Agreement between self-, mother and father proxy-reports on health-related quality of life in adolescents with Tourette syndrome.

This study aimed to investigate agreement and discrepancies between parent proxy- and adolescent self-reports on assessments of adolescents' health-related quality of life (HRQoL), and the role that individual factors may play in parent-adolescent agreement, in a sample of adolescents with Tourette syndrome (TS) compared to a control group of healthy adolescents. Adolescents aged 12-18 years diagnosed with TS were recruited with their parents from primary and secondary referral centres. Adolescent healthy controls were matched for gender and age. Adolescents and each of their parents completed a set of questionnaires including a HRQoL evaluation of adolescent, the 'Vécu et Santé Perçue de l'Adolescent'. Mother-adolescent, father-adolescent and mother-father agreements on adolescents' HRQoL scores were investigated at individual and group level, both in TS and control groups. Data were available for 75 adolescents, 75 mothers and 63 fathers, in the TS group. Agreement between mother, father proxy-reports and TS adolescents self-reports of HRQoL varied from poor to good, without significant difference with the control group. In TS group, mothers and fathers underestimated adolescents' HRQoL in 'Psychological well-being' subscale and mothers underestimated it in 'Physical 'well-being' subscale, while controls overestimated adolescents' HRQoL in these subscales. Larger mother-adolescent discrepancies for 'Psychological well-being' and 'Physical well-being' subscales were associated with internalizing symptoms. Regarding future studies, comprehensive evaluation of the various dimensions of adolescents' HRQoL with TS requires the integration of the perspectives of both adolescents, mothers and fathers. Clinicians should take into account this point to provide comprehensive care and services.

Cross-cultural translation, reliability and validity of the Thai version of the Patient­Reported Outcomes Measurement Information System (PROMIS) Parent Proxy Upper Extremity Short Form 8a in children with congenital upper extremity anomalies.

BACKGROUND: The PROMIS Parent Proxy Upper Extremity Short Form 8a version 2 (PROMIS Parent Proxy UE-SF) is one of the most commonly used self-assessment questionnaires for evaluating function in children with congenital upper extremity anomalies. However, this English questionnaire is difficult for Thai parents to complete. The purpose of this study is to translate the PROMIS Parent Proxy UE-SF into Thai and test its reliability and validity. METHODS: The PROMIS Parent Proxy UE-SF was translated into Thai using FACIT translation methodology. This version and the Thai version of the Michigan Hand Questionnaire (Thai-MHQ) were used to evaluate 30 Thai children with different types of congenital upper extremity anomalies. The reliability and validity of the Thai-PROMIS Parent Proxy UE-SF were evaluated by test-and-retest with the intraclass correlation coefficient (ICC) and Cronbach's alpha coefficient. Correlations between the Thai-PROMIS Parent Proxy UE-SF and Thai-MHQ were analysed by Pearson's correlation coefficients. RESULTS: The children's mean age was 4.47 ± 2.08 years (range 1-9 years). The main diagnoses included thumb duplication (11 children), syndactyly (4 children)4, congenital trigger thumb (3 children) and obstetric brachial plexus palsy (3 children). The children's parents completed the questionnaires, taking 164.23 ± 22.58 s for the Thai-PROMIS and 337.8 ± 49.37 s for the Thai-MHQ. The test-retest reliability of Thai-PROMIS evaluated by ICCs, was 0.9909 (good reliability), and the Cronbach's alpha of all items was 0.923. The Pearson's correlation coefficient between the Thai-PROMIS and Thai-MHQ showed a strong correlation with Domain 2 (activities of daily living, r = 0.7432) and a moderate correlation with the overall Thai-MHQ score (r = 0.699). CONCLUSIONS: The Thai-PROMIS Parent Proxy UE-SF is a valid, reliable and easy-to-use patient-reported outcome measure for assessing function in children with congenital upper extremity anomalies.

Differences in health-related quality of life in children with chronic kidney disease as reported by children and parent proxies.

