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BACKGROUND: Rett syndrome (RTT) is a severe X-linked neurodevelopmental disorder associated with multiple neurologic impairments. Previous studies have shown challenges to the quality of life of individuals with RTT and their caregivers. However, instruments applied to quantify disease burden have not adequately captured the impact of these impairments on affected individuals and their families. Consequently, an international collaboration of stakeholders aimed at evaluating Burden of Illness (BOI) in RTT was organized. METHODS: Based on literature reviews and qualitative interviews with parents of children and adults with RTT, a caregiver questionnaire was constructed to evaluate 22 problems (inclusive of core characteristics, functional impairments, and comorbidities) often experienced with RTT, rated mainly with a 5-level Likert scale. The questionnaire was administered anonymously online to an international sample of 756 caregivers (predominantly parents) of girls and women with RTT. Descriptive statistics were used to identify problems of high frequency and impact on affected individuals and caregivers. Chi-square tests characterized the relationship between problem severity and impact responses, while nonparametric ANOVAs of raw and z-score adjusted scores identified agreement between severity and impact on individual and caregiver. Secondary inferential tests were used to determine the roles of age, clinical type, and country of residence on BOI in RTT. RESULTS: There was variability in reported frequency of problems, with the most prevalent, severe and impactful being those related to the core features of RTT (i.e., communication and fine and gross motor impairments). Chi-square analyses demonstrated interdependence between severity and impact responses, while ANOVAs showed that many problems had disproportionately greater impact than severity, either on affected individuals (e.g., hand stereotypies) or their caregivers (e.g., sleep difficulties, seizures, pain, and behavioral abnormalities). With certain exceptions (e.g., breath-holding, seizures), age, clinical type, or country of residence did not influence these BOI profiles. CONCLUSIONS: Our data demonstrate that core features and related impairments are particularly impactful in RTT. However, problems with mild severity can also have disproportionate impact on affected individuals and, particularly, on their caregivers. Future analyses will examine the role of factors such as treatment outcomes, healthcare services, and healthcare provider's perspectives, in these BOI profiles.

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PURPOSE: Accurate assessment of chronic pain and functional disability in children and adolescents is imperative for guiding pain management interventions. Parents have multifaceted roles in their child's pain experience and frequently provide parent-proxy reports of pain-related functioning. However, cross-informant variance is often observed with limited understanding of contributing factors. This study aims to examine the degree of alignment between child and parent-proxy reports for Patient-Reported Outcomes Measurement Information System (PROMIS) pain interference domain among children with chronic pain and to identify factors associated with improved child-parent agreement. METHODS: This study includes a sample of 127 youth (66.1% female) with mixed etiology chronic pain, ranging in age from 8 to 17 (M = 12.24; SD = 1.598), and their parent. Data was collected at an interdisciplinary pediatric pain clinic and online peer support groups. Measures of demographic, pain intensity, and functioning were collected. RESULTS: Means of parent-proxy reports were significantly lower than child self-reports on the PROMIS (p < 0.05). A statistically significant association between child's pain intensity (ß = 0.953, P < 0.05) and the difference between child self-reported and parent-proxy reported PROMIS functional interference scores was found. CONCLUSION: Parents underestimated pain-related functional disability relative to children's self-reports. The difference between the paired child self-report and parent-proxy report of functional disability was significantly associated with greater child self-reported pain intensity. Although parent-proxy reports in pediatric chronic pain is often used in research and practice, findings underscore the importance of incorporating child and adolescent self-report, when possible, to comprehensively capture the child's pain experience and best inform clinical interventions.

