The failure of success: four lessons learned in five years of research on research integrity and research assessments.

In the past 5 years, we captured the perspectives from a broad array of research stakeholders to better understand the impact that current approaches to success and research assessment may have on the integrity and the quality of research. Here, we translate our findings in four actions that are urgently needed to foster better research. First, we need to address core research structures to overcome systemic problems of the research enterprise; second, we must realign research assessments to value elements that advance and strengthen science; third, we need to remodel, diversify, and secure research careers; and finally, we need to unite and coordinate efforts for change.

Miscarriage: The Pain of Unacknowledged Loss.

These stories of fetal death make us aware of the several losses that attend a miscarriage and-equally important-they highlight the personal, cultural, and organizational constraints that make difficult the kind of caring responses needed to address these losses. In reading these narratives, we feel the grief and frustration caused by the inappropriate, or absent, reactions of caregivers. The seeming meaninglessness of the event led some to look for meaning in religion, while for others, the loss challenged their understandings of God. These stories also point to the importance of rituals-religious or otherwise-for helping the bereaved "carry their sorrow." Careful listening to the experiences of these women can help us make the changes-organizational and personal-that are necessary to provide the care and comfort those living through a miscarriage need.

"Helping fill that gap:" a qualitative study of aging in place after disaster through the lens of home-based care providers.

BACKGROUND: During a disaster, home-based care fills the critical need for continuation of health care. Home-based care is intended to function using existing care delivery models, continuing to provide care for patients wherever they are located, including in shelters and hotels. Home-based care providers are often the closest in contact with their patients -seeing them in place, even throughout a disaster- through which they develop a unique insight into aging in place during a disaster. The purpose of this study was to identify individual and community-level support needs of older adults after a disaster through the lens of home-based care providers. METHODS: Using qualitative inquiry, five focus groups were conducted with home-based care providers (n = 25) who provided in-home care during Hurricane Irma and Hurricane Harvey. Participants were identified by contacting home health agencies listed in an open-source database of agencies participating in Centers for Medicare and Medicaid Services programs. Data were coded using an abductive analytic approach, and larger themes were generated in light of existing theory. RESULTS: The results were distilled into eight themes that related to the importance of community and family, informal and formal supports throughout the disaster management cycle, maintaining autonomy during a disaster, and institutional and systemic barriers to obtaining assistance. CONCLUSIONS: In this study, home-based care providers described the challenges aging adults face in the response and recovery period after a large-scale disaster including maintaining continuity of care, encouraging individual preparedness, and accessing complex governmental support. Listening to home-based care providers offers new and important insights for developing interventions to address social and health needs for older adults aging in place after a large-scale disaster.

Perinatal factors related to pregnancy and childbirth satisfaction: a prospective cohort study.

BACKGROUND: Satisfaction of pregnancy and childbirth is an important quality measure of maternity care. Satisfaction questionnaires generally result in high scores. However, it has been argued that dissatisfaction relies on a different construct. In response to a worldwide call for obstetric care that is more woman-centered, we identified and described the contributors to suboptimal satisfaction with pregnancy and childbirth. METHODS: A prospective subcohort of 739 women from a larger cohort (Expect Study I, n = 2614) received a pregnancy and childbirth satisfaction questionnaire. Scores were transformed to a binary outcome whereby a score <100 points corresponded with less satisfied women. We performed a multiple logistic regression analysis to define independent perinatal factors related to suboptimal satisfaction. RESULTS: Decreased perceived personal well-being, antenatal anxiety, and obstetrician-led care during labor were all independently associated with suboptimal pregnancy and childbirth satisfaction. No difference in satisfaction was found between antenatal care led by a midwife or an obstetrician, but midwife-led antenatal care reduced the odds of suboptimal satisfaction compared to women who were transferred to an obstetrician in the antenatal period. Antenatal anxiety was experienced by 25% of all women and is associated with decreased satisfaction scores. DISCUSSION: Screening and treatment of women suffering from anxiety might improve pregnancy and childbirth satisfaction, but further research is necessary. Women's birthing experience may improve by reducing unnecessary secondary obstetric care.

