{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,23]],"date-time":"2026-10-23T03:02:55Z","timestamp":1792724575032,"version":"4.5.0"},"reference-count":43,"publisher":"SAGE Publications","issue":"2","license":[{"start":{"date-parts":[[2024,12,21]],"date-time":"2024-12-21T00:00:00Z","timestamp":1734739200000},"content-version":"vor","delay-in-days":366,"URL":"http:\/\/www.sagepub.com\/licence-information-for-chorus"}],"funder":[{"DOI":"10.13039\/100000090","name":"Congressionally Directed Medical Research Programs","doi-asserted-by":"publisher","award":["PR151448"],"award-info":[{"award-number":["PR151448"]}],"id":[{"id":"10.13039\/100000090","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000050","name":"National Heart, Lung, and Blood Institute","doi-asserted-by":"publisher","award":["K08 HL127277-01"],"award-info":[{"award-number":["K08 HL127277-01"]}],"id":[{"id":"10.13039\/100000050","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["World J Pediatr Congenit Heart Surg"],"published-print":{"date-parts":[[2024,3]]},"abstract":"<jats:p>Background: Despite surgical advances, children with tetralogy of Fallot\/pulmonary atresia\/major aortopulmonary collaterals (TOF\/PA\/MAPCAs) are subject to chronic right ventricular (RV) pressure and volume overload. Current diagnostic tools do not identify adverse myocardial remodeling and cannot predict progression to RV failure. We sought to identify a noninvasive, circulating signature of the systemic response to right heart stress to follow disease progression. Methods: Longitudinal data were collected from patients with TOF\/PA\/MAPCAs (N\u2009=\u20095) at the time of (1) early RV pressure overload and (2) late RV pressure and volume overload. Plasma protein and microRNA expression were evaluated using high-throughput data-independent mass spectroscopy and Agilent miR Microarray, respectively. Results: At the time of early RV pressure overload, median patient age was 0.34 years (0.02-9.37), with systemic RV pressures, moderate-severe hypertrophy, and preserved systolic function. Late RV pressure and volume overload occurred at a median age of 4.08 years (1.51-10.83), with moderate RV hypertrophy and dilation, and low normal RV function; 277 proteins were significantly dysregulated (log2FC \u22650.6\/\u2264\u22120.6, FDR\u22640.05), predicting downregulation in lipid transport (apolipoproteins), fibrinolytic system, and extracellular matrix structural proteins (talin 1, profilin 1); and upregulation in the respiratory burst. Increasing RV size and decreasing RV function correlated with decreasing structural protein expression. Similarly, miR expression predicted downregulation of extracellular matrix\u2013receptor interactions and upregulation in collagen synthesis. Conclusion: To our knowledge, we show for the first time a noninvasive protein and miR signature reflecting the systemic response to adverse RV myocardial remodeling in TOF\/PA\/MAPCAs which could be used to follow disease progression.<\/jats:p>","DOI":"10.1177\/21501351231213626","type":"journal-article","created":{"date-parts":[[2023,12,21]],"date-time":"2023-12-21T20:32:00Z","timestamp":1703190720000},"page":"162-173","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":3,"title":["A Noninvasive Circulating Signature of Combined Right Ventricular Pressure and Volume Overload in Tetralogy of Fallot\/Pulmonary Atresia\/Major Aortopulmonary Collateral Arteries"],"prefix":"10.1177","volume":"15","author":[{"given":"Katie L.","family":"Clouthier","sequence":"first","affiliation":[{"name":"Department of Pediatrics (Cardiology), Stanford University, Palo Alto, CA, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anne C.","family":"Taylor","sequence":"additional","affiliation":[{"name":"Department of Pediatrics (Cardiology), Stanford University, Palo Alto, CA, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ji","family":"Xuhuai","sequence":"additional","affiliation":[{"name":"Human Immune Monitoring Center and Functional Genomics Facility, Stanford University, Palo Alto, CA, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yuhan","family":"Liu","sequence":"additional","affiliation":[{"name":"Department of Medicine (Quantitative Science Unit), Stanford University, Palo Alto, CA, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sarah","family":"Parker","sequence":"additional","affiliation":[{"name":"Department of Medicine, Cedars-Sinai Medical Center, Los Angeles, CA, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jennifer","family":"Van Eyk","sequence":"additional","affiliation":[{"name":"Department of Medicine, Cedars-Sinai Medical Center, Los Angeles, CA, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sushma","family":"Reddy","sequence":"additional","affiliation":[{"name":"Department of Pediatrics (Cardiology), Stanford University, Palo Alto, CA, USA"},{"name":"Cardiovascular Institute, Stanford University, Los Angeles, CA, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2023,12,21]]},"reference":[{"key":"bibr1-21501351231213626","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(09)60657-7"},{"key":"bibr2-21501351231213626","doi-asserted-by":"publisher","DOI":"10.1161\/CIRCINTERVENTIONS.116.004952"},{"issue":"11","key":"bibr3-21501351231213626","first-page":"153","volume":"110","author":"Frigiola A","year":"2004","journal-title":"Circulation"},{"key":"bibr4-21501351231213626","doi-asserted-by":"publisher","DOI":"10.1161\/CIRCULATIONAHA.113.001600"},{"key":"bibr5-21501351231213626","doi-asserted-by":"publisher","DOI":"10.1378\/chest.12-1246"},{"key":"bibr6-21501351231213626","doi-asserted-by":"publisher","DOI":"10.1016\/j.athoracsur.2013.10.115"},{"key":"bibr7-21501351231213626","doi-asserted-by":"publisher","DOI":"10.1016\/S1672-0229(08)60044-3"},{"key":"bibr8-21501351231213626","doi-asserted-by":"publisher","DOI":"10.1016\/j.molmed.2008.03.006"},{"key":"bibr9-21501351231213626","doi-asserted-by":"publisher","DOI":"10.1161\/CIRCULATIONAHA.107.687947"},{"key":"bibr10-21501351231213626","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0014263"},{"key":"bibr11-21501351231213626","doi-asserted-by":"publisher","DOI":"10.1172\/jci.insight.91625"},{"key":"bibr12-21501351231213626","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.0608791103"},{"key":"bibr13-21501351231213626","doi-asserted-by":"publisher","DOI":"10.1038\/nm1582"},{"issue":"11","key":"bibr14-21501351231213626","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1371\/journal.pone.0241476","volume":"15","author":"Weldy CS","year":"2020","journal-title":"PLoS One"},{"key":"bibr15-21501351231213626","doi-asserted-by":"publisher","DOI":"10.1161\/CIR.0000000000000509"},{"key":"bibr16-21501351231213626","doi-asserted-by":"publisher","DOI":"10.3390\/children9121803"},{"key":"bibr17-21501351231213626","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijcha.2021.100921"},{"key":"bibr18-21501351231213626","doi-asserted-by":"publisher","DOI":"10.1016\/j.trsl.2020.10.001"},{"key":"bibr19-21501351231213626","doi-asserted-by":"publisher","DOI":"10.15252\/msb.20178126"},{"key":"bibr20-21501351231213626","doi-asserted-by":"crossref","unstructured":"Holewinski RJ, Parker SJ, Matlock AD, Venkatraman V, Van Eyk JE. 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