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Genetic disorders pose great challenges for affected individuals and their families, as they must cope with the irreversible nature of the disease and a life-long dependence on medical assistance and treatment. Children and adolescents dealing with Pompe disease (PD) often struggle to keep up with their peers in physical activities. To gain valuable insights into their subjective experiences and better understand their perception and coping related to daily challenges linked to their condition and treatment, the use of standardized questionnaires is crucial. This study introduces the novel PompeQoL 1.0 questionnaire for children and adolescents with PD, designed for comprehensive assessment of both disease-specific FDH and HRQoL through self- and proxy reports. Content validity was ensured through patients' and parents' involvement at the initial stages of development and in subsequent cognitive debriefing process. Participants found the questionnaire easy to understand, answerable, relevant, and comprehensive. Adjustments based on feedback from patients and their parents improved its utility as a patient- and observer-reported outcome measure. After careful item examination, 52 items were selected, demonstrating moderate to excellent test-retest reliability for most scales and initial evidence for satisfactory construct validity. The PompeQoL questionnaire stands as a valuable screening instrument for both clinical and research purposes. Future research should prioritize additional revisions and larger validation studies, focusing on testing the questionnaire in clinical practice and trials. Nevertheless, the PompeQoL 1.0 stands out as the first standardized measure providing insights into disease-specific FDH and HRQoL among children and adolescents with various forms of PD.

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This systematic review investigates Patient-reported Outcome Measures (PROMs) and Observed Reported Outcome Measures (ObsROMs) pertinent to assessing Health-Related Quality of Life (HRQoL) in short-stature paediatric patients, focusing on Achondroplasia (ACH), Growth Hormone Deficiency (GHD), Isolated Growth Hormone Deficiency (IGHD), and Small-for-Gestational-Age (SGA) diagnoses. Utilising rigorous selection criteria, 53 studies published from 1998 to 2023 were analysed, revealing a predominance of European-based research. Notably, the review elucidated the utilisation of disease-specific and generic HRQoL measures, showcasing the multifaceted nature of short-stature conditions and their impact across physical, emotional, and social domains. The Quality of Life in Short Stature Youth (QoLISSY), Paediatric Quality of Life Inventory (PedsQL), and KIDSCREEN emerged as frequently employed instruments, offering nuanced insights into HRQoL perceptions across diverse age demographics. Additionally, the review highlighted the adaptation of adult HRQoL measures for adolescent populations, signalling a need for age-appropriate assessment tools. Furthermore, integrating PROMs and ObsROMs in HRQoL assessment underscored a comprehensive approach, considering both subjective patient perspectives and observed outcomes. Future research directions encompass comprehensive search strategies, longitudinal studies with diverse populations, and the development of age-appropriate HRQoL assessment tools. In conclusion, this review emphasises the importance of comprehensive HRQoL assessment to address the diverse needs of short-stature paediatric patients effectively.

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Food insecurity is associated with a multitude of negative outcomes among adolescents. Despite adolescents' burgeoning autonomy, much of the research has relied on parent-proxy report, with few studies directly comparing adolescents' and parents' perceptions of adolescent food security. In the present study, parent-adolescent dyads (N = 144) completed questionnaires measuring perceptions of adolescent food security. Adolescents (12-17 years) completed the Child Food Security Survey Module. Parents completed the Household Food Security Survey Module. Dyads demonstrated discordant perceptions of adolescent food security on categorical- and item-levels of analysis. Further research is needed to elucidate explanations for these discrepancies using longitudinal designs.

