From opt-out to opt-in consent for secondary use of medical data and residual biomaterial: An evaluation using the RE-AIM framework.

BACKGROUND: Patient records, imaging, and residual biomaterial from clinical procedures are crucial resources for medical research. In the Netherlands, consent for secondary research has historically relied on opt-out consent. For ethical-legal experts who purport passive consent undermines patient autonomy, opt-in consent (wherein affirmative action is required) is seen as the preferred standard. To date, there is little empirical research exploring patient feasibility, organizational consequences, and the potential risks for research based on secondary data. Thus, we applied the RE-AIM framework to evaluate the impact of migrating from an opt-out to an opt-in consent process. METHODS: This evaluation was carried out in Radboud University Medical Center, a large tertiary hospital located in the southeast of the Netherlands. All non-acute, mentally competent patients ≥16 years of age registered between January 13, 2020 and June 30, 2023 were targeted (N = 101,437). In line with the RE-AIM framework, individual and organizational consequences were evaluated across five domains: reach, efficacy, adoption, implementation, and maintenance. RESULTS: 101,437 eligible patients were approached of whom 66,214 (65.3%) consented, 8,059 (7.9%) refused consent and 27,164 (26.8%) had no response. Of the 74,273 patients with a response, 89.1% consented to secondary use. The migration to an opt-in consent system was modestly successful; yet notably, differential response patterns by key sociodemographic characteristics were observed. Adaptions to the process flow improved its effectiveness and resulted in a reasonable response over time. Implementation was most affected by budgetary restraints, thus impeding the iterative approach which could have further improved domain outcomes. CONCLUSION: This evaluation provides an overview of logistical and pragmatic issues encountered when migrating from opt-out to opt-in consent. Response bias remains a major concern. Though not always directly transferable, these lessons can be broadly used to inform other health care organizations of the potential advantages and pitfalls of an opt-in consent system.

Multimorbidity patterns in older persons and their association with self-reported quality of life and limitations in activities of daily living.

BACKGROUND: As populations age, multimorbidity (the presence of two or more chronic morbidities) is increasingly more common. These evolving demographics demand further research into the identification of morbidity patterns in different settings as well as the longitudinal effects of these patterns. METHODS: Prospectively collected data on 12,755 older persons aged 65+ years were derived from The Older Persons and Informal Caregivers Survey Minimum DataSet (TOPICS-MDS, www.topics-mds.eu). Latent class analyses were performed to identify unobserved relationship patterns between morbidities in older persons. Using linear mixed models, the average difference in health-related quality of life (EQ-5D) and general quality of life scores (Cantril's Self Anchoring Ladder) as well as limitations in Activities of Daily Living and Instrumental Activities of Daily Living (ADL/IADL) were examined over a 12-month period. RESULTS: Five multimorbidity patterns were identified: sensory (n = 3882), cardio-metabolic (n = 2627), mental health (n = 920), osteo-articular (n = 4486), and system decline (n = 840). Relative to older persons in the sensory group, multimorbidity patterns did not have a strong effect on health-related quality of life, general quality of life or ADL/IADLs over a one-year period. CONCLUSIONS: The observed multimorbidity patterns are similar to others based on different methodologies and study populations. When examining the effect of such patterns on quality of life, the EQ-5D and Cantril's Ladder may be insufficient outcome measures. Further investigations into the prognostic value of morbidity patterns would be of benefit.

Expanding the boundaries of previously obtained informed consent in research: Views from participants in the Personalised Risk-based Mammascreening study.

