Measuring health-related quality of life in children with suspected genetic conditions: validation of the PedsQL proxy-report versions.

PURPOSE: Measuring health-related quality of life (HRQoL) of children with suspected genetic conditions is important for understanding the effect of interventions such as genomic sequencing (GS). The Pediatric Quality of Life Inventory (PedsQL) is a widely used generic measure of HRQoL in pediatric patients, but its psychometric properties have not yet been evaluated in children undergoing diagnostic GS. METHODS: In this cross-sectional study, we surveyed caregivers at the time of their child's enrollment into GS research studies as part of the Clinical Sequencing Evidence Generating Research (CSER) consortium. To evaluate structural validity of the PedsQL 4.0 Generic Core Scales and PedsQL Infant Scales parent proxy-report versions, we performed a confirmatory factor analysis of the hypothesized factor structure. To evaluate convergent validity, we examined correlations between caregivers' reports of their child's health, assessed using the EQ VAS, and PedsQL scores by child age. We conducted linear regression analyses to examine whether age moderated the association between caregiver-reported child health and PedsQL scores. We assessed reliability using Cronbach's alpha. RESULTS: We analyzed data for 766 patients across all PedsQL age group versions (1-12 months through 13-18 years). Model fit failed to meet criteria for good fit, even after modification. Neither age group (categorical) nor age (continuous) significantly moderated associations between PedsQL scores and caregiver-reported child health. Cronbach's alphas indicated satisfactory internal consistency for most PedsQL scales. CONCLUSION: The PedsQL Generic Core Scales and Infant Scales may be appropriate to measure HRQoL in pediatric patients with suspected genetic conditions across a wide age range. While we found evidence of acceptable internal consistency and preliminary convergent validity in this sample, there were some potential problems with structural validity and reliability that require further attention.

Projection of Care Partners' Preferences in the Proxy Assessments of Everyday Preferences for Persons With Cognitive Impairment.

OBJECTIVE: Bias in surrogate decision-making can occur when proxy decision-makers overestimate the degree to which their preferences are shared by others, resulting in a projection of their beliefs onto others. The purpose of this study is to assess projection of care partners' preferences onto surrogate assessments of everyday preferences for persons with cognitive impairment (CI) and to address clinical and demographic factors as predictors of projection. METHODS: The sample included 116 dyads of persons with CI (Clinical Dementia Rating Scale score ≥ 0.5) and their care partners. The Preferences for Everyday Living Inventory (PELI) was used to assess importance of preferences among persons with CI. Care partners completed two separate PELI assessments: one from the perspective of the persons with CI (i.e., acting as a surrogate decision-maker) and one from their own perspective. To assess for projection of care partners' preferences onto surrogate assessments of preferences for persons with CI, two-step regression with multivariable-adjusted general linear models was used. RESULTS: Significant projection was noted within the PELI domains of autonomous choice, personal growth, and keeping a routine (p < 0.005). More significant cognitive impairment was associated with increased projection within the PELI domains of autonomous choice and personal growth (p < 0.05). CONCLUSION: The results of this study suggest that projection of care partners' own preferences may be a significant source of bias in proxy decision-making regarding everyday preferences for persons with CI, particularly for those with more significant CI.

(Re)Conceptualising 'good' proxy decision-making for research: the implications for proxy consent decision quality.

