The mixed methods appraisal tool (MMAT) was developed for critically appraising different study designs. This study aimed to improve the content validity of three of the five categories of studies in the MMAT by identifying relevant methodological criteria for appraising the quality of qualitative, survey, and mixed methods studies. First, we performed a literature review to identify critical appraisal tools and extract methodological criteria. Second, we conducted a two-round modified e-Delphi technique. We asked three method-specific panels of experts to rate the relevance of each criterion on a five-point Likert scale. A total of 383 criteria were extracted from 18 critical appraisal tools and a literature review on the quality of mixed methods studies, and 60 were retained. In the first and second rounds of the e-Delphi, 73 and 56 experts participated, respectively. Consensus was reached for six qualitative criteria, eight survey criteria, and seven mixed methods criteria. These results led to modifications of eight of the 11 MMAT (version 2011) criteria. Specifically, we reformulated two criteria, replaced four, and removed two. Moreover, we added six new criteria. Results of this study led to improve the content validity of this tool, revise it, and propose a new version (MMAT version 2018).

This paper presents the development of the Warwick Patient Experiences Framework (WaPEF) and describes how it informed the development of the NICE Guidance and Quality Standard, ‘Patient experience in adult NHS services: improving the experience of care for people using adult NHS services’. The WaPEF was developed using a thematic qualitative overview that utilized a systematic review approach. Search strategies were developed, inclusion and exclusion criteria developed and data extracted from papers. The WaPEF identifies seven key generic themes that are important to a high-quality patient experience: patient as active participant, responsiveness of services, an individualized approach, lived experience, continuity of care and relationships, communication, information and support. The WaPEF is the first patient experiences framework with an explicit link to an underpinning patient evidence base, linking themes and sub-themes with specific references. The WaPEF informed the structure and content of the NICE Patient Experiences Guidance. The guidance, published in February 2012, will form a key part of the NHS Outcomes Framework in the UK for the future evaluation of health and social care. The proposed framework could be adapted to other country contexts and settings.

Despite no consensus on the definition of ‘seriousness’, the concept is regularly used in policy and practice contexts to categorise conditions, determine access to genetic technologies and uses of selective pregnancy termination. Whilst attempts have been made to create taxonomies of genetic condition seriousness to inform clinical and policy decision-making, these have often relied on condition appraisals made by health and genetics professionals. The views of people with genetic conditions have been largely under-represented. This study explores the concept of seriousness through the perspectives of people with a range of ‘clinically serious’ conditions (fragile X conditions, spinal muscular atrophy, cystic fibrosis, haemophilia, thalassaemia). Attitudes towards suffering, quality of life (QoL) and selective pregnancy termination were elucidated from 45 in-depth qualitative interviews and 469 postal/online surveys. The majority of participants reported good health/wellbeing, and the capacity for good QoL, despite experiencing suffering with their condition. Notably, participants with later-onset conditions held more negative views of their health and QoL, and were more likely to view their condition as an illness, than those with early-onset conditions. These participants were more likely to see their condition as part of their identity. Whilst most participants supported prenatal screening, there was little support for selective termination. Moreover, social environment emerged as a critical mediator of the experience of the condition. The complex and rich insights of people living with genetic conditions might usefully be incorporated into future genetic taxonomies of ‘seriousness’ to ensure they more accurately reflect the lived reality of those with genetic conditions.

The expressivist objection to prenatal testing is acknowledged as a significant critique of prenatal testing practices most commonly advanced by disability rights supporters. Such writers argue that prenatal testing and selective termination practices are objectionable as they express disvalue not only of the foetus being tested, but also of disabled people as a whole, by focusing exclusively on the disabling trait. While the objection has been widely critiqued on the basis of its theoretical incoherence, this paper highlights the way in which it, nevertheless, is a significant mediator in decisions around the use of reproductive genetic technologies. By drawing on 41 in-depth qualitative interviews (drawn from a sample of 61) conducted in the UK between 2007 and 2009 with families and individuals living with a genetic disease, Spinal Muscular Atrophy (SMA), this paper highlights the ways in which expressivist objections feature prominently in the reproductive decisions of families living with SMA and the significant emotional burden they represent. While the literature on the expressivist objection has focused on the reproductive decisions of those undergoing prenatal testing for a condition of which they have little (or no) prior knowledge, the context of intimate familial relationships and extensive experience with the tested-for condition fundamentally alters the nature and impact of expressivist objections within families living with an inheritable condition. By focussing on the reproductive decisions of families living with SMA and their strategic management of the expressivist objection, this paper will address the call, made primarily by disability rights supporters, for 'experientially based' (as opposed to medical) information about the tested-for disability to be made available to would-be parents considering selective termination. It will be argued that parents' experiential knowledge of the tested-for disability can, in fact, amplify expressivist objections to prenatal testing, and thus paradoxically constrain, rather than facilitate, reproductive decisions.