INTRODUCTION: Understanding participants' concerns and information needs regarding broadened consent is crucial to ensure transparency and participant autonomy. Our study qualitatively examined these issues in women participating in the Personalized RISk-based MAmmascreening study (PRISMA). The original PRISMA informed consent was project-specific (i.e., breast cancer research), limiting the scope of secondary research. We explored participants' needs for broadened consent to preserve informed decision-making while maximising the potential re-use of data. METHODS: Focus groups (FGs) were performed following a semistructured discussion guide. Two independent researchers analysed the data thematically using an inductive approach. FINDINGS: Twenty-three asymptomatic women and 13 women diagnosed with breast cancer were randomly divided into six FGs. Four superordinate themes were identified: (1) Normalization, (2) Attitude towards the pharmaceutical industry, (3) Privacy and (4) Knowledge. Our participants viewed data sharing as an important conduit for advancing medical science. Perceived integrity was more often attributed to noncommercial than commercial parties, with a marked mistrust towards the pharmaceutical industry. Most requested information needs related to data protection. Participants' ideal consent process would confer a range of options; for example, they would be able to choose with whom data can be shared, whether data will be de-identified or anonymous, the expiration date of their consent and how, if requested, general and personal study results would be disclosed. CONCLUSION: Our participants expressed clear information needs and a strong desire to be actively engaged in future data sharing decisions. Given that many researchers collaborate with commercial parties, building public confidence in these institutions would be beneficial. Illustrative examples addressing privacy concerns and clarifying difficult terms would aid consent decision-making. Although our participants displayed great altruism in sharing their data and accepted that broad consent would ultimately facilitate future research, broad consent did not reflect their ideal situation. Dynamic consent may be an option but warrants further feasibility research. PATIENT AND PUBLIC CONTRIBUTION: Women were recruited from the general breast cancer screening population. Their perceptions and information needs, as reported in this study, will not only inform broadened consent for PRISMA but ideally guide other consent templates and decisions regarding consent processes.

Risk of bias assessment of sequence generation: a study of 100 systematic reviews of trials.

BACKGROUND: Systematic reviews of randomised trials guide policy and healthcare decisions. Yet, we observed that some reviews judge randomised trials as high or unclear risk of bias (ROB) for sequence generation, potentially introducing bias. However, to date, the extent of this issue has not been well examined. We evaluated the consistency in the ROB assessment for sequence generation of randomised trials in Cochrane and non-Cochrane reviews, and explored the reviewers' judgement of the quality of evidence for the related outcomes. METHODS: Cochrane intervention reviews (01/01/2017-31/03/2017) were retrieved from the Cochrane Database of Systematic Reviews. We also searched for systematic reviews in ten general medical journals with highest impact factors (01/01/2016-31/03/2017). We examined the proportion of reviews that rated the sequence generation domain as high, low or unclear risk of selection bias. For reviews that had rated any randomised trials as high or unclear risk of bias, we examined the proportion that had assessed the quality of evidence. RESULTS: Overall, 100 systematic reviews were included in our analysis. We evaluated 64 Cochrane reviews which comprised of 984 randomised trials; 0.8% (n = 8) and 52.2% (n = 514) were rated as high and unclear ROB for sequence generation respectively. We further evaluated 36 non-Cochrane reviews which comprised of 1376 trials; 5.8% (n = 80) and 39.6% (n = 545) were rated as high and unclear ROB respectively. Ninety percent (n = 10) of non-Cochrane reviews which rated randomised trials as high ROB for sequence generation did not report an underlying reason. All Cochrane reviews assessed the quality of evidence (GRADE). For the non-Cochrane reviews, only just over half had assessed the quality of evidence. CONCLUSION: Systematic reviews of interventions frequently rate randomised trials as high or unclear ROB for sequence generation. In general, Cochrane reviews were more transparent than non-Cochrane reviews in ROB and quality of evidence assessment. The scientific community should more strongly promote consistent ROB assessment for sequence generation to minimise selection bias and support transparent quality of evidence assessment. Consistency ensures that appropriate conclusions are drawn from the data.

Development of a Patient-Reported Outcome Measure for Geriatric Care: The Older Persons and Informal Caregivers Survey Short Form.

BACKGROUND: The Older Persons and Informal Caregivers Minimum Data Set (TOPICS-MDS) is a standardized data set that was developed to evaluate the quality of multidimensional geriatric care. There is an inherent need to reduce the number of TOPICS-MDS survey items to core outcomes to allow it to be more easily applied as a patient-reported outcome measure in clinical settings. OBJECTIVES: To create a TOPICS-short form (TOPICS-SF) and examine its validity. METHODS: Data in the TOPICS-MDS from persons aged 65 years and older in the Netherlands were used for the following analyses. Multiple linear regression analyses were performed to select the items and to derive domain weights of TOPICS-SF. A priori hypotheses were made on the basis of psychometric properties of the full-length TOPICS-MDS preference-weighted score (TOPICS-CEP). The validity of TOPICS-SF was evaluated by 1) examining the meta-correlation of the TOPICS-SF score with TOPICS-CEP and two quality-of-life measures, that is, the Cantril Ladder score and the EuroQol five-dimensional questionnaire utility index, and 2) performing mixed multiple regression of TOPICS-SF scores across key sociodemographic characteristics. RESULTS: TOPICS-SF scores were strongly correlated with the TOPICS-CEP (r = 0.96) and had stronger correlation with the EuroQol five-dimensional questionnaire utility index compared with the Cantril Ladder (r = 0.61 and 0.38, respectively). TOPICS-SF scores were higher among older persons who were married, living independently, and having higher levels of education. CONCLUSIONS: We have developed the 22-item TOPICS-SF and demonstrated its validity, supporting its use as a patient-reported outcome measure in geriatric care.

