{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,5]],"date-time":"2026-10-05T18:03:18Z","timestamp":1791223398888,"version":"4.1.0"},"reference-count":83,"publisher":"Ovid Technologies (Wolters Kluwer Health)","issue":"1","content-domain":{"domain":["www.ahajournals.org"],"crossmark-restriction":true},"short-container-title":["JAHA"],"published-print":{"date-parts":[[2024,1,2]]},"abstract":"<jats:sec sec-type=\"background\" xml:lang=\"en\">\n            <jats:title>Background<\/jats:title>\n            <jats:p xml:lang=\"en\">Individuals with genetic syndromes can manifest both congenital heart disease (CHD) and cancer attributable to possible common underlying pathways. To date, reliable risk estimates of hematopoietic cancer (HC) among children with CHD based on large population\u2010based data remain scant. This study sought to quantify the risk of HC by the presence of genetic syndrome among children with CHD.<\/jats:p>\n          <\/jats:sec>\n          <jats:sec xml:lang=\"en\">\n            <jats:title>Methods and Results<\/jats:title>\n            <jats:p xml:lang=\"en\">Data sources were the Canadian CHD database, a nationwide database on CHD (1999\u20132017), and the CCR (Canadian Cancer Registry). Standardized incidence ratios were calculated for comparing HC incidences in children with CHD with\u00a0the general pediatric population. A modified Kaplan\u2010Meier curve was used to estimate the cumulative incidence of HC with death as a competing risk. A total of 143\u2009794 children (aged 0\u201317\u2009years) with CHD were followed up from birth to age 18\u2009years for 1\u2009314\u2009603 person\u2010years. Of them, 8.6% had genetic syndromes, and 898 HC cases were observed. Children with known syndromes had a substantially higher risk of incident HC than the general pediatric population (standardized incidence ratio, 13.4 [95% CI, 11.7\u201315.1]). The cumulative incidence of HC was 2.44% (95% CI, 2.11\u20132.76) among children with a syndrome and 0.79% (95% CI, 0.72\u20130.87) among children without a syndrome. Acute myeloid leukemia had a higher cumulative incidence during early childhood than acute lymphoblastic leukemia.<\/jats:p>\n          <\/jats:sec>\n          <jats:sec xml:lang=\"en\">\n            <jats:title>Conclusions<\/jats:title>\n            <jats:p xml:lang=\"en\">This is the first large population\u2010based analysis documenting that known genetic syndromes in children with CHD are a significant predictor of HC. The finding could be essential in informing risk\u2010stratified policy recommendations for cancer surveillance in children with CHD.<\/jats:p>\n          <\/jats:sec>","DOI":"10.1161\/jaha.122.026604","type":"journal-article","created":{"date-parts":[[2023,12,29]],"date-time":"2023-12-29T10:05:56Z","timestamp":1703844356000},"update-policy":"https:\/\/doi.org\/10.1161\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":4,"title":["CanCHD Study of Hematopoietic Cancers in Children With and Without Genetic Syndromes"],"prefix":"10.1161","volume":"13","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-1774-2452","authenticated-orcid":false,"given":"Mohammad Sazzad","family":"Hasan","sequence":"first","affiliation":[{"name":"Department of Epidemiology, Biostatistics and Occupational Health McGill University Montreal Quebec Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2632-4228","authenticated-orcid":false,"given":"Elie","family":"Ganni","sequence":"additional","affiliation":[{"name":"McGill Adult Unit for Congenital Heart Disease Excellence McGill University Health Centre Montreal Quebec Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aihua","family":"Liu","sequence":"additional","affiliation":[{"name":"McGill Adult Unit for Congenital