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Evidence-based literature recognizes that the different degrees of agreement between a child self-report and a proxy-report depend on the characteristics of the domains, the child's age and illness, the proxy's own perspective on QoL, and family attendance during the child's hospitalization. This study aims to determine the degree of agreement between proxy-reports and child self-reports on quality of life (QoL) for children with hematologic malignancy ranging in age from 5 to 18 years who are undergoing treatment. We retrieved clinical QoL data from a study titled "Dynamic change in QoL for Vietnamese children with hematologic malignancy" from April 2021 to December 2022. To evaluate the magnitude of agreement between self-reports and proxy-reports, intraclass correlation coefficients (ICCs) for 259 pairs of measurements were quantified. Using independent t tests, the mean differences between self-reports and proxy-reports were tested. Moderate agreement was consistent through all age groups for five subscales, including physical, psychosocial, pain, nausea, and procedural anxiety (ICCs ranged from 0.53 to 0.74). The weakest agreement appeared in two groups, subjects aged 5-7 years and 13-18 years on six domains (school, treatment anxiety, worry, cognitive problems, perceived physical appearance, and communication) (-0.01 to 0.49). Child self-rating was consistently higher than that of proxies for the physical, emotional, and nausea domains among children aged 5-7 years and for procedural anxiety, treatment anxiety, and cognitive problems among children aged 8-12 years.    Conclusion: The agreement level of self-reports and proxy-reports was differently distributed by child age and the PedsQL domains. The proxy children agreement on QoL among children with hematologic malignancy was divergent according to the different age groups, which could potentially be explained by proxy-child bonding at different stages of childhood development. Our recommendation for future studies is to explore children's age as a potential factor influencing proxy agreement on QoL among children with cancer. What is Known: • Children and their proxies may think differently about quality of life (QoL). • Comparing two sources of data (i.e., child and proxy) on aspects of QoL can help identify the discrepancies between children's perceptions of their QoL and their parents' perceptions. This can be useful in terms of identifying potential areas for improvement or concern and may also be helpful in making decisions about treatment and care. What is New: • Our study results demonstrated that proxies who comprised children aged 5-7 years or 13-18 years reported differently among domains that cannot be expressed verbally or with body language, including cognitive problems, perceived physical appearance, and communication. • Children generally perceived their QoL to be better than their proxies. Therefore, a more comprehensive understanding of children's QoL may require the consideration of multiple sources of data from various perspectives.

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Background and Aim: Malocclusion can negatively impact the quality of life of children. Therefore, this study assesses the impact of proxy-reported malocclusion and oral health-related quality of life among children in Riyadh, Saudi Arabia, from the parents or guardians' perspectives. Materials and Methods: A self-administered electronic questionnaire was used to assess the correlation between proxy-reported malocclusion conditions during the early mixed dentition stage (children age 6-12 years) and oral health-related quality of life using the OHIP-14 measure. All collected data were analyzed using SPSS. Results: Among the 353 participants in the study, anterior open-bite was the most common proxy-reported malocclusion with a prevalence of 19%, followed by unilateral posterior cross bite (13.3%). Furthermore, 31% reported that their children sometimes experienced negative impacts on quality of life from malocclusions. The results also show that OHIP-14 scores were significantly associated with all proxy-reported malocclusion (p < 0.05). The highest OHIP-14 score was found to be significantly associated with the presence of deep-bite from parents' or guardians' perspective. Conclusion: The presence of some proxy-assessed malocclusion was associated with negative impacts on children's oral health-related quality of life. This is very important to consider when assessing the need for orthodontic intervention, especially at this stage as this age is critical in building a child's confidence and self-esteem.

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BACKGROUND: Health-related quality of life (HRQoL) is clearly recognized as a patient-important outcome in patients with traumatic brain injury (TBI). Patient-reported outcomes are therefore often used and supposed to be directly reported by the patients without interpretation of their responses by a physician or anyone else. However, patients with TBI are often unable to self-report because of physical and/or cognitive impairments. Thus, proxy-reported measures, e.g., family members, are often used on the patient's behalf. Yet, many studies have reported that proxy and patient ratings differ and are noncomparable. However, most studies usually do not account for other potential confounding factors that may be associated with HRQoL. In addition, patients and proxies can interpret some items of the patient-reported outcomes differently. As a result, item responses may not only reflect patients' HRQoL but also the respondent's (patient or proxy) own perception of the items. This phenomenon, called differential item functioning (DIF), can lead to substantial differences between patient-reported and proxy-reported measures and compromise their comparability, leading to highly biased HRQoL estimates. Using data from the prospective multicenter continuous hyperosmolar therapy in traumatic brain-injured patients study (240 patients with HRQoL measured with the Short Form-36 (SF-36)), we assessed the comparability of patients' and proxies' reports by evaluating the extent to which items perception differs (i.e., DIF) between patients and proxies after controlling for potential confounders. METHODS: Items at risk of DIF adjusting for confounders were examined on the items of the role physical and role emotional domains of the SF-36. RESULTS: Differential item functioning was evidenced in three out of the four items of the role physical domain measuring role limitations due to physical health problems and in one out of the three items of the role emotional domain measuring role limitations due to personal or emotional problems. Overall, despite an expected similar level of role limitations between patients who were able to respond and those for whom proxies responded, proxies tend to give more pessimistic responses than patients in the case of major role limitations and more optimistic responses than patients in the case of minor limitations. CONCLUSIONS: Patients with moderate-to-severe TBI and proxies seem to have different perceptions of the items measuring role limitations due to physical or emotional problems, questioning the comparability of patient and proxy data. Therefore, aggregating proxy and patient responses may bias HRQoL estimates and alter medical decision-making based on these patient-important outcomes.