BACKGROUND: Correlation between reports of children and parent for health-related quality of life (HRQOL) is not well studied. This study aims to assess the degree of agreement between child self- and parent proxy-rated HRQOL and to identify factors associated with discordance at baseline and during follow-up in Taiwanese children with chronic kidney disease (CKD). METHODS: This study includes pediatric patients aged 5-18 years with confirmed CKD. Participants completed the generic version of the Pediatric Quality of Life Inventory (PedsQL) at baseline and every 6 months during follow-up. Child-parent agreement on HRQOL reports was assessed using intraclass correlation coefficient (ICC). Multivariate regression models were used to determine factors associated with child-parent discordance. RESULTS: Of the 112 child-parent dyads included in the analysis, 97 dyads with 640 patient visits were assessed in 4.5 years. Children reported higher total scores on the physical and psychosocial domains as compared to their parent proxies. ICC was low (< 0.5) for the psychosocial domain and moderate for the physical health domain at initial assessment and slightly increased for the physical health (0.62) and for school functioning (0.51) during follow-up. Development of mineral bone disorder/anemia (ß, 11.75 [3.77-19.72]) and proteinuria (ß, 8.48 [1.15-15.81]) in the follow-up were associated with increased discordance in school functioning, and fathers with chronic disease were associated with increased discordance in social functioning (ß, 4.21 [0.68-7.74]). CONCLUSIONS: Parent proxy consistently estimated lower PedsQL score compared to self-reports of children. Child self-rated psychosocial health domains should be evaluated whenever possible to better elucidate treatment outcome over time. A higher resolution version of the Graphical abstract is available as Supplementary information.

Validation of the simplified Chinese version of PROMIS Parent Proxy-25 Profile in parents of children with cancer.

PURPOSE: To determine the psychometric properties of the Patient-reported Outcomes Measurement Information System Parent Proxy-25 Profile (PROMIS-25) in a sample of Chinese parents of children with cancer. DESIGN AND METHODS: A cross-sectional sample (N = 148) of parents with children aged 5-17 years living with cancer was recruited. Each participant completed sociodemographic and clinical questionnaires and PROMIS-25. The flooring and ceiling effects were calculated. Reliability was determined by the Cronbach's alpha and split-half coefficient. Factor structure was examined by factor analysis. Model fit and graphical plots were assessed to test the assumptions of Rasch model-based item response theory (IRT). Differential item functioning (DIF) was assessed in terms of gender, age, and treatment stage. RESULTS: PROMIS-25 demonstrated some flooring and ceiling effect, excellent reliability (Cronbach's α > 0.7 for all six domains), and the six-domain factor structure was supported. The IRT assumptions were met in terms of unidimensionality, local independence, monotonicity, and measurement equivalence with acceptable DIF in terms of gender, age, diagnosis, and treatment stage. CONCLUSION: PROMIS-25 is a highly reliable and valid instrument for evaluating children with cancer that assesses important health-related quality of life domains of child cancer. PRACTICE IMPLICATION: Chinese parents of children with cancer and healthcare providers could use PROMIS-25 to assess the symptoms of children.

Validation of the Sri Lankan Health-Related Quality-of-Life Index for pre-schoolers (SLHQL-P) and school age (SLHQL-S) children with epilepsy.

INTRODUCTION: Health-related quality of life (HRQL) in children and adolescents remains an underappreciated problem in Asian region, due to lack of culturally sound HRQL measures. This paper describes the validation process of two age-specific, proxy-rated HRQL indexes namely Sri Lankan Health-Related Quality-of-Life Index for preschoolers (SLHQL-P) and school age children (SLHQL-S) with epilepsy. METHODS: Primary caregivers of children and adolescents with epilepsy aged 1-18 years from three districts in Sri Lanka were invited to fill the SLHQL-P (for 1-5 years) and SLHQLS (for 6-18 years). It was re-administered to a subset of consenting primary caregivers after an interval of two weeks. Scientific soundness of SLHQL-P and SLHQL-S were established based on causal indicator model. RESULTS: Total of 98 and 169 primary caregivers responded to SLHQL-P, SLHQL-S respectively. Final questionnaires of SLHQL-P and SLHQL-S consists of 26, 57 items respectively in three multi-item domains measuring the child's physical functioning (6, 9 items), psychological functioning (6, 31 items) and social functioning (14, 17 items) with higher scores reflecting better HRQL. Construct validity was established by several methods including strong relationship with seizure severity. Good test-retest reliability was also demonstrated for both SLHQL-P and SLHQL-S (r = 0.77, 0.938) CONCLUSIONS: Both SLHQL-P and SLHQL-S are feasible, reliable and valid instruments to measure HRQL in children and adolescents with epilepsy in clinical as well as research settings in Sri Lanka.

