Akiko Higuchi

Development of the Japanese version of the Pediatric Quality of Life Inventory™ Brain Tumor Module

BackgroundThe Pediatric Quality of Life Inventory™ (PedsQL™) is a widely-used modular instrument for measuring health-related quality of life in children aged 2 to 18 years. The PedsQL™ Brain Tumor Module is comprised of six scales: Cognitive Problems, Pain and Hurt, Movement and Balance, Procedural Anxiety, Nausea, and Worry. In the present study, we developed the Japanese version of the PedsQL™ Brain Tumor Module and investigated its feasibility, reliability, and validity among Japanese children and their parents.MethodsTranslation equivalence and content validity were verified using the standard back-translation method and cognitive debriefing tests. Participants were recruited from 6 hospitals in Japan and the Childrens Cancer Association of Japan, and questionnaires were completed by 137 children with brain tumors and 166 parents. Feasibility of the questionnaire was determined based on the amount of time required to complete the form and the percentage of missing values. Internal consistency was assessed using Cronbachs coefficient alpha. Test-retest reliability was assessed by retesting 22 children and 27 parents. Factorial validity was verified by exploratory factor analyses. Known-groups validity was described with regard to whole brain irradiation, developmental impairment, infratentorial tumors, paresis, and concurrent chemotherapy. Convergent and discriminant validity were determined using Generic Core Scales and State-Trait Anxiety Inventory for children.ResultsInternal consistency was relatively high for all scales (Cronbachs coefficient alpha > 0.70) except the Pain and Hurt scale for the child-report, and sufficient test-retest reliability was demonstrated for all scales (intraclass correlation coefficient = 0.45-0.95). Factorial validity was supported through exploratory factor analysis (factor-item correlation = 0.33-0.96 for children, 0.55-1.00 for parents). Evaluation of known-groups validity confirmed that the Cognitive Problems scale was sensitive for developmental impairment, the Movement and Balance scale for infratentorial tumors or paresis, and the Nausea scale for a patient currently undergoing chemotherapy. Convergent and discriminant validity with the PedsQL™ Generic Core Scales and State-Trait Anxiety Inventory for children were acceptable.ConclusionsThe Japanese version of the PedsQL™ Brain Tumor Module is suitable for assessing health-related quality of life in children with brain tumors in clinical trials and research studies.

Factors influencing self- and parent-reporting health-related quality of life in children with brain tumors

PurposeHealth-related quality of life (HRQOL) is not only a degree of health but also reflects patient perceptions and expectations of health. For children with brain tumors, better understanding of HRQOL requires the use of complementary reports from parents and interviewer-administered reports for children. Here, we aimed to test whether or not the trait anxiety of children and the psychological distress of their parents influence children’s and parents’ responses to HRQOL questionnaires, and whether or not the report-administration method for children influences children’s responses to HRQOL questionnaires.MethodsOne hundred and thirty-four children aged 5–18 with brain tumors and one of their parents completed the Pediatric Quality of Life Inventory™ (PedsQL™) Brain Tumor Module questionnaires. In addition, the children also completed the State-Trait Anxiety Inventory for Children (STAIC), and the parents also completed the Kessler-10 (K10) and health and sociodemographic characteristics questionnaires. The child questionnaires were administered either by the child (self-administered) or an interviewer. Rater-dependent perceptions about HRQOL were derived from the subscales scores of the PedsQL™ Brain Tumor Module using structural equation modeling based on a multitrait-multimethod model. The STAIC trait-anxiety score, K10 score, report-administration method, and other health and sociodemographic factors related to each child’s or parent’s perceptions were identified through multiple linear regression analyses of the questionnaire responses. We used a path analysis to estimate the change in a PedsQL™ child-reported score that occurs when interviewer-administration changes the child’s perception about HRQOL.ResultsSurveys for 89 children were self-administered while those for 45 were interviewer-administered. The perceptions of the children and parents were calculated by fitting data to the model (chi-squared Pxa0=xa00.087, normed fit indexxa0=xa00.932, comparative fit indexxa0=xa00.978, standardized root mean squared residualxa0=xa00.053, and root mean square error of approximationxa0=xa00.054). The children’s perception of HRQOL was affected by their STAIC trait-anxiety score (bxa0=xa0−0.43, 95% CI [−0.60, −0.25]). The parent’s perception was affected by their child’s treatment status (bxa0=xa00.26, 95% CI [0.09, 0.43]), the parent’sxa0K10 score (bxa0=xa0−0.21, 95% CI [−0.37, −0.04]), and by education level (bxa0=xa00.17, 95% CI [0.00, 0.34]). The change in the child-reported PedsQL™ score in relation to the method of administration ranged from −1.1 (95% CI: −3.5, 1.3) on the procedural anxiety subscale to −2.5 (95% CI: −7.6, 2.6) on the movement and balance subscale.ConclusionChild-reporting of HRQOL is little influenced by the method of administration. Children’s perception about HRQOL tended to be influenced by their trait anxiety, while parents’ perception was influenced by their psychological distress, academic background, and their child’s treatment status.

