = -0.226, p < 0.001). Peer relationships (effect = 1.76, 95% CI [1.50, 2.03]) and depressive symptoms (effect = 0.23, 95% CI [0.14, 0.33]) each mediated the association between sports participation and school adjustment. They also formed a small but statistically significant serial mediation effect pathway (effect = 0.20, 95% CI [0.14, 0.28]). Conclusion This study further clarifies the relationship between adolescent sports participation and school adjustment, and emphasizes the chain mediating role of peer relationships and depressive symptoms. These findings provide empirical evidence for the educational value of adolescent sports participation and offer practical implications for optimizing school physical education to enhance school adjustment and promote mental health.
Abstract Introduction/Background Chronic pain has been dubbed a place where mental and physical health meet. The emotional and social needs of young people experiencing Chronic Pain can be vast, yet families often fall through cracks in services. Other families receive a plethora of support options from psychological practitioners, allied health professionals and social care, but with laser focus on the medical answer and fix decline all potential referrals. Historically psychology received numerous referrals for young people who declined to meet with us, leaving our colleagues and families feeling unheard, frustrated stuck and alone. How to support families/colleagues without seeing children individually? Description/Method Ed (15-year-old male) experiencing paralysis and pain in dominant arm and hand: • extensive investigations and specialist consultations sought, including scans and neurological opinion - nothing medically concerning identified. • Previous referral to another tertiary psychology service made and group therapy offered. • Over the course of the investigations reports of pain and A&E attendance increased, whilst school attendance decreased -> referred to chronic pain service. Input offered at Evelina London: • 2 x Multidisciplinary clinic (chronic pain diagnosis given at first appointment). • 1 x Pain education workshop. • Multiple physiotherapy and occupational therapy appointments. • 3 x referrals to psychology over preceding 9 months with concerns about mood and engagement. Progress: Initial gains in first months • Pain and paralysis reduced to dominant hand only. • Strength and range of motion restored to shoulder and arm. • School attendance increased. However, in recent months A&E attendances and reports of pain were increasing, with service engagement decreasing. Requests for individual psychology for Ed had been made by various team members, but Ed had not taken up offered psychology conversations or appointments. This contributed to expressions of frustration, worry and hopelessness in the team who wanted individual psychology for Ed “to move things forward”. 3rd Multidisciplinary Clinic appointment: With all therapists and doctors in attendance the psychologist took time to explore Ed and his mother's daily lives outside pain, their interests, joys and skills. They explored the family's explanations for pain, experience of services so far, roles they felt investigations, doctors, occupational therapy, physiotherapy and psychology had, could and should play. Amongst other things particular emphasis placed on: • Privileging family's views. • Using patient's language. • Regular summarising. • Placing professionals then family members in listening positions and exploring what each had heard the other say. The psychologist endorsed Ed's view that psychology had no role to play in his care. Discussion/Results Was Ed in desperate need of 1-1 psychology to move forward? For this family the psychological intervention was with both family and healthcare system. The psychologist used a narrative approach, positioned as a curious (almost) "outsider", with the patient as an expert in their own care. Beginning with a foundation of identity outside pain, followed by facilitated witnessing of ideas and experiences for family and healthcare professionals. Thus, new insights, a shared understanding and plan could develop. With this approach it emerged referrals to psychology coupled with no simple medical explanation and treatment fed fears professionals thought "pain was all in Ed's head". This shook confidence in the healthcare team and reinforced messages the family experienced family, school and friends as giving. Around this time Ed's progress stalled. Whilst family described previous negative experiences of psychologists, current physiotherapy was described as developing "strength and mobility", occupational therapy as "really understanding" and giving "useful exercises/advice", cheerleading throughout a difficult journey. Doctors offered (another) detailed review of investigations and physical examination reiterating pain, but not damage were present. The psychologist thoroughly explored and all things psychology might add, none of which the family felt were useful. This choice was thoroughly validated and respected. During the appointment "thinking break" (family not present) the team were surprised by the family's positive interpretations of care offered thus far. It was hypothesised not feeling believed/validated was a very difficult place to "recover" from and ideas generated of what validation/being believed would look like for this family. Given the family's vocalised block to progress was "the hand", perhaps is was a hand specialist rather than a psychologist? The family were pleased to see joint occupational and hand therapy resulting in "full recovery". Throughout the professionals were offered regular psychological consultation and team discussion time. Key learning points/Conclusion There are many ways to peel the chronic pain orange. When faced with often overwhelming needs of young people living with chronic pain and sometimes overwhelming needs of referring health and social care systems it can be hard to hold onto this idea. Initially referrers thought individual psychology was indicated, what was offered and created change was a psychologically informed multidisciplinary appointment in which the family's worries were given space, validated and answered, whilst their right to decline psychological support was validated and bolstered. Alongside ongoing psychological consultation to professionals. Pain is all in my head, no-one believes me, no-one understands. Are powerful ideas that can be detrimental to family-healthcare relationships, whether stated directly, implied or internally voiced. They can represent a significant barrier to families accessing psychology directly. We may wholeheartedly believe people experience pain that cannot reliably be seen or measured externally, but that does not mean we are experienced that way. For me this case highlighted the importance of • checking out beliefs about symptoms. • gauging whether ongoing/regular validation and reassurance is needed and in what form. • even celebrating someone's successes in the face of pain even suggesting someone can move on with their life can be experienced as invalidating/not getting it. Families need a voice in their won paths forward. Is what you thought you said what they thought you meant? Whether with families or professional you can never underestimate the value of checking whether the same thing was heard differently. This is written from the position of a psychologist embedded within a predominantly non-pharmacological multidisciplinary chronic pain service, which informed the intervention. Other cases have invited/necessitated different approaches. It would be interesting and helpful to invite discussion around the variety of ways similar/different road blocks have been bypassed, removed or otherwise.
