Sarah Knight

Processing Speed, Attention, and Working Memory After Treatment for Medulloblastoma: An International, Prospective, and Longitudinal Study

PURPOSE The current study prospectively examined processing speed (PS), broad attention (BA), and working memory (WM) ability of patients diagnosed with medulloblastoma over a 5-year period. PATIENTS AND METHODS The study included 126 patients, ages 3 to 21 years at diagnosis, enrolled onto a collaborative protocol for medulloblastoma. Patients were treated with postsurgical risk-adapted craniospinal irradiation (n = 36 high risk [HR]; n = 90 average risk) followed by four cycles of high-dose chemotherapy with stem-cell support. Patients completed 509 neuropsychological evaluations using the Woodcock-Johnson Tests of Cognitive Abilities Third Edition (median of three observations per patient). RESULTS Linear mixed effects models revealed that younger age at diagnosis, HR classification, and higher baseline scores were significantly associated with poorer outcomes in PS. Patients treated as HR and those with higher baseline scores are estimated to have less favorable outcomes in WM and BA over time. Parent education and marital status were significantly associated with BA and WM baseline scores but not change over time. CONCLUSION Of the three key domains, PS was estimated to have the lowest scores at 5 years after diagnosis. Identifying cognitive domains most vulnerable to decline should guide researchers who are aiming to develop efficacious cognitive intervention and rehabilitation programs, thereby improving the quality of survivorship for the pediatric medulloblastoma population.

Examination of risk factors for intellectual and academic outcomes following treatment for pediatric medulloblastoma

BACKGROUND The aim of this study was to prospectively examine the effects of hearing loss and posterior fossa syndrome (PFS), in addition to age at diagnosis and disease risk status, on change in intellectual and academic outcomes following diagnosis and treatment in a large sample of medulloblastoma patients. METHODS Data from at least 2 cognitive and academic assessments were available from 165 patients (ages 3-21 years) treated with surgery, risk-adapted craniospinal irradiation, and 4 courses of chemotherapy with stem cell support. Patients underwent serial evaluation of cognitive and academic functioning from baseline up to 5 years post diagnosis. RESULTS Serious hearing loss, PFS, younger age at diagnosis, and high-risk status were all significant risk factors for decline in intellectual and academic skills. Serious hearing loss and PFS independently predicted below-average estimated mean intellectual ability at 5 years post diagnosis. Patients with high-risk medulloblastoma and young age at diagnosis (<7 years) exhibited the largest drop in mean scores for intellectual and academic outcomes. CONCLUSIONS Despite a significant decline over time, intellectual and academic outcomes remained within the average range at 5 years post diagnosis for the majority of patients. Future studies should determine if scores remain within the average range at time points further out from treatment. Patients at heightened risk should be closely monitored and provided with recommendations for appropriate interventions.

Neurodevelopmental Outcomes in Infants and Children With Single-Suture Craniosynostosis: A Systematic Review

Children with single-suture craniosynostosis (SSC) are increasingly considered to be at high risk of adverse neurodevelopmental outcomes. This systematic review aimed to synthesise and critically appraise the existing literature on the neurodevelopmental features of SSC, with particular attention to methodological quality. A total of 33 articles based on 27 cohorts met inclusion criteria. In the context of variable methodological design and quality, most neurodevelopmental studies indicated that children with SSC are at increased risk for difficulties in cognitive, language, and motor domains during infancy (both pre- and post-surgery) and childhood. Limited information exists on factors influencing outcome.

Interventions in Pediatric Chronic Fatigue Syndrome/Myalgic Encephalomyelitis: A Systematic Review

PURPOSE A range of interventions have been used for the management of Chronic Fatigue Syndrome/Myalgic Encephalomyelitis (CFS/ME) in children and adolescents. Currently, debate exists as to the effectiveness of these different management strategies. The objective of this review was to synthesize and critically appraise the literature on interventions for pediatric CFS/ME. METHOD CINAHL, PsycINFO and Medline databases were searched to retrieve relevant studies of intervention outcomes in children and/or adolescents diagnosed with CFS/ME. Two reviewers independently selected articles and appraised the quality on the basis of predefined criteria. RESULTS A total of 24 articles based on 21 studies met the inclusion criteria. Methodological design and quality were variable. The majority assessed behavioral interventions (10 multidisciplinary rehabilitation; 9 psychological interventions; 1 exercise intervention; 1 immunological intervention). There was marked heterogeneity in participant and intervention characteristics, and outcome measures used across studies. The strongest evidence was for Cognitive Behavioral Therapy (CBT)-based interventions, with weaker evidence for multidisciplinary rehabilitation. Limited information exists on the maintenance of intervention effects. CONCLUSIONS Evidence for the effectiveness of interventions for children and adolescents with CFS/ME is still emerging. Methodological inadequacies and inconsistent approaches limit interpretation of findings. There is some evidence that children and adolescents with CFS/ME benefit from particular interventions; however, there remain gaps in the current evidence base.

