{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,23]],"date-time":"2026-09-23T14:09:34Z","timestamp":1790172574870,"version":"4.1.0"},"reference-count":44,"publisher":"Wiley","issue":"8","license":[{"start":{"date-parts":[[2019,7,26]],"date-time":"2019-07-26T00:00:00Z","timestamp":1564099200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/100011541","name":"Division of Cancer Epidemiology and Genetics, National Cancer Institute","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100011541","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Ann Clin Transl Neurol"],"published-print":{"date-parts":[[2019,8]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Objectives<\/jats:title><jats:p>Recent evidence showed that myotonic dystrophy type I (DM1) patients are at increased risk of certain cancers, but the risk of benign tumors is unknown. We compared the risk of benign tumors in DM1 patients with matched DM1\u2010free individuals and assessed the association between benign tumors and subsequent cancers.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>We identified 927 DM1 patients and 13,085 DM1\u2010free individuals matched on gender, birth\u2010year, clinic, and clinic\u2010registration year from the UK Clinical Practice Research Datalink, a primary care records database. We used Cox regression models for statistical analyses.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>DM1 patients had elevated risks of thyroid nodules (Hazard Ratio [HR]\u00a0=\u00a010.4; 95% Confidence Interval [CI]\u00a0=\u00a03.91\u201327.52; <jats:italic>P<\/jats:italic>\u00a0&lt;\u00a00.001), benign tumors of the brain or nervous system (HR\u00a0=\u00a08.4; 95% CI\u00a0=\u00a02.48\u201328.47; <jats:italic>P<\/jats:italic>\u00a0&lt;\u00a00.001), colorectal polyps (HR\u00a0=\u00a04.3; 95% CI\u00a0=\u00a01.76\u201310.41; <jats:italic>P<\/jats:italic>\u00a0=\u00a00.001), and possibly uterine fibroids (HR\u00a0=\u00a02.7; 95% CI\u00a0=\u00a01.22\u20135.88; <jats:italic>P<\/jats:italic>\u00a0=\u00a00.01). Pilomatricomas and salivary gland adenomas occurred almost exclusively in DM1 patients (Fisher's exact <jats:italic>P<\/jats:italic>\u00a0&lt;\u00a00.001). The HR for colorectal polyps was elevated in DM1 males but not in females (HR\u00a0=\u00a08.2 vs. 1.3, respectively; <jats:italic>P<\/jats:italic>\u2010heterogeneity\u00a0&lt;\u00a00.001), whereas endocrine and brain tumors occurred exclusively in females. The data suggested an association between benign tumors and subsequent cancer in classic DM1 patients (HR\u00a0=\u00a02.7; 95% CI\u00a0=\u00a00.93\u20137.59; <jats:italic>P<\/jats:italic>\u00a0=\u00a00.07).<\/jats:p><\/jats:sec><jats:sec><jats:title>Interpretation<\/jats:title><jats:p>Our study showed a similar site\u2010specific benign tumor profile to that previously reported for DM1\u2010associated cancers. The possible association between benign tumors and subsequent cancer in classic DM1 patients warrants further investigation as it may guide identifying patients at elevated risk of cancer. Our findings underscore the importance of following population\u2010based screening recommendations in DM1 patients,\u00a0for example<jats:italic>,<\/jats:italic> for\u00a0colorectal cancer.<\/jats:p><\/jats:sec>","DOI":"10.1002\/acn3.50856","type":"journal-article","created":{"date-parts":[[2019,7,26]],"date-time":"2019-07-26T13:04:02Z","timestamp":1564146242000},"page":"1510-1518","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":25,"title":["Benign tumors in myotonic dystrophy type I target disease\u2010related cancer sites"],"prefix":"10.1002","volume":"6","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-5043-8789","authenticated-orcid":false,"given":"Rotana","family":"Alsaggaf","sequence":"first","affiliation":[{"name":"Clinical Genetics Branch, Division of Cancer Epidemiology and Genetics National Cancer Institute Bethesda Maryland"},{"name":"Department of Epidemiology and Public Health University of Maryland Baltimore Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Diane Marie M.","family":"St. George","sequence":"additional","affiliation":[{"name":"Department of Epidemiology and Public Health University of Maryland Baltimore Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Min","family":"Zhan","sequence":"additional","affiliation":[{"name":"Department of Epidemiology and Public Health University of Maryland Baltimore Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ruth M.","family":"Pfeiffer","sequence":"additional","affiliation":[{"name":"Biostatistics Branch, Division of Cancer Epidemiology and Genetics National Cancer Institute Bethesda Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5841-4183","authenticated-orcid":false,"given":"Youjin","family":"Wang","sequence":"additional","affiliation":[{"name":"Clinical Genetics Branch, Division of Cancer Epidemiology and Genetics National Cancer Institute Bethesda Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lesley A.","family":"Anderson","sequence":"additional","affiliation":[{"name":"Centre for Public Health, School of Medicine, Dentistry and Biomedical Science Queen's University Belfast Belfast Northern Ireland United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zhiwei","family":"Liu","sequence":"additional","affiliation":[{"name":"Infections and Immunoepidemiology Branch, Division of Cancer Epidemiology and Genetics National Cancer Institute Bethesda Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jill","family":"Koshiol","sequence":"additional","affiliation":[{"name":"Infections and Immunoepidemiology Branch, Division of Cancer Epidemiology and Genetics National Cancer Institute Bethesda Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrew J.","family":"Bauer","sequence":"additional","affiliation":[{"name":"Division of Endocrinology and Diabetes Children's Hospital of Philadelphia Philadelphia Pennsylvania USA"},{"name":"Department of Pediatrics, The Perelman School of Medicine The University of Pennsylvania Philadelphia Pennsylvania USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kathryn R.","family":"Wagner","sequence":"additional","affiliation":[{"name":"Hugo W. Moser Research Institute at Kennedy Krieger Institute Baltimore Maryland USA"},{"name":"Departments of Neurology and Neuroscience Johns Hopkins University School of Medicine Baltimore Maryland USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mark H.","family":"Greene","sequence":"additional","affiliation":[{"name":"Clinical Genetics Branch, Division of Cancer Epidemiology and Genetics National Cancer Institute Bethesda Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sania","family":"Amr","sequence":"additional","affiliation":[{"name":"Department of Epidemiology and Public Health University of Maryland Baltimore Maryland"},{"name":"Marlene and Stuart Greenebaum Comprehensive Cancer Center University of Maryland Baltimore Maryland USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shahinaz M.","family":"Gadalla","sequence":"additional","affiliation":[{"name":"Clinical Genetics Branch, Division of Cancer Epidemiology and Genetics National Cancer Institute Bethesda Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2019,7,26]]},"reference":[{"key":"e_1_2_8_2_1","unstructured":"BirdTD.Myotonic dystrophy type 1. 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