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BACKGROUND: The Alzheimer's Disease Cooperative Study - Activities of Daily Living Scale for use in Mild Cognitive Impairment (MCI), the ADCS-ADL-MCI, is an evaluation scale with information provided by an informant/caregiver to describe the functional impairment of patients with MCI. As the ADCS-ADL-MCI has yet to undergo a full psychometric evaluation, this study aimed to evaluate the measurement properties of the ADCS-ADL-MCI in subjects with amnestic MCI. METHODS: Measurement properties, including item-level analysis, internal consistency reliability, test-retest reliability, construct validity (convergent/discriminant, known-groups validity), and responsiveness were evaluated using data from the ADCS ADC-008 trial, a 36-month, multicenter, placebo-controlled study in 769 subjects with amnestic MCI (defined by clinical criteria and a global clinical dementia rating, CDR, score of 0.5). Due to most subjects' mild condition at baseline and resulting low variance in scores, psychometric properties were assessed using both baseline and 36-month data. RESULTS: Ceiling effects were not apparent at the total score level, with 3% of the cohort reaching the maximum score of 53, despite most subjects having a relatively high score at baseline (mean score = 46.0 [standard deviation = 4.8]). Item-total correlations were overall weak at baseline, most likely due to low variability in responses; however, at month 36, good item homogeneity was found. Cronbach's alpha values ranged from acceptable (0.64 at baseline) to good (0.87 at month 36), indicating overall very good internal consistency reliability. Further, moderate to good test-retest reliability was found (intraclass correlation coefficients ranging from 0.62-0.73). The analyses also largely supported convergent/discriminant validity, especially at month 36. Finally, the ADCS-ADL-MCI discriminated well between groups showing good known-groups validity, and was responsive in patients who indicated a longitudinal change in other instruments. CONCLUSIONS: This study provides a comprehensive psychometric evaluation of the ADCS-ADL-MCI. Findings suggest that the ADCS-ADL-MCI is a reliable, valid and responsive measure capable of capturing functional abilities in patients with amnestic MCI. TRIAL REGISTRATION: ClinicalTrials.gov Identifier: NCT00000173.