Heart Disease Excellence McGill University Health Centre Montreal Quebec Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6400-3775","authenticated-orcid":false,"given":"Liming","family":"Guo","sequence":"additional","affiliation":[{"name":"McGill Adult Unit for Congenital Heart Disease Excellence McGill University Health Centre Montreal Quebec Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrew S.","family":"Mackie","sequence":"additional","affiliation":[{"name":"Division of Cardiology, Stollery Children\u2019s Hospital and Department of Pediatrics University of Alberta Edmonton Alberta Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1606-401X","authenticated-orcid":false,"given":"Jay S.","family":"Kaufman","sequence":"additional","affiliation":[{"name":"Department of Epidemiology, Biostatistics and Occupational Health McGill University Montreal Quebec Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1784-8206","authenticated-orcid":false,"given":"Ariane J.","family":"Marelli","sequence":"additional","affiliation":[{"name":"Department of Epidemiology, Biostatistics and Occupational Health McGill University Montreal Quebec Canada"},{"name":"McGill Adult Unit for Congenital Heart Disease Excellence McGill University Health Centre Montreal Quebec Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"276","reference":[{"key":"e_1_3_1_2_2","doi-asserted-by":"publisher","DOI":"10.1161\/CIRCULATIONAHA.113.008396"},{"key":"e_1_3_1_3_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.cjca.2019.09.015"},{"key":"e_1_3_1_4_2","doi-asserted-by":"publisher","DOI":"10.1001\/jamanetworkopen.2019.6762"},{"key":"e_1_3_1_5_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.cjca.2019.09.023"},{"key":"e_1_3_1_6_2","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0116844"},{"key":"e_1_3_1_7_2","doi-asserted-by":"publisher","DOI":"10.1136\/hrt.2008.160309"},{"key":"e_1_3_1_8_2","doi-asserted-by":"publisher","DOI":"10.1093\/eurheartj\/ehl014"},{"key":"e_1_3_1_9_2","doi-asserted-by":"publisher","DOI":"10.1158\/1055-9965.EPI-10-0542"},{"key":"e_1_3_1_10_2","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.2235592100"},{"key":"e_1_3_1_11_2","doi-asserted-by":"publisher","DOI":"10.1093\/eurheartj\/ehr281"},{"key":"e_1_3_1_12_2","doi-asserted-by":"publisher","DOI":"10.1503\/cmaj.100463"},{"key":"e_1_3_1_13_2","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa0901249"},{"key":"e_1_3_1_14_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpeds.2013.10.074"},{"key":"e_1_3_1_15_2","doi-asserted-by":"publisher","DOI":"10.1007\/s10654-018-0357-0"},{"key":"e_1_3_1_16_2","doi-asserted-by":"publisher","DOI":"10.1001\/jamanetworkopen.2019.10584"},{"key":"e_1_3_1_17_2","doi-asserted-by":"publisher","DOI":"10.1038\/bjc.2014.103"},{"key":"e_1_3_1_18_2","doi-asserted-by":"publisher","DOI":"10.1161\/CIRCULATIONAHA.113.005425"},{"key":"e_1_3_1_19_2","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pmed.1003903"},{"key":"e_1_3_1_20_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.lanepe.2022.100407"},{"key":"e_1_3_1_21_2","doi-asserted-by":"publisher","DOI":"10.3322\/caac.21132"},{"key":"e_1_3_1_22_2","doi-asserted-by":"publisher","DOI":"10.1136\/bmj.f2360"},{"key":"e_1_3_1_23_2","doi-asserted-by":"publisher","DOI":"10.1093\/ije\/29.3.424"},{"key":"e_1_3_1_24_2","doi-asserted-by":"publisher","DOI":"10.3324\/haematol.2018.187716"},{"key":"e_1_3_1_25_2","doi-asserted-by":"publisher","DOI":"10.1088\/0952-4746\/33\/1\/1"},{"key":"e_1_3_1_26_2","doi-asserted-by":"publisher","DOI":"10.1017\/S1047951112002144"},{"key":"e_1_3_1_27_2","doi-asserted-by":"publisher","DOI":"10.1161\/CIRCRESAHA.116.309140"},{"key":"e_1_3_1_28_2","doi-asserted-by":"publisher","DOI":"10.1016\/S1054-8807(00)00050-8"},{"key":"e