OBJECTIVE: Pathogenic desmoplakin (DSP) gene variants are associated with the development of a distinct form of arrhythmogenic cardiomyopathy known as DSP cardiomyopathy. Patients harbouring these variants are at high risk for sustained ventricular arrhythmia (VA), but existing tools for individualized arrhythmic risk assessment have proven unreliable in this population.
METHODS: Patients from the multi-national DSP-ERADOS (Desmoplakin SPecific Effort for a RAre Disease Outcome Study) Network patient registry who had pathogenic or likely pathogenic DSP variants and no sustained VA prior to enrolment were followed longitudinally for the development of first sustained VA event. Clinically guided, step-wise Cox regression analysis was used to develop a novel clinical tool predicting the development of incident VA. Model performance was assessed by c-statistic in both the model development cohort (n = 385) and in an external validation cohort (n = 86).
RESULTS: In total, 471 DSP patients [mean age 37.8 years, 65.6% women, 38.6% probands, 26% with left ventricular ejection fraction (LVEF) < 50%] were followed for a median of 4.0 (interquartile range: 1.6-7.3) years; 71 experienced first sustained VA events {2.6% [95% confidence interval (CI): 2.0, 3.5] events/year}. Within the development cohort, five readily available clinical parameters were identified as independent predictors of VA and included in a novel DSP risk score: female sex [hazard ratio (HR) 1.9 (95% CI: 1.1-3.4)], history of non-sustained ventricular tachycardia [HR 1.7 (95% CI: 1.1-2.8)], natural logarithm of 24-h premature ventricular contraction burden [HR 1.3 (95% CI: 1.1-1.4)], LVEF < 50% [HR 1.5 (95% CI: .95-2.5)], and presence of moderate to severe right ventricular systolic dysfunction [HR 6.0 (95% CI: 2.9-12.5)]. The model demonstrated good risk discrimination within both the development [c-statistic .782 (95% CI: .77-.80)] and external validation [c-statistic .791 (95% CI: .75-.83)] cohorts. The negative predictive value for DSP patients in the external validation cohort deemed to be at low risk for VA (<5% at 5 years; n = 26) was 100%.
CONCLUSIONS: The DSP risk score is a novel model that leverages readily available clinical parameters to provide individualized VA risk assessment for DSP patients. This tool may help guide decision-making for primary prevention implantable cardioverter-defibrillator placement in this high-risk population and supports a gene-first risk stratification approach.

UNASSIGNED: This study aimed to assess visual acuity (VA) in Congenital Zika Syndrome (CZS)-children to evaluate visual loss. To that end we evaluated 41 CZS - children, from Rio de Janeiro using Teller Acuity Cards.
UNASSIGNED: To asses VA, we evaluated 41 CZS - children, from Rio de Janeiro using Teller Acuity Cards. The children had Zika virus-infection confirmed by reverse transcription-polymerase chain reaction (RT-PCR) or clinical evaluation.
UNASSIGNED: In 39 out of 41 (95%) children, the VA scores were below normative values, while in 10 cases, VA was only marginally below normal; in the remaining 29 cases, VA was more than 0.15 logMAR below the lower limit. There was no correlation between VA and the cognitive domain tasks, although there was a correlation between VA and motor domain tasks. Thirty-seven children performed at least one task in the cognitive set, while fourteen children did not perform any task in the motor set. Children with VA above the lower limit performed better in the cognitive and motor tasks.
UNASSIGNED: We concluded that ZIKV- infected children with CZS were highly VA impaired which correlated with motor performance, but not with cognitive performance. Part of the children had VA within the normal limits and displayed better performance in the cognitive and motor sets. Therefore, even if heavily impaired, most children had some degree of VA and visual function.

Sleep-wake disturbances frequently present in Veterans with mild traumatic brain injury (mTBI). These TBI-related sleep impairments confer significant burden and commonly exacerbate other functional impairments. Therapies to improve sleep following mTBI are limited and studies in Veterans are even more scarce. In our previous pilot work, morning bright light therapy (MBLT) was found to be a feasible behavioral sleep intervention in Veterans with a history of mTBI; however, this was single-arm, open-label, and non-randomized, and therefore was not intended to establish efficacy. The present study, LION (light vs ion therapy) extends this preliminary work as a fully powered, sham-controlled, participant-masked randomized controlled trial (NCT03968874), implemented as fully remote within the VA (target n=120 complete). Randomization at 2:1 allocation ratio to: 1) active: MBLT (n=80), and 2) sham: deactivated negative ion generator (n=40); each with identical engagement parameters (60-min duration; within 2-hrs of waking; daily over 28-day duration). Participant masking via deception balanced expectancy assumptions across arms. Outcome measures were assessed following a 14-day baseline (pre-intervention), following 28-days of device engagement (post-intervention), and 28-days after the post-intervention assessment (follow-up). Primary outcomes were sleep measures, including continuous wrist-based actigraphy, self-report, and daily sleep dairy entries. Secondary/exploratory outcomes included cognition, mood, quality of life, circadian rhythm via dim light melatonin onset, and biofluid-based biomarkers. Participant drop out occurred in <10% of those enrolled, incomplete/missing data was present in <15% of key outcome variables, and overall fidelity adherence to the intervention was >85%, collectively establishing feasibility and acceptability for MBLT in Veterans with mTBI.

