Childhood-onset hypertrophic cardiomyopathy (HCM) is far less common than adult-onset disease, thus natural history is not well characterized. We aim to describe the characteristics and outcomes of childhood-onset HCM. We performed an observational cohort study of 7677 HCM patients from the Sarcomeric Human Cardiomyopathy Registry (SHaRe). Hypertrophic cardiomyopathy patients were stratified by age at diagnosis [18 years (adulthood)] and assessed for composite endpoints reflecting heart failure (HF), life-threatening ventricular arrhythmias, atrial fibrillation (AF), and an overall composite that also included stroke and death. Stratifying by age of diagnosis, 184 (2.4%) patients were diagnosed in infancy; 1128 (14.7%) in childhood; and 6365 (82.9%) in adulthood. Childhood-onset HCM patients had an ∼2%/year event rate for the overall composite endpoint, with ventricular arrhythmias representing the most common event in the 1st decade following baseline visit, but HF and AF becoming more common by the end of the 2nd decade. Sarcomeric variants were more common in childhood-onset HCM (63%) and carried a worse prognosis than non-sarcomeric disease, including a greater than two-fold increased risk of HF [HR_adj 2.39 (1.36–4.20), P = 0.003] and 67% increased risk of the overall composite outcome [HR_adj 1.67 (1.16–2.41), P = 0.006]. When compared with adult-onset HCM, childhood-onset was 36% more likely to develop life-threatening ventricular arrhythmias [HR_adj 1.36 (1.03–1.80)] and twice as likely to require transplant or ventricular assist device [HR_adj 1.99 (1.23–3.23)]. Patients with childhood-onset HCM are more likely to have sarcomeric disease, carry a higher risk of life-threatening ventricular arrythmias, and have greater need for advanced HF therapies. These findings provide insight into the natural history of disease and can help inform clinical risk stratification. Natural history of sarcomeric HCM may differ depending on age of diagnosis with HCM.

Background. Crowdsourcing, the process of shifting individual tasks to a large group, may enhance human immunodeficiency virus (HIV) testing interventions. We conducted a noninferiority, randomized controlled trial to compare first-time HIV testing rates among men who have sex with men (MSM) and transgender individuals who received a crowdsourced or a health marketing HIV test promotion video. Methods. Seven hundred twenty-one MSM and transgender participants (≥16 years old, never before tested for HIV) were recruited through 3 Chinese MSM Web portals and randomly assigned to 1 of 2 videos. The crowdsourced video was developed using an open contest and formal transparent judging while the evidence-based health marketing video was designed by experts. Study objectives were to measure HIV test uptake within 3 weeks of watching either HIV test promotion video and cost per new HIV test and diagnosis. Results. Overall, 624 of 721 (87%) participants from 31 provinces in 217 Chinese cities completed the study. HIV test uptake was similar between the crowdsourced arm (37% [114/307]) and the health marketing arm (35% [111/317]). The estimated difference between the interventions was 2.1% (95% confidence interval, −5.4% to 9.7%). Among those tested, 31% (69/225) reported a new HIV diagnosis. The crowdsourced intervention cost substantially less than the health marketing intervention per first-time HIV test (US$131 vs US$238 per person) and per new HIV diagnosis (US$415 vs US$799 per person). Conclusions. Our nationwide study demonstrates that crowdsourcing may be an effective tool for improving HIV testing messaging campaigns and could increase community engagement in health campaigns. Clinical Trials Registration. NCT02248558.

Background: Crowdsourcing is used increasingly in health and medical research. Crowdsourcing is the process of aggregating crowd wisdom to solve a problem. The purpose of this systematic review is to summarize quantitative evidence on crowdsourcing to improve health. Methods: We followed Cochrane systematic review guidance and systematically searched seven databases up to September 4th 2019. Studies were included if they reported on crowdsourcing and related to health or medicine. Studies were excluded if recruitment was the only use of crowdsourcing. We determined the level of evidence associated with review findings using the GRADE approach. Results: We screened 3508 citations, accessed 362 articles, and included 188 studies. Ninety-six studies examined effectiveness, 127 examined feasibility, and 37 examined cost. The most common purposes were to evaluate surgical skills (17 studies), to create sexual health messages (seven studies), and to provide layperson cardio-pulmonary resuscitation (CPR) out-of-hospital (six studies). Seventeen observational studies used crowdsourcing to evaluate surgical skills, finding that crowdsourcing evaluation was as effective as expert evaluation (low quality). Four studies used a challenge contest to solicit human immunodeficiency virus (HIV) testing promotion materials and increase HIV testing rates (moderate quality), and two of the four studies found this approach saved money. Three studies suggested that an interactive technology system increased rates of layperson initiated CPR out-of-hospital (moderate quality). However, studies analyzing crowdsourcing to evaluate surgical skills and layperson-initiated CPR were only from high-income countries. Five studies examined crowdsourcing to inform artificial intelligence projects, most often related to annotation of medical data. Crowdsourcing was evaluated using different outcomes, limiting the extent to which studies could be pooled. Conclusions: Crowdsourcing has been used to improve health in many settings. Although crowdsourcing is effective at improving behavioral outcomes, more research is needed to understand effects on clinical outcomes and costs. More research is needed on crowdsourcing as a tool to develop artificial intelligence systems in medicine. Trial registration: PROSPERO: CRD42017052835. December 27, 2016.

Background: The impact of sex on phenotypic expression in hypertrophic cardiomyopathy (HCM) has not been well characterized in genotyped cohorts. Methods: Retrospective cohort study from an international registry of patients receiving care at experienced HCM centers. Sex-based differences in baseline characteristics and clinical outcomes were assessed. Results: Of 5873 patients (3788 genotyped), 2226 (37.9%) were women. At baseline, women were older (49.0±19.9 versus 42.9±18.4 years, P <0.001) and more likely to have pathogenic/likely pathogenic sarcomeric variants (HCM patients with a sarcomere mutation; 51% versus 43%, P <0.001) despite equivalent utilization of genetic testing. Age at diagnosis varied by sex and genotype despite similar distribution of causal genes. Women were 3.6 to 7.1 years older at diagnosis ( P <0.02) except for patients with MYH7 variants where age at diagnosis was comparable for women and men (n=492; 34.8±19.2 versus 33.3±16.8 years, P =0.39). Over 7.7 median years of follow-up, New York Heart Association III-IV heart failure was more common in women (hazard ratio, 1.87 [CI, 1.48–2.36], P <0.001), after controlling for their higher burden of symptoms and outflow tract obstruction at baseline, reduced ejection fraction, HCM patients with a sarcomere mutation, age, and hypertension. All-cause mortality was increased in women (hazard ratio, 1.50 [CI, 1.13–1.99], P <0.01) but neither implantable cardioverter-defibrillator utilization nor ventricular arrhythmia varied by sex. Conclusions: In HCM, women are older at diagnosis, partly modified by genetic substrate. Regardless of genotype, women were at higher risk of mortality and developing severe heart failure symptoms. This points to a sex-effect on long-term myocardial performance in HCM, which should be investigated further.

HIV self-testing offers an alternative to facility-based testing that could expand HIV testing among men who have sex with men (MSM). We organized an online survey of MSM in China to better understand the frequency and correlates of HIV self-testing. A total of 1342 individuals completed the survey. About 20.3% of MSM reported previous HIV self-testing. Self-testing was correlated with being married, having 6 or greater male anal sex partners in the past 3 months, and having HIV tested within 12 months in the multivariable analysis. Our study suggests that HIV self-testing may be able to reach subgroups of high-risk MSM and enable more frequent HIV testing.