Correction: Examining the construct and known-group validity of a composite endpoint for The Older Persons and Informal Caregivers Survey Minimum Data Set (TOPICS-MDS); A large-scale data sharing initiative.

[This corrects the article DOI: 10.1371/journal.pone.0173081.].

What do women want? Valuing women's preferences and estimating demand for alternative models of maternity care using a discrete choice experiment.

In many countries, there has been a considerable shift towards providing a more woman-centred maternity service, which affords greater consumer choice. Maternity service provision in Ireland is set to follow this trend with policymakers committed to improving maternal choice at hospital level. However, women's preferences for maternity care are unknown, as is the expected demand for new services. In this paper, we used a discrete choice experiment (DCE) to (1) investigate women's strengths of preference for different features of maternity care; (2) predict market uptake for consultant- and midwifery-led care, and a hybrid model of care called the Domiciliary In and Out of Hospital Care scheme; and (3) calculate the welfare change arising from the provision of these services. Women attending antenatal care across two teaching hospitals in Ireland were invited to participate in the study. Women's preferred model of care resembled the hybrid model of care, with considerably more women expected to utilise this service than either consultant- or midwifery-led care. The benefit of providing all three services proved considerably greater than the benefit of providing two or fewer services. From a priority setting perspective, pursuing all three models of care would generate a considerable welfare gain, although the cost-effectiveness of such an approach needs to be considered.

A Cost-Benefit Analysis of Two Alternative Models of Maternity Care in Ireland.

BACKGROUND: The Irish government has committed to expand midwifery-led care alongside consultant-led care nationally, although very little is known about the potential net benefits of this reconfiguration. OBJECTIVES: To formally compare the costs and benefits of the major models of care in Ireland, with a view to informing priority setting using the contingent valuation technique and cost-benefit analysis. METHODS: A marginal payment scale willingness-to-pay question was adopted from an ex ante perspective. 450 pregnant women were invited to participate in the study. Cost estimates were collected primarily, describing the average cost of a package of care. Net benefit estimates were calculated over a 1-year cycle using a third-party payer perspective. RESULTS: To avoid midwifery-led care, women were willing to pay €821.13 (95% CI 761.66-1150.41); to avoid consultant-led care, women were willing to pay €795.06 (95% CI 695.51-921.15). The average cost of a package of consultant- and midwifery-led care was €1,762.12 (95% CI 1496.73-2027.51) and €1018.47 (95% CI 916.61-1120.33), respectively. Midwifery-led care ranked as the best use of resources, generating a net benefit of €1491.22 (95% CI 989.35-1991.93), compared with €123.23 (95% CI -376.58 to 621.42) for consultant-led care. CONCLUSIONS: While both models of care are cost-beneficial, the decision to provide both alternatives may be constrained by resource issues. If only one alternative can be implemented then midwifery-led care should be undertaken for low-risk women, leaving consultant-led care for high-risk women. However, pursuing one alternative contradicts a key objective of government policy, which seeks to improve maternal choice. Ideally, multiple alternatives should be pursued.

Measurement properties of the EQ-5D across four major geriatric conditions: Findings from TOPICS-MDS.