Euthanasia and Assisted Suicide of Persons With Dementia in the Netherlands.

OBJECTIVE: To describe the characteristics of persons with dementia receiving euthanasia/assisted suicide (EAS) and how the practice is regulated in the Netherlands. DESIGNS: Qualitative directed content analysis of dementia EAS reports published by the Dutch euthanasia review committees between 2011 and October 5, 2018. RESULTS: Seventy-five cases were reviewed: 59 concurrent requests and 16 advance requests. Fifty-three percent (40/75) were women, and 48% (36/75) had Alzheimer disease. Advance request EAS patients were younger, had dementia longer, and more frequently had personal experience with dementia. Some concurrent request EAS patients were quite impaired: 15% (9/59) were deemed incompetent by at least one physician; in 24% (14/59), patients' previous statements or current body language were used to assess competence. In 39% (29/75), patients' own physicians declined to perform EAS; in 43% (32/75), the physician performing EAS was new to them. Physicians disagreed about patients' eligibility in 21% (16/75). All advance request and 14 (25%) concurrent request patients had an advance euthanasia directive but the conditions of applicability often lacked specificity. In 5 of 16 advance request EAS and 2 of 56 concurrent request EAS cases, EAS procedure was modified (e.g., premedication). Twenty-five percent (4/16) of advance request cases did not meet legal due care criteria, in particular the "unbearable suffering" criterion. CONCLUSIONS: Advance and concurrent request EAS cases differ in age, duration of illness, and past experience. Advance request EAS cases were complicated by ambiguous directives, patients being unaware of the EAS procedure, and physicians' difficulty assessing "unbearable suffering." Notably, some concurrent request patients were quite impaired yet deemed competent by appeals to previous statements.

Conscience reconsidered: The moral work of navigating participation in abortion care on labor and delivery.

How do caregivers make decisions about participating in morally contested care, such as abortion? Debates about conscience in the delivery of health care generally assume that participation decisions stem from religious beliefs and moral values. Few studies have examined this question in the context of everyday practice. Drawing on 50 interviews with the staff of a labor and delivery unit offering abortion care-including nurses, maternal-fetal medicine specialists, obstetrics and gynecology residents, and anesthesiologists-we show that respondents have varied definitions of "participation" in abortion care and that participation decisions are driven by an array of factors beyond personal beliefs. We present a conceptual model of "moral work" that shows conscience to be an emerging, iterative process influenced not only by beliefs-religious and non-religious-but also by personal and work experiences and social and institutional contexts. Our study brings new insights into understanding conscience and participation in contested care.

Interpretations of the Term "Actionable" when Discussing Genetic Test Results: What you Mean Is Not What I Heard.

In genomic medicine, the familiarity and inexactness of the term "actionable" can lead to multiple interpretations and mistaken beliefs about realistic treatment options. As part of a larger study focusing on public attitudes toward policies for the return of secondary genomic results, we looked at how members of the lay public interpret the term "medically actionable" in the context of genetic testing. We also surveyed a convenience sample of oncologists as part of a separate study and asked them to define the term "medically actionable." After being provided with a definition of the term, 21 out of 60 (35%) layperson respondents wrote an additional action not specified in the provided definition (12 mentioned "cure" and 9 mentioned environment or behavioral change) and 17 (28%) indicated "something can be done" with no action specified. In contrast, 52 surveyed oncologists did not mention environment, behavioral change, or cure. Based on our findings, we propose that rather than using the term "actionable" alone, providers should also say "what they mean" to reduce miscommunication and confusion that could negatively impact medical decision-making. Lastly, to guide clinicians during patient- provider discussion about genetic test results, we provide examples of phrasing to facilitate clearer communication and understanding of the term "actionable."