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Cerebral palsy (CP) is an early onset, non-progressive, neuromotor disorder. Adolescence is the transition from childhood to adulthood when changes in physical and emotional aspects and self-perception occur further imposing an impact to quality of life (QoL) in individuals with CP. Cerebral Palsy Quality of Life (CP QoL) Teen is a questionnaire examining different domains of QoL for adolescents with CP. This study is aimed at translating and validating self-report and proxy-report CP QoL-Teen (HK). Prior approval of translation has been obtained. Forward and backward translations were performed following standardized translation procedures. Participants and their caregivers were asked to complete self-report and proxy-report CP QoL-Teen (HK), and Child Health Questionnaire (CHQ). Internal consistency and test-retest reliability were assessed by Cronbach's alpha and intraclass correlation coefficient (ICC), respectively. Concurrent validity was evaluated by Spearman's rank correlation between subscales of CP QoL-Teen (HK) and CHQ as well as expanded and revised version of Gross Motor Function Classification System (GMFCS-E&R). Ninety-six participants completed the study. Of these, twenty participants completed CP QoL-Teen (HK) twice. Cronbach's α of CP QoL-Teen (HK) ranged from 0.84 to 0.95 suggesting excellent internal consistency. Moderate to excellent test-retest reliability were demonstrated in all subscales of CP QoL-Teen (HK) (self-report: ICC = 0.46-0.8; proxy-report: ICC = 0.40-0.72, p < 0.05). Weak to moderate association between subscales of CP QoL-Teen (HK) and CHQ (self-report: rs = 0.24-0.61; proxy-report: rs = - 0.41-0.60) was reported. CONCLUSION: This study showed that CP QoL-Teen (HK) has good psychometric properties. It is a valid and reliable tool to assess quality of life of adolescents with CP. WHAT IS KNOWN: • Cerebral Palsy Quality of life-Teen (CP QoL-Teen) is a validated tool with strong psychometric properties and clinical utility in gauging the QoL in adolescents with CP during their transition from childhood to adulthood when changes in physical and emotional aspects and self-perception occur. Yet, a locally validated tool is lacking in measuring the QoL for adolescents with CP in Hong Kong. WHAT IS NEW: • The Chinese translated version CP QoL-Teen (HK) is a valid and reliable tool to assess quality of life of adolescents with CP tailoring to the local cultural and social background with good psychometric properties being demonstrated.

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BACKGROUND: Venous access port is commonly used during cancer treatment in children, yet little is known about how children experience such needle insertion procedures. AIM: To study distress before and pain after venous access port needle insertion among children and adolescents with cancer. A second aim was to explore associations between their self-report of procedure-related distress and pain with proxy reports by parents and nurses. METHOD: The sample included 43 children/adolescents, aged 1-16 years with cancer, treated at two Norwegian university hospitals. The patient, parent(s), and the nurse performing the procedure completed developmentally appropriate 11-point distress and pain scales before and immediately after the venous access port procedure. Data were analysed using descriptive statistics and non-parametric correlations. ETHICAL ISSUES: The ethical code of conduct was followed and conformed to the ethical guidelines adopted by the Regional Committee for Medicine and Health Research and the data protector officer at the hospitals. RESULTS: For the youngest children (1-5 years), the median distress proxy score was 8 (range 0-9) and pain proxy score 4 (range 0-10). Median distress and pain scores for children aged 6-12 years were 3 (range 0-9) and 1 (range 0-10), respectively, and for the adolescents (age 13-16) 0 (range 0-6) and 1 (range 0-5), respectively. Patients' self-reported distress and pain correlated highly with parents' (distress: rho = 0.83, p < 0.001, pain: rho = 0.92, p < 0.001) and with nurses' proxy ratings (distress: rho = 0.89, p < 0.001, pain: rho = 0.88, p < 0.001). CONCLUSION: There were individual age differences in experienced distress/pain associated with venous access port needle insertion, with a trend for younger children to experience higher levels of distress/ pain than the older children. Children's self-report of distress/ pain concurred with both parental and nurse proxy reports.

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PURPOSE: The aim of this study is to assess the level of agreement between adolescents' self-assessment and parent-proxy reports on health-related quality of life (HRQOL) in Jamaican adolescents with chronic illness. METHODS: A cross-sectional study was conducted, recruiting adolescents living with a chronic illness (ALCIs)-asthma, human immunodeficiency virus, insulin-dependent diabetes mellitus, or sickle cell disease and age/sex-matched healthy adolescents. Data were collected on HRQOL from adolescents and parents using the Pediatric Quality of Life Scale. Parent-adolescent agreement was determined at group level (Wilcoxon signed-rank test) and individual level (intraclass correlation coefficient). RESULTS: Two hundred twenty-six (226) parent/adolescent pairs participated: 130 ALCIs and 96 healthy peers; mean age 14.9 ± 2.8 years; 58% females. Adolescents with and without chronic illness reported similar HRQOL; parent-proxies reported better HRQOL for healthy adolescents compared to ALCIs. Intraclass correlation demonstrated higher levels of parent-adolescent correlation for ALCIs than healthy adolescents (ALCIs: 0.11-0.34; healthy adolescents: 0.01-0.10). At group level, analyses demonstrated better parent-proxy rating of QOL in all of the scores with the exception of the general health score. Parent-proxies overestimated QOL for asthma and insulin-dependent diabetes mellitus but not for sickle cell disease and human immunodeficiency virus. Linear regression modeling revealed that female sex and living with chronic illness were significant predictors of agreement. DISCUSSION: Parent-proxies overestimated adolescents' QOL compared to adolescents' report regardless of whether the adolescent was living with a chronic illness or not. As such, health care providers should elicit feedback from the adolescent wherever possible and proxy reports should be used as complementary information rather than primary source.