BACKGROUND: As populations age, chronic geriatric conditions linked to progressive organ failure jeopardize health-related quality of life (HRQoL). Thus, this research assessed the validity and applicability of the EQ-5D (a common HRQoL instrument) across four major chronic geriatric conditions: hearing issues, joint damage, urinary incontinence, or dizziness with falls. METHODS: The study sample comprised 25,637 community-dwelling persons aged 65 years and older residing in the Netherlands (Data source: TOPICS-MDS, www.topics-mds.eu ). Floor and ceiling effects were examined. To assess convergent validity, random effects meta-correlations (Spearman's rho) were derived between individual EQ-5D domains and related survey items. To further examine construct validity, the association between sociodemographic characteristics and EQ-5D summary scores were assessed using linear mixed models. Outcomes were compared to the overall study population as well as a 'healthy' subgroup reporting no major chronic conditions. RESULTS: Whereas ceiling effects were observed in the overall study population and the 'healthy' subgroup, such was not the case in the geriatric condition subgroups. The majority of hypotheses regarding correlations between survey items and sociodemographic associations were supported. EQ-5D summary scores were lower in respondents who were older, female, widowed/single, lower educated, and living alone. Increasing co-morbidity had a clear negative effect on EQ-5D scores. CONCLUSION: This study supported the construct validity of the EQ-5D across four major geriatric conditions. For older persons who are generally healthy, i.e. reporting few to no chronic conditions, the EQ-5D confers poor discriminative ability due to ceiling effects. Although the overall dataset initially suggested poor discriminative ability for the EQ-5D, such was not the case within subgroups presenting with major geriatric conditions.

Examining the construct and known-group validity of a composite endpoint for The Older Persons and Informal Caregivers Survey Minimum Data Set (TOPICS-MDS); A large-scale data sharing initiative.

BACKGROUND: Preference-weighted multi-faceted endpoints have the potential to facilitate comparative effectiveness research that incorporates patient preferences. The Older Persons and Informal Caregivers Survey-Composite endpoint (TOPICS-CEP) is potentially a valuable outcome measure for evaluating interventions in geriatric care as it combines multiple outcomes relevant to older persons in a single metric. The objective of this study was to validate TOPICS-CEP across different study settings (general population, primary care and hospital). METHODS: Data were extracted from TOPICS Minimum Dataset (MDS), a pooled public-access national database with information on older persons throughout the Netherlands. Data of 17,603 older persons were used. Meta-correlations were performed between TOPICS-CEP indexed scores, EuroQol5-D utility scores and Cantril's ladder life satisfaction scores. Mixed linear regression analyses were performed to compare TOPICS-CEP indexed scores between known groups, e.g. persons with versus without depression. RESULTS: In the complete sample and when stratified by study setting TOPICS-CEP and Cantril's ladder were moderately correlated, whereas TOPICS-CEP and EQ-5D were highly correlated. Higher mean TOPICS-CEP scores were found in persons who were: married, lived independently and had an education at university level. Moreover, higher mean TOPICS-CEP scores were found in persons without dementia, depression, and dizziness with falls, respectively. Similar results were found when stratified by subgroup. CONCLUSION: This study supports that TOPICS-CEP is a robust measure which can potentially be used in broad settings to identify the effect of intervention or of prevention in elderly care.

What women want: Exploring pregnant women's preferences for alternative models of maternity care.

Depending on obstetric risk, maternity care may be provided in one of two locations at hospital level: a consultant-led unit (CLU) or a midwifery-led unit (MLU). Care in a MLU is sparsely provided in Ireland, comprising as few as two units out of a total 21 maternity units. Given its potential for greater efficiencies of care and cost-savings for the state, there has been an increased interest to expand MLUs in Ireland. Yet, very little is known about women's preferences for midwifery-led care, and whether they would utilise this service when presented with the choice of delivering in a CLU or MLU. This study seeks to involve women in the future planning of maternity care by investigating their preferences for care and subsequent motivations when choosing place of birth. Qualitative research is undertaken to explore maternal preferences for these different models of care. Women only revealed a preference for the MLU when co-located with a CLU due to its close proximity to medical services. However, the results suggest women do not have a clear preference for either model of care, but rather a hybrid model of care which encompasses features of both consultant- and midwifery-led care.

Quantitative cardiotocography to improve fetal assessment during labor: a preliminary randomized controlled trial.

