Evidence map›Paper›PMID 28972003›Full record

ArticleCirculation2017

Latent Rheumatic Heart Disease: Identifying the Children at Highest Risk of Unfavorable Outcome.

Andrea Beaton, Twalib Aliku, Alyssa Dewyer, Marni Jacobs, Jiji Jiang, Chris T Longenecker, Sulaiman Lubega, Robert McCarter, Mariana Mirabel, Grace Mirembe and 6 more

Open access · greenAbstract read
In one paragraph

Article in Circulation, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 39 papers, 5 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
39citing papers in PubMed, 5 pooled it
6.7field-weighted citation impact, top 3% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

39 citing papers in PubMed, 5 syntheses or guidelines pooled it, 72 citations in OpenAlex.

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4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

16 authors at 2 institutions in 4 countries.

Andrea BeatonChildren's National Health System, Washington DC (A.B., A.D., M.J., J.J., R.M., A.S., C.S.). Gulu University, Uganda (T.A.). Case Western Reserve University, Cleveland, OH (C.T.L.). Uganda Heart Institute, Kampala (S.L., J.N., E.O., E.T., P.L.). INSERM U970 (French National Institute of Health and Medical Research), Paris-Cardiovascular Research Center PARCC, France (M.M.). Joint Clinical Research Centers, Kampala, Uganda (G.M.). abeaton@childrennational.org.
Twalib AlikuChildren's National Health System, Washington DC (A.B., A.D., M.J., J.J., R.M., A.S., C.S.). Gulu University, Uganda (T.A.). Case Western Reserve University, Cleveland, OH (C.T.L.). Uganda Heart Institute, Kampala (S.L., J.N., E.O., E.T., P.L.). INSERM U970 (French National Institute of Health and Medical Research), Paris-Cardiovascular Research Center PARCC, France (M.M.). Joint Clinical Research Centers, Kampala, Uganda (G.M.).
Alyssa DewyerChildren's National Health System, Washington DC (A.B., A.D., M.J., J.J., R.M., A.S., C.S.). Gulu University, Uganda (T.A.). Case Western Reserve University, Cleveland, OH (C.T.L.). Uganda Heart Institute, Kampala (S.L., J.N., E.O., E.T., P.L.). INSERM U970 (French National Institute of Health and Medical Research), Paris-Cardiovascular Research Center PARCC, France (M.M.). Joint Clinical Research Centers, Kampala, Uganda (G.M.).
Marni JacobsChildren's National Health System, Washington DC (A.B., A.D., M.J., J.J., R.M., A.S., C.S.). Gulu University, Uganda (T.A.). Case Western Reserve University, Cleveland, OH (C.T.L.). Uganda Heart Institute, Kampala (S.L., J.N., E.O., E.T., P.L.). INSERM U970 (French National Institute of Health and Medical Research), Paris-Cardiovascular Research Center PARCC, France (M.M.). Joint Clinical Research Centers, Kampala, Uganda (G.M.).
Jiji JiangChildren's National Health System, Washington DC (A.B., A.D., M.J., J.J., R.M., A.S., C.S.). Gulu University, Uganda (T.A.). Case Western Reserve University, Cleveland, OH (C.T.L.). Uganda Heart Institute, Kampala (S.L., J.N., E.O., E.T., P.L.). INSERM U970 (French National Institute of Health and Medical Research), Paris-Cardiovascular Research Center PARCC, France (M.M.). Joint Clinical Research Centers, Kampala, Uganda (G.M.).
Chris T LongeneckerChildren's National Health System, Washington DC (A.B., A.D., M.J., J.J., R.M., A.S., C.S.). Gulu University, Uganda (T.A.). Case Western Reserve University, Cleveland, OH (C.T.L.). Uganda Heart Institute, Kampala (S.L., J.N., E.O., E.T., P.L.). INSERM U970 (French National Institute of Health and Medical Research), Paris-Cardiovascular Research Center PARCC, France (M.M.). Joint Clinical Research Centers, Kampala, Uganda (G.M.).
Sulaiman LubegaChildren's National Health System, Washington DC (A.B., A.D., M.J., J.J., R.M., A.S., C.S.). Gulu University, Uganda (T.A.). Case Western Reserve University, Cleveland, OH (C.T.L.). Uganda Heart Institute, Kampala (S.L., J.N., E.O., E.T., P.L.). INSERM U970 (French National Institute of Health and Medical Research), Paris-Cardiovascular Research Center PARCC, France (M.M.). Joint Clinical Research Centers, Kampala, Uganda (G.M.).