People who are unable to make decisions about participating in research rely on proxies to make a decision based on their wishes and preferences. However, patients rarely discuss their preferences about research and proxies find it challenging to determine what their wishes would be. While the process of informed consent has traditionally been the focus of research to improve consent decisions, the more conceptually complex area of what constitutes 'good' proxy decision-making for research has remained unexplored. Interventions are needed to improve and support proxy decision-making for research but are hampered by a lack of understanding about what constitutes decision quality in this context. A global increase in conditions associated with cognitive impairment such as dementia has led to an urgent need for more research into these conditions. The COVID-19 pandemic and subsequent necessity to conduct research with large numbers of critically ill patients has made this need even more pressing. Much of the empirical research centres on the desire to improve decision accuracy, despite growing evidence that authenticity is more reflective of the aim of proxy decisions and concerns about the methodological flaws in authenticity-focused studies. Such studies also fail to take account of the impact of decision-making on proxies, or the considerable body of research on improving the quality of healthcare decisions. This paper reports a concept synthesis of the literature that was conducted to develop the first conceptualisation of 'good' proxy decisions about research participation. Elements of decision quality were identified across three stages of decision-making: proxy preparedness for decision-making which includes knowledge and understanding, and values clarification and preference elicitation; the role of uncertainty, decisional conflict, satisfaction and regret in the decision-making process; and preference linked outcomes and their effect. This conceptualisation provides an essential first step towards the future development of interventions to enhance the quality of proxy decision-making and ensure proxy decisions represent patients' values and preferences.

Discrepancies between proxy estimates and patient reported, health related, quality of life: minding the gap between patient and clinician perceptions in heart failure.

PURPOSE: Health related quality of life (HRQoL) is rarely routinely measured in the clinical setting. In the absence of patient reported data, clinicians rely on proxy and informal estimates to support clinical decisions. This study compares clinician estimates (proxy) with patient reported HRQoL in patients with advanced heart failure and examines factors influencing discrepancies. METHODS: Seventy-five patients with heart failure, (22 females, 53 males) completed the EQ-5D-5L questionnaire. Thirty-nine clinicians (11 medical, 23 nursing, 5 allied health) completed the proxy version (V1) producing 194 dyads. Correlation was assessed using Spearman's rank tests, systematic bias was examined with Bland-Altman analyses. Inter-rater agreement at the domain level, was investigated using linear weighted Kappa statistics while factors influencing the IRG were explored using independent student t-tests, analysis of variance and regression. RESULTS: There was a moderate positive correlation between clinician HRQoL estimates and patient reported utility (r = 0.38; p < .0005). Mean clinician estimates were higher than patient reported utility (0.60 vs 0.54; p = 0.008), with significant underestimation of reported problems apparent in three of the five EQ-5D-5L domains. Patient sex (female), depressed mood and frailty were all associated with an increased inter-rater gap. CONCLUSION: Clinicians in this sample overestimated HRQoL. Factors affecting the inter-rater gap, including sex and depression, support formal HRQoL screening to enhance clinical conversations and decision making. The discrepancy also supports regulatory restriction on the use of expert opinion in the development of QALYs in health economic analysis.

Patient and proxy reports regarding the experience of treatment decision-making in cancer care.

OBJECTIVE: Shared decision-making, including the elicitation of patient preferences regarding treatment decisions, is considered part of high-quality cancer care. However, patients may not be able to self-report due to illness, and therefore proxy reports may be used. We sought to determine the difference between proxy and patient reports about patient decisions and preferences among patients who received or were scheduled for chemotherapy using data from a large, population-based survey of patients with incident lung or colorectal cancer. METHODS: Of 3573 patients who received or were scheduled for chemotherapy, 3108 self-reported and 465 had proxies reporting on their behalf about preferred and actual decision roles regarding this treatment. Preferred and actual decision roles were assessed using the Control Preferences Scale, and categorized as shared, patient-controlled, or doctor-controlled. Multivariable logistic regression models were used to assess the association between patient and proxy responses and whether preferences were met. The models adjusted for sociodemographic and clinical variables and patient/proxy-reported health status. RESULTS: Sixty-three percent of all respondents reported actual roles in decisions that matched their preferred roles (role attainment). Proxies and patients were similarly likely to report role attainment (65% vs 63%). In adjusted analyses, proxies were more likely report role attainment (OR = 1.27, 95%CI = 1.02-1.59), but this difference was smaller if health variables were excluded from the model (OR = 1.14, 95%CI = 0.92-1.41). CONCLUSION: Most patients' preferences for treatment participation were met. Surveys from proxies appear to yield small differences on the reports of attainment of preferred treatment decision-making roles in cancer care vs surveys from patients.

Assessing the effect of child's gender on their father-mother perception of the PedsQL™ 4.0 questionnaire: an iterative hybrid ordinal logistic regression/item response theory approach with Monte Carlo simulation.

