The use of proxy measurement of internal states in people with intellectual disability: A scoping review.

BACKGROUND: The measurement of internal states of people with an intellectual disability is challenging. Given these difficulties, proxy respondents are sometimes used. However, the literature provides some caution about the validity of the use of proxy respondents. AIMS: This review aims to identify the available evidence regarding the use of proxy respondents in the measurement of internal states of people with intellectual disabilities. METHOD: The Arksey and O'Malley scoping review framework was used for this review, refined with the Joanna Briggs Institute Methodology. RESULTS: Results indicate there are some differences in the findings reported with respect to the efficacy of the use of proxy respondents. These may be due to a number of inconsistencies and weaknesses in examinations of their utility. CONCLUSIONS: If proxy-reports are to be useful in understanding the internal states of people with intellectual disability, researchers need to address the shortcomings revealed by this review.

Reliability of proxy reports on patient reported outcomes measures in stroke: An updated systematic review.

OBJECTIVES: With the rising global burden of stroke-related morbidity, and increased focus on patient-centered healthcare, patient reported outcome measures (PROMs) are increasingly used to inform healthcare decision-making. Some stroke patients with cognitive or motor impairments are unable to respond to PROMs, so proxies may respond on their behalf; the reliability of which remains unclear. The aim of the study is to update a 2010 systematic review to investigate the inter-rater reliability of proxy respondents answering PROMs for stroke patients. MATERIALS AND METHODS: Studies on the reliability of proxy respondents in stroke were searched within CINAHL, Embase, PsycInfo, and WoS databases (01/07/22, 08/07/22). Fifteen studies were included for review. ICC and k-statistic were extracted for PROMs scales and categorized as poor (0.80). Bias was assessed using the CCAT. RESULTS: Five studies reported PROMs with inter-rater reliability scores ranging from 0.80. Two studies reported activities of daily living (ADLs) scores ranging from 0.41 to 0.80 and 8 studies reported quality of life (QoL) measures with scores ranging from 0.80. Subcategories of these scales included physical (ICC/k-statistic 0.41- >0.8), cognitive (ICC/k-statistic 0.40-0.80), communication (ICC/k-statistic <0.4-0.80,) and psychological (ICC/k-statistic <0.40-0.60) measures. CONCLUSIONS: Proxy respondents are reliable sources for PROM reports on physical domains in ADLs, PROMs and QoL scales. Proxy reports for measures of communication and psychological domains had greater variability in reliability scores, ranging from poor to substantial; hence, caution should be applied when interpreting proxy reports for these domains.

EMPOWER: A Multi-Site Pilot Trial to Reduce Distress in Surrogate Decision-Makers in the ICU.

CONTEXT: Efforts to reduce the psychological distress of surrogate decision-makers of critically ill patients have had limited success, and some have even exacerbated distress. OBJECTIVES: The aim of this study was to determine the feasibility, acceptability, and preliminary efficacy of EMPOWER (Enhancing and Mobilizing the POtential for Wellness and Resilience), an ultra-brief (∼2-hour), 6-module manualized psychological intervention for surrogates. METHODS: Surrogates who reported significant anxiety and/or an emotionally close relationship with the patient (n=60) were randomized to receive EMPOWER or enhanced usual care (EUC) at one of three metropolitan hospitals. Participants completed evaluations of EMPOWER's acceptability and measures of psychological distress pre-intervention, immediately post-intervention, and at 1- and 3-month follow-up assessments. RESULTS: Delivery of EMPOWER appeared feasible, with 89% of participants completing all 6 modules, and acceptable, with high ratings of satisfaction (mean=4.5/5, SD = .90). Compared to EUC, intent-to-treat analyses showed EMPOWER was superior at reducing peritraumatic distress (Cohen's d = -0.21, small effect) immediately post-intervention and grief intensity (d = -0.70, medium-large effect), posttraumatic stress (d = -0.74, medium-large effect), experiential avoidance (d = -0.46, medium effect), and depression (d = -0.34, small effect) 3 months post-intervention. Surrogate satisfaction with overall critical care (d = 0.27, small effect) was higher among surrogates randomized to EMPOWER. CONCLUSIONS: EMPOWER appeared feasible and acceptable, increased surrogates' satisfaction with critical care, and prevented escalation of posttraumatic stress, grief, and depression 3 months later.

How do surrogates make treatment decisions for patients with dementia: An experimental survey study.