OBJECTIVE: To evaluate the effectiveness of a computerized decision support system, referred to as "quantitative cardiotocography" (qCTG), to reduce adverse birth outcomes compared to conventional CTG with fetal blood sampling. STUDY DESIGN: A preliminary parallel randomized control trial in a tertiary maternity hospital (Sofia, Bulgaria) was conducted with a sample size of 360 women per trial arm (N=720). Women in labor were recruited between March 2008 and March 2011. Unadjusted relative risks were derived to assess the effect of qCTG on outcomes of interest. A ROC curve was derived to determine the sensitivity and specificity of qCTG to detect acidemia (Clinical trial registration: Current Controlled Trials, http://www.controlled-trials.com/, ISRCTN46449237). MAIN OUTCOME MEASURES: Primary outcomes were hypoxia (cord-artery blood pH<7.20), acidemia (umbilical-artery blood pH<7.05), cesarean delivery, and forceps extraction. Secondary outcomes were Apgar score <7 at five minutes, neonatal seizures, and admission to the neonatal intensive care unit (NICU). RESULTS: Reduced risks were observed for all outcomes of interest in women monitored using qCTG. There was a significant reduction in hypoxia (RR: 0.53; 0.33, 0.84), acidemia (RR: 0.31; 95% CI: 0.12, 0.84), cesarean delivery (95% CI: 0.45, 0.85), and admission to the NICU (RR: 0.33; 95% CI: 0.14, 0.77) in women monitored using qCTG versus conventional CTG. CONCLUSION: qCTG may reduce risk of adverse birth outcomes; however, the small sample size and long recruitment period in this trial may overstate the benefits of this intervention. Further large-scale randomized control trials with sufficient sample size to detect rare adverse events are required prior to the adoption of qCTG in daily clinical practice.

Reliability and Validity of the International Dementia Alliance Schedule for the Assessment and Staging of Care in Ireland.

OBJECTIVE: To independently assess the inter-rater reliability of the International Dementia Alliance (IDEAL) schedule in Ireland and to examine criterion validity for cognitive functioning and caregiver distress. METHODS: Two raters independently completed the IDEAL schedule based on an interview with persons diagnosed with dementia and their informal caregivers. The Clinical Dementia Rating (CDR) scale and the Zarit Burden Interview were used as reference standards for criterion validity. Intraclass coefficients (ICCs) and correlations (Spearman's ρ) were derived. RESULTS: ICCs for the IDEAL subdomains ranged from 0.77 to 1.00, indicating robust agreement between raters. Strong correlations between the CDR and the IDEAL cognitive functioning domain (ρ = 0.82) and overall summary score (ρ = 0.77) were observed. The Zarit Burden Interview and the IDEAL carer distress subdomain were moderately correlated (ρ = 0.56). CONCLUSION: This study confirmed that the IDEAL schedule is a valid and reliable instrument in the Irish population and further supports the international use of the IDEAL schedule.

Rasch analysis reveals comparative analyses of activities of daily living/instrumental activities of daily living summary scores from different residential settings is inappropriate.

OBJECTIVES: To internally validate a 15-item dichotomous activities of daily living (ADL) and instrumental activities of daily living (IADL) index. STUDY DESIGN AND SETTING: Data were extracted from The Older Persons and Informal Caregivers Survey Minimum DataSet (TOPICS-MDS). Using Rasch modeling, six aspects of the ADL/IADL scale were assessed: (1) overall fit, (2) internal consistency, (3) individual item and person fit, (4) local dependency, (5) targeting, and (6) differential item functioning (DIF) (RUMM 2030). All analyses were stratified by living situation [community-dwelling (n = 21,926) or residential care facility (n = 2,458)]. RESULTS: In both settings, "eating" was the easiest activity on the scale and "performing household tasks" was the most difficult activity. However, based on the location on the logit scale, the level of difficulty for certain items varied between residential settings, suggesting summary scores are not equivalent between these settings. DIF by gender and age group was observed for several items, indicating potential measurement bias in the scale. CONCLUSION: Unless adjustments are undertaken, ADL/IADL summary scores retrieved from older persons residing in the community or residential care facilities should not be directly compared. This 15-item scale is poorly targeted for a community-dwelling older population, underscoring the need for items with improved discriminative ability.

[TOPICS-MDS: a versatile resource for generating scientific and social knowledge for elderly care]. / TOPICS-MDS: Veelzijdige bron voor wetenschappelijke en maatschappelijke kennisgeneratie ten behoeve van de ouderenzorg.