Human Biospecimens Come from People.

Contrary to the revised Common Rule, and contrary to the views of many bioethicists and researchers, we argue that broad consent should be sought for anticipated later research uses of deidentified biospecimens and health information collected during medical care. Individuals differ in the kinds of risk they find concerning and in their willingness to permit use of their biospecimens for future research. For this reason, asking their permission for unspecified research uses is a fundamental expression of respect for them as persons and should be done absent some compelling moral consideration to the contrary. We examine three moral considerations and argue that each of them fails: that there is a duty of easy rescue binding on all, that seeking consent creates a selection bias that undermines the validity of biospecimen research, and that seeking and documenting consent will be prohibitively expensive.

Biobanks and the Moral Concerns of Donors: A Democratic Deliberation.

Do members of the public believe that biobanks should accommodate the moral concerns of donors about the types of research done with their biospecimens? The answer to this question is critical to the future of genomic and precision medicine, endeavors that rely on a public willing to share their biospecimens and medical data. To explore public attitudes regarding the requirements of consent for biobank donations, we organized three democratic deliberations involving 180 participants. The deliberative sessions involved small group discussions informed by presentations given by experts in both biobank research and ethics. We found that participants had a sophisticated understanding of the ethical problems of biobank consent and the complexity of balancing donor concerns while promoting research important to the future of health care. Our research shows how deliberative methods can offer policy makers creative ideas for accommodating the moral concerns of donors in the biobank consent process.

Encouraging Participation And Transparency In Biobank Research.

Medical biobanks often struggle to obtain sustainable funding. Commercialization of specimens is one solution, but disclosure of commercial interests to potential contributors can be dissuasive. Recent revisions to the federal human subjects research regulations will soon mandate such commercialization disclosure in some circumstances, which raises questions about implications for practice. In this nationally representative, probability-based survey sample of the US adult population, we found that 67 percent of participants agreed that clear notification of potential commercialization of biospecimens is warranted, but only 23 percent were comfortable with such use. Sixty-two percent believed that profits should be used only to support future research, and 41 percent supported sharing profits with the public. We also considered other factors related to disclosure in our analysis and argue for a "disclosure plus" standard: informing potential contributors that their biospecimens might be accessed by commercial organizations and explaining how profits would be used to both enhance transparency and facilitate contributors' altruistic motivations.

Effect of deliberation on the public's attitudes toward consent policies for biobank research.

In this study, we evaluate the effect of education and deliberation on the willingness of members of the public to donate tissue to biobank research and on their attitudes regarding various biobank consent policies. Participants were randomly assigned to a democratic deliberation (DD) group, an education group that received only written materials, and a control group. Participants completed a survey before the deliberation and two surveys post-deliberation: one on (or just after) the deliberation day, and one 4 weeks later. Subjects were asked to rate 5 biobank consent policies as acceptable (or not) and to identify the best and worst policies. Analyses compared acceptability of different policy options and changes in attitudes across the three groups. After deliberation, subjects in the DD group were less likely to find broad consent (defined here as consent for the use of donations in an unspecified range of future research studies, subject to content and process restrictions) and study-by-study consent acceptable. The DD group was also significantly less likely to endorse broad consent as the best policy (OR = 0.34), and more likely to prefer alternative consent options. These results raise ethical challenges to the current widespread reliance on broad consent in biobank research, but do not support study-by-study consent.

Birth place preferences and women's expectations and experiences regarding duration and pain of labor.

