Physical functioning in pediatric survivors of childhood posterior fossa brain tumors.

BACKGROUND: Survival rates for children diagnosed with posterior fossa brain tumors (PFBTs) have improved significantly over the past several decades, and long-term functioning assessments have become priorities. These evaluations have occurred frequently in adults but only rarely in children. This study describes a cross-sectional assessment of physical functioning in pediatric survivors of PFBTs using the Bruininks-Osteretsky Test of Motor Performance, Second Edition (BOT-2). METHODS: Primary analyses compared BOT-2 scores to normative data using 1-sample t tests for each gross motor subscale (Bilateral Coordination, Balance, Running Speed/Agility, Strength) and motor-area composite (Body Coordination and Strength and Agility). Second, the cohort was stratified by diagnostic or treatment variables. Group differences and groups vs norms were evaluated using independent 2-sample and 1-sample t tests, respectively. Primary analyses compared BOT-2 scores with normative data using 1-sample t tests for each gross motor subscale (Bilateral Coordinationcoordination, Balance, Running Speed/Agility, Strength) and motor-area composite (Body Coordination and Strength and Agility). Second, the cohort was stratified by diagnostic or treatment variables. Group differences and groups vs norms were evaluated using independent 2-sample and 1-sample t tests, respectively. RESULTS: Mean age of 30 participants was 11.4 years (range, 4.9y-18.2y), and mean time from diagnosis was 6.1 years (range, 1.1y-16.7y). Cerebellar astrocytoma (43.3%) and medulloblastoma (40%) were the most common diagnoses. As a group, significantly decreased functioning, compared with norms, was observed in Balance (P < .001) and Running Speed/Agility (P = .005). Specifically in Balance, 21 (70%) participants performed below or well-below average. Participants with a non-astrocytoma performed significantly lower than norms in all areas, independent of age at diagnosis. Survivors with tumors infiltrating the vermis demonstrated significantly lower Body Coordination than norms (P < .001). CONCLUSIONS: Pediatric survivors of PFBTs demonstrated decreased physical functioning, most notably in Balance. These data underscore the need for further research and implementation of physical activity programs aimed specifically at approaches to minimize physical limitations.

Long-term quality of life in children treated for posterior fossa brain tumors.

OBJECT: In the face of increasing survival, quality of life (QOL) has become an important indicator of treatment success in children with posterior fossa brain tumors (PFBTs). The authors' objective was to assess the long-term QOL in survivors of PFBT. METHODS: The authors conducted a cross-sectional study of children who, between birth and age 18 years at diagnosis, had previously been treated at their institution for a PFBT. At the time of assessment for this study, children were between 5 and 19 years old and had received standard treatment for PFBT ending at least 6 months before the assessment. The QOL was measured with the Pediatric Quality of Life Inventory (PedsQL) generic score scales and the Health Utilities Index Mark 3 (HUI3). Multivariate analyses were used to assess several variables (patient related, treatment related, and socioeconomic) for association with QOL. RESULTS: A total of 62 children participated in the study (median age at assessment 11.9 years, interquartile range [IQR] 7.8-14.8, and median age at tumor diagnosis of 4.9 years, IQR 2.5-6.9). Median time since active treatment for their PFBT was 5.2 years (IQR 2.4-10.1). Tumor types included cerebellar pilocytic astrocytoma (45.2%), medulloblastoma (30.6%), ependymoma (11.3%), and brainstem astrocytoma (11.3%). Adjuvant therapy included chemotherapy (40.3%) or radiotherapy (14.5% focal and 21.0% craniospinal radiotherapy). Permanent treatment for hydrocephalus was required in 38.7% of the patients. Tumors recurred in 11.3%, requiring repeat treatment in these patients. The median HUI3 utility score was 0.91 (IQR 0.71-1.00) and the median PedsQL total score was 78.3 (IQR 64.1-92.4). Only the following variables were significantly associated with decreased QOL in multivariable model testing (all p < 0.05): need for permanent hydrocephalus treatment, large ventricle size, decreased family functioning, and lower family income. CONCLUSIONS: As a group, long-term survivors of pediatric PFBT appear to have QOL indicators that are similar to those of the general population, although a reasonable minority of patients experience poor outcomes. Although several confounding variables likely remain in this retrospective study, important associations with QOL include the presence of hydrocephalus and socioeconomic factors. The study sample size, however, was limited and the presence of other important factors cannot be excluded.