BACKGROUND: This study aimed at investigating the possible confounding effect of children's gender on the parents' dyads perception of their child HRQoL at both item and scale levels of PedsQLTM4.0 questionnaire. METHODS: The PedsQL™ 4.0 Generic Core Scales were completed by 573 children and their father-and-mother dyads. An iterative hybrid ordinal logistic regression/item response theory model with Monte Carlo simulation was used to detect differential item functioning (DIF) invariance across mothers/fathers and daughter/sons. RESULTS: Assessing DIF across mother-daughter, father-daughter, mother-son, and father-son dyads revealed that although parents and their children perceived the meaning of some items of PedsQLTM4.0 instrument differently, the pattern of fathers' and mothers' report does not vary much across daughters and sons. CONCLUSION: In the Persian version of PedsQLTM4.0, the child's gender is not a confounding factor in the mothers' and fathers' report with respect to their daughters' and sons' HRQoL. Hence, paternal proxy-reports can be included in studies, along with maternal proxy-reports, and the reports can be combined short of concerning children gender, when looking at parent-child agreement.

Assessing the proxy response bias of EQ-5D-3 L in general population: a study based on a large-scale representative household health survey using propensity score matching.

BACKGROUND: Proxy respondent-someone who assists the intended respondent or responds on their behalf-are widely applied in the measurement of health-related quality of life (HRQL). However, proxies may not provide the same responses as the intended respondents, which may bias the findings. OBJECTIVES: To determine whether the use of proxies is related to socio-demographic characteristics of the intended respondent, and to assess the possible proxy response bias of Chinese version of EQ-5D-3 L in general population. METHODS: A cross-sectional study based on a provincially representative sample from 2013 National Health Service Survey (NHSS) in Shaanxi, China was performed. HRQL was measured by Chinese version of EQ-5D-3 L. Propensity score matching (PSM) was used to get matched pairs of self-reports and proxy-reports. Before and after PSM, univariate logistic and linear models including the indicator of proxy response as the only independent variable, were employed to assess the possible proxy response bias of the dimensional and overall health status of EQ-5D-3 L respectively. RESULTS: 19.9% of the responses involved a proxy. Before PSM, the proxy-report group was younger in age and reported less unhealthy lifestyle, lower prevalence of disease, and less hospitalization than the self-report group. After PSM, it showed that the proxy-report group was statistically more likely to report health problem on each dimension of EQ-5D-3 L, with odds ratios larger than one comparing with self-report group. The means of EQ-5D-3 L index and EQ VAS of proxy-report group were 0.022 and 0.834 lower than self-report group. CONCLUSIONS: Significantly negative proxy response bias was found in Chinese EQ-5D-3 L in general population, and the magnitude of the bias was larger in physical dimensions than psychological dimensions after using PSM to control confounders.

Self and parent-proxy rated health-related quality of life (HRQoL) in youth with obesity: are parents good surrogates?

PURPOSE: Consideration of health-related quality of life (HRQoL) and wellbeing outcomes is important to guide healthcare services for youth with obesity, yet youth perspectives may differ from their parents. This study compared youth and parental HRQoL reports and evaluated levels of concordance across HRQoL domains and as a function of youth age, youth gender and parent informant (mother and father). METHODS: 376 youths with obesity, recruited from community (N = 223) and hospital settings (N = 153), and their parents (N = 190 mothers; N = 91 fathers), completed the PedsQL. Parental and youth agreement across subgroup dyads (mother; father; child gender; child age) were evaluated using Wilcoxon signed-rank test, intra-correlations coefficients (ICCs) and Bland-Altman plots. RESULTS: Compared to norms, HRQoL levels (youth self-report and parental proxy reports) were lower in all domains. Both mother and fathers' HRQoL reports were significantly lower than youths, most notably in physical HRQoL. Youth-parent concordance ranged from poor to moderate (ICC = 0.230-0.618), with lowest agreement for Physical HRQOL. Mothers were better proxies with ICCs being significant in all domains. Youth-father ICCs were significant only for Social (ICC = 0.428) and School (ICC = 0.303) domains. Girl-mother agreement was significant across all domains, while girl-father agreement was significant only in the Social domain (ICC = 0.653). Both mothers and fathers were poor raters for boys, and younger youths (aged ≤ 12), with non-significant ICCs in most HRQoL domains. CONCLUSIONS: Parents are poor surrogates for youth HRQoL. Clinicians should be cognizant that parents are not necessarily accurate proxies for youths, and exercise caution when interpreting parent-proxy scores.