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To encourage person-centered care, the Centers for Medicare and Medicaid require nursing homes to measure resident preferences using the Preferences Assessment Tool (PAT). No known research has examined the implications of respondent type (i.e., resident, proxy, staff) on preference importance; therefore, the purpose of this study was to compare the importance of preferences depending on which respondent completed the PAT. Participants included 16,111 Veterans discharged to community-based skilled nursing facilities after hospitalization for heart failure. A majority (95%) of residents completed the PAT compared to proxy (3%) and staff (2%). Proxy responders were both more and less likely to indicate individual preferences as important compared to residents. Staff members were consistently less likely to indicate all preferences as important compared to residents. Findings from this study emphasize the need for proxy and staff to find methods to better understand residents' preferences when residents are not able to participate in assessments.

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OBJECTIVE: To test the effectiveness of an 8-month school-based multicomponent intervention (MOVI-KIDS) in improving health-related quality of life (HRQoL) in schoolchildren. STUDY DESIGN: A randomized cluster trial was conducted including 1168 children aged 4-6 years who attended 21 schools in two Spanish provinces (Cuenca and Ciudad Real). MOVI-KIDS study is a multicomponent physical activity intervention, which consisted of (i) 3 × 60-min sessions/week, (ii) educational materials for parents and teachers, and (iii) school playground modifications. The parent's proxy report of the KINDL-R Spanish version (6 subdimensions and a total score), and the KINDL-R self-reported by children (total score) was used to measure HRQoL. Mixed linear regression models were conducted to test differences in each HRQoL dimension between intervention and control groups, controlling for baseline values, cardiorespiratory fitness, and socioeconomic status, by gender. RESULTS: The boys in the intervention group presented better scores on total HRQoL than the control group in both the parent (ß = 1.46; 95% CI: 0.23-2.70) and self-reported (ß = 2.13; 95% CI: 0.53-3.74) versions, as well as on the emotional well-being dimension (ß = 2.43; 95% CI: 0.48-4.36). There was no significant effect of the intervention on physical well-being, self-esteem, family, and friends. In girls, no statistically significant differences were found between those who participated in MOVI-KIDS and those who did not. CONCLUSION: Our data support gender differences in the effect of MOVI-KIDS, such that while in boys the intervention was successful in increasing total scores of HRQoL, as well as emotional well-being scores, the intervention was not effective in improving girls' HRQoL.

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Billions of children/adolescents experienced unprecedented changes in their daily lives that impacted their health-related quality of life (HRQoL) during the first wave of the coronavirus disease 2019. The purpose of this study was to describe child-parent discrepancies in reporting on HRQoL and explore factors associated with such discrepancies at the end of the first lockdown in France. A cross-sectional study was conducted among French school-aged children from 8 to 18 years and their parents living in the Grand Est region in France during the first wave of the epidemic. The impact of individual, self-reported health status and environmental data on discrepant parent-child reports of HRQoL was assessed by multinomial multivariable logistic regression models. A total of 471 parent-child pairs were included. Among 50% of the discordant pairs, parents underestimated HRQoL more frequently than they overestimated it. Home location, social support score, children's education level, parents' education level, tensions and conflicts with neighbors reported by children, whether they had access to a garden, and parents' professional activity were significantly associated with parental overestimation (adjustedOR from 2.08 to 11.61; p < 0.05). Factors associated with parental underestimation were children's education level, SF12 score, home location, the child's gender, parent's level of education, the presence of noise in the residence reported by children, whether a household member was infected with COVID-19, whether they had access to a garden, and family structure (adjustedOR from 1.60 to 4.0; p < 0.05). This study revealed differences between child-reported and parent-reported HRQoL. The COVID-19 pandemic accentuated the discrepancies in observable dimensions and attenuated them in unobservable dimensions of HRQoL but did not impact the directional discrepancy; parents underestimated their child's HRQoL more. These discrepancies appear to be explained by parent and child sociodemographic factors.