Comparison of Hydrocephalus Outcome Questionnaire scores to neuropsychological test performance in school-aged children.

OBJECT: The Hydrocephalus Outcome Questionnaire (HOQ) is an established means of measuring quality of life, but the cognitive component of this questionnaire has never been formally compared with gold-standard neuropsychological test scores. The authors hypothesized that the HOQ Cognitive Health score would demonstrate a relatively strong correlation with neuropsychological test scores, whereas much weaker correlations would be seen for HOQ Physical and Social-Emotional Health scores. METHODS: A cross-sectional study of children with long-standing hydrocephalus presenting to The Hospital for Sick Children's Neurosurgery Clinic was performed between July 2006 and September 2008. Participating children and families completed the HOQ and a battery of 21 standard neuropsychological tests and questionnaires. Pearson correlation analysis was then performed. RESULTS: A total of 83 patients (81% participation) was accrued; the mean age was 11.5 ± 3.4 years (mean ± SD) at the time of assessment. The mean age at hydrocephalus treatment was 1.3 ± 2.6 years. The mean overall HOQ score was 0.69 ± 0.21. The HOQ Cognitive score had a moderate or strong correlation with 19 (90%) of 21 neuropsychological test scores, much more so than the HOQ Social-Emotional score (5 moderate or strong correlations, 24%) and the HOQ Physical score (1 moderate correlation, 5%). For 19 neuropsychological tests (90%), the HOQ Cognitive score had a stronger correlation than the other scores. The HOQ Cognitive score had particularly strong correlations with the Verbal IQ, List Learning, Behavior Problems, and Metacognitive Abilities components. CONCLUSIONS: Data from a wide-ranging representative sample of children with long-standing hydrocephalus provide added evidence of the validity of the HOQ Cognitive score and the overall domain structure of the HOQ itself.

Author self-citation in the general medicine literature.

BACKGROUND: Author self-citation contributes to the overall citation count of an article and the impact factor of the journal in which it appears. Little is known, however, about the extent of self-citation in the general clinical medicine literature. The objective of this study was to determine the extent and temporal pattern of author self-citation and the article characteristics associated with author self-citation. METHODOLOGY/PRINCIPAL FINDINGS: We performed a retrospective cohort study of articles published in three high impact general medical journals (JAMA, Lancet, and New England Journal of Medicine) between October 1, 1999 and March 31, 2000. We retrieved the number and percentage of author self-citations received by the article since publication, as of June 2008, from the Scopus citation database. Several article characteristics were extracted by two blinded, independent reviewers for each article in the cohort and analyzed in multivariable linear regression analyses. Since publication, author self-citations accounted for 6.5% (95% confidence interval 6.3-6.7%) of all citations received by the 328 articles in our sample. Self-citation peaked in 2002, declining annually thereafter. Studies with more authors, in cardiovascular medicine or infectious disease, and with smaller sample size were associated with more author self-citations and higher percentage of author self-citation (all p≤0.01). CONCLUSIONS/SIGNIFICANCE: Approximately 1 in 15 citations of articles in high-profile general medicine journals are author self-citations. Self-citation peaks within about 2 years of publication and disproportionately affects impact factor. Studies most vulnerable to this effect are those with more authors, small sample size, and in cardiovascular medicine or infectious disease.

Quality of life after endoscopic third ventriculostomy and cerebrospinal fluid shunting: an adjusted multivariable analysis in a large cohort.