_1_3_1_29_2","doi-asserted-by":"publisher","DOI":"10.1002\/ijc.30126"},{"key":"e_1_3_1_30_2","doi-asserted-by":"publisher","DOI":"10.1289\/ehp.9023"},{"key":"e_1_3_1_31_2","doi-asserted-by":"publisher","DOI":"10.1007\/s11864-017-0485-x"},{"key":"e_1_3_1_32_2","doi-asserted-by":"publisher","DOI":"10.1615\/CritRevOncog.v16.i1-2.40"},{"key":"e_1_3_1_33_2","doi-asserted-by":"publisher","DOI":"10.1038\/bjc.2015.75"},{"key":"e_1_3_1_34_2","doi-asserted-by":"publisher","DOI":"10.1002\/bdr2.1390"},{"key":"e_1_3_1_35_2","doi-asserted-by":"publisher","DOI":"10.1161\/CIRCULATIONAHA.106.627224"},{"key":"e_1_3_1_36_2","volume-title":"Cancer Incidence in Five Continents","author":"Parkin D","year":"2002","unstructured":"Parkin D, Whelan S, Ferlay J, Teppo L, Thomas D. Cancer Incidence in Five Continents. Vol 8. International Agency for Cancer; 2002."},{"key":"e_1_3_1_37_2","doi-asserted-by":"publisher","DOI":"10.1111\/j.1600-6143.2009.02973.x"},{"key":"e_1_3_1_38_2","unstructured":"Rothman K Boice JD. Epidemiologic analysis with a programmable calculator. US Department of Health Education and Welfare Public Health Service National Institutes of Health. 1979:31\u201332."},{"key":"e_1_3_1_39_2","volume-title":"Epidemiologic Analysis with a Programmable Calculator","author":"Rothman KJ","year":"1982","unstructured":"Rothman KJ, Boice JD Jr. Epidemiologic Analysis with a Programmable Calculator. Kenneth Rothman; 1982."},{"key":"e_1_3_1_40_2","doi-asserted-by":"publisher","DOI":"10.1002\/bimj.4710350716"},{"key":"e_1_3_1_41_2","volume-title":"Statistics in Epidemiology: Methods, Techniques and Applications","author":"Sahai H","year":"1995","unstructured":"Sahai H, Khurshid A. Statistics in Epidemiology: Methods, Techniques and Applications. CRC Press; 1995."},{"key":"e_1_3_1_42_2","unstructured":"Breslow NE Day NE. Statistical methods in cancer research volume 2\u2013the design and analysis of cohort studies. IARC Scientific Publications. Statistical Methods in Cancer Research. 1986."},{"key":"e_1_3_1_43_2","doi-asserted-by":"publisher","DOI":"10.1136\/jech.38.1.85"},{"key":"e_1_3_1_44_2","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1097-0258(20000615\/30)19:11\/12<1495::AID-SIM441>3.0.CO;2-E"},{"key":"e_1_3_1_45_2","doi-asserted-by":"publisher","DOI":"10.1002\/ijc.21518"},{"key":"e_1_3_1_46_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(99)05264-2"},{"key":"e_1_3_1_47_2","doi-asserted-by":"publisher","DOI":"10.1161\/CIRCULATIONAHA.115.019137"},{"key":"e_1_3_1_48_2","doi-asserted-by":"publisher","DOI":"10.1038\/bjc.2014.526"},{"key":"e_1_3_1_49_2","doi-asserted-by":"publisher","DOI":"10.1038\/ejhg.2011.37"},{"key":"e_1_3_1_50_2","doi-asserted-by":"publisher","DOI":"10.1146\/annurev.pathmechdis.3.121806.154300"},{"key":"e_1_3_1_51_2","doi-asserted-by":"publisher","DOI":"10.1038\/bcj.2015.52"},{"key":"e_1_3_1_52_2","doi-asserted-by":"publisher","DOI":"10.1016\/S2352-3026(18)30106-6"},{"key":"e_1_3_1_53_2","doi-asserted-by":"publisher","DOI":"10.1038\/bjc.2015.415"},{"key":"e_1_3_1_54_2","doi-asserted-by":"publisher","DOI":"10.1007\/s00247-020-04803-0"},{"key":"e_1_3_1_55_2","doi-asserted-by":"publisher","DOI":"10.3390\/ijerph13111057"},{"key":"e_1_3_1_56_2","doi-asserted-by":"publisher","DOI":"10.2307\/3579149"},{"key":"e_1_3_1_57_2","doi-asserted-by":"publisher","DOI":"10.1080\/09553009414551761"},{"key":"e_1_3_1_58_2","doi-asserted-by":"publisher","DOI":"10.1182\/asheducation-2016.1.293"},{"key":"e_1_3_1_59_2","doi-asserted-by":"publisher","DOI":"10.1080\/09553009414550081"},{"key":"e_1_3_1_60_2","doi-asserted-by":"publisher","DOI":"10.1088\/0952-4746\/36\/1\/N1"},{"key":"e_1_3_1_61_2","doi-asserted-by":"publisher","DOI":"10.1158\/1055-9965.EPI-22-0393"},{"key":"e_1_3_1_62_2","doi-asserted-by":"publisher","DOI":"10.24095\/hpcdp.38.3.01"},{"key":"e_1_3_1_63_2","doi-asserted-by":"publisher","DOI":"10.1111\/pan.12705"},{"key":"e_1_3_1_64_2","doi-asserted-by":"publisher","DOI":"10.1007\/s12308-015-0256-1"},{"key":"e_1_3_1_65_2","first-page":"268","article-title":"Myelodysplastic syndromes: the pediatric point of view","volume":"80","author":"Locatelli F","year":"1995","unstructured":"Locatelli F, Zecca M, Pession A, Maserati E, De Stefano P, Severi F. Myelodysplastic syndromes: the pediatric point of view. Haematologica. 