Against the family veto in organ procurement: Why the wishes of the dead should prevail when the living and the deceased disagree on organ donation.

The wishes of registered organ donors are regularly set aside when family members object to donation. This genuine overruling of the wishes of the deceased raises difficult ethical questions. A successful argument for providing the family with a veto must (a) provide reason to disregard the wishes of the dead, and (b) establish why the family should be allowed to decide. One branch of justification seeks to reconcile the family veto with important ideas about respecting property rights, preserving autonomy, and preventing harm. These arguments are ultimately unsuccessful. Another branch of arguments is consequentialist, pointing out the negative consequences of removing the veto. Whether construed as concerning family distress or as a potential drop in the organs available, these arguments are unsuccessful; the first fails to recognize the tremendous distress associated with waiting for an organ, while the second has little supporting evidence. A final section considers and rejects whether combining some of the arguments just examined could justify the family veto. We should thus remove the family veto in organ donation.

Against autonomy: How proposed solutions to the problems of living wills forgot its underlying principle.

Significant criticisms have been raised regarding the ethical and psychological basis of living wills. Various solutions to address these criticisms have been advanced, such as the use of surrogate decision makers alone or data science-driven algorithms. These proposals share a fundamental weakness: they focus on resolving the problems of living wills, and, in the process, lose sight of the underlying ethical principle of advance care planning, autonomy. By suggesting that the same sweeping solutions, without opportunities for choice, be applied to all, individual patients are treated as population-level groups-as a theoretical patient who represents a population, not the specific patient crafting his or her individualized future care plans. Instead, advance care planning can be improved through a multimodal approach that both mitigates cognitive biases and allows for customization of the decision-making process by allowing for the incorporation of a variety of methods of advance care planning.

Factors underlying surrogate medical decision-making in middle eastern and east Asian women: a Q-methodology study.

BACKGROUND: It is not clear how lay people prioritize the various, sometimes conflicting, interests when they make surrogate medical decisions, especially in non-Western cultures. The extent such decisions are perspective-related is also not well documented. METHODS: We explored the relative importance of 28 surrogate decision-making factors to 120 Middle-Eastern (ME) and 120 East-Asian (EA) women from three perspectives, norm-perception (N), preference as patient (P), and preference as surrogate decision-maker (S). Each respondent force-ranked (one to nine) 28 opinion-items according to each perspective. Items' ranks were analyzed by averaging-analysis and Q-methodology. RESULTS: Respondents' mean (SD) age was 33.2 (7.9) years; all ME were Muslims, 83% of EA were Christians. "Trying everything possible to save patient," "Improving patient health," "Patient pain and suffering," and/or "What is in the best interests of patient" were the three most-important items, whereas "Effect of caring for patient on all patients in society," "Effect of caring for patient on patients with same disease," and/or "Cost to society from caring for patient" were among the three least-important items, in each ME and EA perspectives. P-perspective assigned higher mean ranks to family and surrogate's needs and burdens-related items, and lower mean rank to "Fear of loss" than S-perspective (p<0.001). ME assigned higher mean ranks to "Medical facts" and "Surrogate own wishes for patient" and lower mean rank to "Family needs" in all perspectives (p<0.001). Q-methodology identified models that were relatively patient's preference-, patient's religious/spiritual beliefs-, or emotion-dependent (all perspectives); medical facts-dependent (N- and S-perspectives), financial needs-dependent (P- and S-perspectives), and family needs-dependent (P-perspective). CONCLUSIONS: 1) Patient's health was more important than patient's preference to ME and EA women; society interest was least important. 2) Family and surrogate's needs/ burdens were more important, whereas fear of loss was less important to respondents as patients than as surrogate decision-makers. 3) Family needs were more important to EA than ME respondents, the opposite was true for medical facts and surrogate's wishes for patient. 4) Q-methodology models that relatively emphasized various surrogate decision-making factors overlapped the ME and EA women' three perspectives.