OBJECT: Quality of life (QOL) studies comparing treatment with endoscopic third ventriculostomy (ETV) and CSF shunting are very limited. The authors compared QOL outcomes following these 2 treatments in a large cohort of children with hydrocephalus by using multivariable statistical techniques to adjust for possible confounder variables. METHODS: The families of children between 5 and 18 years of age with previously treated hydrocephalus at 3 Canadian pediatric neurosurgery centers completed measures of QOL: the Hydrocephalus Outcome Questionnaire (HOQ) and the Health Utilities Index Mark 3 (HUI3). Medical records and recent brain imaging studies were reviewed. A linear regression analysis was performed with the QOL measures as the dependent variable. In multivariable analyses, the authors assessed the independent effect of initial hydrocephalus treatment (ETV vs shunting) while adjusting for the treatment center, current patient age, age at initial treatment, etiology of hydrocephalus, total number of days spent in the hospital for initial treatment, total number of days spent in the hospital for subsequent hydrocephalus complications, functioning ETV at follow-up assessment, frequency of seizures, and current ventricle size. RESULTS: Data from 603 patients were available for analysis. Fifty-eight patients had undergone ETV as their primary treatment and 545 had undergone CSF shunting. Endoscopic third ventriculostomy patients were slightly younger at the follow-up assessment, were older at the first surgery, and spent fewer days in the hospital for hydrocephalus complications. Without adjustment for any confounders, treatment with ETV was associated with significantly higher HOQ physical scores and HUI3 scores. After multivariable adjustment, however, there was no significant difference in any outcome measure. A functioning ETV at the time of the follow-up assessment was not significant in any model. CONCLUSIONS: Treatment with either ETV or CSF shunting does not appear to be associated with any substantial difference in QOL outcome after adjusting for prognostic factors. Further study is needed to definitively determine the relative QOL benefit of either procedure, if any.

Does treatment with endoscopic third ventriculostomy result in less concern among parents of children with hydrocephalus?

PURPOSE: A possible benefit of endoscopic third ventriculostomy (ETV) is that families might harbor less concern and anxiety compared to shunt. This has not yet been demonstrated, however. Our goal was to compare parental concern in a large sample of children with hydrocephalus treated with ETV or shunt, using our previously developed measure of parental concern, the Hydrocephalus Concerns Questionnaire for Parents (HCQ-P). METHODS: The parents of children 5-18 years old with previously treated hydrocephalus at three Canadian pediatric neurosurgery centers completed the HCQ-P. HCQ-P scores were compared between those who were initially treated with ETV and those initially treated with shunt. A multivariable linear regression analysis was used to adjust for center, current age, age at initial hydrocephalus surgery, seizures, etiology, hydrocephalus complications, and quality of life. RESULTS: Six hundred three families participated (58 ETV [9.6%], 545 shunt [90.4%]). In unadjusted comparison, ETV parents had lower overall concern (HCQ-P = 0.41 versus 0.51, p = 0.02). After adjustment for multiple patient factors, ETV parents still had lower concern (p = 0.03) but the only questions for which there was a still a statistically significant difference were those related to concerns about shunt/ETV complications. CONCLUSIONS: Parents of children who have had ETV experience less concern than those who have had shunt and this is due almost exclusively to less concern about hydrocephalus treatment complications. While this could be interpreted as a beneficial aspect of ETV treatment, it remains important for neurosurgeons to ensure that parents are not being overly complacent about the possibility of ETV failure requiring urgent treatment.

Quality of life in obstructive hydrocephalus: endoscopic third ventriculostomy compared to cerebrospinal fluid shunt.

PURPOSE: In the current literature, there are essentially no comparisons of quality of life (QOL) outcome after endoscopic third ventriculostomy (ETV) and shunt in childhood hydrocephalus. Our objective was to compare QOL in children with obstructive hydrocephalus, treated with either ETV or shunt. METHODS: A cross-sectional survey was conducted at SickKids, Toronto of children between ages five and 18 years, with obstructive hydrocephalus due to aqueductal obstruction and no other brain abnormalities. Measures of QOL were the Hydrocephalus Outcome Questionnaire and the Health Utilities Index Mark 3. A subset of patients was given the Wechsler Intelligence Scales for Children (WISC-IV). RESULTS: A total of 47 of 59 (80%) eligible patients participated (24 had ETV as primary treatment, 23 had shunt as primary treatment), with a mean age of 12.1 years (standard deviation 3.9) at assessment. The ETV group was older at initial surgery (p < 0.001) and had larger ventricle size at last follow-up (p = 0.047). In all QOL measures, there were no significant differences between the ETV group and shunt group (all p > or = 0.09). Treatment failure, hydrocephalus complications, and the presence of a functioning ETV at assessment were not associated with QOL differences. Among the 11 children (six ETV, five shunt) who were given the WISC-IV, there were no significant differences between the scores of the ETV group and shunt group (all p > or = 0.11). CONCLUSIONS: This is the first study to provide a meaningful comparison of QOL after ETV and shunt in children. These preliminary results suggest that there is no obvious difference in QOL after ETV and shunt.