Despite the growing need for surrogate decision-making for older adults, little is known about how surrogates make decisions and whether advance directives would change decision-making. We conducted a nationally representative experimental survey that cross-randomized cognitive impairment, gender, and characteristics of advance care planning among hospitalized older adults through a series of vignettes. Our study yielded three main findings: first, respondents were much less likely to recommend life-sustaining treatments for patients with dementia, especially after personal exposure. Second, respondents were more likely to ignore patient preferences for life-extending treatment when the patient had dementia, and choose unwanted life-extending treatments for patients without dementia. Third, in scenarios where the patient's wishes were unclear, respondents were more likely to choose treatments that matched their own preferences. These findings underscore the need for improved communication and decision-making processes for patients with cognitive impairment and highlight the importance of choosing a surrogate decision-maker with similar treatment preferences.

What Counts as a Surrogate Decision?

When patients lose decision-making capacity, others must make surrogate decisions on their behalf. What counts as a surrogate decision might seem self-evident. But as clinician-researchers in the field of advance care planning, we have found that it is not always so clear-cut. In this paper, we describe how and why this is a matter of concern, a novel approach for assessing whether a surrogate decision occurred, and findings from this assessment.

Parents underestimate fatigue in younger children aged 5-7 years with asthma but not in older children.

AIM: To assess fatigue in children aged 2-17 years with asthma from both child and parent perspectives and describe associated factors. METHODS: Fatigue scores were self-reported by children aged 5-17 years old and proxy-reported by parents or carers for all children. The Pediatric Quality of Life Inventory Multidimensional Fatigue Scale was used. Fatigue scores ranged from 0 to 100, higher scores meant less fatigue. RESULTS: There were 527 children and parents enrolled. The mean overall fatigue score by self-report was 72.7 ± 15.8 and by proxy report was 75.8 ± 16.3. Self-reported fatigue score was lower in children aged 5-7 years (71.5 ± 15.9) compared to proxy-reported score (76.3 ± 15.5). Proxy and self-reported fatigue scores were similar between parents and older children. Fatigue scores were lower in association with poor asthma control and receipt of social support. Lower self-reported, but not proxy-reported, fatigue score was related to asthma severity. Lower proxy-reported, but not self-reported, fatigue score was related to the child being older and having shortness of breath. CONCLUSION: Parents underestimated the fatigue of younger children aged 5-7 years, but fatigue scores were similar between parents and older children. Both clinical and social factors are associated with fatigue in children with asthma.

The Mediating Role of Proxy Respondents on the Relationship Between Cognitive Function and Self-Rated Health.

BACKGROUND AND OBJECTIVES: Proxy respondents are an important tool in survey research, especially among people with cognitive impairment. However, proxy respondents may be unable to accurately answer subjective survey instruments for cognitively impaired persons. This study investigates the mediating effect of proxy status on the relationship between cognitive impairment and subjectively rated health. RESEARCH DESIGN AND METHODS: Respondents from the 2018 wave of the Health and Retirement Study (N = 17,146) were included, and the Baron and Kenny method assessed the potential mediating role of having a proxy on subjectively rated health. Subjectively rated health and proxy status were dichotomously coded, and relationships between cognitive impairment, proxy status, and subjectively rated health were assessed using logistic regression. RESULTS: Findings indicate that cognitive impairment is inversely associated with subjectively rated health, and it is directly associated with having a proxy. They also indicate that having a proxy is inversely related to subjectively rated health. When including proxy status and cognitive impairment in the same model, cognitive impairment no longer predicted subjectively rated health. This indicates that proxy status perfectly mediated the relationship between cognitive impairment and subjectively rated health. DISCUSSION AND IMPLICATIONS: Measuring the experiences of people with cognitive impairment is challenging in survey research due to the limiting features of cognitive impairment. Although having a proxy respondent helps address attrition-related measurement challenges in survey research, findings indicate that proxies report worse subjectively rated health for people with cognitive impairment compared to individuals with comparable levels of cognitive impairment without a proxy. Future research may benefit from exploring optimal proxy-respondent characteristics.

Against an Inflexible, Prioritized List for Default Surrogate Selection.

AbstractSurrogate selection can be extremely consequential for patients. Most surrogates are selected by default, so we should care about whether legal provisions for default surrogate selections are ethically justified. Most U.S. states use an inflexible, prioritized list of relationships, that is, a hierarchical list where eligible classes of higher-ranked individuals must be selected before lower-ranked individuals. I argue that while some inflexible, prioritized lists may roughly reflect the order that many patients would select, there is a significant minority that inflexible lists systematically disempower. This is morally unacceptable given the availability of less morally problematic alternatives. One alternative is a flexible, prioritized list, which provides conditions for lower-ranked individuals to be selected ahead of higher-ranked ones. I argue that since all the U.S. states that currently have an inflexible, prioritized list systematically disempower a significant proportion of their residents, they have good reason to adopt a flexible, prioritized list instead. Furthermore, the Universal Law Commission currently recommends that states adopt an inflexible, prioritized list, so they have good reason to change their recommendation.

