This study examined rates of sustainability, defined as program continuation, and factors associated with sustainability 6 years after full implementation of five evidence-based practices in 49 sites in the National Implementing Evidence-Based Practices Project. Based on interviews with agency leaders and state leaders, 47 % of sites sustained the practice for 6 years, 16 % restarted the practice after a period of discontinuation, and 37 % discontinued the practice permanently. Agency leaders from discontinuing sites identified inadequate financial support, lack of prioritization, and workforce issues as barriers to continuation. Adequate financing, ongoing supervision, and monitoring of fidelity and outcome may promote long-term sustainability.

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Rationale: Sirolimus therapy stabilizes lung function and reduces the size of chylous effusions and lymphangioleiomyomas in patients with lymphangioleiomyomatosis.Objectives: To determine whether sirolimus has beneficial effects on lung function, cystic areas, and adjacent lung parenchyma; whether these effects are sustained; and whether sirolimus is well tolerated by patients.Methods: Lung function decline over time, lung volume occupied by cysts (cyst score), and lung tissue texture in the vicinity of the cysts were quantified with a computer-aided diagnosis system in 38 patients. Then we compared cyst scores from the last study on sirolimus with studies done on sirolimus therapy. In 12 patients, we evaluated rates of change in lung function and cyst scores off and on sirolimus.Measurements and Main Results: Sirolimus reduced yearly declines in FEV 1 (22.3 6 0.1 vs. 1.0 6 0.3% predicted; P , 0.001) and diffusing capacity of carbon monoxide (22.6 6 0.1 vs. 0.9 6 0.2% predicted; P , 0.001). Cyst scores 1.2 6 0.8 years (30.5 6 11.9%) and 2.5 6 2 years (29.7 6 12.1%) after initiating sirolimus were not significantly different from pretreatment values (28.4 6 12.5%). In 12 patients followed for 5 years, a significant reduction in rates of yearly decline in FEV 1 (21.4 6 0.2 vs. 0.3 6 0.4% predicted; P = 0.025) was observed. Analyses of 104 computed tomography scans showed a nonsignificant (P = 0.23) reduction in yearly rates of change of cyst scores (1.8 6 0.2 vs. 0.3 6 0.3%; P = 0.23) and lung texture features. Despite adverse events, most patients were able to continue sirolimus therapy.Conclusions: Sirolimus therapy slowed down lung function decline and increase in cystic lesions. Most patients were able to tolerate sirolimus therapy.Keywords: sirolimus; lung cystic destruction; LAM Lymphangioleiomyomatosis (LAM) is a multisystem disease affecting almost exclusively women, which is characterized by cystic lung destruction, renal angiomyolipomas, and lymphangioleiomyomas (1-4). Patients with LAM frequently present with dyspnea on exertion; recurrent pneumothoraces; chylous effusions; incidental detection of lung cysts on imaging studies; and, less frequently, bleeding angiomyolipomas (1, 2). The clinical course of LAM is variable but LAM is generally considered to be a chronic disease with a median transplant-free survival time spanning more than a decade (5). LAM lesions are characterized by proliferation of a neoplastic LAM cell, which has features both of smooth muscle and melanocytic cells (4). The histologic features of LAM consist of cysts and LAM cells in the walls of cysts and along blood vessels, lymphatics, and bronchioles, causing airways narrowing, vascular wall thickening, lymphatic disruption, and venous occlusion (4).The inherited form of LAM associated with tuberous sclerosis complex (TSC) is caused by mutations in the TSC1 or TSC2 suppressor genes (6-8). Sporadic LAM has been associated only with mutations of TSC2 (6-8). TSC1 and TSC2 encode, respectively, hamartin and tuberin, two Corres...

