Body weight, behaviours of concern, and social contact in adults and adolescents with Prader-Willi syndrome in full-time care services: Findings from pooled international archival data.

BACKGROUND: Prader-Willi syndrome (PWS) is a complex genetic neurodevelopmental condition characterised by a range of debilitating and lifelong symptoms. The many physical and behavioural challenges that arise with adults with PWS often necessitate full-time (i.e., 24-hour) professional care support. However, despite the fact that many clinicians regard full-time PWS-specific care to represent best practice, relatively few studies have directly examined the benefits of such services. The purpose of this paper is to use archival data to investigate the impact of full-time care services on people with PWS, and to assemble a large statistical dataset on which robust analyses of improvements in weight, BMI, and behavioural outcomes can be based. METHODS: Information collated by the International PWS Organisation (IPWSO), an international non-profit membership organisation supporting national PWS associations around the world, was combined into a single anonymised dataset for statistical analysis. Data were supplied by service-providers from several countries who provide full-time support to people with PWS. The dataset included details on the specific services provided, basic demographic information on service recipients, including weight, body mass index (BMI), and observational records relating to behaviours of concern (BOC; consisting of temper outbursts, skin-picking, egocentrism, inflexibility, and striving for dominance). RESULTS: A total of 193 people with PWS (ranging in age from < 10 yrs to > 50 yrs; 93% of whom were > 18 yrs), residing in 11 services across 6 countries, were represented in the dataset. On average, people with PWS showed significant reductions in weight and BMI after joining a full-time care service, with improvements within one year of entering, which were cumulative over time and independent of age or initial weight at entry. Similar cumulative improvements over time were seen for BOC within one year and were unrelated to age or severity of BOC at entry. The degree to which services are specialised for residents with PWS appeared to confer particular benefits, with people living in PWS-exclusive services showing the greatest improvements in weight, BMI, and BOC. Reductions in BOC were associated with greater, rather than less, social contact, suggesting that these improvements were not achieved at the expense of broader freedoms, such as the opportunity to meet with families and friends. CONCLUSIONS: We conclude that full-time care services have a high likelihood of enhancing the lives of people with PWS within one year with long-lasting benefits, especially if those services are exclusive and specialised around the particular needs of PWS.

Psychophysiological responses to psychological stress exposure and neural correlates in adults with mental disorders: a scoping review.

Introduction: The dysregulation of psychophysiological responses to mental stressors is a common issue addressed in individuals with psychiatric conditions, while brain circuit abnormalities are often associated with psychiatric conditions and their manifestations. However, to our knowledge, there is no systematic overview that would comprehensively synthesize the literature on psychophysiological responses during laboratory-induced psychosocial stressor and neural correlates in people with mental disorders. Thus, we aimed to systematically review the existing research on psychophysiological response during laboratory-induced stress and its relationship with neural correlates as measured by magnetic resonance imaging techniques in mental disorders. Methods: The systematic search was performed on PubMed/Medline, EBSCOhost/PsycArticles, Web of Science, and The Cochrane Library databases during November 2021 following the PRISMA guidelines. Risk of bias was evaluated by employing the checklists for cross-sectional and case-control studies from Joanna Briggs Institute (JBI) Reviewers Manual. Results: Out of 353 de-duplicated publications identified, six studies were included in this review. These studies were identified as representing two research themes: (1) brain anatomy and psychophysiological response to mental stress in individuals with mental disorders, and (2) brain activity and psychophysiological response to mental stress in individuals with mental disorders. Conclusions: Overall, the evidence from studies exploring the interplay between stress psychophysiology and neural correlates in mental disorders is limited and heterogeneous. Further studies are warranted to better understand the mechanisms of how psychophysiological stress markers interplay with neural correlates in manifestation and progression of psychiatric illnesses.

The Role of Psychotherapy in the Care of Patients with Myalgic Encephalomyelitis/Chronic Fatigue Syndrome.

Myalgic encephalomyelitis/chronic fatigue (ME/CFS) is a post-infectious, chronic disease that can lead to severe impairment and, even, total disability. Although the disease has been known for a long time, and has been coded in the ICD since 1969 (G93.3), medical research has not yet been able to reach a consensus regarding its physiological basis and how best to treat it. Against the background of these shortcomings, psychosomatic disease models have been developed and psychotherapeutic treatments have been derived from them, but their empirical testing has led to sobering results. According to the current state of research, psychotherapy and psychosomatic rehabilitation have no curative effect in the treatment of ME/CFS. Nevertheless, we see numerous patients in practices and outpatient clinics who suffer severely as a result of their illness and whose mental well-being and coping strategies would benefit from psychotherapeutic help. In this article, we outline a psychotherapeutic approach that serves this need, taking into account two basic characteristics of ME/CFS: firstly, the fact that ME/CFS is a physical illness and that curative treatment must therefore be physical; and secondly, the fact that post exertional malaise (PEM) is a cardinal symptom of ME/CFS and thus warrants tailored psychotherapeutic attention.