Robert McCarterChildren's National Health System, Washington DC (A.B., A.D., M.J., J.J., R.M., A.S., C.S.). Gulu University, Uganda (T.A.). Case Western Reserve University, Cleveland, OH (C.T.L.). Uganda Heart Institute, Kampala (S.L., J.N., E.O., E.T., P.L.). INSERM U970 (French National Institute of Health and Medical Research), Paris-Cardiovascular Research Center PARCC, France (M.M.). Joint Clinical Research Centers, Kampala, Uganda (G.M.).
Mariana MirabelChildren's National Health System, Washington DC (A.B., A.D., M.J., J.J., R.M., A.S., C.S.). Gulu University, Uganda (T.A.). Case Western Reserve University, Cleveland, OH (C.T.L.). Uganda Heart Institute, Kampala (S.L., J.N., E.O., E.T., P.L.). INSERM U970 (French National Institute of Health and Medical Research), Paris-Cardiovascular Research Center PARCC, France (M.M.). Joint Clinical Research Centers, Kampala, Uganda (G.M.).
Grace MirembeChildren's National Health System, Washington DC (A.B., A.D., M.J., J.J., R.M., A.S., C.S.). Gulu University, Uganda (T.A.). Case Western Reserve University, Cleveland, OH (C.T.L.). Uganda Heart Institute, Kampala (S.L., J.N., E.O., E.T., P.L.). INSERM U970 (French National Institute of Health and Medical Research), Paris-Cardiovascular Research Center PARCC, France (M.M.). Joint Clinical Research Centers, Kampala, Uganda (G.M.).
Judith NamuyongaChildren's National Health System, Washington DC (A.B., A.D., M.J., J.J., R.M., A.S., C.S.). Gulu University, Uganda (T.A.). Case Western Reserve University, Cleveland, OH (C.T.L.). Uganda Heart Institute, Kampala (S.L., J.N., E.O., E.T., P.L.). INSERM U970 (French National Institute of Health and Medical Research), Paris-Cardiovascular Research Center PARCC, France (M.M.). Joint Clinical Research Centers, Kampala, Uganda (G.M.).
Emmy OkelloChildren's National Health System, Washington DC (A.B., A.D., M.J., J.J., R.M., A.S., C.S.). Gulu University, Uganda (T.A.). Case Western Reserve University, Cleveland, OH (C.T.L.). Uganda Heart Institute, Kampala (S.L., J.N., E.O., E.T., P.L.). INSERM U970 (French National Institute of Health and Medical Research), Paris-Cardiovascular Research Center PARCC, France (M.M.). Joint Clinical Research Centers, Kampala, Uganda (G.M.).
Amy ScheelChildren's National Health System, Washington DC (A.B., A.D., M.J., J.J., R.M., A.S., C.S.). Gulu University, Uganda (T.A.). Case Western Reserve University, Cleveland, OH (C.T.L.). Uganda Heart Institute, Kampala (S.L., J.N., E.O., E.T., P.L.). INSERM U970 (French National Institute of Health and Medical Research), Paris-Cardiovascular Research Center PARCC, France (M.M.). Joint Clinical Research Centers, Kampala, Uganda (G.M.).
Emmanuel TenywaChildren's National Health System, Washington DC (A.B., A.D., M.J., J.J., R.M., A.S., C.S.). Gulu University, Uganda (T.A.). Case Western Reserve University, Cleveland, OH (C.T.L.). Uganda Heart Institute, Kampala (S.L., J.N., E.O., E.T., P.L.). INSERM U970 (French National Institute of Health and Medical Research), Paris-Cardiovascular Research Center PARCC, France (M.M.). Joint Clinical Research Centers, Kampala, Uganda (G.M.).
Craig SableChildren's National Health System, Washington DC (A.B., A.D., M.J., J.J., R.M., A.S., C.S.). Gulu University, Uganda (T.A.). Case Western Reserve University, Cleveland, OH (C.T.L.). Uganda Heart Institute, Kampala (S.L., J.N., E.O., E.T., P.L.). INSERM U970 (French National Institute of Health and Medical Research), Paris-Cardiovascular Research Center PARCC, France (M.M.). Joint Clinical Research Centers, Kampala, Uganda (G.M.).
Peter LwabiChildren's National Health System, Washington DC (A.B., A.D., M.J., J.J., R.M., A.S., C.S.). Gulu University, Uganda (T.A.). Case Western Reserve University, Cleveland, OH (C.T.L.). Uganda Heart Institute, Kampala (S.L., J.N., E.O., E.T., P.L.). INSERM U970 (French National Institute of Health and Medical Research), Paris-Cardiovascular Research Center PARCC, France (M.M.). Joint Clinical Research Centers, Kampala, Uganda (G.M.).
Inserm · FRChildren's National · US

