Episodic disability questionnaire (EDQ) measurement properties among adults living with HIV in Canada, Ireland, United Kingdom, and United States.

BACKGROUND: The Episodic Disability Questionnaire (EDQ) is a generic 35-item patient-reported outcome measure of presence, severity and episodic nature of disability. We assessed the measurement properties of the Episodic Disability Questionnaire (EDQ) with adults living with HIV. METHODS: We conducted a measurement study with adults living with HIV in eight clinical settings in Canada, Ireland, United Kingdom, and United States. We electronically administered the EDQ followed by three reference measures (World Health Organization Disability Assessment Schedule; Patient Health Questionnaire; Social Support Scale) and a demographic questionnaire. We administered the EDQ only 1 week later. We assessed the internal consistency reliability (Cronbach's alpha; > 0.7 acceptable), and test-retest reliability (Intra Class Correlation Coefficient; > 0.7 acceptable). We estimated required change in EDQ domain scores to be 95% certain that a change was not due to measurement error (Minimum Detectable Change (MDC95%)). We evaluated construct validity by assessing 36 primary hypotheses of relationships between EDQ scores and scores on the reference measures (> 75% hypotheses confirmed indicated validity). RESULTS: Three hundred fifty nine participants completed the questionnaires at time point 1, of which 321 (89%) completed the EDQ approximately 1 week later. Cronbach's alpha for internal consistency ranged from 0.84 (social domain) to 0.91 (day domain) for the EDQ severity scale, and 0.72 (uncertainty domain) to 0.88 (day domain) for the EDQ presence scale, and 0.87 (physical, cognitive, mental-emotional domains) to 0.89 (uncertainty domain) for the EDQ episodic scale. ICCs for test-retest reliability ranged from 0.79 (physical domain) to 0.88 (day domain) for the EDQ severity scale and from 0.71 (uncertainty domain) to 0.85 (day domain) for the EDQ presence scale. Highest precision was demonstrated in the severity scale for each domain (MDC95% range: 19-25 out of 100), followed by the presence (MDC95% range: 37-54) and episodic scales (MDC95% range:44-76). Twenty-nine of 36 (81%) construct validity hypotheses were confirmed. CONCLUSIONS: The EDQ possesses internal consistency reliability, construct validity, and test-retest reliability, with limited precision when administered electronically with adults living with HIV across in clinical settings in four countries. Given the measurement properties, the EDQ can be used for group level comparisons for research and program evaluation in adults living with HIV.

Baclofen and Tizanidine Adverse Effects Observed Among Community-Dwelling Adults Above the Age of 50 Years: A Systematic Review.

OBJECTIVE: This review highlights adverse effects of baclofen and tizanidine in older community-dwelling adults. DATA SOURCES: A literature search was conducted, including search terms of "adverse effect," "baclofen," "elderly," "falls," "fractures," and "tizanidine." Studies were included if they described community-dwelling adults aged 50 years and older who received oral baclofen or tizanidine. The Federal Drug Administration Adverse Event Reporting System (FAERS) data were compiled for adverse effect incidence. STUDY SELECTION AND DATA EXTRACTION: The literature search was completed in July 2019 and updated in June 2023. Reviews performed by 2 independent reviewers yielded 15 records. FAERS identified 486 (baclofen) and 305 (tizanidine) adverse effects of interest. DATA SYNTHESIS: Two retrospective cohort studies evaluating baclofen use in older adults showed increased hospitalizations for encephalopathy in chronic kidney disease (7.2% vs 0.1%) and end-stage renal disease (daily dose 20 mg or more; relative risk [RR] 19.8, 95% CI = [14.0-28.0]). Other articles were case reports; 10 articles reported dyskinesias, encephalopathy or disorientation, and drowsiness associated with baclofen, and 5 articles reported bradycardia and/or hypotension with tizanidine. The FAERS Public Dashboard revealed 12.1% and 28.7% overall incidence of adverse effects of interest, with a 27.8% and 29.2% incidence of falls for baclofen and tizanidine, respectively. Baclofen and tizanidine are associated with concerning adverse effects in older adults. Alternative agents should be considered, but, if necessary, providers should start at lower doses and increase slowly. CONCLUSIONS: This review highlights the importance of using baclofen and tizanidine with caution in older adults.

HIV in MOTION: a community of practice on physical rehabilitation for and by people living with HIV and their allies.