When Should Providers Defer versus Impose Their Views?

AbstractThis piece discusses perhaps the most agonizing ethical decision ethics consultants and other providers encounter. This is the extent to which providers should defer decisions to patients or to their proxy decision makers as opposed to imposing their own views as to what they think is ethically right. It discusses the most difficult issues these providers may encounter, especially when they wish to depart from authoritative bodies' standards or guidelines, and it presents initial steps providers may take to help patients and their families work together to resolve these dilemmas more harmoniously. It highlights how providers may inadvertently impose flawed biases on patients and families. Finally, it discusses how providers should take initiative with both parties to offer to help appeal when these avenues already exist and seek to establish the appellate procedures when they are absent.

Opportunities and challenges of self-binding directives: A comparison of empirical research with stakeholders in three European countries.

BACKGROUND: Self-binding directives (SBDs) are psychiatric advance directives that include a clause in which mental health service users consent in advance to involuntary hospital admission and treatment under specified conditions. Medical ethicists and legal scholars identified various potential benefits of SBDs but have also raised ethical concerns. Until recently, little was known about the views of stakeholders on the opportunities and challenges of SBDs. AIMS: This article aims to foster an international exchange on SBDs by comparing recent empirical findings on stakeholders' views on the opportunities and challenges of SBDs from Germany, the Netherlands, and the United Kingdom. METHOD: Comparisons between the empirical findings were drawn using a structured expert consensus process. RESULTS: Findings converged on many points. Perceived opportunities of SBDs include promotion of autonomy, avoidance of personally defined harms, early intervention, reduction of admission duration, improvement of the therapeutic relationship, involvement of persons of trust, avoidance of involuntary hospital admission, addressing trauma, destigmatization of involuntary treatment, increase of professionals' confidence, and relief for proxy decision-makers. Perceived challenges include lack of awareness and knowledge, lack of support, undue influence, inaccessibility during crisis, lack of cross-agency coordination, problems of interpretation, difficulties in capacity assessment, restricted therapeutic flexibility, scarce resources, disappointment due to noncompliance, and outdated content. Stakeholders tended to focus on practical challenges and did not often raise fundamental ethical concerns. CONCLUSIONS: Stakeholders tend to see the implementation of SBDs as ethically desirable, provided that the associated challenges are addressed.

Development of consensus-based considerations for use of adult proxy reporting: an ISOQOL task force initiative.

AIMS: Many large-scale population-based surveys, research studies, and clinical care allow for inclusion of proxy reporting as a strategy to collect outcomes when patients are unavailable or unable to provide reliable self-report. Prior work identified an absence of methodological guidelines regarding proxy reporting in adult populations, including who can serve as a proxy, and considerations for data collection, analysis, and reporting. The primary objective of this work by the ISOQOL Proxy Task Force was to review documents and clinical outcome assessment measures with respect to proxy reporting and to develop, through consensus, considerations for proxy reporting. METHODS: We assembled an international group with clinically relevant and/or methodological expertise on proxy use in adult populations. We conducted a targeted review of documentation based on regulatory, non-regulatory, professional society, and individual measure sources. Using a standardized collection form, proxy-related information was extracted from each source including definitions of a proxy, characteristics of a proxy, domains addressable or addressed by a proxy, and observer-reporting. RESULTS: The definition of proxy was inconsistent across 39 sources, except regulatory documents which defined a proxy as a person other than the patient who reports on an outcome as if she/he were the patient. While proxy report was discouraged in regulatory documentation, it was acknowledged there were instances where self-report was impossible. Many documentation sources indicated proxies would be well-justified in certain contexts, but did not indicate who could act as a proxy, when proxies could be used, what domains of patient health they could report on, or how data should be reported. Observer-reported outcomes were typically defined as those based on observed behaviors, however there was not a consistent differentiation between proxy and observer reporting. Based on information extracted from these resources, we developed a checklist of considerations when including proxy-reported measures or using proxies in study design, data collection, analysis, interpretation and reporting of proxy reported data. CONCLUSION: Our targeted review highlights a lack of clarity in capturing, interpreting and reporting data from proxies in adult populations. We provide a checklist of considerations to assist researchers and clinicians with including proxies in research studies and clinical care. Lastly, our review identified areas where further guidance and future research are necessary.