Patient-centered care during the last year of life: adaptation and validation of the German PACIC short form for bereaved persons as proxies (PACIC-S9-proxy).

BACKGROUND: Providing patient-centered care (PCC) during the last year of life (LYOL) can be challenging due to the complexity of the patients' medical, social and psychological needs, especially in case of chronic illnesses. Assessing PCC can be helpful in identifying areas for improvements. Since not all patients can be surveyed, a questionnaire for proxy informants was developed in order to retrospectively assess patient-centeredness in care during the whole LYOL. This study aimed to evaluate the feasibility and validity of an adapted version of the German Patient Assessment of Chronic Illness Care (PACIC) for surveying bereaved persons in order to assess PCC during the decedents' LYOL. METHODS: The German PACIC short form (11 items) was adapted to a nine-item version for surveying bereaved persons on the decedent's LYOL (PACIC-S9-Proxy). Items were rated on a five-point Likert scale. The PACIC adaptation and validation was part of a cross-sectional survey in the region of Cologne. Participants were recruited through self-selection and active recruitment by practice partners. Sociodemographic characteristics and missing data were analyzed using descriptive statistics. An exploratory factor analysis was conducted in order to assess the structure of the PACIC-S9-Proxy. Internal consistency was estimated using Cronbach's alpha. RESULTS: Of the 351 informants who participated in the survey, 230 (65.52%) considered their decedent to have suffered from chronic illness prior to death. 193 of these informants (83.91%) completed ≥5 items of the questionnaire and were included in the analysis. The least answered item was item (74.09%) was item 4 (encouragement to group & classes for coping). The most frequently answered item (96.89%) was item 2 (satisfaction with care organization). Informants rated the item" Given a copy of their treatment plan" highest (mean 3.96), whereas "encouragement to get to a specific group or class to cope with the condition" (mean 1.74) was rated lowest. Cronbach's alpha was 0.84. A unidimensional structure of the questionnaire was found (Kaiser-Meyer-Olkin 0.86, Bartlett's test for sphericity p < 0.001), with items' factor loadings ranging from 0.46 to 0.82. CONCLUSIONS: The nine-item questionnaire can be used as efficient tool for assessing PCC during the LYOL retrospectively and by proxies. TRIAL REGISTRATION: The study was registered in the German Clinical Trials Register ( DRKS00011925 ) on 13 June 2017.

Do caregiver proxy reports and congruence of client-proxy activity participation goals relate to quality of life in people with aphasia?

