Neonatal systemic gene therapy restores cardiorespiratory function in a rat model of Pompe disease.

Absence of functional acid-α-glucosidase (GAA) leads to early-onset Pompe disease with cardiorespiratory and neuromuscular failure. A novel Pompe rat model ( ) was used to test the hypothesis that neonatal gene therapy with adeno-associated virus serotype 9 (AAV9) restores cardiorespiratory neuromuscular function across the lifespan. Temporal vein administration of AAV9-DES-GAA or sham (saline) injection was done on post-natal day 1; rats were studied at 6-12 months old.

Implementing and Evaluating a National Integrated Digital Registry and Clinical Decision Support System in Early Intervention in Psychosis Services (Early Psychosis Informatics Into Care): Co-Designed Protocol.

Background: Early intervention in psychosis (EIP) services are nationally mandated in England to provide multidisciplinary care to people experiencing first-episode psychosis, which disproportionately affects deprived and ethnic minority youth. Quality of service provision varies by region, and people from historically underserved populations have unequal access. In other disease areas, including stroke and dementia, national digital registries coupled with clinical decision support systems (CDSSs) have revolutionized the delivery of equitable, evidence-based interventions to transform patient outcomes and reduce population-level disparities in care.

A Scoping Review of Measures Used in Early Intervention Services for Psychosis.

Objective: The early intervention service (EIS) model for psychosis has been implemented with increasing frequency; yet, improving outcomes across domains for all patients remains challenging. Measurement-based care can strengthen outcomes by optimizing interventions and promoting alignment with standards, but it is still not widely deployed in EIS. The authors conducted a scoping review by systematically identifying and synthesizing measures used in EIS related to purpose (i.

Show me you care: A patient- and family-reported measure of care experiences in early psychosis services.

Aim: Despite their emphasis on engagement, there has been little research on patients' and families' experiences of care in early intervention services for psychosis. We sought to compare patients' and families' experiences of care in two similar early psychosis services in Montreal, Canada and Chennai, India. Because no patient- or family-reported experience measures had been used in a low- and middle-income context, we created a new measure, Show me you care.

Whose responsibility? Part 1 of 2: A scale to assess how stakeholders apportion responsibilities for addressing the needs of persons with mental health problems.

Background: Individuals with mental health problems have multiple, often inadequately met needs. Responsibility for meeting these needs frequently falls to patients, their families/caregivers, and governments. Little is known about stakeholders' views of who should be responsible for these needs and there are no measures to assess this construct.

Whose responsibility? Part 2 of 2: views of patients, families, and clinicians about responsibilities for addressing the needs of persons with mental health problems in Chennai, India and Montreal, Canada.

Background: Individuals with mental health problems have many insufficiently met support needs. Across sociocultural contexts, various parties (e.g.

Adapting, updating and translating the Social Functioning Scale to assess social, recreational and independent functioning among youth with psychosis in diverse sociocultural contexts.

Aim: To compare social, recreational and independent functioning among persons with psychosis across two geo-cultural contexts, we adapted the well-established Social Functioning Scale (SFS) and translated it into French and Tamil. We present the development and psychometric testing of this adaptation, the SFS-Early Intervention.

Methods: Sixteen items were added to reflect contemporary youth activities (e.

Fit for purpose: Conception and psychometric evaluation of developmentally appropriate measures to assess functional recovery in first-episode psychosis across geo-cultural contexts.

Objective: Given the paucity of functional recovery measures for young people with psychosis, we developed and conducted psychometric testing of the Functional Outcomes Interview (FOI) and the Roles and Aspirations Among Youth scale (RAY; a much-needed patient-reported outcome measure).

Method: Both measures were developed in English, Tamil, and French through discussions with professionals, participants, and families at early psychosis programs in Canada and India. The FOI assesses the number and tenure of functional roles and allows the nuanced assessment of each role in terms of performance, need for support, and quality of social contacts.

Engagement in specialized early intervention services for psychosis as an interplay between personal agency and critical structures: A qualitative study.

Background: Specialized early intervention programs for individuals experiencing a first episode of psychosis prioritize service engagement, generally operationalized as attendance, treatment completion, therapeutic alliance, and treatment adherence. However, there are critical theoretical and methodological gaps in understanding how service users experience and define their engagement with the service.

Objectives: This study aimed to explore how current and former service users define their engagement with a specialized early intervention program.