Abstract Background Phubbing (phone snubbing) occurs when attention to a smartphone displaces attention to someone who is physically present. Among school-age children, family-based intervention may be relevant because patterns of technology use develop within everyday caregiver relationships. Objective This study examined whether a phubbing-adapted Child–Parent Relationship Therapy (CPRT) program was associated with lower phubbing scores in primary school children. Methods: The quasi-experimental pretest–posttest study included 30 children aged 10–12 years with elevated phubbing screening scores. Children entered an intervention group ( n = 15) or a waitlist control group ( n = 15). Mothers in the intervention group attended 10 weekly CPRT sessions of approximately 2 hours each. Children completed the Persian Generic Scale of Phubbing at baseline and posttest. Separate ANCOVA models compared posttest scores after adjustment for the corresponding baseline score, and Holm correction was applied across the total score and four subscales. Results Mean total scores decreased by 7.20 points in the intervention group and increased by 2.40 points in the control group. Adjusted posttest means were 42.13 and 51.73, respectively, F (1, 27) = 29.73, Holm-adjusted p < .001, partial η² = .524. Adjusted differences also favored the intervention for nomophobia, interpersonal conflict, self-isolation, and problem acknowledgement (Holm-adjusted ps ≤ .006; partial η² = .263–.423). Sensitivity analyses produced the same overall pattern. Conclusions: Participation in the program was associated with lower short-term child-reported phubbing scores. Larger randomized studies with prespecified screening rules, independent fidelity assessment, and follow-up are needed before drawing causal or long-term conclusions.
Introduction To investigate parental type 1 diabetes-related anxiety in response to genetic screening at birth and islet autoantibody test results in at-risk children participating in the prevention study Primary Oral Insulin Trial (POInT). Methods Children were screened at birth for genetic risk for type 1 diabetes and enrolled between 4-7 months of age in the POInT study. Parental anxiety regarding their child's risk of type 1 diabetes was measured by the State Anxiety Inventory (SAI) at 8-11, 18, and 36 months of age. Blood samples were analyzed for islet autoantibodies at each visit and results and information on risk of type 1 diabetes was communicated to the parents. Linear regression was used to investigate factors associated with anxiety. Results Type 1 diabetes related anxiety was considered high among 58.5% of the mothers and 41.9% for the fathers when the child was between 8-11 months old, regardless of autoantibody status. Mean anxiety scores decreased over time among parents of children with negative autoantibody status. Receiving information about their child's positive islet autoantibody status increased parental anxiety and their anxiety scores remained high over time. Anxiety was also found to be associated with country of residence, having a first degree relative with type 1 diabetes, and parental study satisfaction for both mothers and fathers at all time-points. First degree relatives were more anxious than those from the general population and parents more satisfied with participation in the study were less anxious. Parents from Poland and Belgium were more anxious than parents from Germany and Sweden. Conclusion Type 1 diabetes related anxiety is common among parents participating in genetic screening at birth and receiving information on high genetic risk after screening at birth, declining over time. Information on islet autoantibody positivity increases type 1 diabetes related anxiety. Having a first degree relative and being a mother increase the risk of anxiety, while fathers and parents satisfied with participation are less anxious. These findings emphasize the importance of close follow-up and individualized support of families with a child at high genetic risk for type 1 diabetes.
