{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T16:04:20Z","timestamp":1787069060632,"version":"build-2736575974"},"reference-count":38,"publisher":"Ovid Technologies (Wolters Kluwer Health)","issue":"5","license":[{"start":{"date-parts":[[2022,5,1]],"date-time":"2022-05-01T00:00:00Z","timestamp":1651363200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["www.ahajournals.org"],"crossmark-restriction":true},"short-container-title":["Circ: Arrhythmia and Electrophysiology"],"published-print":{"date-parts":[[2022,5]]},"abstract":"<jats:sec>\n                    <jats:title>Background:<\/jats:title>\n                    <jats:p>Maximal left ventricular wall thickness (MLVWT) is a risk factor for sudden cardiac death (SCD) in hypertrophic cardiomyopathy (HCM). In adults, the severity of left ventricular hypertrophy has a nonlinear relationship with SCD, but it is not known whether the same complex relationship is seen in childhood. The aim of this study was to describe the relationship between left ventricular hypertrophy and SCD risk in a large international pediatric HCM cohort.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods:<\/jats:title>\n                    <jats:p>The study cohort comprised 1075 children (mean age, 10.2 years [\u00b14.4]) diagnosed with HCM (1\u201316 years) from the International Paediatric Hypertrophic Cardiomyopathy Consortium. Anonymized, noninvasive clinical data were collected from baseline evaluation and follow-up, and 5-year estimated SCD risk was calculated (HCM Risk-Kids).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results:<\/jats:title>\n                    <jats:p>\n                      MLVWT\n                      <jats:italic>Z<\/jats:italic>\n                      score was &lt;10 in 598 (58.1%), \u226510 to &lt;20 in 334 (31.1%), and \u226520 in 143 (13.3%). Higher MLVWT\n                      <jats:italic>Z<\/jats:italic>\n                      scores were associated with heart failure symptoms, unexplained syncope, left ventricular outflow tract obstruction, left atrial dilatation, and nonsustained ventricular tachycardia. One hundred twenty-two patients (71.3%) with MLVWT\n                      <jats:italic>Z<\/jats:italic>\n                      score \u226520 had coexisting risk factors for SCD. Over a median follow-up of 4.9 years (interquartile range, 2.3\u20139.3), 115 (10.7%) had an SCD event. Freedom from SCD event at 5 years for those with MLVWT\n                      <jats:italic>Z<\/jats:italic>\n                      scores &lt;10, \u226510 to &lt;20, and \u226520 was 95.6%, 87.4%, and 86.0, respectively. The estimated SCD risk at 5 years had a nonlinear, inverted U-shaped relationship with MLVWT\n                      <jats:italic>Z<\/jats:italic>\n                      score, peaking at\n                      <jats:italic>Z<\/jats:italic>\n                      score +23. The presence of coexisting risk factors had a summative effect on risk.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions:<\/jats:title>\n                    <jats:p>In children with HCM, an inverted U-shaped relationship exists between left ventricular hypertrophy and estimated SCD risk. The presence of additional risk factors has a summative effect on risk. While MLVWT is important for risk stratification, it should not be used either as a binary variable or in isolation to guide implantable cardioverter defibrillator implantation decisions in children with HCM.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1161\/circep.121.010075","type":"journal-article","created":{"date-parts":[[2022,5,2]],"date-time":"2022-05-02T05:00:14Z","timestamp":1651467614000},"update-policy":"https:\/\/doi.org\/10.1161\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":21,"title":["Relationship Between Maximal Left Ventricular Wall Thickness and Sudden Cardiac Death in Childhood Onset Hypertrophic Cardiomyopathy"],"prefix":"10.1161","volume":"15","author":[{"given":"Gabrielle","family":"Norrish","sequence":"first","affiliation":[{"name":"Centre for Inherited Cardiovascular Diseases, Great Ormond Street Hospital, London, United Kingdom (G.N., E.F., E.C., J.P.K.)."},{"name":"Institute of Cardiovascular Sciences (G.N., C.O., P.M.E., J.P.K.), University College London, United Kingdom."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tao","family":"Ding","sequence":"additional","affiliation":[{"name":"Department of Statistical Science (T.D., R.Z.O.), University College London, United Kingdom."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0808-2763","authenticated-orcid":true,"given":"Ella","family":"Field","sequence":"additional","affiliation":[{"name":"Centre for Inherited Cardiovascular Diseases, Great Ormond Street Hospital, London, United Kingdom (G.N., E.F., E.C., J.P.K.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2612-1014","authenticated-orcid":true,"given":"Elena","family":"Cervi","sequence":"additional","affiliation":[{"name":"Centre for Inherited Cardiovascular Diseases, Great Ormond Street Hospital, London, United Kingdom (G.N., E.F., E.C., J.P.K.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0300-1941","authenticated-orcid":true,"given":"Lidia","family":"Zi\u00f3\u0142kowska","sequence":"additional","affiliation":[{"name":"The Children\u2019s Memorial Health Institute, Warsaw, Poland (L.Z.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1751-9266","authenticated-orcid":true,"given":"Iacopo","family":"Olivotto","sequence":"additional","affiliation":[{"name":"Careggi University Hopsital, Florence, Italy (I.O.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Diala","family":"Khraiche","sequence":"additional","affiliation":[{"name":"Necker\u2013Enfants Malades Hospital, Paris, France (D.K.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8291-9517","authenticated-orcid":true,"given":"Giuseppe","family":"Limongelli","sequence":"additional","affiliation":[{"name":"Monaldi Hospital, Naples, Italy (G.L.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aris","family":"Anastasakis","sequence":"additional","affiliation":[{"name":"Onassis Cardiac Surgery Center, Athens, Greece (A.A.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7324-8697","authenticated-orcid":true,"given":"Robert","family":"Weintraub","sequence":"additional","affiliation":[{"name":"Royal Children\u2019s Hospital, Melbourne, Australia (R.W.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0295-0440","authenticated-orcid":true,"given":"Elena","family":"Biagini","sequence":"additional","affiliation":[{"name":"Cardiology Unit, S. 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