UNASSIGNED: The Department of Veterans Affairs (VA) Office of Rural Health (ORH) supports national VA program offices\' efforts to expand health care to rural Veterans through its Enterprise-Wide Initiatives (EWIs) program. In 2017, ORH selected Reach, Effectiveness, Adoption, Implementation, and Maintenance (RE-AIM), an implementation science framework, to structure the EWI evaluation and reporting process. As part of its mandate to improve EWI program evaluation, the Center for the Evaluation of Enterprise-Wide Initiatives conducted a qualitative evaluation to better understand EWI team\' perceptions of, and barriers and facilitators to, the EWI evaluation process.
UNASSIGNED: We conducted 43 semi-structured interviews with 48 team members (e.g., evaluators, program office leads, and field-based leads) representing 21 EWIs from April-December 2020. Questions focused on participants\' experiences using strategies targeting each RE-AIM dimension. Interviews were inductively analyzed in MAXQDA. We also systematically reviewed 51 FY19-FY20 EWI annual reports to identify trends in misapplications of RE-AIM.
UNASSIGNED: Participants had differing levels of experience with RE-AIM. While participants understood ORH\'s rationale for selecting a common framework to structure evaluations, the perceived misalignment between RE-AIM and EWIs\' work emerged as an important theme. Concerns centered around 3 sub-themes: (1) (Mis)Alignment with RE-AIM Dimensions, (2) (Mis)Alignment between RE-AIM and the EWI, and (3) (Mis)Alignment with RE-AIM vs. other Theories, Models, or Frameworks. Participants described challenges differentiating between and operationalizing dimensions in unique contexts. Participants also had misconceptions about RE-AIM and its relevance to their work, e.g., that it was meant for established programs and did not capture aspects of initiative planning, adaptations, or sustainability. Less commonly, participants shared alternative models or frameworks to RE-AIM. Despite criticisms, many participants found RE-AIM useful, cited training as important to understanding its application, and identified additional training as a future need.
UNASSIGNED: The selection of a shared implementation science framework can be beneficial, but also challenging when applied to diverse initiatives or contexts. Our findings suggest that establishing a common understanding, operationalizing framework dimensions for specific programs, and assessing training needs may better equip partners to integrate a shared framework into their evaluations.

BACKGROUND: American Indian and Alaska Natives serve in the military at one of the highest rates of all racial and ethnic groups. For Veterans, the already significant healthcare disparities Natives experience are aggravated by barriers to accessing care, care navigation, and coordination of health care within the Veterans Health Administration (VHA) between the VHA and tribal health systems. To mitigate these barriers, the VHA is developing a patient navigation program designed specifically for rural Native Veterans. We describe formative work aimed at understanding and addressing barriers to VHA care from the perspective of rural Native Veterans and those who facilitate their care.
METHODS: Thirty-four individuals participated in semi-structured interviews (22 Veterans, 6 family members, and 6 Veteran advocates) drawn from 9 tribal communities across the US.
RESULTS: Participants described many barriers to using the VHA, including perceptions of care scarcity, long travel distances to the VHA, high travel costs, and bureaucratic barriers including poor customer service, scheduling issues, and long waits for appointments. Many Veterans preferred IHS/tribal health care over the VHA due to its proximity, simplicity, ease of use, and quality.
CONCLUSIONS: Rural Native Veterans must see a clear benefit to using the VHA given the many obstacles to its use. Veteran recommendations for addressing barriers to VHA care within a navigation program include assistance enrolling in, scheduling, and navigating VHA systems; paperwork assistance; cost reimbursement; and care coordination with the IHS/tribal health care.