The role of parental health and distress in assessing children's health status.

PURPOSE: The purpose of the study was to examine the contributions of parents' health and distress to parent's and children's assessments of children's health. METHODS: We used baseline data from a longitudinal study of 364 children (ages 4-12) about to undergo surgery and their parents in a Southern California pediatric hospital. We used the 20-item child self-reported CHRIS 2.0 general health and the parallel parent-reported measure of the child's health, along with a measure of parental distress about the child's health were administered in the perioperative period. Other measures included parents' physical and mental health, quality of life, distress over their child's health, and number and extent of other health problems of the child and siblings. RESULTS: On average, parents' reports about the child were consistently and statistically significantly higher than children's self-reports across all sub-dimensions of the CHRIS 2.0 measure. Parents' personal health was positively associated with their reports of the child's health. More distressed parents were closer to the child's self-reports, but reported poorer personal health. CONCLUSION: Parent-child differences in this study of young children's health were related to parental distress. Exploring the nature of the gap between parents and children in assessments of children's health could improve effective clinical management for the child and enhance family-centered pediatric care. Future studies are needed to assess the generalizability of CHRIS 2.0 to other health settings and conditions and to other racial/ethnic groups.

The effects of child anxiety and depression on concordance between parent-proxy and self-reported health-related quality of life for pediatric liver transplant patients.

BACKGROUND: HRQOL is a key outcome following pediatric LT. Parent-proxy reports may substitute for patients unable to report their own HRQOL. This study compared parent-proxy and self-reported HRQOL in children who have undergone LT. METHODS: Pediatric LT recipients between the ages of 8 and 18 years, and a parent, completed self and proxy versions of the PeLTQL questionnaire, PedsQL Generic and Transplant modules, and standardized measures of depression and anxiety. RESULTS: Data from 129 parent-patient dyads were included. Median parent age was 44 years, and most (89%) were mothers. Median patient age was 2.5 years at LT and 13.6 years at the time of study participation. Parents had significantly lower scores than patients on PedsQL total generic (70.8 ± 18.5 and 74.3 ± 19.0, p = .01), PeLTQL coping and adjustment (63.0 ± 15.6 and 67.3 ± 16.2, p < .01), and social-emotional (66.3 ± 14.9 and 71.9 ± 15.6, p < .001) domains. Higher patient anxiety and depression were related to larger absolute differences between parent-proxy and self-reported scores on all HRQOL measures (all p < .05). In this disparity, parents reported higher HRQOL scores than their child as self-reported anxiety and depression scores increased. CONCLUSIONS: Differences in concordance between parent-proxy and self-reported HRQOL scores can be more prominent when children have more symptoms of anxiety and depression. Children's mental health symptoms should be queried, if feasible, when interpreting differences in parent and child reports of HRQOL.

Evaluation of the TRxANSITION Index-Parent Version for Assessment of Readiness to Transition to Adult Care Among Youth with Chronic Conditions.

PURPOSE: To develop and evaluate a parent-proxy measure of youth HCT readiness: the TRxANSITION Index-Parent Version. DESIGN AND METHODS: We recruited parents (77% female) and youth (ages 12 to 25) to complete transition readiness measures during outpatient clinic visits. The TRxANSITION Index-Parent Version contains two domains: the Parent Knowledge Domain assessing a parent's knowledge of their youth's illness, and the Parent Proxy Domain, which provides a parental perspective regarding a youth's transition readiness skills. We evaluated the TRxANSITION Index - Parent Version for differences between parent and youth reports of HCT readiness, associations between parent's score and youth's characteristics, and item-category, item-sub-index, and sub-index category correlations. RESULTS: Data from 93 parents-youth dyads were analyzed. Parents scored significantly higher than youth in the Parent Knowledge Domain and similarly in the Parent Proxy Domain. Parents of daughters had significantly higher scores in the Parent Knowledge Domain than parents of sons and reported similar scores to Parents of sons in the Parent Proxy Domain. Only the self-management sub-index significantly correlated with youth's age. The sub-index-domain, item-sub-index, and item-domain correlations assessed were generally large in magnitude (r > 0.5). CONCLUSIONS: The TRxANSITION Index-Parent Version shows promise as a means of assessing parent knowledge of a youth's illness and may provide an accurate proxy assessment of a youth HCT readiness skills. PRACTICE IMPLICATIONS: Obtaining parental perspective on a youth's HCT readiness may provide useful clinical information during the transition process.