BACKGROUND: Achieving activity participation goals is a key factor in quality of life (QOL) for people with aphasia (PWA), but expressing participation goals can be difficult for many of them. Proxy reports by caregivers may not accurately reflect the interests and participation goals of PWA, and discrepancies in these goals between PWA and their caregivers may affect QOL, based on the assumption that caregivers' awareness of their loved ones' unique participation goals may be important to increasing PWA activity participation. AIMS: To examine everyday activities valued by PWA using the Life Interests and Values (LIV) Cards; to measure congruence between PWA and their caregivers on life participation goals; and to measure how congruence of PWA-caregiver participation goals related to QOL. METHODS & PROCEDURES: A convenience sample of 25 PWA completed the LIV Card assessment and the Stroke Aphasia Quality of Life Scale-39 to assess participation goals and QOL. Participation goals were also evaluated with respect to age, time post-onset and aphasia severity. A total of 12 caregivers were administered the LIV Cards to calculate agreement between PWA-proxy activity reports and the relationship between agreement and QOL. OUTCOMES & RESULTS: PWA endorsed wanting to participate more in a wide range of activities, with common interests in walking/running, going to the beach and eating out, among others. PWA-caregiver activity agreement was fair to moderate with point-to-point agreement averaging 70%. However, no relationship between degree of congruence in PWA-proxy pairs and QOL was found. CONCLUSIONS & IMPLICATIONS: PWA have a variety of activity participation goals that can be integrated into intervention plans. Dependence on proxy respondents should be reduced as much as possible to support self-determination for PWA. What this paper adds What is already known on the subject Achieving activity participation goals is a key factor in QOL for PWA, but communicating about participation goals can be difficult for many of them. Because proxy reports by caregivers may not accurately reflect the interests and participation goals of PWA, this study examined how both PWA and their caregivers responded to an aphasia-friendly assessment for determining participation goals, and then compared level of agreement about these goals to QOL. Because activity participation is known to be an important factor in QOL, the reason for investigating how agreement relates to QOL is that caregivers' awareness of their loved ones' unique participation goals likely facilitates increased participation by PWA in their ongoing desired activities. The relationship between PWA-caregiver agreement regarding participation goals and QOL in PWA had not yet been investigated before this study. What this paper adds to existing knowledge This study adds additional as well as confirmatory information to the existing literature about life participation goals of community-dwelling individuals with chronic aphasia. Top activities endorsed by a group of 25 PWA are reported within four activity domains (home and community activities, creative and relaxing activities, physical activities, and social activities). Results indicated that agreement between PWA and their caregiver proxies on PWA's most desired activities was < 50%. However, the level of agreement between caregivers and proxies on participation goals was not significantly related to QOL in this sample. What are the potential or actual clinical implications of this work? PWA have a variety of participation goals that can be integrated into intervention plans to be carried out with clinicians, caregivers and family members. The use of proxy respondents when determining participation goals should be reduced as much as possible to support self-determination for PWA. Use of the LIV Cards, a picture-based sorting-task assessment, reduces the need for proxy responders and guesswork about the specific participation goals of PWA.

Exploring Family Decisions to Refuse Organ Donation at Imminent Death.

Communication about organ donation at the time of imminent death is a meaningful, yet less understood, area of health communication. We employed a multiple goals framework to explore family normative perceptions of organ donation and the conversational goal tensions experienced during a family member's imminent death. Semi-structured interviews were conducted with 14 family members who refused to donate when approached by an organ procurement coordinator (OPC) upon the imminent death of a family member. Thematic analysis revealed that family members described their decisions to refuse donation as (a) last acts of love, (b) responses to unnecessary requests, and (c) consistent with the known beliefs of the patient. Participants described several goal tensions operating within the organ donation conversation itself, including (a) the management of frequent requests, (b) pressure to donate, and (c) enduring unwanted requests from the OPC. Communication goals frameworks offer practical insights for improving organ-related conversations.

A Multicenter Study of the Causes and Consequences of Optimistic Expectations About Prognosis by Surrogate Decision-Makers in ICUs.

OBJECTIVES: Optimistic expectations about prognosis by surrogate decision-makers in ICUs are common, but there are few data about the causes and clinical consequences. Our objective was to determine the causes of optimistic expectations about prognosis among surrogates and whether it is associated with more use of life support at the end of life. DESIGN: Prospective, multicenter cohort study from 2009 to 2012. SETTING: Twelve ICUs from multiple regions of the United States. SUBJECTS: The surrogates and physicians of 275 incapacitated ICU patients at high risk of death. INTERVENTIONS: None. MEASUREMENTS AND MAIN RESULTS: Surrogates and physicians completed a validated instrument assessing their prognostic expectations for hospital survival. We determined the proportion of surrogates with optimistic expectations, defined as a prognostic estimate that was at least 20% more optimistic than the physician's, then determined how frequently this arose from surrogates miscomprehending the physicians' prognosis versus holding more hopeful beliefs compared with the physician. We used multivariable regression to examine whether optimistic expectations were associated with length of stay, stratified by survival status, and time to withdrawal of life support among nonsurvivors. Overall, 45% of surrogates (95% CI, 38-51%) held optimistic expectations about prognosis, which arose from a combination of misunderstanding the physician's prognostic expectations and from holding more hopeful beliefs compared with the physician. Optimistic expectations by surrogates were associated with significantly longer duration of ICU treatment among nonsurvivors before death (ß coefficient = 0.44; 95% CI, 0.05-0.83; p = 0.027), corresponding to a 56% longer ICU stay. This difference was associated with a significantly longer time to withdrawal of life support among dying patients whose surrogates had optimistic prognostic expectations compared with those who did not (ß coefficient = 0.61; 95% CI, 0.16-1.07; p = 0.009). CONCLUSIONS: The prevalent optimism about prognosis among surrogates in ICUs arises both from surrogates' miscomprehension of physicians' prognostications and from surrogates holding more hopeful beliefs. This optimism is associated with longer duration of life support at the end of life.