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BACKGROUND: Views can be collected from individuals (self-report) or others on their behalf (proxy-report). OBJECTIVE: We aimed to review the literature on methods and statistical approaches used to evaluate observer versus self-report responses from individuals with intellectual disability or Down syndrome. METHODS: A series of key questions related to statistical approaches and data collection methods were formulated a priori to inform the search strategy and review process. These addressed the topics of self-report in individuals with intellectual disability, including Down syndrome. Using the National Library of Medicine database, PubMed, detailed literature searches were performed. The quality of available evidence was then evaluated, the existing literature was summarised, and knowledge gaps and research needs were identified. RESULTS: Fifty relevant original articles were identified which addressed at least one key question. Study details, including: research design, internal validity, external validity, and relevant results are presented. Review of studies of individuals with intellectual disability which used a variety of statistical approaches showed mixed agreement between self-report and proxy-report. CONCLUSION: Few studies identified to-date have used self-report from individuals with Down syndrome, but lessons from the existing intellectual disability literature can guide researchers to incorporate self-report from individuals with Down syndrome in the future.

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BACKGROUND: Proxy report is essential for patients unable to complete patient-reported outcome (PRO) measures themselves and potentially beneficial when the caregiver perspective can complement patient report. In this study, we examine agreement between self-report by older adults and proxy report by their caregivers when completing PROs for pain, anxiety, depression, and other symptoms/impairments. METHODS: Four PROs were administered by telephone to older adults and their caregivers followed by re-administration within 24 h in a random subgroup. The PROs included the PHQ-9 depression, GAD-7 anxiety, PEG pain, and SymTrak multi-dimensional symptom and functional status scales. RESULTS: The sample consisted of 576 older adult and caregiver participants (188 patient-caregiver dyads, 200 patients without identified caregiver). The four measures had good internal (Cronbach's alpha, 0.76 to 0.92) and test-retest (ICC, 0.63 to 0.92) reliability whether completed by patients or caregivers. Total score and item-level means were relatively similar for both patient and caregiver reports. Agreement for total score as measured by intraclass correlation coefficient (ICC) was better for SymTrak-23 (0.48) and pain (0.58) than for anxiety (0.28) and depression (0.25). Multinomial modeling showed higher (worse) patient-reported scale scores were associated with caregiver underreporting, whereas higher caregiver task difficulty was associated with overreporting. CONCLUSION: When averaged over individuals at the group level, proxy reports of PRO scores by caregivers tend to approximate patient reports. For individual patients, proxy report should be interpreted more cautiously for psychological symptoms as well as when patient-reported symptoms are more severe, or caregiver task difficulty is high.

Assessment of patient-reported outcomes (e.g., symptoms, functional status, and other quality of life domains) by a proxy on behalf of the patient is helpful when patient self-report is not possible or when a complementary perspective may inform care. Determining how well older adult patients and their caregivers agree on the patient's pain, depression, anxiety, and functioning is important in investigating and managing these core clinical domains. In this study, patient-caregiver agreement was evaluated for four commonly-used scales that assess depression (PHQ-9), anxiety (GAD-7), pain (PEG), and multi-dimensional symptom and functional impairment (SymTrak). Total score and item-level scores were similar between patients and caregivers when averaged at the group level. Agreement was higher for more physically oriented domains (pain and SymTrak) than psychological conditions (depression and anxiety). Higher (worse) patient-reported scale scores were associated with caregiver underreporting, whereas higher caregiver task difficulty was associated with overreporting. Proxy report may be sufficiently accurate in research when studying group differences. However, proxy reports should be interpreted more cautiously in individual patients with psychological symptoms or higher symptom severity, or where there is caregiver diffficulty.