INTRODUCTION: We know a great deal about how childbirth is affected by setting; we know less about how the experience of birth is shaped by the attitudes women bring with them to the birthing room. In order to better understand how women frame childbirth, we examined the relationship between birth place preference and expectations and experiences regarding duration of labor and labor pain in healthy nulliparous women. METHODS: A prospective cohort study (2007-2011) of 454 women who preferred a home birth (n = 179), a midwife-led hospital birth (n = 133) or an obstetrician-led hospital birth (n = 142) in the Netherlands. Data were collected using three questionnaires (before 20 weeks gestation, 32 weeks gestation and 6 weeks postpartum) and medical records. Analyses were performed according to the initial preferred place of birth. RESULTS: Women who preferred a home birth were significantly less likely to be worried about the duration of labor (OR 0.5, 95%CI 0.2-0.9) and were less likely to expect difficulties with coping with pain (OR 0.4, 95%CI 0.2-0.8) compared with women who preferred an obstetrician-led birth. We found no significant differences in postpartum accounts of duration of labor. When compared to women who preferred an obstetrician-led birth, women who preferred a home birth were significantly less likely to experience labor pain as unpleasant (OR 0.3, 95%CI 0.1-0.7). Women who preferred a midwife-led birth - either home or hospital - were more likely to report that it was not possible to make their own choices regarding pain relief compared to women who preferred obstetrician-led care (OR 4.3, 95%CI 1.9-9.8 resp. 3.4, 95%CI 1.5-7.7). Compared to women who preferred a midwife-led hospital birth, women who preferred a home birth had an increased likelihood of being dissatisfied about the management of pain relief (OR 2.5, 95%CI 1.1-6.0). DISCUSSION: Our findings suggest a more natural orientation toward birth with the acceptance of labor pain as part of giving birth in women with a preference for a home birth. Knowledge about women's expectations and experiences will help caregivers to prepare women for childbirth and will equip them to advise women on birth settings that fit their cognitive frame.

An Approach to measuring Integrated Care within a Maternity Care System: Experiences from the Maternity Care Network Study and the Dutch Birth Centre Study.

INTRODUCTION: Integrated care is considered to be a means to reduce costs, improve the quality of care and generate better patient outcomes. At present, little is known about integrated care in maternity care systems. We developed questionnaires to examine integrated care in two different settings, using the taxonomy of the Rainbow Model of Integrated Care. The aim of this study was to explore the validity of these questionnaires. METHODS: We used data collected between 2013 and 2015 from two studies: the Maternity Care Network Study (634 respondents) and the Dutch Birth Centre Study (56 respondents). We assessed the feasibility, discriminative validity, and reliability of the questionnaires. RESULTS: Both questionnaires showed good feasibility (overall missing rate < 20%) and reliability (Cronbach's Alpha coefficient > 0.70). Between-subgroups post-hoc comparisons showed statistically significant differences on integration profiles between regional networks (on all items, dimensions of integration and total integration score) and birth centres (on 50% of the items and dimensions of integration). DISCUSSION: Both questionnaires are feasible and can discriminate between sites with different integration profiles in The Netherlands. They offer an opportunity to better understand integrated care as one step in understanding the complexity of the concept.

Factors influencing the clinical decision-making of midwives: a qualitative study.

BACKGROUND: Although midwives make clinical decisions that have an impact on the health and well-being of mothers and babies, little is known about how they make those decisions. Wide variation in intrapartum decisions to refer women to obstetrician-led care suggests that midwives' decisions are based on more than the evidence based medicine (EBM) model - i.e. clinical evidence, midwife's expertise, and woman's values - alone. With this study we aimed to explore the factors that influence clinical decision-making of midwives who work independently. METHODS: We used a qualitative approach, conducting in-depth interviews with a purposive sample of 11 Dutch primary care midwives. Data collection took place between May and September 2015. The interviews were semi-structured, using written vignettes to solicit midwives' clinical decision-making processes (Think Aloud method). We performed thematic analysis on the transcripts. RESULTS: We identified five themes that influenced clinical decision-making: the pregnant woman as a whole person, sources of knowledge, the midwife as a whole person, the collaboration between maternity care professionals, and the organisation of care. Regarding the midwife, her decisions were shaped not only by her experience, intuition, and personal circumstances, but also by her attitudes about physiology, woman-centredness, shared decision-making, and collaboration with other professionals. The nature of the local collaboration between maternity care professionals and locally-developed protocols dominated midwives' clinical decision-making. When midwives and obstetricians had different philosophies of care and different practice styles, their collaborative efforts were challenged. CONCLUSION: Midwives' clinical decision-making is a more varied and complex process than the EBM framework suggests. If midwives are to succeed in their role as promoters and protectors of physiological pregnancy and birth, they need to understand how clinical decisions in a multidisciplinary context are actually made.