Background: This paper describes the design, implementation, and evaluation of a community of practice (CoP), HIV in MOTION (HIM), to advance physical activity rehabilitation interventions with adults living with HIV, clinicians, researchers, and representatives from community-based organizations. We attracted a diverse audience of geographically dispersed people living with HIV, clinicians, exercise personnel, and trainees to eight HIM community of practice events that featured the clinical, research, and lived experience of people living with HIV. HIV in MOTION had (a) a domain related to physical rehabilitation, exercise, and social participation for people living with HIV; (b) a community of diverse individuals; and (c) a practice, that is, a series of sustained interactions online and offline, synchronous, and asynchronous. Our team included six diverse people living with HIV, two coordinators, and three academic researchers who planned, prepared, implemented, and evaluated each online session. To evaluate the HIV in MOTION CoP, we employed an evaluation framework composed of five criteria: Goals and Scope, Context and Structure, Process and Activities, Outcomes, and Impact. We collected quantitative and qualitative evaluative data using online evaluation, audiovisual archiving, and participant observations during the debriefing with all members of our team. Results: We widened the Goals and Scope of the HIV in MOTION CoP to include the HIV narrative of lived experiences, including autopathography, and participant storytelling. In matters of Context and Structure, we received explicit satisfaction with our governance and leadership. Also, being flexible to fit online formats was a productive strategy that made the HIV in MOTION CoP sessions agile and amenable to audiovisual archiving. Our indicators of success in Process, Activities, and Outcomes included participant retention online, elicited verbal interventions and comments in the chat room, and a rate of three repeat visits online. The indicators of success of Impact were the presence of voluntary and unscripted autopathography, the patient storytelling and how it reportedly caused changes in the participants, and the "legitimate peripheral participation" of emerging research and clinical students. In conclusion, we recommend our form of CoP for mixing the knowledge of diverse persons in this area. However, we recommend considering budget and burnout as serious challenges to sustainability.

Piloting an online telecoaching community-based exercise intervention with adults living with HIV: protocol for a mixed-methods implementation science study.

INTRODUCTION: Our aim is to evaluate the implementation of an online telecoaching community-based exercise (CBE) intervention with the goal of reducing disability and enhancing physical activity and health among adults living with HIV. METHODS AND ANALYSIS: We will conduct a prospective longitudinal mixed-methods two-phased intervention study to pilot the implementation of an online CBE intervention with ~30 adults (≥18 years) living with HIV who consider themselves safe to participate in exercise. In the intervention phase (0-6 months), participants will take part in an online CBE intervention involving thrice weekly exercise (aerobic, resistance, balance and flexibility), with supervised biweekly personal training sessions with a fitness instructor, YMCA membership providing access to online exercise classes, wireless physical activity monitor to track physical activity and monthly online educational sessions on topics related to HIV, physical activity and health. In the follow-up phase (6-12 months), participants will be encouraged to continue independent exercise thrice weekly. Quantitative assessment: Bimonthly, we will assess cardiopulmonary fitness, strength, weight, body composition and flexibility, followed by administering self-reported questionnaires to assess disability, contextual factor outcomes (mastery, engagement in care, stigma, social support), implementation factors (cost, feasibility, technology), health status and self-reported physical activity. We will conduct a segmented regression analyses to describe the change in level and trend between the intervention and follow-up phases. Qualitative assessment: We will conduct online interviews with a subsample of ~10 participants and 5 CBE stakeholders at baseline (month 0), postintervention (month 6) and end of follow-up (month 12) to explore experiences, impact and implementation factors for online CBE. Interviews will be audiorecorded and analysed using content analytical techniques. ETHICS AND DISSEMINATION: Protocol approved by the University of Toronto Research Ethics Board (Protocol # 40410). Knowledge translation will occur in the form of presentations and publications in open-access peer-reviewed journals. TRIAL REGISTRATION NUMBER: NCT05006391.

Conceptualising the episodic nature of disability among adults living with Long COVID: a qualitative study.

INTRODUCTION: Our aim was to describe episodic nature of disability among adults living with Long COVID. METHODS: We conducted a community-engaged qualitative descriptive study involving online semistructured interviews and participant visual illustrations. We recruited participants via collaborator community organisations in Canada, Ireland, UK and USA.We recruited adults who self-identified as living with Long COVID with diversity in age, gender, race/ethnicity, sexual orientation and duration since initial COVID infection between December 2021 and May 2022. We used a semistructured interview guide to explore experiences of disability living with Long COVID, specifically health-related challenges and how they were experienced over time. We asked participants to draw their health trajectory and conducted a group-based content analysis. RESULTS: Among the 40 participants, the median age was 39 years (IQR: 32-49); majority were women (63%), white (73%), heterosexual (75%) and living with Long COVID for ≥1 year (83%). Participants described their disability experiences as episodic in nature, characterised by fluctuations in presence and severity of health-related challenges (disability) that may occur both within a day and over the long-term living with Long COVID. They described living with 'ups and downs', 'flare-ups' and 'peaks' followed by 'crashes', 'troughs' and 'valleys', likened to a 'yo-yo', 'rolling hills' and 'rollercoaster ride' with 'relapsing/remitting', 'waxing/waning', 'fluctuations' in health. Drawn illustrations demonstrated variety of trajectories across health dimensions, some more episodic than others. Uncertainty intersected with the episodic nature of disability, characterised as unpredictability of episodes, their length, severity and triggers, and process of long-term trajectory, which had implications on broader health. CONCLUSION: Among this sample of adults living with Long COVID, experiences of disability were described as episodic, characterised by fluctuating health challenges, which may be unpredictable in nature. Results can help to better understand experiences of disability among adults living with Long COVID to inform healthcare and rehabilitation.