ShareDisk: A novel visual tool to assess perceptions about who should be responsible for supporting persons with mental health problems.

Objectives: Views on who bears how much responsibility for supporting individuals with mental health problems may vary across stakeholders (patients, families, clinicians) and cultures. Perceptions about responsibility may influence the extent to which stakeholders get involved in treatment. Our objective was to report on the development, psychometric properties and usability of a first-ever tool of this construct.

"Everyone Has a Role": Perspectives of Service Users With First-Episode Psychosis, Family Caregivers, Treatment Providers, and Policymakers on Responsibility for Supporting Individuals With Mental Health Problems.

Varying perceptions of who should be responsible for supporting individuals with mental health problems may contribute to their needs remaining unmet. A qualitative descriptive design was used to explore these perceptions among key stakeholders. Focus groups were conducted with 13 service users, 12 family members, and 18 treatment providers from an early psychosis intervention program in Montreal, Canada.

Experience with Event Timing Does not Alter Emergent Timing: Further Evidence for Robustness of Event and Emergent Timing.

Although, event and emergent timings are thought of as mutually exclusive, significant correlations between tapping and circle drawing (Baer, Thibodeau, Gralnick, Li, & Penhune, 2013 ; Studenka, Zelaznik, & Balasubramaniam, 2012 ; Zelaznik & Rosenbaum, 2010 ) suggest that emergent timing may not be as robust as once thought. We aimed to test this hypothesis in both a younger (18-25) and older (55-100) population. Participants performed one block of circle drawing as a baseline, then six blocks of tapping, followed by circle drawing.

Who should be responsible for supporting individuals with mental health problems? A critical literature review.

Background: Individuals with mental health problems have many support needs that are often inadequately met; however, perceptions of who should be responsible for meeting these needs have been largely unexplored. Varying perceptions may influence whether, how, and to what extent relevant stakeholders support individuals with mental health problems.

Aims: To critically evaluate the literature to determine who different stakeholders believe should be responsible for supporting individuals with mental health problems, what factors shape these perceptions, and how they relate to one another.

Positive Changes Experienced After a First Episode of Psychosis: A Systematic Review.

Objective: Although the negative consequences associated with first-episode psychosis (FEP) have been well investigated, relatively less is known about positive changes that people may experience after FEP. Existing literature is disparate and in need of synthesis. Such a synthesis can inform the design of mental health services that foster strengths, hope, and optimism.

The role of multiple internal timekeepers and sources of feedback on interval timing.

The aim of this experiment was to document the role of multiple internal clock mechanisms and external sources of temporal feedback on reducing timing variability when two fingers tap instead of one (a phenomenon known as the bimanual advantage). Previous research documents a reduction in timed interval variability when two effectors time instead of one. In addition, interval variability decreases with multiple sources of feedback.

Examination of cultural competence in service providers in an early intervention programme for psychosis in Montreal, Quebec: Perspectives of service users and treatment providers.

Aim: To better understand cultural competence in early intervention for psychosis, we compared service users' and service providers' perceptions of the importance of providers being culturally competent and attentive to aspects of culture.

Methods: At a Canadian early intervention programme, a validated scale was adapted to assess service user (N = 51) and provider (N = 30) perceptions of service providers' cultural competence and the importance accorded thereto.

Results: Analyses of variance revealed that the importance of service providers being culturally competent was rated highest by service providers, followed by visible minority service users, followed by white service users.

Post-traumatic growth following a first episode of psychosis: a scoping review.

Aim: A first-episode psychosis (FEP) is a traumatic experience that can often result in great suffering. However, in addition to suffering, persons affected by FEP may also experience post-traumatic growth (PTG) or the perception that good has followed psychosis. Although much is known concerning the negative outcomes following FEP, little attention has been given to the state of scientific knowledge on PTG following FEP.

Diagnostic stability of first-episode psychotic disorders and persistence of comorbid psychiatric disorders over 1 year.

Objective: Diagnostic stability is an important indicator of the reliability and validity of psychiatric diagnoses and has implications in clinical practice and research. While several studies have investigated the diagnostic stability of first-episode psychosis (FEP) disorders, less is known about psychiatric comorbidity in FEP and the persistence of such comorbid conditions over time. Our study aimed to confirm the diagnostic stability of FEP disorders and determine the variation in persistence of comorbid substance use disorders (SUDs), mood disorders, and anxiety disorders over 1 year.