Parent-Reported Social Outcomes After Treatment for Pediatric Embryonal Tumors: A Prospective Longitudinal Study

PURPOSE To examine longitudinal parent-reported social outcomes for children treated for pediatric embryonal brain tumors. PATIENTS AND METHODS Patients (N=220) were enrolled onto a multisite clinical treatment protocol. Parents completed the Child Behavior Checklist/6-18 at the time of their childs diagnosis and yearly thereafter. A generalized linear mixed effects model regression approach was used to examine longitudinal changes in parent ratings of social competence, social problems, and withdrawn/depressed behaviors with demographic and treatment factors as covariates. RESULTS During the 5-year period following diagnosis and treatment, few patients were reported to have clinically elevated scores on measures of social functioning. Mean scores differed significantly from population norms, yet remained within the average range. Several factors associated with unfavorable patterns of change in social functioning were identified. Patients with high-risk treatment status had a greater increase in parent-reported social problems (P=.001) and withdrawn/depressed behaviors (P=.01) over time compared with average-risk patients. Patients with posterior fossa syndrome had greater parent-reported social problems over time (P=.03). Female patients showed higher withdrawn/depressed scores over time compared with male patients (P<.001). Patient intelligence, age at diagnosis, and parent education level also contributed to parent report of social functioning. CONCLUSION Results of this study largely suggest positive social adjustment several years after diagnosis and treatment of a pediatric embryonal tumor. However, several factors, including treatment risk status and posterior fossa syndrome, may be important precursors of long-term social outcomes. Future research is needed to elucidate the trajectory of social functioning as these patients transition into adulthood.

Fatigue in Child Chronic Health Conditions: A Systematic Review of Assessment Instruments

BACKGROUND AND OBJECTIVE: Fatigue is common in chronic health conditions in childhood, associated with decreased quality of life and functioning, yet there are limited data to compare assessment instruments across conditions and childhood development. Our objective was to describe fatigue assessment instruments used in children with chronic health conditions and critically appraise the evidence for the measurement properties of identified instruments. METHODS: Data sources included Medline, Cumulative Index to Nursing and Allied Health Literature, and PsycINFO (using the EBSCOhost platform). Study selection included quantitative assessment of fatigue in children with health conditions. Data extraction was as follows: (1) study design, participant and fatigue instruments, (2) measurement properties of fatigue instruments, (3) methodological quality of included studies, and (4) synthesis of the quality of evidence across studies for the measurement properties of fatigue instruments. RESULTS: Twenty fatigue assessment instruments were identified (12 child reports, 7 parent reports, 1 staff report), used in 89 studies. Fatigue was assessed in over 14 health conditions, most commonly in children with cancer and chronic fatigue syndrome. Evidence for the measurement properties of instruments varied, and overall quality was low. Two fatigue instruments demonstrated strong measurement properties for use in children with diverse health conditions and children with cancer. CONCLUSIONS: The review is limited to children younger than 18 years and results are specific to health conditions described, limiting generalizability of findings to other populations. Evidence for the measurement properties of fatigue instruments varied according to the population in which instruments were used and informant. Further evidence is required for assessment of fatigue in younger children, and children with particular health conditions.

Working Memory Abilities Among Children Treated for Medulloblastoma: Parent Report and Child Performance

OBJECTIVE We investigated the 5-year postsurgical developmental trajectory of working memory (WM) in children with medulloblastoma using parent and performance-based measures. METHOD This study included 167 patients treated for medulloblastoma. Serial assessments of WM occurred at predetermined time points for 5 years. RESULTS There was a subtle, statistically significant increase in parental concern about WM, coupled with a statistically significant decrease in age-standardized scores on performance-based measures. However, whole-group mean scores on both parent and performance-based measures remained in the age-expected range. Posterior fossa syndrome was consistently associated with poorer WM. Younger age at treatment and higher treatment intensity were associated with greater negative change in WM performance only. CONCLUSIONS Most children treated for medulloblastoma display WM within the age-appropriate range according to parent report and performance. However, the subtle negative changes over time and identified subgroups at increased risk highlight the need for ongoing monitoring of this population.

Motor functioning during and following treatment with chemotherapy for pediatric acute lymphoblastic leukemia

This systematic review evaluated empirical studies examining motor skills in children during and following treatment for acute lymphoblastic leukemia. Most studies indicated that children on‐treatment display poorer gross and fine motor abilities than healthy peers, but generally have intact visual‐motor integration skills. Studies have reported gross motor difficulties in 5–54% of survivors. There is some limited evidence for long‐term fine motor deficits. The evidence for visual‐motor integration difficulties in the survivor population is less consistent. Larger studies with a longitudinal design are needed to further specify the onset and timing of motor difficulties and ascertain risk factors. Pediatr Blood Cancer 2013;60:1261–1266.

Paediatric chronic fatigue syndrome: complex presentations and protracted time to diagnosis.

The diagnosis and management of paediatric chronic fatigue syndrome (CFS) remain ongoing challenges for paediatric clinicians, particularly given its unknown aetiology and the little research on effective treatments for this condition. The aim of this study was to describe the presenting features of new patients attending a specialist chronic fatigue clinic at a tertiary‐level Australian childrens hospital.

Gross and fine motor skills in children treated for acute lymphoblastic leukaemia

Objective: Chemotherapy treatment for acute lymphoblastic leukaemia (ALL) may disrupt motor development, with suggestions that gross and fine motor deficits are different depending on time since treatment. Methods: Thirty-seven participants aged between 2.5 to 5 years at the time of diagnosis were assessed using the Movement Assessment Battery for Children, 2nd Edition (MABC-2) and the Bruininks–Oseretsky Test of Motor Proficiency, 2nd Edition, Short Form (BOT-2 SF), and divided into groups (i.e. months-off-treatment): (1) 0–12, (2) 13–24, and (3) 25–60 for comparison. Results: MABC-2 and BOT-2 SF mean total scores fell within the average range. Twenty-six percent of the sample performed in the impaired range on the MABC-2. Group 2 had significantly lower Manual Dexterity scores than the normative population and lower BOT-2 SF scores than Group 1. Conclusion: Most children treated for ALL display appropriate motor skills, yet around a quarter experience general motor difficulties. Time-off-treatment did not affect the prevalence of motor impairments on any measure.