Regular HIV testing enables early identification and treatment of HIV among at-risk men who have sex with men (MSM). Characterizing HIV testing needs for Internet-using MSM informs development of Internet-facilitated testing interventions. In this systematic review we analyze HIV testing patterns among Internet-using MSM in the United States who report, through participation in an online study or survey, their HIV status as negative or unknown and identify demographic or behavioral risk factors associated with testing. We systematically searched multiple electronic databases for relevant English-language articles published between January 1, 2005 and December 16, 2014. Using meta-analysis, we summarized the proportion of Internet-using MSM who had ever tested for HIV and the proportion who tested in the 12 months preceding participation in the online study or survey. We also identified factors predictive of these outcomes using meta-regression and narrative synthesis. Thirty-two studies that enrolled 83,186 MSM met our inclusion criteria. Among the studies reporting data for each outcome, 85 % (95 % CI 82–87 %) of participants had ever tested, and 58 % (95 % CI 53–63 %) had tested in the year preceding enrollment in the study, among those for whom those data were reported. Age over 30 years, at least a college education, use of drugs, and self-identification as being homosexual or gay were associated with ever having tested for HIV. A large majority of Internet-using MSM indicated they had been tested for HIV at some point in the past. A smaller proportion—but still a majority—reported they had been tested within the year preceding study or survey participation. MSM who self-identify as heterosexual or bisexual, are younger, or who use drugs (including non-injection drugs) may be less likely to have ever tested for HIV. The overall findings of our systematic review are encouraging; however, a subpopulation of MSM may benefit from targeted outreach. These findings indicate unmet needs for HIV testing among Internet-using MSM and identify subpopulations that might benefit from targeted outreach, such as provision of HIV self-testing kits.

Common facilitators and barriers to implementation of integrated dual disorders treatment emerged across sites. The results confirmed the importance of the use of the consultant-trainer in the model of implementation, as well as the need for intensive activities at multiple levels to facilitate implementation. Further research on service implementation is needed, including but not limited to clarifying strategies to overcome barriers.

Few studies have examined predictors of long-term sustainability of evidence-based practices in mental health. This study used assessments of five evidence-based practices implemented in 49 sites in eight states at baseline and years 2, 4, and 8. Program characteristics, implementation characteristics, reinforcement activities, and sustainability factors were used to predict program survival status. The majority of predictors were not significant. Supervisor turnover in year 4 predicted survival status in year 8, but site characteristics, fidelity at implementation, quality improvement activities, and post-implementation activities had little impact on long-term program survival. This study extends previous sustainability research by examining the long-term impact of internal program factors over a substantial period of time using longitudinal prediction. Future research should also consider the influence of external factors such as financial policies.

Purpose: To determine if mosaic tuberous sclerosis complex (TSC) can be stratified into subtypes that correspond with prognosis and extent of disease. Methods: Next-generation sequencing of skin tumor and other samples was used to identify patients with mosaic pathogenic variants in TSC1 or TSC2 . Extent of disease, onset age, and family history of TSC were determined through retrospective analysis of patient records. Results: The median number of disease findings and age at penetrance differed between mosaic patients with asymmetrically distributed facial angiofibromas (4 findings, 24y, n=7), mosaic patients with bilaterally symmetric facial angiofibromas (8 findings, 10y, n=12), and germline TSC patients (10 findings, 4y, n=29). Cutaneous and internal organ involvement positively correlated in mosaic (R=0.62, p=0.005), but not germline (R=−0.24, p=0.24) TSC. Variant allele fraction (VAF) in the blood (range: 0-19%) positively correlated with the number of major features (R=0.55, p=0.028). Five had a TSC2 variant identified in the skin that was below detection in the blood. One of 12 children from a mosaic parent had TSC. Conclusion: The phenotype of mosaic TSC ranged from mild to indistinguishable from germline disease. Patients with mosaicism and asymmetric facial angiofibromas exhibited fewer findings, later onset, and lower VAF in the blood.

ClinicalTrials.gov; No.: NCT01687179; URL: www.clinicaltrials.gov.