External Validation Study of First Trimester Obstetric Prediction Models (Expect Study I): Research Protocol and Population Characteristics.

BACKGROUND: A number of first-trimester prediction models addressing important obstetric outcomes have been published. However, most models have not been externally validated. External validation is essential before implementing a prediction model in clinical practice. OBJECTIVE: The objective of this paper is to describe the design of a study to externally validate existing first trimester obstetric prediction models, based upon maternal characteristics and standard measurements (eg, blood pressure), for the risk of pre-eclampsia (PE), gestational diabetes mellitus (GDM), spontaneous preterm birth (PTB), small-for-gestational-age (SGA) infants, and large-for-gestational-age (LGA) infants among Dutch pregnant women (Expect Study I). The results of a pilot study on the feasibility and acceptability of the recruitment process and the comprehensibility of the Pregnancy Questionnaire 1 are also reported. METHODS: A multicenter prospective cohort study was performed in The Netherlands between July 1, 2013 and December 31, 2015. First trimester obstetric prediction models were systematically selected from the literature. Predictor variables were measured by the Web-based Pregnancy Questionnaire 1 and pregnancy outcomes were established using the Postpartum Questionnaire 1 and medical records. Information about maternal health-related quality of life, costs, and satisfaction with Dutch obstetric care was collected from a subsample of women. A pilot study was carried out before the official start of inclusion. External validity of the models will be evaluated by assessing discrimination and calibration. RESULTS: Based on the pilot study, minor improvements were made to the recruitment process and online Pregnancy Questionnaire 1. The validation cohort consists of 2614 women. Data analysis of the external validation study is in progress. CONCLUSIONS: This study will offer insight into the generalizability of existing, non-invasive first trimester prediction models for various obstetric outcomes in a Dutch obstetric population. An impact study for the evaluation of the best obstetric prediction models in the Dutch setting with respect to their effect on clinical outcomes, costs, and quality of life-Expect Study II-is being planned. TRIAL REGISTRATION: Netherlands Trial Registry (NTR): NTR4143; http://www.trialregister.nl/trialreg/admin/rctview.asp?TC=4143 (Archived by WebCite at http://www.webcitation.org/6t8ijtpd9).

Developing quality indicators for assessing quality of birth centre care: a mixed- methods study.

BACKGROUND: Birth centres are described as settings where women with uncomplicated pregnancies can give birth in a home-like environment assisted by midwives and maternity care assistants. If complications arise or threaten, the woman is referred to a maternity unit of a hospital where an obstetrician will take over responsibility. In the last decade, a number of new birth centres have been established in the Netherlands, based on the assumption that birth centres provide better quality of care since they offer a better opportunity for more integrated care than the existing system with independent primary and secondary care providers. At present, there is no evidence for this assumption. The Dutch Birth Centre Study is designed to present evidence-based recommendations for organization and functioning of future birth centres in the Netherlands. A necessary first step in this evaluation is the development of indicators for measuring the quality of the care delivered in birth centres in the Netherlands. The aim of this study is to identify a comprehensive set of structure and process indicators to assess quality of birth centre care. METHODS: We used mixed methods to develop a set of structure and process quality indicators for evaluating birth centre care. Beginning with a literature review, we developed an exhaustive list of determinants. We then used a Delphi study to narrow this list, calling on experts to rate the determinants for relevance and feasibility. A multidisciplinary expert panel of 63 experts, directly or indirectly involved with birth centre care, was invited to participate. RESULTS: A panel of 42 experts completed two Delphi rounds rating determinants of the quality of birth centre care based on their relevance (to the setting) and feasibility (of use). A set of 30 determinants for structure and process quality indicators was identified to assess the quality of birth centre care in the Netherlands. CONCLUSIONS: We identified 30 determinants for structure and process quality indicators concerning birth centre care. This set will be validated during the evaluation of birth centres in the Dutch Birth Centre Study.

