{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,30]],"date-time":"2026-09-30T01:57:07Z","timestamp":1790733427469,"version":"4.1.0"},"reference-count":27,"publisher":"Wiley","issue":"18","license":[{"start":{"date-parts":[[2018,9,12]],"date-time":"2018-09-12T00:00:00Z","timestamp":1536710400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/100000054","name":"National Cancer Institute","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000054","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":["acsjournals.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Cancer"],"published-print":{"date-parts":[[2018,9,15]]},"abstract":"<jats:sec>\n                    <jats:title>BACKGROUND<\/jats:title>\n                    <jats:p>\n                      In the United States, only approximately 0.4% of all melanomas are diagnosed in patients aged &lt;20 years. To the authors' knowledge, melanoma in pediatric members of melanoma\u2010prone families has not been fully investigated to date. The objective of the current study was to evaluate pediatric patients with melanoma with extensive follow\u2010up in melanoma\u2010prone families with and without cyclin\u2010dependent kinase inhibitor 2A (\n                      <jats:italic>CDKN2A<\/jats:italic>\n                      ) mutations.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>METHODS<\/jats:title>\n                    <jats:p>\n                      For this non\u2010population\u2010based study, families were followed prospectively for up to 40 years. A total of 60 families with\u2009\u2265\u20093 patients with melanoma were included for analysis: 30\n                      <jats:italic>CDKN2A<\/jats:italic>\n                      mutation\u2010positive (\n                      <jats:italic>CDKN2A<\/jats:italic>\n                      +) and 30\n                      <jats:italic>CDKN2A<\/jats:italic>\n                      mutation\u2010negative (\n                      <jats:italic>CDKN2A<\/jats:italic>\n                      \u2010) families. Age at the time of first melanoma and number of melanomas were obtained for each patient and summarized by family or sets (\n                      <jats:italic>CDKN2A<\/jats:italic>\n                      \u2009+\u2009vs\n                      <jats:italic>CDKN2A<\/jats:italic>\n                      \u2010). For set comparisons and categorical variables (occurrence of melanoma in pediatric patients, number of melanomas, number of patients with single or multiple melanomas), the Pearson chi\u2010square or Fisher exact test was used.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>RESULTS<\/jats:title>\n                    <jats:p>\n                      Regardless of\n                      <jats:italic>CDKN2A<\/jats:italic>\n                      status, melanoma\u2010prone families were found to have 6\u2010fold to 28\u2010fold higher percentages of patients with pediatric melanoma compared with the general population of patients with melanoma in the United States. Within\n                      <jats:italic>CDKN2A<\/jats:italic>\n                      \u2009+\u2009families, pediatric patients with melanoma were significantly more likely to have multiple melanomas compared with their relatives who were diagnosed at age &gt;20 years (71% vs 38%, respectively;\n                      <jats:italic>P<\/jats:italic>\n                      \u2009=\u2009.004).\n                      <jats:italic>CDKN2A<\/jats:italic>\n                      \u2009+\u2009families had significantly higher percentages of pediatric patients with melanoma compared with\n                      <jats:italic>CDKN2A<\/jats:italic>\n                      \u2010 families (11.1% vs 2.5%;\n                      <jats:italic>P<\/jats:italic>\n                      \u2009=\u2009.004).\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>CONCLUSIONS<\/jats:title>\n                    <jats:p>These observations have implications for the prevention of melanoma as well as clinical care for its early detection. Children in melanoma\u2010prone families should have careful sun protection from an early age and skin surveillance to reduce their risk of melanoma.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/cncr.31641","type":"journal-article","created":{"date-parts":[[2018,9,12]],"date-time":"2018-09-12T11:33:17Z","timestamp":1536751997000},"page":"3715-3723","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":21,"title":["Pediatric melanoma in melanoma\u2010prone families"],"prefix":"10.1002","volume":"124","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-7538-3582","authenticated-orcid":false,"given":"Alisa M.","family":"Goldstein","sequence":"first","affiliation":[{"name":"Human Genetics Program, Division of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health, Department of Health and Human Services Bethesda Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kelsey