How Should We Capture Health State Utility in Dementia? Comparisons of DEMQOL-Proxy-U and of Self- and Proxy-Completed EQ-5D-5L.

BACKGROUND: Dementia-specific and proxy-completed preference-based measures have been proposed for use in intervention studies involving people living in residential care, in instances where generic, self-reported preference-based measures have been deemed inappropriate. OBJECTIVE: This study was conducted to investigate the construct validity, criterion validity, and responsiveness of DEMQOL-Proxy-U and of self- and proxy-completed EQ-5D-5L. METHODS: The analysis used a 3-wave, individual-level data set of 1004 people living with dementia in residential care that included self-completed EQ-5D-5L and formal-carer and informal-carer proxy-completed EQ-5D-5L and DEMQOL-Proxy-U utility values, in addition to other nonutility cognitive measures (Functional Assessment Staging [FAST], Clinical Dementia Rating [CDR], Cohen-Mansfield Agitation Inventory [CMAI]) and health-related quality of life (HRQOL) measures (nursing home version of the Quality of Life with Alzheimer's disease scale [QOL-AD-NH], Quality of Life in Late-Stage Dementia [QUALID] scale). Construct validity, criterion validity, and responsiveness were assessed using correlation, Bland-Altman plots, and panel data regression models. RESULTS: Self-completed EQ-5D-5L failed to reflect clinically important differences and changes in FAST, CDR, and CMAI but did capture the resident's own view of HRQOL (QOL-AD-NH). As dementia severity increased, collection of EQ-5D-5L-proxy and DEMQOL-Proxy-U data was more feasible than collection of self-completed EQ-5D-5L. These formal-carer and informal-carer proxy measures also better reflected changes in FAST, CDR, and CMAI but did not capture the resident's own view of HRQOL (QOL-AD-NH), despite adequately capturing the proxy's own view of the resident's HRQOL (QUALID). This indicates discrepancies between a proxy's view and resident's view of the impact that tangible declines in health, cognition, or functional abilities have on HRQOL. The EQ-5D-5L-proxy and DEMQOL-Proxy-U were generally poor substitutes. Regardless of which proxy completed it, the EQ-5D-5L-proxy was typically more responsive than the DEMQOL-Proxy-U to changes in CDR, FAST, and CMAI, indicating that use of the DEMQOL-Proxy-U is not always justified. CONCLUSION: Disparities in the measurement properties of different utility measures mean that choices about how to measure utility in trials could affect economic evaluation outcomes and hence how resources are allocated for dementia care.

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.

'It's a tough decision': a qualitative study of proxy decision-making for research involving adults who lack capacity to consent in UK.