Comparisons of citations in Web of Science, Scopus, and Google Scholar for articles published in general medical journals.

CONTEXT: Until recently, Web of Science was the only database available to track citation counts for published articles. Other databases are now available, but their relative performance has not been established. OBJECTIVE: To compare the citation count profiles of articles published in general medical journals among the citation databases of Web of Science, Scopus, and Google Scholar. DESIGN: Cohort study of 328 articles published in JAMA, Lancet, or the New England Journal of Medicine between October 1, 1999, and March 31, 2000. Total citation counts for each article up to June 2008 were retrieved from Web of Science, Scopus, and Google Scholar. Article characteristics were analyzed in linear regression models to determine interaction with the databases. MAIN OUTCOME MEASURES: Number of citations received by an article since publication and article characteristics associated with citation in databases. RESULTS: Google Scholar and Scopus retrieved more citations per article with a median of 160 (interquartile range [IQR], 83 to 324) and 149 (IQR, 78 to 289), respectively, than Web of Science (median, 122; IQR, 66 to 241) (P < .001 for both comparisons). Compared with Web of Science, Scopus retrieved more citations from non-English-language sources (median, 10.2% vs 4.1%) and reviews (30.8% vs 18.2%), and fewer citations from articles (57.2% vs 70.5%), editorials (2.1% vs 5.9%), and letters (0.8% vs 2.6%) (all P < .001). On a log(10)-transformed scale, fewer citations were found in Google Scholar to articles with declared industry funding (nonstandardized regression coefficient, -0.09; 95% confidence interval [CI], -0.15 to -0.03), reporting a study of a drug or medical device (-0.05; 95% CI, -0.11 to 0.01), or with group authorship (-0.29; 95% CI, -0.35 to -0.23). In multivariable analysis, group authorship was the only characteristic that differed among the databases; Google Scholar had significantly fewer citations to group-authored articles (-0.30; 95% CI, -0.36 to -0.23) compared with Web of Science. CONCLUSION: Web of Science, Scopus, and Google Scholar produced quantitatively and qualitatively different citation counts for articles published in 3 general medical journals.

Comparing children's and parents' perspectives of health outcome in paediatric hydrocephalus.

This study exampled the properties of a child-completed version of the Hydrocephalus Outcome Questionnaire (cHOQ) and compared these with parental responses to the HOQ (parent version). This was a cross-sectional study in the outpatient clinics at three Canadian paediatric hospitals (Toronto, Vancouver, and Halifax). All cognitively-capable children with previously treated hydrocephalus who were aged between 6 and 19 years were eligible. Parents completed the HOQ and the Health Utilities Index Mark 3; children completed the cHOQ. A total of 273 children participated (146 males, 127 females; mean age 14 y 1 mo, SD 2 y 7 mo). Internal consistency of the cHOQ was 0.93 and test-retest reliability was 0.86 (95% confidence interval 0.78-0.92). Mother-child agreement and father-child agreement were 0.57 (0.40-0.68) and 0.62 (0.48-0.73) respectively. Agreement was higher for assessments of physical health, but lower for assessments of cognitive health and social-emotional health. There was greater parent-child agreement for older children. When there was disagreement, it seemed that children tended to rate their health better than their parents did. In older children with hydrocephalus, the cHOQ appears to be a scientifically reliable means of assessing long-term outcome. The differences in child and parent perceptions of health need to be appreciated when conducting outcome studies in this population.

Medical, social, and economic factors associated with health-related quality of life in Canadian children with hydrocephalus.