Episodic Disability Questionnaire (EDQ) measurement properties among adults living with HIV in Canada, Ireland, United Kingdom, and United States.

Background: The Episodic Disability Questionnaire (EDQ) is a generic 35-item patient-reported outcome measure of presence, severity and episodic nature of disability. We assessed the measurement properties of the Episodic Disability Questionnaire (EDQ) with adults living with HIV. Methods: We conducted a measurement study with adults living with HIV in eight clinical settings in Canada, Ireland, United Kingdom, and United States. We electronically administered the EDQ followed by three reference measures (World Health Organization Disability Assessment Schedule; Patient Health Questionnaire; Social Support Scale) and a demographic questionnaire. We administered the EDQ only 1 week later. We assessed the internal consistency reliability (Cronbach's alpha; >0.7 acceptable), and test-retest reliability (Intra Class Correlation Coefficient; >0.7 acceptable). We estimated required change in EDQ domain scores to be 95% certain that a change was not due to measurement error (Minimum Detectable Change (MDC95%)). We evaluated construct validity by assessing 36 primary hypotheses of relationships between EDQ scores and scores on the reference measures (> 75% hypotheses confirmed indicated validity). Results: 359 participants completed the questionnaires at time point 1, of which 321 (89%) completed the EDQ approximately 1 week later. Cronbach's alpha for internal consistency ranged from 0.84 (social domain) to 0.91 (day domain) for the EDQ severity scale, and 0.72 (uncertainty domain) to 0.88 (day domain) for the EDQ presence scale, and 0.87 (physical, cognitive, mental-emotional domains) to 0.89 (uncertainty domain) for the EDQ episodic scale. ICCs for test-retest reliability ranged from 0.79 (physical domain) to 0.88 (day domain) for the EDQ severity scale and from 0.71 (uncertainty domain) to 0.85 (day domain) for the EDQ presence scale. Highest precision was demonstrated in the severity scale for each domain (MDC95% range: 19-25 out of 100), followed by the presence (MDC95% range: 37-54) and episodic scales (MDC95% range:44-76). Twenty-nine of 36 (81%) construct validity hypotheses were confirmed. Conclusions: The EDQ possesses internal consistency reliability, construct validity, and test-retest reliability, with limited precision when administered electronically with adults living with HIV across in clinical settings in four countries. Given the measurement properties, the EDQ can be used for group level comparisons for research and program evaluation in adults living with HIV.

Long COVID and episodic disability: advancing the conceptualisation, measurement and knowledge of episodic disability among people living with Long COVID - protocol for a mixed-methods study.

INTRODUCTION: As the prevalence of Long COVID increases, there is a critical need for a comprehensive assessment of disability. Our aims are to: (1) characterise disability experiences among people living with Long COVID in Canada, UK, USA and Ireland; and (2) develop a patient-reported outcome measure to assess the presence, severity and episodic nature of disability with Long COVID. METHODS AND ANALYSIS: In phase 1, we will conduct semistructured interviews with adults living with Long COVID to explore experiences of disability (dimensions, uncertainty, trajectories, influencing contextual factors) and establish an episodic disability (ED) framework in the context of Long COVID (n~10 each country). Using the conceptual framework, we will establish the Long COVID Episodic Disability Questionnaire (EDQ). In phase 2, we will examine the validity (construct, structural) and reliability (internal consistency, test-retest) of the EDQ for use in Long COVID. We will electronically administer the EDQ and four health status criterion measures with adults living with Long COVID, and readminister the EDQ 1 week later (n~170 each country). We will use Rasch analysis to refine the EDQ, and confirm structural and cross-cultural validity. We will calculate Cronbach's alphas (internal consistency reliability), and intraclass correlation coefficients (test-retest reliability), and examine correlations for hypotheses theorising relationships between EDQ and criterion measure scores (construct validity). Using phase 2 data, we will characterise the profile of disability using structural equation modelling techniques to examine relationships between dimensions of disability and the influence of intrinsic and extrinsic contextual factors. This research involves an academic-clinical-community partnership building on foundational work in ED measurement, Long COVID and rehabilitation. ETHICS AND DISSEMINATION: This study was approved by the University of Toronto Research Ethics Board. Knowledge translation will occur with community collaborators in the form of presentations and publications in open access peer-reviewed journals and presentations.