Validation of the parent-proxy version of the pediatric Charcot-Marie-Tooth disease quality of life instrument for children aged 0-7 years.

OBJECTIVE: To evaluate the parent-proxy version of the pediatric Charcot Marie Tooth specific quality of life (pCMT-QOL) outcome instrument for children aged 7 or younger with CMT. We have previously developed and validated the direct-report pCMT-QOL for children aged 8-18 years and a parent proxy version of the instrument for children 8-18 years old. There is currently no CMT-QOL outcome measure for children aged 0-7 years old. METHODS: Testing was conducted in parents or caregivers of children aged 0-7 years old with CMT evaluated at participating INC sites from the USA, United Kingdom, and Australia. The development of the instrument was iterative, involving identification of relevant domains, item pool generation, prospective pilot testing and clinical assessments, structured focus group interviews, and psychometric testing. The parent-proxy instrument was validated rigorously by examining previously identified domains and undergoing psychometric tests for children aged 0-7. RESULTS: The parent-proxy pCMT-QOL working versions were administered to 128 parents/caregivers of children aged 0-7 years old between 2010 and 2016. The resulting data underwent rigorous psychometric analysis, including factor analysis, internal consistency, and convergent validity, and longitudinal analysis to develop the final parent-proxy version of the pCMT-QOL outcome measure for children aged 0-7 years old. CONCLUSIONS: The parent-proxy version of the pCMT-QOL outcome measure, known as the pCMT-QOL (0-7 years parent-proxy) is a valid and sensitive proxy measure of health-related QOL for children aged 0-7 years with CMT.

Black and white proxy experiences and perceptions that influence advanced dementia care in nursing homes: The ADVANCE study.

BACKGROUND: Regional, facility, and racial variability in intensity of care provided to nursing home (NH) residents with advanced dementia is poorly understood. MATERIALS AND METHODS: Assessment of Disparities and Variation for Alzheimer's disease NH Care at End of life (ADVANCE) is a multisite qualitative study of 14 NHs from four hospital referral regions providing varied intensity of advanced dementia care based on tube-feeding and hospital transfer rates. This report explored the perceptions and experiences of Black and White proxies (N = 44) of residents with advanced dementia to elucidate factors driving these variations. Framework analyses revealed themes and subthemes within the following a priori domains: understanding of advanced dementia and care decisions, preferences related to end-of-life care, advance care planning, decision-making about managing feeding problems and acute illness, communication and trust in NH providers, support, and spirituality in decision-making. Matrix analyses explored similarities/differences by proxy race. Data were collected from June 1, 2018 to July 31, 2021. RESULTS: Among 44 proxies interviewed, 19 (43.1%) were Black, 36 (81.8%) were female, and 26 (59.0%) were adult children of residents. In facilities with the lowest intensity of care, Black and White proxies consistently reported having had previous conversations with residents about wishes for end-of-life care and generally better communication with providers. Black proxies held numerous misconceptions about the clinical course of advanced dementia and effectiveness of treatment options, notably tube-feeding and cardiopulmonary resuscitation. Black and White proxies described mistrust of NH staff but did so towards different staffing roles. Religious and spiritual beliefs commonly thought to underlie preferences for more intense care among Black residents, were rarely, but equally mentioned by race. CONCLUSIONS: This report refuted commonly held assumptions about religiosity and spirituality as drivers of racial variations in advanced dementia care and revealed several actionable facility-level factors, which may help reduce these variations.

Proxy Decision-Making for Clinical Research in Nursing Home Residents with Dementia: A Qualitative Analysis.

OBJECTIVES: The benefit-risk ratio of many interventions remains unclear in older adults with dementia. Efforts for more representative trial inclusion are made; however, recruiting and particularly gaining informed consent remains complex. For research participation, dementia compels the designation of a legal guardian (LG) to give proxy consent. To advance future trial development, we aimed to provide more insights into the factors that affect the proxy decision-making process in dementia research. DESIGN: A qualitative analysis of semi-structured interviews about proxy decision-making on participation in dementia research. SETTING AND PARTICIPANTS: LGs of nursing home residents that gave (n = 19) and refrained from giving (n = 18) proxy consent for a clinical trial (the Danton study) in the Netherlands. METHODS: Verbatim transcripts were thematically analyzed by using a preliminary deductive framework with room for induction of additional emerging themes, being an overall abductive approach. Based on that theme list, related factors of the decision-making process were grouped into overarching levels and merged into a step-by-step process. RESULTS: When discussing proxy decision-making on the participation of an older adult with dementia in a clinical trial, LGs described interconnected factors on the level of the study and patient. Past experiences and attitudes of the LG influenced the weighing of these study- and patient-related factors, leading to a preliminary decision. Other proxies and treating health care professionals (HCPs) were named as important other stakeholders of the decision-making process. CONCLUSIONS AND IMPLICATIONS: When giving proxy consent for research participation, LGs weigh study- and patient-related factors, leading to an initial benefit-risk evaluation. This weighing process is influenced by LG-related factors and can be modulated by other proxies or treating HCPs, leading to a definitive decision. Although insights into these underlying mechanisms could facilitate the proxy decision-making process for both LGs and researchers, treating HCPs could act as an independent party.