OBJECTIVE: To study the factors associated with health-related quality of life (HRQL) in Canadian children with hydrocephalus, using a comprehensive model of determinants of child health, including socioeconomic factors. STUDY DESIGN: A cross-sectional study was performed between November 2005 and November 2006 at 3 Canadian pediatric hospitals. Parents of children with hydrocephalus age 5 to 18 years completed the Hydrocephalus Outcome Questionnaire (HOQ) and the Health Utilities Index Mark 3 (HUI-3). RESULTS: A consecutive sample of 340 subjects participated from a total of 366 eligible children (mean age, 11.6 +/- 3.6 years; mean time from the diagnosis of hydrocephalus, 10.0 +/- 4.6 years). Adjusted multivariate linear regression models demonstrated that the most important determinants of poorer HRQL included lower family income, lower parental education, worse family functioning, seizures, myelomeningocele, and prolonged treatment for cerebrospinal fluid shunt obstruction. CONCLUSIONS: Despite a national universal health care system, socioeconomic disparities remain important as determinants of HRQL. Given the absence of a parallel private health care system in Canada, this suggests that the impact of socioeconomic factors is related to issues other than access to care.

Characteristics associated with citation rate of the medical literature.

BACKGROUND: The citation rate for articles is viewed as a measure of their importance and impact; however, little is known about what features of articles are associated with higher citation rate. METHODOLOGY/PRINCIPAL FINDINGS: We conducted a cohort study of all original articles, regardless of study methodology, published in the Lancet, JAMA, and New England Journal of Medicine, from October 1, 1999 to March 31, 2000. We identified 328 articles. Two blinded, independent reviewers extracted, in duplicate, nine variables from each article, which were analyzed in both univariable and multivariable linear least-squares regression models for their association with the annual rate of citations received by the article since publication. A two-way interaction between industry funding and an industry-favoring result was tested and found to be significant (p = 0.02). In our adjusted analysis, the presence of industry funding and an industry-favoring result was associated with an increase in annual citation rate of 25.7 (95% confidence interval, 8.5 to 42.8) compared to the absence of both industry funding and industry-favoring results. Higher annual rates of citation were also associated with articles dealing with cardiovascular medicine (13.3 more; 95% confidence interval, 3.9 to 22.3) and oncology (12.6 more; 95% confidence interval, 1.2 to 24.0), articles with group authorship (11.1 more; 95% confidence interval, 2.7 to 19.5), larger sample size and journal of publication. CONCLUSIONS/SIGNIFICANCE: Large trials, with group authorship, industry-funded, with industry-favoring results, in oncology or cardiology were associated with greater subsequent citations.

Quality of life in children with hydrocephalus: results from the Hospital for Sick Children, Toronto.

OBJECT: Children with hydrocephalus face several quality of life (QOL) issues that have been poorly studied. The authors' aim was to quantify the QOL for children with hydrocephalus and identify predictors of long-term outcome, using a reliable and validated outcome measure: the Hydrocephalus Outcome Questionnaire (HOQ). METHODS: All children (5-18 years old) with treated hydrocephalus attending the neurosurgery outpatient clinic at the Hospital for Sick Children were asked to participate. The patient's QOL was measured by the parent-completed HOQ. Predictor variables were extracted from the medical records. Multivariable linear regression was used to identify those predictor variables that were significantly associated with outcome. RESULTS: There was an 89% participation rate, with a total of 346 children participating (mean age 11.7 years, mean duration since diagnosis 9.9 years). Their mean HOQ Overall Health score was 0.68 (on a scale of 0 [worst QOL] to 1.0 [best QOL]). On multivariable analysis, the following predictors were associated with a worse overall QOL: increased seizure frequency, increased length of stay (LOS) in the hospital for the initial treatment of hydrocephalus, increased LOS for treatment of shunt infection and shunt overdrainage, increased number of proximal shunt catheters in situ, and increased distance of the family residence from the pediatric neurosurgical center. CONCLUSIONS: For the first time, these results establish baseline QOL values for a typical large group of children many years after their diagnosis of hydrocephalus, by using a validated and reproducible outcome measure. Many of the factors that adversely impact QOL appear to be related to shunt complications and might, therefore, be modifiable.