Developed as part of the National Care for the Elderly Programme (NPO), TOPICS-MDS is a uniform, national database on the health and wellbeing of the older persons and caregivers who participated in NPO-funded projects. TOPICS-MDS Consortium has gained extensive experience in constructing a standardized questionnaire to collect relevant health care data on quality of life, health services utilization, and informal care use. A proactive approach has been undertaken not only to ensure the standardization and validation of instruments but also the infrastructure for external data requests. Efforts have been made to promote scientifically and socially responsible use of TOPICS-MDS; data has been available for secondary use since early 2014. Through this data sharing initiative, researchers can explore health issues in a broader framework which may have not been possible within individual NPO projects; this broader framework is highly relevant for influencing health policy. In this article, we provide an overview of the development and on-going progress of TOPICS-MDS. We further describe how information derived from TOPICS-MDS can be applied to facilitate future scientific innovations and public health initiatives to improve care for frail older persons and their caregivers.

Parental decision making around perinatal autopsy: a qualitative investigation.

BACKGROUND: Decades of decline in uptake rates of perinatal autopsies has limited investigation into the causes and risk factors for stillbirth. AIMS: This study aimed to qualitatively explore perinatal autopsy decision-making processes in parents who experienced antepartum and intrapartum stillbirths. MATERIAL AND METHODS: A qualitative semi-structured interview format was utilized. The line of questioning centred on how parents came to decide on consenting or declining to have a perinatal autopsy undertaken. Interpretative phenomenological analysis was employed as the analytic strategy. Purposive sampling was used to recruit 10 parents who either consented or declined autopsy from a large tertiary maternity hospital in Cork Ireland, where there were 30 stillbirths in 2011. RESULTS: Findings revealed four superordinate themes influencing parents' decision-making which varied with type of stillbirth experienced. Those parents who experienced antepartum stillbirths were more likely to consent; thus, knowing that the child was stillborn prior to delivery rather than on the day of delivery was associated with consent. In fact, these parents had more time for meaning-making; those consenting wanted to rule out self-blame and were fearful about future pregnancies. Parents who declined autopsy wanted to protect their infant from further harm. Interestingly, parents' knowledge and understanding of the autopsy itself were acquired primarily from public discourse. CONCLUSION: Parents' decision-making regarding autopsy is profoundly affected by their emotional response to stillbirth; clinicians and other health professionals may play a key role, especially if they can address parental concerns regarding the invasiveness of the autopsy procedure.

[Frailty, disability and multi-morbidity: the relationship with quality of life and healthcare costs in elderly people]. / Kwetsbaarheid, dagelijkse beperkingen en ziektelast: samenhang met kwaliteit van leven en zorggebruik bij ouderen.

OBJECTIVE: To assess the independent and combined impact of frailty, multi-morbidity, and activities of daily living (ADL) limitations on self-reported quality of life and healthcare costs in elderly people. DESIGN: Cross-sectional, descriptive study. METHOD: Data came from The Older Persons and Informal Caregivers Minimum DataSet (TOPICS-MDS), a pooled dataset with information from 41 projects across the Netherlands from the Dutch national care for the Elderly programme. Frailty, multi-morbidity and ADL limitations, and the interactions between these domains, were used as predictors in regression analyses with quality of life and healthcare costs as outcome measures. Analyses were stratified by living situation (independent or care home). Directionality and magnitude of associations were assessed using linear mixed models. RESULTS: A total of 11,093 elderly people were interviewed. A substantial proportion of elderly people living independently reported frailty, multi-morbidity, and/or ADL limitations (56.4%, 88.3% and 41.4%, respectively), as did elderly people living in a care home (88.7%, 89.2% and 77,3%, respectively). One-third of elderly people living at home (31.9%) reported all three conditions compared with two-thirds of elderly people living in a care home (68.3%). In the multivariable analysis, frailty had a strong impact on outcomes independently of multi-morbidity and ADL limitations. Elderly people experiencing problems across all three domains reported the poorest quality-of-life scores and the highest healthcare costs, irrespective of their living situation. CONCLUSION: Frailty, multi-morbidity and ADL limitations are complementary measurements, which together provide a more holistic understanding of health status in elderly people. A multi-dimensional approach is important in mapping the complex relationships between these measurements on the one hand and the quality of life and healthcare costs on the other.