Cortisol response to psychosocial stress, mental distress, fatigue and quality of life in coronary artery disease patients.

We aimed to explore the relationship between cortisol response to psychosocial stress, mental distress, fatigue and health related quality of life (HRQoL) in individuals with coronary artery disease (CAD) after recent acute coronary syndrome (ACS). A cross-sectional study initially included 113 subjects (88% men, 53 ± 7 years) 1-3 weeks after ACS. Cortisol response was assessed by measuring salivary cortisol during Trier Social Stress Test. Mental distress was measured with Hospital Anxiety and Depression Scale, State-Trait Anxiety Inventory, and Type D Scale-14. Fatigue symptoms were evaluated using Multidimensional Fatigue Inventory 20-items, while HRQoL was assessed with 36-Item Short Form Medical Outcome Questionnaire. After conducting multivariable linear regression analyses, diminished cortisol response sampled after Public speech (T3-T1, + 15 min) was significantly associated with higher anxiety symptoms (ß = -0.224; p = 0.035), while diminished cortisol response sampled after preparation time (T2-T1, + 10 min) was significantly linked with the presence of Type D personality (ß = -0.290; p = 0.006; ß = -0.282; p = 0.008 respectively), even after controlling for confounders (i.e., sex, age, education, New York Heart Association functional class, beta-blockers and baseline levels of cortisol measures). We found that mental distress, but not fatigue and HRQoL, was linked with blunted cortisol response during anticipation time of psychosocial stress, independently of potential covariates.

Smokers show an altered hemodynamic profile to active stress: Evidence of a dysregulated stress response in young adults.

Tobacco smoking has been associated with lower cardiovascular reactivity to psychological stress in middle-aged samples, but its impact on cardiovascular reactivity to stress in young adults remains unclear. The present study examined whether young healthy adults showed differing cardiovascular stress reaction profiles depending on their smoking status. Across two laboratory studies (N = 64 and N = 114), we asked participants to complete cognitive stress-tasks while undergoing continuous hemodynamic monitoring. In both studies, there was not a statistically signification association between systolic blood pressure, diastolic blood pressure, or heart rate reactivity to stress (all ps > .05). However, examination of the underlying hemodynamic profile of the stress response suggested differences between non-smokers and smokers in both studies. In Study 1, non-smokers exhibited the expected myocardial response to the active stress-task; however, smokers exhibited a mixed hemodynamic profile. In Study 2, smokers evidenced a weaker myocardial profile to the active stress-tasks compared to non-smokers. However, the examination of the continuous hemodynamic profile score (HP) did not identify statistical differences. These results highlight that any level of the smoking habit is associated with an altered hemodynamic profile in response to stress in smokers, which may have important implications for long-term cardiovascular health. The findings also suggest that controlling for smoking behavior in reactivity research examining blood pressure and heart rate responses to stress in young adults is not necessary.

Response to Adamson et al. (2020): 'Cognitive behavioural therapy for chronic fatigue and chronic fatigue syndrome: Outcomes from a specialist clinic in the UK'.

In a paper published in the Journal of the Royal Society of Medicine, Adamson et al. (2020) interpret data as showing that cognitive behavioural therapy leads to improvement in patients with chronic fatigue syndrome and chronic fatigue. Their research is undermined by several methodological limitations, including: (a) sampling ambiguity; (b) weak measurement; (c) survivor bias; (d) missing data and (e) lack of a control group. Unacknowledged sample attrition renders statements in the published Abstract misleading with regard to points of fact. That the paper was approved by peer reviewers and editors illustrates how non-rigorous editorial processes contribute to systematic publication bias.

Fatigue Is Associated With Diminished Cardiovascular Response to Anticipatory Stress in Patients With Coronary Artery Disease.