Self-reported quality of life and degree of youth-parent agreement: A long-term follow-up of childhood-onset epilepsy.

OBJECTIVE: To prospectively delineate self-reported health-related quality of life (HRQOL) of adolescents and young adults (AYAs) 8 and 10 years after an epilepsy diagnosis and evaluate the degree of AYA-parent agreement in ratings of AYA's HRQOL. METHODS: Data came from the Health-Related Quality of Life in Children with Epilepsy Study (HERQULES), a 10-year longitudinal study of children, aged 4-12 years, with newly diagnosed epilepsy. Epilepsy-specific HRQOL was self-reported by AYA 8 and 10 years after diagnosis and by parents at multiple time points throughout the 10-year follow-up. Measurers of HRQOL over time were analyzed using a linear mixed-effect model approach. AYA-parent agreement was evaluated using intraclass correlation coefficient (ICC) and Bland-Altman plots. RESULTS: A total of 165 AYAs participated at long-term follow-up. There was considerable heterogeneity among AYA's HRQOL, and as a group, there was no significant change in HRQOL from the 8- to 10-year follow-up. Household income at the time of diagnosis, seizure control at follow-up, and a history of emotional problems (anxiety/depression) were independent predictors of HRQOL at follow-up. AYA-parent agreement on AYA's HRQOL was moderate (ICC 0.62, 95% CI 0.51-0.71), although considerable differences were observed at the individual level. AYA-parent agreement varied with AYA's and parent's age, seizure control, and family environment. SIGNIFICANCE: In the long-term after a diagnosis of epilepsy, AYAs report stable HRQOL over time at the group level, although notable individual differences exist. Seizure control, anxiety/depression, and family environment meaningfully impact AYA's long-term HRQOL. AYA and parent reports on HRQOL are similar at the group level, although they cannot be used interchangeably, given the large individual differences observed.

Self-reported and parent proxy reported functional impairment among pediatric cancer survivors and controls.

BACKGROUND: A unique and limiting component in the research on functional impairment among children has been the exclusive use of parent proxy reports about child functioning; and there is limited information regarding the impact of pediatric cancer treatment on children's day-to-day functioning and how this is related to neurocognitive functioning. The objective of the current study was to examine a novel measure of self-reported functional impairment, and explore the relationship between self-reported and parent-reported child functional impairment in pediatric cancer survivors compared to controls. METHODS: A cross-sectional cohort of survivors (n = 26) and controls (n = 53) were recruited. Survivors were off treatment an average of 6.35 years (SD = 5.38; range 1-15 years) and demonstrated an average "medium" Central Nervous System treatment intensity score. Participants completed measures of functional impairment (FI), intellectual assessment (RIST) and executive functions (NIH Examiner), while parents reported on children's functional impairment. RESULTS: Survivors were similar to controls in functional impairment. Regardless of group membership, self-reported FI was higher than parent-reported FI, although they were correlated and parent report of FI significantly predicted self-reported FI. Across groups, increased impairment was associated with four of seven Examiner scores. CONCLUSIONS: Research regarding self-reported functional impairment of cancer survivors and its association with parent-reported functional impairment and neurocognitive deficits has been limited. Our results suggest that self-reported FI appears to be a reasonable and viable outcome measure that corresponds with and adds incremental validity to parent reported FI. While low treatment intensity may confer relative sparing of functional impairment among survivors, children report higher FI levels than parents, suggesting that FI can be of clinical utility. In conclusion, pediatric cancer survivors should be screened for self-reported functional difficulties.

Agreement Between Parent Proxy Reports and Self-Reports of Adolescent Emotional Distress.

Parents' recognition of adolescents' emotional distress is a significant determinant of early detection and treatment of mental disorders. However, there is dearth of research exploring parent-adolescent agreement regarding adolescents' emotional distress. This cross-sectional, school-based study compared parents' proxy reports and self-reports of adolescent's emotional distress among 289 parent-adolescent dyads in Korea. Findings revealed low agreement between adolescents' and parents' reports of depression, anxiety, and anger, with an average polychoric r of .25 to .27. The agreement was particularly low for high school students, boys, and father-adolescent dyads. Additionally, parents tended to underestimate adolescents' emotional distress symptoms; a significant percentage of adolescents experiencing symptoms were rated in the normal range by parents, particularly high school students experiencing anger. Interventions are needed to help adolescents learn to manage and express their negative emotions. Moreover, parent education programs that improve parents' recognition of emotional distress and appropriate help-seeking behaviors are needed.