C.","family":"Stidd","sequence":"additional","affiliation":[{"name":"Human Genetics Program, Division of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health, Department of Health and Human Services Bethesda Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xiaohong R.","family":"Yang","sequence":"additional","affiliation":[{"name":"Human Genetics Program, Division of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health, Department of Health and Human Services Bethesda Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mary C.","family":"Fraser","sequence":"additional","affiliation":[{"name":"Human Genetics Program, Division of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health, Department of Health and Human Services Bethesda Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Margaret A.","family":"Tucker","sequence":"additional","affiliation":[{"name":"Human Genetics Program, Division of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health, Department of Health and Human Services Bethesda Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2018,9,12]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1146\/annurev-genom-091212-153429"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1093\/jnci\/djq363"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1001\/archderm.137.11.1493"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmedgenet-2015-103150"},{"key":"e_1_2_8_6_1","volume-title":"SEER Cancer Statistics Review, 1975\u20102014","author":"Howlader N","year":"2017"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1159\/000247489"},{"key":"e_1_2_8_8_1","doi-asserted-by":"crossref","first-page":"1915","DOI":"10.1001\/jama.1986.03380140085027","article-title":"Familial malignant melanoma","volume":"256","author":"Kopf AW","year":"1986","journal-title":"JAMA"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1001\/archderm.142.4.485"},{"key":"e_1_2_8_10_1","first-page":"84","volume-title":"WHO Classification of Tumors: Pathology and Genetics of Skin Tumours","author":"Barnhill RL","year":"2006"},{"key":"e_1_2_8_11_1","first-page":"339","volume-title":"New Malignancies Among Cancer Survivors: SEER Cancer Registries, 1973\u20102000","author":"Curtis RE","year":"2006"},{"key":"e_1_2_8_12_1","doi-asserted-by":"publisher","DOI":"10.1038\/jid.2008.159"},{"key":"e_1_2_8_13_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.2012-2520"},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.1111\/pcmr.12574"},{"key":"e_1_2_8_15_1","doi-asserted-by":"publisher","DOI":"10.1158\/1055-9965.EPI-06-0270"},{"key":"e_1_2_8_16_1","doi-asserted-by":"publisher","DOI":"10.1158\/0008-5472.CAN-06-0494"},{"key":"e_1_2_8_17_1","doi-asserted-by":"publisher","DOI":"10.1186\/1897-4287-12-20"},{"key":"e_1_2_8_18_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM199803263381305"},{"key":"e_1_2_8_19_1","doi-asserted-by":"publisher","DOI":"10.1038\/sj.jid.5700689"},{"key":"e_1_2_8_20_1","doi-asserted-by":"publisher","DOI":"10.1097\/01.cmr.0000131014.79262.bf"},{"key":"e_1_2_8_21_1","unstructured":"ClinVar.https:\/\/www.ncbi.nlm.nih.gov\/clinvar\/variation\/127523\/#clinical-assertions. Accessed November 4 2017."},{"key":"e_1_2_8_22_1","doi-asserted-by":"publisher","DOI":"10.1038\/gim.2017.26"},{"key":"e_1_2_8_23_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature19057"},{"key":"e_1_2_8_24_1","doi-asserted-by":"publisher","DOI":"10.1016\/0190-9622(95)91284-3"},{"key":"e_1_2_8_25_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3476(83)80777-X"},{"key":"e_1_2_8_26_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2015.05.022"},{"key":"e_1_2_8_27_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10897-017-0185-5"},{"key":"e_1_2_8_28_1","doi-asserted-by":"publisher","DOI":"10.1038\/gim.2017.216"}],"container-title":["Cancer"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fcncr.31641","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/acsjournals.onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/cncr.31641","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,30]],"date-time":"2025-10-30T22:46:57Z","timestamp":1761864417000},"score":1,"resource":{"primary":{"URL":"https:\/\/acsjournals.onlinelibrary.wiley.com\/doi\/10.1002\/cncr.31641"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2018,9,12]]},"references-count":27,"journal-issue":{"issue":"18","published-print":{"date-parts":[[2018,9,15]]}},"alternative-id":["10.1002\/cncr.31641"],"URL":"https:\/\/doi.org\/10.1002\/cncr.31641","archive":["Portico"],"relation":{},"ISSN":["0008-543X","1097-0142"],"issn-type":[{"value":"0008-543X","type":"print"},{"value":"1097-0142","type":"electronic"}],"subject":[],"published":{"date-parts":[[2018,9,12]]},"assertion":[{"value":"2018-03-16","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2018-06-11","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2018-09-12","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}