Targeted Echocardiographic Screening for Latent Rheumatic Heart Disease in Northern Uganda: Evaluating Familial Risk Following Identification of an Index Case
Identifieur interne : 000D71 ( Main/Curation ); précédent : 000D70; suivant : 000D72Targeted Echocardiographic Screening for Latent Rheumatic Heart Disease in Northern Uganda: Evaluating Familial Risk Following Identification of an Index Case
Auteurs : Twalib Aliku [Ouganda] ; Craig Sable [États-Unis] ; Amy Scheel [États-Unis] ; Alison Tompsett [États-Unis] ; Peter Lwabi [Ouganda] ; Emmy Okello [Ouganda] ; Robert Mccarter [États-Unis] ; Marshall Summar [États-Unis] ; Andrea Beaton [États-Unis]Source :
- PLoS Neglected Tropical Diseases [ 1935-2727 ] ; 2016.
Abstract
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.
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%).
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.
Url:
DOI: 10.1371/journal.pntd.0004727
PubMed: 27294545
PubMed Central: 4905680
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Health System, Washington, District of Columbia, United States of America</addr-line></nlm:aff><country xml:lang="fr">États-Unis</country><wicri:regionArea>Division of Cardiology, Children’s National Health System, Washington, District of Columbia</wicri:regionArea><wicri:noRegion>District of Columbia</wicri:noRegion></affiliation></author><author><name sortKey="Tompsett, Alison" sort="Tompsett, Alison" uniqKey="Tompsett A" first="Alison" last="Tompsett">Alison Tompsett</name><affiliation wicri:level="1"><nlm:aff id="aff002"><addr-line>Division of Cardiology, Children’s National Health System, Washington, District of Columbia, United States of America</addr-line></nlm:aff><country xml:lang="fr">États-Unis</country><wicri:regionArea>Division of Cardiology, Children’s National Health System, Washington, District of Columbia</wicri:regionArea><wicri:noRegion>District of Columbia</wicri:noRegion></affiliation></author><author><name sortKey="Lwabi, Peter" sort="Lwabi, Peter" uniqKey="Lwabi P" first="Peter" last="Lwabi">Peter Lwabi</name><affiliation wicri:level="1"><nlm:aff id="aff003"><addr-line>Uganda Heart Institute, Kampala, Uganda</addr-line></nlm:aff><country xml:lang="fr">Ouganda</country><wicri:regionArea>Uganda Heart Institute, Kampala</wicri:regionArea><wicri:noRegion>Kampala</wicri:noRegion></affiliation></author><author><name sortKey="Okello, Emmy" sort="Okello, Emmy" uniqKey="Okello E" first="Emmy" last="Okello">Emmy Okello</name><affiliation wicri:level="1"><nlm:aff id="aff003"><addr-line>Uganda Heart Institute, Kampala, Uganda</addr-line></nlm:aff><country xml:lang="fr">Ouganda</country><wicri:regionArea>Uganda Heart Institute, Kampala</wicri:regionArea><wicri:noRegion>Kampala</wicri:noRegion></affiliation><affiliation wicri:level="1"><nlm:aff id="aff004"><addr-line>School of Medicine, Makerere University, Kampala, Uganda</addr-line></nlm:aff><country xml:lang="fr">Ouganda</country><wicri:regionArea>School of Medicine, Makerere University, Kampala</wicri:regionArea><wicri:noRegion>Kampala</wicri:noRegion></affiliation></author><author><name sortKey="Mccarter, Robert" sort="Mccarter, Robert" uniqKey="Mccarter R" first="Robert" last="Mccarter">Robert Mccarter</name><affiliation wicri:level="1"><nlm:aff id="aff005"><addr-line>Division of Biostatistics and Informatics, Children’s National Health System, Washington, District of Columbia, United States of America</addr-line></nlm:aff><country xml:lang="fr">États-Unis</country><wicri:regionArea>Division of Biostatistics and Informatics, Children’s National Health System, Washington, District of Columbia</wicri:regionArea><wicri:noRegion>District of Columbia</wicri:noRegion></affiliation></author><author><name sortKey="Summar, Marshall" sort="Summar, Marshall" uniqKey="Summar M" first="Marshall" last="Summar">Marshall Summar</name><affiliation wicri:level="1"><nlm:aff id="aff006"><addr-line>Division of Genetics and Metabolism, Children’s National Health System, Washington, District of Columbia, United States of America</addr-line></nlm:aff><country xml:lang="fr">États-Unis</country><wicri:regionArea>Division of Genetics and Metabolism, Children’s National Health System, Washington, District of Columbia</wicri:regionArea><wicri:noRegion>District of Columbia</wicri:noRegion></affiliation></author><author><name sortKey="Beaton, Andrea" sort="Beaton, Andrea" uniqKey="Beaton A" first="Andrea" last="Beaton">Andrea Beaton</name><affiliation wicri:level="1"><nlm:aff id="aff002"><addr-line>Division of Cardiology, Children’s National Health System, Washington, District of Columbia, United States of America</addr-line></nlm:aff><country xml:lang="fr">États-Unis</country><wicri:regionArea>Division of Cardiology, Children’s National Health System, Washington, District of Columbia</wicri:regionArea><wicri:noRegion>District of Columbia</wicri:noRegion></affiliation></author></analytic><series><title level="j">PLoS Neglected Tropical Diseases</title><idno type="ISSN">1935-2727</idno><idno type="eISSN">1935-2735</idno><imprint><date when="2016">2016</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><sec id="sec001"><title>Background</title><p>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.</p></sec><sec id="sec002"><title>Methodology/Principal Findings</title><p>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%).</p></sec><sec id="sec003"><title>Conclusions/Significance</title><p>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.</p></sec></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Carapetis, Jr" uniqKey="Carapetis J">JR Carapetis</name></author><author><name sortKey="Steer, Ac" uniqKey="Steer A">AC Steer</name></author><author><name sortKey="Mulholland, Ek" uniqKey="Mulholland E">EK Mulholland</name></author><author><name sortKey="Weber, M" uniqKey="Weber M">M Weber</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Carapetis, Jr" uniqKey="Carapetis J">JR Carapetis</name></author><author><name sortKey="Mcdonald, M" uniqKey="Mcdonald M">M McDonald</name></author><author><name sortKey="Wilson, Nj" uniqKey="Wilson N">NJ Wilson</name></author></analytic></biblStruct><biblStruct><analytic><author><name 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sortKey="Nkepu, S" uniqKey="Nkepu S">S Nkepu</name></author><author><name sortKey="Meiring, A" uniqKey="Meiring A">A Meiring</name></author></analytic></biblStruct></listBibl></div1></back></TEI></record>Pour manipuler ce document sous Unix (Dilib)
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