1995;80:268\u2013279.","journal-title":"Haematologica"},{"key":"e_1_3_1_66_2","doi-asserted-by":"publisher","DOI":"10.3390\/ijms17060838"},{"key":"e_1_3_1_67_2","first-page":"216","article-title":"Myelodysplastic syndromes in children: where are we today?","volume":"12","author":"Rau A","year":"2012","unstructured":"Rau A, Shreedhara A, Kumar S. Myelodysplastic syndromes in children: where are we today? Ochsner J. 2012;12:216\u2013220.","journal-title":"Ochsner J"},{"key":"e_1_3_1_68_2","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2018-10-844621"},{"key":"e_1_3_1_69_2","doi-asserted-by":"publisher","DOI":"10.3390\/jcm4010127"},{"key":"e_1_3_1_70_2","doi-asserted-by":"publisher","DOI":"10.1111\/bjh.12041"},{"key":"e_1_3_1_71_2","doi-asserted-by":"publisher","DOI":"10.3978\/j.issn.2224-4336.2015.04.08"},{"key":"e_1_3_1_72_2","doi-asserted-by":"publisher","DOI":"10.1177\/2040620712471368"},{"key":"e_1_3_1_73_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.kjms.2016.05.011"},{"key":"e_1_3_1_74_2","doi-asserted-by":"publisher","DOI":"10.3389\/fonc.2016.00041"},{"key":"e_1_3_1_75_2","doi-asserted-by":"publisher","DOI":"10.1093\/bfgp\/elt036"},{"key":"e_1_3_1_76_2","doi-asserted-by":"publisher","DOI":"10.1186\/2050-7771-1-23"},{"key":"e_1_3_1_77_2","doi-asserted-by":"publisher","DOI":"10.1161\/CIRCRESAHA.117.309186"},{"key":"e_1_3_1_78_2","doi-asserted-by":"publisher","DOI":"10.1161\/CIRCULATIONAHA.117.029138"},{"key":"e_1_3_1_79_2","doi-asserted-by":"publisher","DOI":"10.1542\/peds.71.2.235"},{"key":"e_1_3_1_80_2","doi-asserted-by":"publisher","DOI":"10.1093\/ije\/22.4.584"},{"key":"e_1_3_1_81_2","doi-asserted-by":"publisher","DOI":"10.1093\/ije\/dyz009"},{"key":"e_1_3_1_82_2","volume-title":"Data Quality Study of the 2015\u20132016 Discharge Abstract Database: A Focus on Hospital Harm","author":"Canadian Institute for Health Information","year":"2016","unstructured":"Canadian Institute for Health Information . Data Quality Study of the 2015\u20132016 Discharge Abstract Database: A Focus on Hospital Harm. CIHI; 2016."},{"key":"e_1_3_1_83_2","doi-asserted-by":"publisher","DOI":"10.1002\/pds.3446"},{"key":"e_1_3_1_84_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.annepidem.2016.08.016"}],"container-title":["Journal of the American Heart Association"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.ahajournals.org\/doi\/full\/10.1161\/JAHA.122.026604","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,5,13]],"date-time":"2024-05-13T12:20:15Z","timestamp":1715602815000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.ahajournals.org\/doi\/10.1161\/JAHA.122.026604"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,1,2]]},"references-count":83,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2024,1,2]]}},"alternative-id":["10.1161\/JAHA.122.026604"],"URL":"https:\/\/doi.org\/10.1161\/jaha.122.026604","relation":{},"ISSN":["2047-9980"],"issn-type":[{"value":"2047-9980","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,1,2]]},"assertion":[{"value":"2022-04-29","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-10-23","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-12-29","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e026604"}}