Aliku Twalib, Sable Craig, Scheel Amy, Tompsett Alison, Lwabi Peter, Okello Emmy, McCarter Robert, Summar Marshall, Beaton Andrea
School of Medicine, Gulu University, Gulu, Uganda.
Division of Cardiology, Children's National Health System, Washington, District of Columbia, United States of America.
PLoS Negl Trop Dis. 2016 Jun 13;10(6):e0004727. doi: 10.1371/journal.pntd.0004727. eCollection 2016 Jun.
Echocardiographic screening for detection of latent RHD has shown potential as a strategy to decrease the burden of disease. However, further research is needed to determine optimal implementation strategies. RHD results from a complex interplay between environment and host susceptibility. Family members share both and relatives of children with latent RHD may represent a high-risk group. The objective of this study was to use echocardiographic family screening to determine the relative risk of RHD among first-degree relatives of children with latent RHD compared to the risk in first-degree relatives of healthy peers.
METHODOLOGY/PRINCIPAL FINDINGS: Previous school-based screening data were used to identify RHD positive children and RHD negative peers. All first-degree relatives ≥ 5 years were invited for echocardiography screening (2012 World Heart Federation Criteria). Sixty RHD positive cases (30 borderline/30 definite RHD) and 67 RHD negative cases were recruited. A total of 455/667 (68%) family members were screened. Definite RHD was more common in childhood siblings of RHD positive compared to RHD negative (p = 0.05). Children with any RHD were 4.5 times as likely to have a sibling with definite RHD, a risk that increased to 5.6 times when considering only cases with definite RHD. Mothers of RHD positive and RHD negative cases had an unexpectedly high rate of latent RHD (9.3%).
CONCLUSIONS/SIGNIFICANCE: Siblings of RHD positive cases with RHD are more likely to have definite RHD and the relative risk is highest if the index case has definite RHD. Future screening programs should consider implementation of sibling screening following detection of an RHD positive child. Larger screening studies of adults are needed, as data on prevalence of latent RHD outside of childhood are sparse. Future studies should prioritize implementation research to answer questions of how RHD screening can best be integrated into existing healthcare structures, ensuring practical and sustainable screening programs.
超声心动图筛查潜在风湿性心脏病（RHD）已显示出作为减轻疾病负担策略的潜力。然而，需要进一步研究以确定最佳实施策略。RHD是环境与宿主易感性之间复杂相互作用的结果。家庭成员共享这两者，患有潜在RHD的儿童的亲属可能代表高危人群。本研究的目的是使用超声心动图家庭筛查来确定患有潜在RHD的儿童的一级亲属中RHD的相对风险与健康同龄人的一级亲属中的风险相比。
方法/主要发现：以前基于学校的筛查数据用于识别RHD阳性儿童和RHD阴性同龄人。所有≥5岁的一级亲属均被邀请进行超声心动图筛查（2012年世界心脏联合会标准）。招募了60例RHD阳性病例（30例临界/30例确诊RHD）和67例RHD阴性病例。总共455/667（68%）名家庭成员接受了筛查。与RHD阴性儿童的童年兄弟姐妹相比，RHD阳性儿童的童年兄弟姐妹中确诊RHD更为常见（p = 0.05）。任何患有RHD的儿童有确诊RHD的兄弟姐妹的可能性是4.5倍，仅考虑确诊RHD病例时，这一风险增加到5.6倍。RHD阳性和RHD阴性病例的母亲中潜在RHD的发生率出乎意料地高（9.3%）。
结论/意义：患有RHD的RHD阳性病例的兄弟姐妹更有可能患有确诊RHD，如果索引病例患有确诊RHD，相对风险最高。未来的筛查计划应考虑在检测到RHD阳性儿童后实施兄弟姐妹筛查。需要对成年人进行更大规模的筛查研究，因为儿童期以外潜在RHD患病率的数据很少。未来的研究应优先进行实施研究，以回答如何将RHD筛查最佳地纳入现有医疗保健结构的问题，确保切实可行且可持续的筛查计划。