Assessing the sensibility and utility of a short-form version of the HIV Disability Questionnaire in clinical practice settings in Canada, Ireland and the USA: a mixed methods study.

OBJECTIVES: The Short-Form HIV Disability Questionnaire (SF-HDQ) was developed to measure the presence, severity and episodic nature of health challenges across six domains. Our aim was to assess the sensibility, utility and implementation of the SF-HDQ in clinical practice. DESIGN: Mixed methods study design involving semistructured interviews and questionnaire administration. PARTICIPANTS: We recruited adults living with HIV and HIV clinicians in Canada, Ireland and the USA. METHODS: We electronically administered the SF-HDQ followed by a Sensibility Questionnaire (face and content validity, ease of usage, format) and conducted semistructured interviews to explore the utility and implementation of the SF-HDQ in clinical practice. The threshold for sensibility was a median score of >5/7 (adults living with HIV) and>4/7 (HIV clinicians) for ≥80% of items. Qualitative interview data were analysed using directed content analysis. RESULTS: Median sensibility scores were >5 (adults living with HIV; n=29) and >4 (HIV clinicians; n=16) for 18/19 (95%) items. Interview data indicated that the SF-HDQ represents the health-related challenges of living with HIV and other concurrent health conditions; captures the daily episodic nature of HIV; and is easy to use. Clinical utility included measuring health challenges and change over time, guiding referral to specialists and services, setting goals, facilitating communication and fostering a multidisciplinary approach to care. Considerations for implementation included flexible, person-centred approaches to administration, and communicating scores based on personal preferences. CONCLUSIONS: The SF-HDQ possesses sensibility and utility for use in clinical settings with adults living with HIV and HIV clinicians in three countries.

[Hyper-IgE syndrome. Lessons from function and defects of STAT-3 or DOCK-8]. / Síndrome hiper-IgE. Lecciones de la función y defectos de STAT-3 o DOCK-8.

In the classification of primary immunodeficiencies, hyper-IgE syndrome, identified with OMIM code # 147060 in the Online Mendelian Inheritance in Man catalog, belongs to the group of syndromes associated with combined immunodeficiencies. It is characterized by elevated levels of IgE, eosinophilia, recurrent skin abscesses, pneumonia, lung parenchyma lesions, recurrent infections, rashes in newborns, eczema, sinusitis, otitis, and mucocutaneous candidiasis. Hyper-IgE syndrome can be transmitted by autosomal dominant or autosomal recessive modes of inheritance. Hyper-IgE syndrome in its dominant form includes non-immunological manifestations like characteristic facies, pathological dentition, scoliosis, bone disorders, and joint hyperextensibility. The reported cause of the dominant form is the loss of function of the signal transducer and activator of transcription 3 (STAT-3, with MIM # 102582). Mutations in dedicator of cytokines 8 (DOCK-8) is the most common cause of the autosomal recessive form of hyper-IgE syndrome.

En la Clasificación de las Inmunodeficiencias Primarias, el síndrome hiper-IgE, identificado con el código OMIM #147060 en el Catálogo Online Mendelian Inheritance in Man, pertenece al grupo de las inmunodeficiencias combinadas asociadas a síndromes. Se caracteriza por elevación de la concentración de IgE, eosinofilia, abscesos recurrentes en piel, neumonías, lesiones en parénquima pulmonar, infecciones recurrentes, erupciones en el recién nacido, eccema, sinusitis, otitis y candidiasis mucocutáneas. El síndrome hiper-IgE puede ser transmitido hereditariamente en forma autosómica dominante o autosómica recesiva. El síndrome hiper-IgE en su forma dominante incluye manifestaciones no inmunológicas como facies característica, dentición patológica, escoliosis, alteraciones óseas e hiperextensibilidad articular. La causa identificada en la forma dominante es la pérdida de la función del transductor de señales y activador de la transcripción 3 (STAT-3, MIM #102582). Las mutaciones en la proteína dedicada a la citocinesis 8 (DOCK-8) representan la mayoría de las causas de la forma autosómica recesiva del síndrome hiper-IgE.