Impact of late effects on health-related quality of life in survivors of pediatric brain tumors: motility disturbance of limb(s), seizure, ocular/visual impairment, endocrine abnormality, and higher brain dysfunction.

Background: Survivors of pediatric brain tumors are often affected by late effects, such as motility disturbance of limb(s), seizure, ocular/visual impairment, endocrine abnormality, and higher brain dysfunction, resulting from the disease and its treatment. Appropriate provision of supportive care will require understanding the effects of these experiences on survivors’ health-related quality of life (HRQOL). Objective: The aim of this study was to identify the relationships between late effects and specific aspects of the HRQOL of pediatric brain tumor survivors. Methods: We distributed questionnaires for measuring HRQOL to 138 survivors and their parents at 8 hospitals and 1 clinic in Japan and simultaneously surveyed late effects using information provided by the survivors’ attending physicians. We compared the HRQOL of survivors with and survivors without specific late effects. Results: A total of 106 survivors and their parents returned the questionnaires to the researchers. The HRQOL of survivors 18 years or older was negatively affected by all 5 late effects, indicating that their higher impairment was associated with diminished HRQOL. The HRQOL of survivors aged 12 to 17 years was negatively affected by 2 late effects (ocular/visual impairment and motility disturbance of the limbs). A part of the HRQOL subdomain (motor and cognitive functioning) of survivors aged 12 to 17 years was positively related to ocular/visual impairment. Conclusions: Five late effects influenced the HRQOL of pediatric brain tumor survivors. Implications for Practice: Nurses and other health professionals should provide specific care designed to support aspects of HRQOL affected by late effects. For example, survivors with ocular/visual impairment may be expected to require additional emotional support, and those with seizures or endocrine abnormalities may be expected to require additional support for sleep disorders.

Cancer-specific health-related quality of life in children with brain tumors

PurposeTo understand the influence of disease and treatment on the health-related quality of life (HRQOL) of children with brain tumors, compared to the HRQOL of children with other cancers, from the viewpoints of children and parents.MethodsA total of 133 children aged 5–18xa0years and 165 parents of children aged 2–18 completed questionnaires of the Pediatric Quality of Life Inventory Cancer Module (Pain and Hurt, Nausea, Procedural Anxiety, Treatment Anxiety, Worry, Cognitive Problems, Perceived Physical Appearance, and Communication scales); higher scores indicate a better HRQOL. The Cancer Module scores, weighted by age and treatment status, were compared to those obtained in a previous study of children with other cancers (mostly leukemia).ResultsThe weighted mean scores for Pain and Hurt (effect size dxa0=xa00.26) and Nausea (dxa0=xa00.23) from child reports and the scores for Nausea (dxa0=xa00.28) from parent reports were higher for children with brain tumors than scores for children with other cancers. The scores for Procedural Anxiety (dxa0=xa0−0.22) and Treatment Anxiety (dxa0=xa0−0.32) from parent reports were lower for parents of children with brain tumors than the scores for parents of children with other cancers. The child-reported Pain and Hurt score of the Cancer Module was higher (dxa0=xa00.29) and in less agreement (intraclass correlation coefficientxa0=xa00.43) with scores from the Brain Tumor Module, indicating that assessments completed with the Cancer Module misesteem pain and hurt problems in children with brain tumors.ConclusionsThe profiles of cancer-specific HRQOL in children with brain tumors differ from those of children with other cancers; we therefore suggest that these children receive specific psychological support.