Parent Proxy Discrepancy Groups of Quality of Life in Childhood Epilepsy.

OBJECTIVES: To study the extent to which parents are able to serve as true proxies for their children with epilepsy using a more granular approach than has been found in any study to date. METHODS: Proxy resemblance to the child was based on discrepancy in z-centered child minus parent scores of matching quality-of-life (QOL) domains for 477 dyads. Latent class mixed models (LCMMs) were built, with child's age as the independent variable for epilepsy-specific and generic QOL. Data were obtained from the QUALITÉ Canadian cohort, which recruited children with epilepsy aged 8 to 14 years at baseline and their parents. RESULTS: Both epilepsy-specific and generic LCMMs produced latent classes representing proxies that were overly positive, overly negative, or in agreement relative to their children with posterior probabilities of 79% to 84%. The "agreement" classes had N = 411 and N = 349 in the epilepsy-specific and generic LCMMs, respectively. The epilepsy-specific LCMM had a small unique class of N = 5 with a posterior probability of 88% called "growing discrepancy." CONCLUSIONS: Most parents of children with epilepsy can serve as valid proxies for their children on QOL scales. Poorer parental adaptation is more related to overly negative proxies, whereas low peer support from the child's perspective is more related to overly positive proxies.

Assessing the measurement invariance of the RCADS-25 questionnaire across gender and child-parent dyads in the presence of multilevel data.

PURPOSE: This study aims to assess the measurement invariance of the short version of the Revised Child Anxiety and Depression Scale (RCADS-25) across male and female adolescents along with child self-reports and parent proxy-reports. METHODS: The sample consisted of 2039 adolescents and their parents from 51 public and private schools in Shiraz (southern Iran). The measurement invariance of the RCADS-25 was investigated by the multilevel multiple-indicators multiple-causes model. In addition, the reliability of the questionnaire was examined by Cronbach's alpha coefficient, and its validity was evaluated by exploratory and categorical confirmatory factor analysis. RESULTS: Although the RCADS-25 includes five (20%) non-invariant items across male and female adolescents, their impact is negligible at the scale level. In addition, 10 out of 25 items (40%) were non-invariant across self- and proxy-reports, which did not have substantial effect at the scale level. CONCLUSION: Based on our findings, the instrument can be used, albeit with caution, for comparing depression and anxiety scores between male and female adolescents as well as child self-reports and parent proxy-reports.

Longitudinal study of quality of life among children with acute respiratory infection and cough.

PURPOSE: Acute respiratory infections (ARIs), and associated symptoms such as cough, are frequently experienced among children and impose a burden on families (e.g., use of medical resources and time off work/school). However, there are little data on changes in, and predictors of, quality of life (QoL) over the duration of an ARI with cough (ARIwC) episode. We therefore aimed to determine cough-specific QoL and identify its influencing factors among children with ARIwC, at the time of presentation to a pediatric emergency department (ED), and over the following 4 weeks. METHODS: Data from 283 children aged < 15 years were included in our analyses. We used the validated parent-proxy children's acute cough-specific QoL questionnaire (PAC-QoL) at each time-point. Linear regression and mixed effect modeling were used to identify factors influencing QoL at baseline and over the follow-up period. RESULTS: Median PAC-QoL at baseline was 2.7 (IQR 2.1-3.6) and significantly improved by Day-7 (4.9, IQR 3.8-6.1) and Day-14 (6.59, IQR 5.1-7.0), both p < 0.001. The improvements in median PAC-QoL between Days-14, -21, and -28 were not significant. Regression modeling identified that day-cough severity, night-cough severity, and financial concerns had the highest impact on both baseline, and follow-up, PAC-QoL scores. There were five additional independent significant factors at baseline and six at follow-up. CONCLUSIONS: Quality of life is considerably impaired at presentation to ED, but improves significantly by Days-7 and -14. As cough severity and financial concerns had the highest impact on QoL, effectively managing cough to reduce the clinical and financial burden on children and families is important.

Social adjustment and repressive adaptive style in survivors of pediatric cancer.