UNASSIGNED: Human astrovirus (HAstV) is an important pathogen of acute gastroenteritis (AGE) in children. This study was aimed at investigating the diversity and epidemiology of classic and novel HAstV in outpatient children aged 0-16 years old with AGE in Shanghai.
UNASSIGNED: From May 2020 to December 2022, a total of 1,482 stool samples were collected from children diagnosed as AGE from the Children\'s Hospital of Fudan University. HAstV was identified using pan-astrovirus consensus primers by Reverse transcription PCR.
UNASSIGNED: During the study period, 3.3% (49/1,482) of specimens were identified as HAstV, with a detection rate of 2.5% (37/1,482) for classic HAstV and 0.8% (12/1,482) for novel HAstV. Among the 12 novel HAstV strains, 11 (91.7%) belonged to the HAstV-MLB and 1 (8.3%) was HAstV-VA. Genotyping revealed six circulating genotypes. Strain HAstV-1 was predominant in the study population with a detection rate of 1.8% (26/1,482) followed by HAstV-MLB1 (0.7%, 10/1,482) and HAstV-4 (0.6%, 9/1,482). Of note, all the HAstV-4 strains detected in this study were close to one astrovirus strain isolated from Bactrian camels with 99.0-100.0% amino acid sequences identity. In this study, HAstV was detected in all age groups with the highest detection rate of HAstV-positive specimens observed in children older than 73 months (5.7%, 12/209).
UNASSIGNED: This study provided useful information and contributed to the molecular epidemiology of both classic and novel HAstV, which were simultaneously characterized and reported for the first time in Shanghai.

In this paper, the authors present a clinical picture of the diagnosis and current treatment regimens of optic pathway glioma in the pediatric population, with an emphasis on the role of an ophthalmic diagnosis in the differentiation and monitoring of lesions. Glioma is the most common optic nerve tumor in children.
METHODS: Articles in PubMed, Scholar and Website were reviewed, taking into account current standards of management related to sporadic or NF1-related optic glioma, epidemiology, location, course of the disease, clinical manifestations, histological types of the tumor, genetic predisposition, diagnostic ophthalmic tests currently applicable in therapeutic monitoring of the tumor, neurological diagnosis, therapeutic management and prognosis. The importance of current screening recommendations, in line with standards, was emphasized.
RESULTS: Glioma occurs in children most often in the first decade of life. Initially, they may be asymptomatic, and clinically ophthalmic changes are associated with the organ of vision or with systemic changes. Gliomas associated with the NF1 mutation have a better prognosis for sporadic gliomas. Diagnosis includes radiological imaging methods/MRI/ophthalmology/OCT and visual acuity log MAR assessment. The basis of treatment is clinical observation. In the case of disease progression, surgical treatment, chemotherapy and targeted therapy are used.
CONCLUSIONS: Further research into novel techniques for detecting gliomas would allow for early monitoring of the disease.

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BACKGROUND: It is unclear whether interventions designed to increase housing stability can also lead to improved health outcomes such as reduced risk of death and suicide morbidity. The objective of this study was to estimate the potential impact of temporary financial assistance (TFA) for housing-related expenses from the US Department of Veterans Affairs (VA) on health outcomes including all-cause mortality, suicide attempt, and suicidal ideation.
METHODS: We conducted a retrospective national cohort study of Veterans who entered the VA Supportive Services for Veteran Families (SSVF) program between 10/2015 and 9/2018. We assessed the association between TFA and health outcomes using a multivariable Cox proportional hazards regression approach with inverse probability of treatment weighting. We conducted these analyses on our overall cohort as well as separately for those in the rapid re-housing (RRH) and homelessness prevention (HP) components of SSVF. Outcomes were all-cause mortality, suicide attempt, and suicidal ideation at 365 and 730 days following enrollment in SSVF.
RESULTS: Our analysis cohort consisted of 41,969 unique Veterans with a mean (SD) duration of 87.6 (57.4) days in the SSVF program. At 365 days following SSVF enrollment, TFA was associated with a decrease in the risk of all-cause mortality (HR: 0.696, p < 0.001) and suicidal ideation (HR: 0.788, p < 0.001). We found similar results at 730 days (HR: 0.811, p = 0.007 for all-cause mortality and HR: 0.881, p = 0.037 for suicidal ideation). These results were driven primarily by individuals enrolled in the RRH component of SSVF. We found no association between TFA and suicide attempts.
CONCLUSIONS: We find that providing housing-related financial assistance to individuals facing housing instability is associated with improvements in important health outcomes such as all-cause mortality and suicidal ideation. If causal, these results suggest that programs to provide housing assistance have positive spillover effects into other important aspects of individuals\' lives.

Faith-based organizations (FBOs) are often \"gatekeepers\" to mental health care for congregants at risk of mental illness and suicide, especially U.S. military Veterans, but data to inform better collaboration are needed. We conducted focus groups with clergy in Los Angeles County to understand the mental health support FBOs provide and barriers to collaboration with the mental healthcare system. Clergy detailed strategies used to support the mental health of Veteran congregants. Barriers included stigma, limits in clergy training, and incomplete knowledge about community and VA mental health resources. Results suggest strategies to improve collaboration between FBOs and the mental healthcare system in Los Angeles County.