BACKGROUND: Fatigue and psychophysiological reactions to mental stress are known to be problematic in coronary artery disease (CAD) patients. Currently, studies exploring the relationship between fatigue and cardiovascular reactivity to stress are scarce and inconsistent. The current study aimed to investigate the links between cardiovascular response to mental stress and fatigue in CAD patients after acute coronary syndrome (ACS). METHODS: The cross-sectional study investigated 142 CAD patients (85% males, 52 ± 8 years) within 2-3 weeks after recent myocardial infarction or unstable angina pectoris. Fatigue symptoms were measured using Multidimensional Fatigue Inventory 20-items, while cardiovascular reactivity to stress [i.e., systolic (S) and diastolic (D) blood pressure (ΔBP), and heart rate (ΔHR)] was evaluated during Trier Social Stress Test (TSST). In addition, participants completed psychometric measures, including the Hospital Anxiety and Depression scale and the Type D Scale-14. Multivariable linear regression analyses were completed to evaluate associations between fatigue and cardiovascular response to TSST, while controlling for confounders. RESULTS: After controlling for baseline levels of cardiovascular measures, age, gender, education, heart failure severity, arterial hypertension, smoking history, use of nitrates, anxiety and depressive symptoms, Type D Personality, perceived task difficulty, and perceived task efforts, cardiovascular reactivity to anticipatory stress was inversely associated with both global fatigue (ΔHR: ß = -0.238; p = 0.04) and mental fatigue (ΔSBP: ß = -0.244; p = 0.04; ΔHR ß = -0.303; p = 0.01) as well as total fatigue (ΔSBP: ß = -0.331; p = 0.01; ΔHR: ß = -0.324; p = 0.01). CONCLUSION: In CAD patients after ACS, fatigue was linked with diminished cardiovascular function during anticipation of a mental stress challenge, even after inclusion of possible confounders. Further similar studies exploring other psychophysiological stress responses are warranted.

Cardiovascular Stress Reactivity and Health: Recent Questions and Future Directions.

OBJECTIVE: High cardiovascular reactions to psychological stress are associated with the development of hypertension, systemic atherosclerosis, and cardiovascular disease. However, it has become apparent that low biological stress reactivity also may have serious consequences for health, although less is known about the mechanisms of this. The objectives of this narrative review and opinion article are to summarize and consider where we are now in terms of the usefulness of the reactivity hypothesis and reactivity research, given that both ends of the reactivity spectrum seem to be associated with poor health, and to address some of the key criticisms and future challenges for the research area. METHODS: This review is authored by the members of a panel discussion held at the American Psychosomatic Society meeting in 2019, which included questions such as the following: How do we measure high and low reactivity? Can high reactivity ever indicate better health? Does low or blunted reactivity simply reflect less effort on task challenges? Where does low reactivity originate from, and what is a low reactor? RESULTS: Cardiovascular (and cortisol) stress reactivity are used as a model to demonstrate an increased understanding of the different individual pathways from stress responses to health/disease and show the challenges of how to understand and best use the reconstruction of the long-standing reactivity hypothesis given recent data. CONCLUSIONS: This discussion elucidates the gaps in knowledge and key research issues that still remain to be addressed in this field, and that systematic reviews and meta-analyses continue to be required.

An integrative network analysis framework for identifying molecular functions in complex disorders examining major depressive disorder as a test case.

In addition to the psychological depressive phenotype, major depressive disorder (MDD) patients are also associated with underlying immune dysregulation that correlates with metabolic syndrome prevalent in depressive patients. A robust integrative analysis of biological pathways underlying the dysregulated neural connectivity and systemic inflammatory response will provide implications in the development of effective strategies for the diagnosis, management and the alleviation of associated comorbidities. In the current study, focusing on MDD, we explored an integrative network analysis methodology to analyze transcriptomic data combined with the meta-analysis of biomarker data available throughout public databases and published scientific peer-reviewed articles. Detailed gene set enrichment analysis and complex protein-protein, gene regulatory and biochemical pathway analysis has been undertaken to identify the functional significance and potential biomarker utility of differentially regulated genes, proteins and metabolite markers. This integrative analysis method provides insights into the molecular mechanisms along with key glycosylation dysregulation underlying altered neutrophil-platelet activation and dysregulated neuronal survival maintenance and synaptic functioning. Highlighting the significant gap that exists in the current literature, the network analysis framework proposed reduces the impact of data gaps and permits the identification of key molecular signatures underlying complex disorders with multiple etiologies such as within MDD and presents multiple treatment options to address their molecular dysfunction.

Monitoring treatment harm in myalgic encephalomyelitis/chronic fatigue syndrome: A freedom-of-information study of National Health Service specialist centres in England.