Illness-related parental stress and quality of life in children with kidney diseases.

BACKGROUND: This cross-sectional study investigated quality of life (QoL) and illness-related parental stress in children with kidney diseases by (1) comparing mean levels of these two variables between several kidney disease categories; (2) exploring correlations between QoL and parental stress; and (3) describing which disease category reports lowest QoL and highest parental stress. METHODS: We included 295 patients with a kidney disease (0-18 years) and their parents, followed at 6 reference centers for pediatric nephrology. Children's QoL was assessed by the PedsQL™ 4.0 Generic Core Scales, and illness-related stress by the Pediatric Inventory for Parents. All patients were divided into 5 kidney disease categories according to the multidisciplinary care program criteria prescribed by the Belgian authorities: (1) structural kidney diseases, (2) tubulopathies and metabolic diseases, (3) nephrotic syndrome, (4) acquired diseases with proteinuria and hypertension, and (5) kidney transplantation. RESULTS: Child self-reports showed no differences in QoL between kidney disease categories, in contrast to parent proxy reports. Parents of transplant patients reported lower QoL in their child and more parental stress compared with the 4 non-transplant categories. QoL and parental stress were negatively correlated. Lowest QoL and highest parental stress scores were mainly found in transplant patients. CONCLUSIONS: This study showed lower QoL and higher parental stress in pediatric transplant patients compared with non-transplants, based on parent reports. Higher parental stress is associated with worse QoL in the child. These results highlight the importance of multidisciplinary care for children with kidney diseases, with special attention to transplant patients and their parents. A higher resolution version of the Graphical abstract is available as Supplementary information.

Alzheimer's, Advance Directives, and Interpretive Authority.

Philosophers have debated whether the advance directives of Alzheimer's patients should be enforced, even if patients seem content in their demented state. The debate raises deep questions about the nature of human autonomy and personal identity. But it tends to proceed on the assumption that the advance directive's terms are clear, whereas in practice they are often vague or ambiguous, requiring the patient's healthcare proxy to make difficult judgment calls. This practical wrinkle raises its own, distinct but related, philosophical question: what criteria may the proxy bring to bear when making such interpretive judgments on which the patient's life may depend? After defending a general policy of enforcing advance directives on normative (rather than metaphysical) grounds, I argue that when advance directives are vague, a patient's proxy may permissibly make her own fresh evaluation of the patient's life as a whole and, in so doing, consider how the patient's character as a demented person contributes or fails to contribute to that life.

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.

An evaluation of surrogate decision maker health literacy in the neurology ward and neuroscience ICU.

PURPOSE: There has been little investigation into surrogate comprehension of education provided by the neuroscience healthcare team. We sought to evaluate 1) surrogate understanding about a patient's clinical condition and 2) the relationship between how surrogates and the neuroscience team perceive surrogate comprehension of a patient's condition. METHODS: We prospectively surveyed surrogates of patients who lacked decision making capacity while admitted to the neurology ward or neuroscience ICU for >48 h from 10/2018-05/2021. The survey investigated the surrogate's communication with the neuroscience team and understanding of the hospitalization. A member of the neuroscience team was asked to provide clinical data about the patient and indicate how well they perceived the surrogate understood the situation. RESULTS: We surveyed 50 surrogates at a median of 10 (IQR 5-17) days after hospitalization. There were 38 (76%) surrogates who correctly identified the reason for admission, and 21 (42%) who correctly identified all the assessments/interventions performed. Nearly all surrogates rated their understanding of the patient's medical condition as excellent (47%) or good (49%). There was no to slight agreement (kappa = 0.133) between surrogate self-perception of understanding and neuroscience team perception of the surrogate's understanding. Although only 20% of surrogates used institutional electronic education materials, 74% discussed the hospitalization with friends/family who work in healthcare. CONCLUSION: Objective and subjective assessments of surrogate comprehension demonstrate that there is a need to improve communication with the surrogates of patients with neurological conditions. Neuroscience healthcare teams must be taught how to educate surrogates and assess their understanding.