Correction to: Current situation of cancer among adolescents and young adults in Japan

In the original publication, Tablesxa04 and 5 have not been published in a readable format. The corrected clear version is given in this Correction.

Employment status and termination among survivors of pediatric brain tumors: a cross-sectional survey

BackgroundSome childhood cancer survivors experience employment difficulties. This study aimed to describe pediatric brain-tumor survivors’ employment status.MethodsA cross-sectional, observational study was conducted, with questionnaires distributed to 101 pediatric brain-tumor survivors (aged 15xa0years or older) and their attending physicians from nine institutions in Japan. We compared category and time-series histories for participants’ first-time employment using national census information. Factors related to delayed employment or early employment termination were examined using survival-time analyses.ResultsExcluding students and homemakers, 38 brain-tumor survivors (median age 27xa0years, with 15xa0years since diagnosis) were of working age. Of these, 12 (32%) were unemployed and 9 (24%) had never been employed. First-time employment occurred later for brain-tumor survivors than the general population, particularly in those with lower educational levels. The number of brain-tumor survivors whose first job was terminated within the first year was higher than that for the general population, particularly in male survivors and germ cell-tumor survivors. Brain-tumor survivors described their working patterns (irregular), job types (specialist or professional), reasons for early termination (unsuitable job), and thoughts about working (they wished to serve their communities but lacked confidence).ConclusionBrain-tumor survivors are associated with high unemployment rates and multiple unemployment-related factors. Education and welfare systems should identify individual methods of social participation for this group.

Current situation of cancer among adolescents and young adults in Japan

BackgroundCancer is rare among adolescents and young adults (AYA). Affected persons need generation-specific attention and care; however, no nationwide study has investigated the medical care structure for AYA cancer treatment in Japan.MethodsWe conducted a nationwide survey of AYA cancer for frequency of AYA patients, type of cancer, medical facilities, and certified cancer professionals. Data were collected from 14,713 patients at 218 Core Cancer Treatment Hospitals.ResultsThe average proportion of AYA cancer patients to all cancer patients was 3.6%. The median number of patients aged 15 to 24xa0years per hospital was small (nu2009=u20095, range 1–51). The most frequent primary site of AYA cancer was the cervix uteri, but when cancer in situ was excluded, the hematopoietic malignancies were the most frequent cancer in males and females aged 15–24xa0years. In the age group 25–39 years, testicular and breast cancers were the most frequent cancers in males and females, respectively. Certified cancer professionals and facilities are necessary for appropriate care of AYA cancer patients, but the availability of such professionals varied greatly among hospitals. Hospitals with few AYA cancer patients were less likely to employ such physicians.ConclusionsThe present findings suggest that medical care for AYA cancer in Japan requires further refinement and a multidisciplinary approach.

iMusta4SLC: Database for the structural property and variations of solute carrier transporters

Membrane transporter proteins play important roles in transport of nutrients into the cell, in transport of waste out of the cell, in maintenance of homeostasis, and in signal transduction. Solute carrier (SLC) transporter is the superfamily, which has the largest number of genes (>400 in humans) in membrane transporter and consists of 52 families. SLC transporters carry a wide variety of substrates such as amino acids, peptides, saccharides, ions, neurotransmitters, lipids, hormones and related materials. Despite the apparent importance for the substrate transport, the information of sequence variation and three-dimensional structures have not been integrated to the level of providing new knowledge on the relationship to, for instance, diseases. We, therefore, built a new database named iMusta4SLC, which is available at http://cib.cf.ocha.ac.jp/slc/, that connected the data of structural properties and of pathogenic mutations on human SLC transporters. iMusta4SLC helps to investigate the structural features of pathogenic mutations on SLC transporters. With this database, we found that the mutations at the conserved arginine were frequently involved in diseases, and were located at a border between the membrane and the cytoplasm. Especially in SLC families 2 and 22, the conserved residues formed a large cluster at the border. In SLC2A1, one third of the reported pathogenic missense mutations were found in this conserved cluster.