Temper outbursts are a severe problem for people with Prader-Willi Syndrome (PWS). Previous reports indicate that vagus nerve stimulation (VNS) may reduce maladaptive behaviour in neurodevelopmental disorders, including PWS. We systematically investigated the effectiveness of transcutaneous VNS (t-VNS) in PWS. Using a non-blind single case repeat measures modified ABA design, with participants as their own controls, t-VNS was evaluated in five individuals with PWS [three males; age 22–41 (M = 26.8)]. After a baseline phase, participants received four-hours of t-VNS daily for 12 months, followed by one month of daily t-VNS for two-hours. The primary outcome measure was the mean number of behavioural outbursts per day. Secondary outcomes included findings from behavioural questionnaires and both qualitative and goal attainment interviews. Four of the five participants who completed the study exhibited a statistically significant reduction in number and severity of temper outbursts after approximately nine months of daily four-hour t-VNS. Subsequent two-hour daily t-VNS was associated with increased outbursts for all participants, two reaching significance. Questionnaire and interview data supported these findings, the latter indicating potential mechanisms of action. No serious safety issues were reported. t-VNS is an effective, novel and safe intervention for chronic temper outbursts in PWS. We propose these changes are mediated through vagal projections and their effects both centrally and on the functioning of the parasympathetic nervous system. These findings challenge our present biopsychosocial understanding of such behaviours suggesting that there is a single major mechanism that is modifiable using t-VNS. This intervention is potentially generalizable across other clinical groups. Future research should address the lack of a sham condition in this study along with the prevalence of high drop out rates, and the potential effects of different stimulation intensities, frequencies and pulse widths.
Introduction People with Down's syndrome (DS) have a high prevalence of early‐onset Alzheimer's disease. Early markers of Alzheimer's disease pathology identifiable before clinical change are needed for the evaluation of preventative treatments. The retina, an extension of the brain, may provide a noninvasive imaging site. Methods Forty‐nine adults with DS and 36 age‐matched controls completed retinal nerve fibre layer (RNFL) assessments using optical coherence tomography. RNFL thickness was analyzed in relation to cognitive status and age and previously acquired cortical thickness and cerebral amyloid β binding data in a subgroup. Results RNFL thickness was greater in the DS group and did not show age‐related thinning. RNFL correlated positively with cognitive scores and cortical thickness and was reduced in participants with positive cerebral amyloid β binding. Discussion Increased RNFL in adults with DS may represent early Alzheimer's disease–related changes. Thinning was present in those with cerebral amyloid β binding, independent of age.
Purpose -The purpose of this paper is to ascertain what efforts Adult Safeguarding Leads (ASLs), generic advocates, and Independent Mental Capacity Advocates are making to involve service users in decisions about protective measures, and to investigate whether the Adult Safeguarding service is delivering outcomes, which are valued by its users. Design/methodology/approach -Semi-structured interviews with a sample of key stakeholders. Findings -ASLs are making efforts to involve service users in the complex and demanding process of safeguarding. These efforts, however, are shaped by their understandings of the difference between "residential" and "community" settings.Research limitations/implications -The study is based in a single County Council, albeit in a large county, and involves a limited number of service users. Practical implications -Clarification is needed of what it may mean to adopt a person-centred approach to adult safeguarding, and the responsibilities of ASLs when individuals with capacity to make decisions about this aspect of their lives are unwilling to engage with the safeguarding process. Originality/value -The findings improve the understanding of how ASLs understand their responsibilities towards the users of their services and endeavour to involve them in the adult safeguarding process. Based on this understanding, those with responsibility for managing Adult Safeguarding services should be better able to support improvements in professional practice.
Structured care in general practice improves intermediate outcomes for people with type 2 diabetes. Further improvements need to be made to reach international targets.
Test-retest reliability is essential to the development and validation of psychometric tools. Here we respond to the article by Karlsen et al. (Applied Neuropsychology: Adult, 2020), reporting testretest reliability on the Cambridge Neuropsychological Test Automated Battery (CANTAB), with results that are in keeping with prior research on CANTAB and the broader cognitive assessment literature. However, after adopting a high threshold for adequate test-retest reliability, the authors report inadequate reliability for many measures. In this commentary we provide examples of stable, trait-like constructs which we would expect to remain highly consistent across longer time periods, and contrast these with measures which show acute within-subject change in response to contextual or psychological factors. Measures characterized by greater true within-subject variability typically have lower test-retest reliability, requiring adequate powering in research examining group differences and longitudinal change. However, these measures remain sensitive to important clinical and functional outcomes. Setting arbitrarily elevated test-retest reliability thresholds for test adoption in cognitive research limits the pool of available tools and precludes the adoption of many well-established tests showing consistent contextual, diagnostic, and treatment sensitivity. Overall, test-retest reliability must be balanced with other theoretical and practical considerations in study design, including test relevance and sensitivity.
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