PURPOSE: The aim of the study was to explore the relationship between repressive adaptive style and self-reports of social adjustment in survivors of pediatric cancer compared to their siblings. We hypothesized that there would be a greater proportion of repressors among survivors of pediatric cancer compared to siblings, and that repressive adaptive style would be significantly associated with more positive self-reports of social adjustment. METHODS: We utilized a cross-sectional approach. Seventy-seven families participated. Survivors of pediatric cancer (n = 77, 48% male; 8-18 years of age) and one sibling (n = 50, 48% male; 8-18 years of age) completed measures assessing repressive adaptive style and social adjustment. As well, one parent from each family completed a socio-demographic questionnaire. Questionnaire packages were mailed to eligible families who agreed to participate, and were mailed back to investigators in a pre-addressed, pre-stamped envelope. RESULTS: Chi-square analyses revealed there was no significant difference in the proportion of repressors among survivors and siblings. Social adjustment scores were subjected to a two (group: survivor, sibling) by two (repressor, nonrepressor) ANCOVA with gender and age as covariates. There was a significant main effect of repressive adaptive style (F = 5.69, p < .05, η2 = 0.05) with a modest effect. Survivors and siblings with a repressive style reported significantly higher social adjustment scores (M = 106.91, SD = 11.69) compared to nonrepressors (M = 99.57, SD = 13.45). CONCLUSIONS: Repressive adaptive style explains some of the variance in survivors and siblings' self-reports of social adjustment. Future research should aim to better understand the role of the repressive adaptive style in survivors and siblings of children with cancer.

Validation of the PedsQL Epilepsy Module: A pediatric epilepsy-specific health-related quality of life measure.

OBJECTIVE: To validate a brief and reliable epilepsy-specific, health-related quality of life (HRQOL) measure in children with various seizure types, treatments, and demographic characteristics. METHODS: This national validation study was conducted across five epilepsy centers in the United States. Youth 5-18 years and caregivers of youth 2-18 years diagnosed with epilepsy completed the PedsQL Epilepsy Module and additional questionnaires to establish reliability and validity of the epilepsy-specific HRQOL instrument. Demographic and medical data were collected through chart reviews. Factor analysis was conducted, and internal consistency (Cronbach's alphas), test-retest reliability, and construct validity were assessed. RESULTS: Questionnaires were analyzed from 430 children with epilepsy (Mage = 9.9 years; range 2-18 years; 46% female; 62% white: non-Hispanic; 76% monotherapy, 54% active seizures) and their caregivers. The final PedsQL Epilepsy Module is a 29-item measure with five subscales (i.e., Impact, Cognitive, Sleep, Executive Functioning, and Mood/Behavior) with parallel child and caregiver reports. Internal consistency coefficients ranged from 0.70-0.94. Construct validity and convergence was demonstrated in several ways, including strong relationships with seizure outcomes, antiepileptic drug (AED) side effects, and well-established measures of executive, cognitive, and emotional/behavioral functioning. SIGNIFICANCE: The PedsQL Epilepsy Module is a reliable measure of HRQOL with strong evidence of its validity across the epilepsy spectrum in both clinical and research settings.

Predictors of health-related quality of life in children with chronic heart disease.

OBJECTIVE: Chronic paediatric heart disease is often associated with residual symptoms, persisting functional restrictions, and late sequelae for psychosocial development. It is, therefore, increasingly important to evaluate the health-related quality of life of children and adolescents with chronic heart disease. The aim of this study was to determine medical and socio-demographic variables affecting health-related quality of life in school-aged children and adolescents with chronic heart disease. Patients and methods The Pediatric Cardiac Quality of Life Inventory was administered to 375 children and adolescents and 386 parental caregivers. Medical information was obtained from the charts. The socio-demographic information was provided by the patients and caregivers. RESULTS: Greater disease severity, low school attendance, current cardiac medication, current parental employment, uncertain or limited prognosis, history of connection to a heart-lung machine, number of nights spent in a hospital, and need for treatment in a paediatric aftercare clinic independently contributed to lower health-related quality of life (self-report: R2=0.41; proxy-report: R2=0.46). High correlations between self-reports and parent-proxy reports indicated concordance regarding the evaluation of a child's health-related quality of life. CONCLUSIONS: Beyond medical treatment, integration into school is important to increase health-related quality of life in children and adolescents surviving with chronic heart disease. Regular screening of health-related quality of life is recommended to identify patients with special needs.

Behavioral Problems and Childhood Epilepsy: Parent vs Child Perspectives.