BACKGROUND: Research into dementia and other conditions connected with cognitive impairments is essential but conducting research with populations who lack capacity to provide consent involves a number of ethical, legal and practical challenges. In England and Wales, family members can act as a consultee or legal representative on behalf of someone who lacks capacity. However, there is a paucity of research about how family members make decisions concerning research participation. OBJECTIVE: To explore family members' experiences of proxy decision-making for research. Understanding how proxy decisions are made could lead to interventions to support greater inclusion of individuals in research who have impaired decision-making capacity. METHODS: Semi-structured interviews were conducted with a purposive sample of 17 family members who had experience as a proxy for making decisions about participation in research, including those who had agreed to participation and those who declined. Thematic analysis was used to examine experiences and generate findings for research practice and to develop future supportive interventions. RESULTS: Proxy decision-making is highly contextualised. Proxies balance a number of factors when deciding about research participation, including the person's values and preferences, within the specific context of the study, and the practicalities of being involved. Proxies use these factors to construct a decision that is authentic to the person they care for. CONCLUSIONS: Proxy decision-making for research is a complex process with inter-woven layers of decision-making. Decisions can be problematic for some proxies who may benefit from decision support to make an informed decision about research participation on behalf of a family member.

Patient versus proxy response on global health scales: no meaningful DIFference.

PURPOSE: Assessment of outcomes from a proxy is often substituted for the patient's self-report when the patient is unable or unwilling to report their status. Research has indicated that proxies over-report symptoms on the patient's behalf. This study aimed to quantify the extent of proxy-introduced bias on the Patient-Reported Outcomes Measurement Information System Global Health (PROMIS GH) scale for mental (GMH) and physical (GPH) scores. METHODS: This retrospective cohort study included incident stroke patients seen in a cerebrovascular clinic who completed PROMIS GH between 10/12/15 and 6/6/18. Differential item functioning (DIF) evaluated measurement invariance of patient versus proxy responses. DIF impact was assessed by comparing the initial score to the DIF-adjusted score. Subgroup analyses evaluated DIF within strata of stroke severity, measured by modified Rankin Scale (≤ 1, 2, 3+), and time since stroke (≤ 30, 31-90, > 90 days). RESULTS: Of 1351 stroke patients (age 60.5 ± 14.9, 45.1% female), proxy help completing PROMIS GH was required by 406 patients (30.1%). Proxies indicated significantly worse response to all items. No items for GMH or GPH were identified as having meaningful DIF. In subgroup analyses, no DIF was found by severity or 31-90 days post-stroke. In patients within 30 and > 90 days of stroke, DIF was detected for 2 items. Accounting for DIF had negligible effects on scores. CONCLUSIONS: Our findings revealed the overestimation of symptoms by proxies is a real difference and not the result of measurement non-invariance. PROMIS GH items do not perform differently or have spuriously inflated severity estimates when administered to proxies instead of patients.

Exploring the views of being a proxy from the perspective of unpaid carers and paid carers: developing a proxy version of the Adult Social Care Outcomes Toolkit (ASCOT).

BACKGROUND: Outcomes-based policy and administration of public services present a compelling argument for the value of outcomes data. However, there are a number of challenges inherent in collecting these data from people who are unable to complete a paper-based survey or interview due to cognitive or communication impairments. In this paper, we explore the views of being a proxy from the perspective of unpaid carers and paid carers who may be asked to act as a proxy on behalf of the person(s) they care for. We consider the key issues that need to be addressed when adapting an instrument designed to measure social care outcomes, the Adult Social Care Outcomes Tool (ASCOT), into a proxy-report tool. METHODS: Participants took part in either a focus group (35 paid carers in eight focus groups), or a one-to-one interview (eight unpaid carers). All participants were recruited via carer organisations and care providers. Transcripts, field notes and audio data collected during focus groups and interviews were analysed using a thematic framework approach. RESULTS: Participants agreed that any person acting as a proxy would need to be very familiar with the care recipient, as well as their needs and care provision. A number of provisions for proxy respondents were proposed to improve face validity and acceptability of completing a questionnaire by proxy, and to ensure that any potential bias is reduced in the design of the questionnaire. These included: providing two sets of response options for each proxy perspective (the proxy themselves and the proxy view of how they think the care recipient would respond); a comments box to help people explain why they have selected a given response option (especially where these indicate unmet need); and providing clear guidance for the proxy respondent on how they should complete the questionnaire. CONCLUSIONS: This study has shown some of the challenges involved in assessing outcomes by proxy and explored some potential ways these can be mitigated. The findings highlight the benefits of developing and testing proxy measures in a robust way to widen participation in social care research.