Typology of birth centres in the Netherlands using the Rainbow model of integrated care: results of the Dutch Birth Centre Study.

BACKGROUND: The goal of integrated care is to offer a continuum of care that crosses the boundaries of public health, primary, secondary, and tertiary care. Integrated care is increasingly promoted for people with complex needs and has also recently been promoted in maternity care systems to improve the quality of care. Especially when located near an obstetric unit, birth centres are considered to be ideal settings for the realization of integrated care. At present, however, we know very little about the degree of integration in these centres and we do not know if increased levels of integration improve the quality of the care delivered. The Dutch Birth Centre Study is designed to evaluate birth centres and their contribution to the Dutch maternity care system. The aim of this particular sub-study is to classify birth centres in clusters with similar characteristics based on integration profiles, to support the evaluation of birth centre care. METHODS: This study is based on the Rainbow Model of Integrated Care. We used a survey followed by qualitative interviews in 23 birth centres in the Netherlands to determine which integration profiles can be distinguished and to describe their discriminating characteristics. Cluster analysis was used to classify the birth centres. RESULTS: Birth centres were classified into three clusters: 1)"Mono-disciplinary-oriented birth centres" (n = 10): which are mainly owned by primary care organizations and established as physical facilities to provide an alternative birthplace for low risk births; 2) "Multi-disciplinary-oriented birth centres" (n = 6): which are mainly multi-disciplinary oriented and can be regarded as facilities to give birth, with a focus on integrated birth care; 3) "Mixed Cluster of birth centres" (n = 7): which have a range of organizational forms that differentiate them from centres in the other clusters. CONCLUSION: We identified a recognizable classification, with similar characteristics between birth centres in the clusters. The results of this study can be used to relate integration profiles of birth centres to quality of care, costs, and perinatal outcomes. This assessment makes it possible to develop recommendations with regard to the type and degree of integration of Dutch birth centres in the future.

Public's Views toward Return of Secondary Results in Genomic Sequencing: It's (Almost) All about the Choice.

The therapeutic use of genomic sequencing creates novel and unresolved questions about cost, clinical efficacy, access, and the disclosure of sequencing results. The disclosure of the secondary results of sequencing poses a particularly challenging ethical problem. Experts disagree about which results should be shared and public input - especially important for the creation of disclosure policies - is complicated by the complex nature of genetics. Recognizing the value of deliberative democratic methods for soliciting informed public opinion on matters like these, we recruited participants from a clinical research site for an all-day deliberative democracy (DD) session. Participants were introduced to the clinical and ethical issues associated with genomic sequencing, after which they discussed the tradeoffs and offered their opinions about policies for the return of secondary results. Participants (n = 66; mean age = 57 (SD = 15); 70% female; 76% white) were divided into 10 small groups (5 to 8 participants each) allowing interactive deliberation on policy options for the return of three categories of secondary results: 1) medically actionable results; 2) risks for adult-onset disorders identified in children; and 3) carrier status for autosomal recessive disorders. In our qualitative analysis of the session transcripts, we found that while participants favored choice and had a preference for making information available, they also acknowledged the risks (and benefits) of learning such information. Our research reveals the nuanced reasoning used by members of the public when weighing the pros and cons of receiving genomic information, enriching our understanding of the findings of surveys of attitudes regarding access to secondary results.