Funding

Clinical and Translational Science Institute at Children's NationalUL1TR000075 · NCATS · CHILDREN'S RESEARCH INSTITUTE · PI GUAY-WOODFORD, LISA MARIE · 2012 to 2015
$13.7M
Research_ProjectU54HD090257 · NICHD · CHILDREN'S RESEARCH INSTITUTE · PI VITTORIO GALLO, VITTORIO GALLO VITTORIO GALLO · 2016 to 2020
$5.9M
Pericardial fat, inflammation, and structural heart disease in HIVK23HL123341 · NHLBI · CASE WESTERN RESERVE UNIVERSITY · PI LONGENECKER, CHRIS TODD · 2014 to 2018
$885k
Clinical and Translational Science Institute at Children's NationalKL2TR000076 · NCATS · CHILDREN'S RESEARCH INSTITUTE · PI GUAY-WOODFORD, LISA MARIE · 2012 to 2014
$756k
NCATS NIH HHS KL2 TR000076NCATS NIH HHS UL1 TR000075NHLBI NIH HHS K23 HL123341NHLBI NIH HHS L40 HL124634NICHD NIH HHS U54 HD090257
6 · The paper itself

Abstract

backgroundScreening echocardiography has emerged as a potentially powerful tool for early diagnosis of rheumatic heart disease (RHD). The utility of screening echocardiography hinges on the rate of RHD progression and the ability of penicillin prophylaxis to improve outcome. We report the longitudinal outcomes of a cohort of children with latent RHD and identify risk factors for unfavorable outcomes.

methodsThis was a prospective natural history study conducted under the Ugandan RHD registry. Children with latent RHD and ≥1 year of follow-up were included. All echocardiograms were re-reviewed by experts (2012 World Heart Federation criteria) for inclusion and evidence of change. Bi- and multivariable logistic regression, Kaplan-Meier analysis, and Cox proportional hazards models, as well, were developed to search for risk factors for unfavorable outcome and compare progression-free survival between those treated and not treated with penicillin. Propensity and other matching methods with sensitivity analysis were implemented for the evaluation of the penicillin effect.

resultsBlinded review confirmed 227 cases of latent RHD: 164 borderline and 63 definite (42 mild, 21 moderate/severe). Median age at diagnosis was 12 years and median follow-up was 2.3 years (interquartile range, 2.0-2.9). Penicillin prophylaxis was prescribed in 49.3% with overall adherence of 84.7%. Of children with moderate-to-severe definite RHD, 47.6% had echocardiographic progression (including 2 deaths), and 9.5% had echocardiographic regression. Children with mild definite and borderline RHD showed 26% and 9.8% echocardiographic progression and 45.2% and 46.3% echocardiographic improvement, respectively. Of those with mild definite RHD or borderline RHD, more advanced disease category, younger age, and morphological mitral valve features were risk factors for an unfavorable outcome.

conclusionsLatent RHD is a heterogeneous diagnosis with variable disease outcomes. Children with moderate to severe latent RHD have poor outcomes. Children with both borderline and mild definite RHD are at substantial risk of progression. Although long-term outcome remains unclear, the initial change in latent RHD may be evident during the first 1 to 2 years following diagnosis. Natural history data are inherently limited, and a randomized clinical trial is needed to definitively determine the impact of penicillin prophylaxis on the trajectory of latent RHD.

Indexed as

EchocardiographyAdolescentAge FactorsAnti-Bacterial AgentsChildDisease-Free SurvivalDisease ProgressionEarly DiagnosisFemaleHumansKaplan-Meier EstimateLogistic ModelsMaleMultivariate AnalysisOdds RatioPenicillinsAnti-Bacterial AgentsPenicillinsechocardiographyoutcome assessment (health care)pediatricsrheumatic heart diseasescreening

Identifiers

PMID28972003
PMCPMC5716883
OpenAlexW2756791488

What OpenQuestion holds

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.