The use of graded exercise therapy and cognitive behavioural therapy for myalgic encephalomyelitis/chronic fatigue syndrome has attracted considerable controversy. This controversy relates not only to the disputed evidence for treatment efficacy but also to widespread reports from patients that graded exercise therapy, in particular, has caused them harm. We surveyed the National Health Service-affiliated myalgic encephalomyelitis/chronic fatigue syndrome specialist clinics in England to assess how harms following treatment are detected and to examine how patients are warned about the potential for harms. We sent 57 clinics standardised information requests under the United Kingdom's Freedom of Information Act. Data were received from 38 clinics. Clinics were highly inconsistent in their approaches to the issue of treatment-related harm. They placed little or no focus on the potential for treatment-related harm in their written information for patients and for staff. Furthermore, no clinic reported any cases of treatment-related harm, despite acknowledging that many patients dropped out of treatment. In light of these findings, we recommend that clinics develop standardised protocols for anticipating, recording, and remedying harms, and that these protocols allow for therapies to be discontinued immediately whenever harm is identified.

Cynical hostility relates to a lack of habituation of the cardiovascular response to repeated acute stress.

Hostility is associated with increased risk for cardiovascular disease. Heightened cardiovascular reactivity to psychological stress has been proposed as a potential mechanism. Recent work has emphasized a need to measure cardiovascular reactivity across multiple stress exposures to assess potential habituation over time. The aims of the current study were (a) to examine the relationship between each of the three main components of hostility (i.e., emotional, cognitive, and behavioral) and cardiovascular reactivity at two separate stress testing visits and (b) to examine the relationship between hostility components and cardiovascular reactivity habituation. This study utilized previously collected data from the Pittsburgh Cold Study 3. One hundred and ninety-six participants (Mean (SD)[range] age = 29.9 (10.8)[18-55] years, 42.9% female, 67.3% Caucasian) completed 2 separate, identical laboratory sessions, consisting of a 20-min baseline and 15-min stress (Trier Social Stress Test). Heart rate and systolic/diastolic blood pressure were recorded throughout. Reactivity was calculated separately for heart rate, systolic, and diastolic blood pressure (stress-baseline). Participants also completed a modified version of the Cook-Medley Hostility Scale. Results indicated that greater cognitive hostility (i.e., cynicism) was associated with blunted cardiovascular reactivity at Visit 1 and less cardiovascular reactivity habituation between visits, even when controlling for confounding variables. No significant relationships to cardiovascular reactivity or habituation were found for emotional (i.e., hostile affect) or behavioral (i.e., aggressive responding) components. Outcomes for total hostility did not survive adjustment for confounders. These results identify a potential pathway through which hostility, particularly cynicism, contributes to disease risk.

Trait and state approach-motivated positive affects interactively influence stress cardiovascular recovery.

One of the potential mechanisms of positive affect (PA) in promoting physical health is by speeding up stress cardiovascular recovery. However, little is known whether high and low approach-motivated PA (AMPA) equally speed stress cardiovascular recovery and whether these effects are affected by personality traits that are associated with approach motivation. The present study investigated the impact of trait AMPA (assessed by sensation seeking) and state AMPA (elicited by positive stimuli) on stress cardiovascular recovery. After completion of Sensation Seeking Scale (SSS-V), 165 undergraduate students were subjected to a stress protocol, and then randomized to watch high AMPA, low AMPA and neutral pictures. Baseline, stress exposure, and post-stress cardiovascular data were collected. Results showed that compared to neutral and state low AMPA conditions, state high AMPA prolonged stress HR recovery. Moreover, individuals low in sensation seeking exhibited delayed SBP and DBP recovery under state high AMPA condition relative to state low AMPA and neutral conditions. Individuals high in sensation seeking exhibited comparable stress BP recovery across the three conditions. These findings suggest that the association between PA and stress cardiovascular recovery is contingent on approach motivational intensity, which might have health implications.

Trait dominance and cardiovascular functioning during social stress.

Cardiovascular reactivity (CVR) to stress has been found to be an important indicator of future ill health, and individual differences in personality have been posited to explain disparities in outcomes. Dominance is associated with forceful persons who desire hierarchy in social interactions. This study investigated dominance and CVR during social or asocial stressors. Sixty-one women, categorized as low, moderate, or high in dominance using the Jackson Personality Research Form, completed a social or asocial stressor while undergoing cardiovascular measurement during baseline, stressor, and recovery phases. A 3 × 2 × 3 analysis of covariance revealed a significant Phase × Stressor × Dominance interaction for systolic blood pressure (SBP). Women with lower and moderate dominance-but not women with higher dominance-exhibited greater SBP responses to stress in the social condition compared with the asocial condition. No significant difference was found for women with higher dominance, indicative of blunted SBP during the social stressor. During recovery, women with lower dominance had marginally elevated SBP in the social condition compared with the asocial condition. The current study extends prior knowledge of the association between dominance and CVR, such that greater dominance was associated with blunted SBP and lower dominance was associated with attenuated recovery to social stress.

Vulnerability to stress: Personality facet of vulnerability is associated with cardiovascular adaptation to recurring stress.