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PURPOSE: The main purpose was to study young patients' self-reports on dental fear over a 5-year period, prospectively. Also, to compare these to professionals' proxy reports for dental fear during invasive and non-invasive dental visits. The research question was, to what extent the self-reports and dental professionals' proxy reports are congruent, and if there were patient age-dependent differences. METHODS: 3134 patients from 11 public dental clinics, representing urban and rural areas, were invited. Four age cohorts were formed: 3, 7, 11, and 15 years of age and followed between the years 2008 and 2012. Dental examinations (non-invasive) and restorative treatments + extractions (invasive) were registered. During the treatments, self-reports regarding fear and professional proxy reports were registered: Not afraid at all = 0, little nervous = 1, quite afraid = 2, very scared = 3, terrified = 4. RESULTS: 2363 patients completed the cohort periods (51% girls and 49% boys). In all, 9708 dental examinations, restorations and extractions were performed. The fear prevalence increased with the invasiveness of the dental procedure; 7-56%. For dental examinations and restorations, fear declined with ascending age. The highest fear prevalence was reported for dental extractions. Younger children reported fear more frequently than older children, p < 0.001. Frequent inconsistencies between self-reports and proxy reports were observed among the younger children (16%) compared to the older children (8%), p < 0.001. CONCLUSION: Non-congruence was observed for self-reports and proxy reports regarding all age cohorts.

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BACKGROUND: Mental health conditions are common among individuals with intellectual disability. Under recognition of mental health disorders leading to unmet treatment needs is common in this population. This article addresses one major contributing factor, the lack of cognitively accessible self-report measures for individuals with intellectual disability. METHOD: In this literature-informed overview of the state of the field, we discuss the need for, and complexities of, including individuals with intellectual disability in mental health assessments. RESULTS: With appropriate supports, many individuals with intellectual disability can respond to mental health questions. We discuss evidence-based strategies to make mental health assessments more accessible. CONCLUSION: We highlight the need to engage individuals with intellectual disability to provide first-hand information about their health and well-being. New instruments and research procedures should be developed in partnership with individuals with intellectual disability. Self-report may be essential to advancing the science of mental health research.

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PURPOSE: To analyse: 1) the differences in health-related quality of life (HRQoL) between typically developing (TD) children and children with developmental coordination disorder (DCD) according to parents' and children's perception, and 2) the differences and level of agreement between parents and children's perceptions in HRQoL in both children's samples. METHODS: Cross-sectional analysis in 115 Spanish schoolchildren 4-to-7 years. Motor competence and HRQoL were assessed using the MACB-2 and the KINDL-R questionnaire, respectively. RESULTS: ANCOVA model showed that children with DCD children obtained lower scores in physical well-being, friends, school and total HRQoL dimensions than TD peers after controlling for covariates (p < 0.05). Moreover, parents' perception scores in HRQoL were lower in children with DCD than in TD peers (79.7 vs 84.8; p = 0.022). Student T-tests for repeated-measures showed non-significant differences between children and parents' perceptions in mean HRQoL scores, by motor competence categories. The intraclass correlations coefficients between parents and children's perception of HRQoL was moderate in DCD category (0.62; p = 0.024) and small in TD category (0.29; p = 0.049). CONCLUSIONS: Children under 6 years old with DCD have lower HRQoL scores than their TD peers. No differences were found between children's and parents' perceptions in total HRQoL, although the perceptions of children and parents in DCD category showed a significantly higher level of agreement than TD children. Interventions aimed at promoting motor skills in school settings during the preschool age seem necessary to improve children's quality of life.

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