OBJECTIVE: To test whether the reported association between pediatric epilepsy and behavioral problems may be distorted by the use of parental proxy report instruments. STUDY DESIGN: Children in the Connecticut Study of Epilepsy were assessed 8-9 years after their epilepsy diagnosis (time-1) with the parent-proxy Child Behavior Check List (CBCL) (ages 6-18 years) or the Young Adult Self-Report (≥18 years of age). For children <18 years of age, parents also completed the Child Health Questionnaire, which contains scales for impact of child's illness on the parents. The same study subjects completed the Adult Self-Report 6-8 years later (time-2). Sibling controls were also tested. Case-control differences were examined for evidence suggesting more behavioral problems in cases with epilepsy than in controls based on proxy- vs self-report measures. RESULTS: At time-1, parent-proxy CBCL scores were significantly higher (worse) for cases than controls (n = 140 matched pairs). After adjustment for Child Health Questionnaire scales reflecting parent emotional and time impact, only 1 case-control difference on the CBCL remained significant. Self-reported Young Adult Self-Report scores did not differ between cases and controls (n = 42 pairs). At time-2, there were no significant self-reported case-control differences on the Adult Self-Report (n = 105 pairs). CONCLUSIONS: Parent-proxy behavior measures appear to be influenced by the emotional impact of epilepsy on parents. This may contribute to apparent associations between behavioral problems and childhood epilepsy. Self-report measures in older adolescents (>18 years of age) and young adults do not confirm parental perceptions. Evidence suggesting more behavioral problems in children with epilepsy should be interpreted in light of the source of information.

Conceptualizing a quality of life framework for girls with Rett syndrome using qualitative methods.

Rett syndrome is a neurodevelopmental disorder mainly affecting females and associated with a mutation on the MECP2 gene. There has been no systematic evaluation of the domains of quality of life (QOL) in Rett syndrome. The aims of this study were to explore QOL in school-aged children with Rett syndrome and compare domains with those identified in other available QOL scales. The sample comprised 21 families registered with the Australian Rett Syndrome Database whose daughter with Rett syndrome was aged 6-18 years. Semi-structured telephone interviews were conducted with each parent caregiver (19 mothers, 2 fathers) to investigate aspects of their daughter's life that were satisfying or challenging to her. Qualitative thematic analysis using a grounded theory framework was conducted, and emerging domains compared with those in two generic and three disability parent-report child QOL measures. Ten domains were identified: physical health, body pain, and discomfort, behavioral and emotional well-being, communication skills, movement and mobility, social connectedness, variety of activities, provision of targeted services, stability of daily routines, and the natural environment. The two latter domains were newly identified and each domain contained elements not represented in the comparison measures. Our data articulated important aspects of life beyond the genetic diagnosis. Existing QOL scales for children in the general population or with other disabilities did not capture the QOL of children with Rett syndrome. Our findings support the construction of a new parent-report measure to enable measurement of QOL in this group.

Psychometric evaluation and wording effects on the Chinese version of the parent-proxy Kid-KINDL.

BACKGROUND: The pediatric quality of life (QoL) questionnaire, the child-rated Kid-KINDL, has wording effects. However, no studies have examined for its parallel questionnaire, the parent-proxy Kid-KINDL. This study aimed to examine the psychometric properties and wording effects of the parent-proxy Kid-KINDL. METHODS: Parents with 8- to 12-year-old children (n = 247) completed the parent-proxy Kid-KINDL, 83 of them completed it again 7-14 days later, and 241 of their children completed the child-rated Kid-KINDL. Internal consistency was examined using Cronbach's α; test-retest reliability and concurrent validity, using Pearson correlation coefficients (r); construct validity and wording effects, using confirmatory factor analyses (CFAs). RESULTS: The internal consistency of the parent-proxy Kid-KINDL total score was acceptable (α = .86). Test-retest reliability (r = .33-.60) and concurrent validity (r = .27-.42) were acceptable or nearly acceptable for all subscales and the total score. The CFA models simultaneously accounting for QoL traits and wording effects had satisfactory fit indices, and outperformed the model accounting only for QoL traits. However, four subscales had unsatisfactory internal consistency, which might be attributable to wording effects. CONCLUSION: When children are unable to complete a QoL questionnaire, the parent-proxy Kid-KINDL can substitute with all due cautions to wording effects and inconsistent reliability among different raters.