It is increasingly suggested that personality traits are critical to understanding patterns of cardiovascular stress adaptation. However, studies have focused on higher-order traits with no research having examined underlying facet effects to repeated stress. The examination of facets provides a more granular examination, which has the potential to identify specific personality components that are relevant within the context of psychophysiological stress adaptation. This study objective was to determine if the underlying facets which encapsulate the dimension of emotional stability, are associated with cardiovascular adaptation to recurring stress. Continuous cardiovascular monitoring and psychometric measures were collated from 79 healthy young male and female adults, across a protocol of recurring active stress tasks. Multiple regression analysis revealed that the facet of vulnerability was associated with systolic and diastolic blood pressure adaptation across the protocol. More specifically, vulnerability was negatively associated with adaptation to recurring stress, such that those highest in vulnerability displayed a sensitization to the recurring stressor. No significant effects emerged for any other facet. Importantly, this research adds to the existing literature examining stress adaptation and has implications for future research on the relevance of examining facet effects. This study is the first to implicate the personality facet of vulnerability which encapsulates an individual's tendency to feel unable to cope with stress and becoming hopeless when faced with emergency situations, in the context of cardiovascular stress adaptation. Taken together, this study suggests that the facet of vulnerability is a critical component to consider in the context of cardiovascular stress adaptation.

Effects of emotions on heart rate asymmetry.

Heart rate asymmetry (HRA) is an index that accounts for an uneven contribution of decelerations and accelerations to the heart rate variability (HRV). Clinical studies indicated that HRA measures have additive clinical value over the more frequently used HRV indexes. Despite the abundance of studies on psychological influences on HRV, little is known whether psychological factors influence HRA. Based on previous research regarding HRA and stress, we expected that negative emotions compared to positive emotions would decrease the contribution of decelerations to HRV. Thirty female participants watched three clips that produced negative emotions, positive emotions, and neutral affect. Besides electrocardiogram, we measured several physiological and behavioral responses to ascertain the affective impact of the clips. Using the RR interval time series, we calculated HRV and HRA indexes. We found that HRA differentiated between positive emotions and negative emotions reactivity. Positive emotions produced a higher number of decelerations in short-term variability to the total short-term variability (C1d ) compared to negative emotions. Moreover, C1d correlated with subjective ratings of affect. In sum, the results of this study indicated that HRA is sensitive to psychological influences. HRA indexes are likely to contribute to a more nuanced physiological differentiation between emotions.

High-approach and low-approach positive affect influence physiological responses to threat and anger.

Positive affect has been related to faster cardiovascular recovery from stress. Although the family of positive affective states is diverse, no study examined whether high-approach positive affect (e.g., desire) has a different impact on peripheral physiological processes than more frequently studied low-approach positive affect (e.g., amusement). Building upon prior work on emotions and motivation, we expected that after controlling for arousal and valence, positive affect with higher motivational intensity would facilitate weaker recovery when compared to positive affect with lower motivational intensity. Across two experiments (N = 179 for Study 1, N = 220 for Study 2), we measured cardiovascular, respiratory, and electrodermal responses to positive stimuli that differed in approach intensity. We measured responses during recovery from stress and during reactivity to threat and anger. These studies partially replicated previous findings regarding the soothing function of positive affect (e.g., in respect to diastolic blood pressure recovery and reactivity). However, we found that high-approach and low-approach positive affect produced comparable effects. In summary, these findings suggest that positive valence rather than motivational intensity produces the main soothing effect on peripheral physiology.

Openness to experience and stress responsivity: An examination of cardiovascular and underlying hemodynamic trajectories within an acute stress exposure.

The personality trait openness to experience has been implicated in health, and in particular cardiovascular wellbeing. In a sample of 62 healthy young female adults, the role of openness in cardiovascular responsivity during a stress exposure was examined. Traditionally, methodologies have averaged a stress exposure into a single reading. This may be limited in that it does not consider patterns of cardiovascular adaptation within a stress exposure. Continuous cardiovascular data were reduced to mean 10 second readings, with phases determined through examinations of shifts in responsivity between each 10 second pairing. Analyses revealed a significant linear interaction for openness across the entire exposure for systolic blood pressure, and cardiac output. A significant between-subjects effect for heart rate also emerged. Contrary to their lower counterparts, those highest in openness exhibited an increasingly myocardial hemodynamic response profile throughout the exposure. Comparisons of responsivity suggests adaptive stress response trajectories for those highest in openness. This study also provides evidence that an attenuation of myocardial responsivity may underpin blunted responsivity. This study provides a potential mechanism in reported openness-health associations.