Clinical features of biliary tract cancer in Japanese individuals with Lynch syndrome
BackgroundBiliary tract cancer (BTC) is a Lynch syndrome (LS)-associated cancer with a high mortality rate. This study aimed to clarify the clinical features of BTC in individuals with LS and to discuss its management. MethodsWe obtained data from genetically verified Japanese individuals with LS wh...
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| Veröffentlicht in: | Journal of gastrointestinal oncology 2022-10, Vol.13 (5), p.2532-2538 |
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| creator | Kanaya, Nobuhiko Aoki, Hideki Morito, Toshiaki Taniguchi, Fumitaka Shigeyasu, Kunitoshi Tamura, Chieko Sugano, Kokichi Akagi, Kiwamu Ishida, Hideyuki Tanakaya, Kohji |
| description | BackgroundBiliary tract cancer (BTC) is a Lynch syndrome (LS)-associated cancer with a high mortality rate. This study aimed to clarify the clinical features of BTC in individuals with LS and to discuss its management. MethodsWe obtained data from genetically verified Japanese individuals with LS who were diagnosed at a single institution, between January 2003 and April 2021. Moreover, 21 individuals with sporadic BTC (n=15) and LS associated BTC (n=6) underwent microsatellite instability (MSI) testing. ResultsAmong 92 individuals with LS, 6 individuals with MLH1 variants developed BTCs (10 lesions, male/female, 2:1). The median age at diagnosis of initial BTC was 69 years (range, 34-78 years). Histological examination revealed a predominance of differentiated adenocarcinoma (89%). Then, 2 individuals had multiple BTCs. All available 7 BTC lesions showed high-frequency of microsatellite instability (MSI-H). MLH1 carriers showed a 7.2% cumulative risk of BTC development at an age of 70 years. Five of the six individuals died of BTC. ConclusionsMSI analysis could facilitate LS identification in individuals with BTC. Surveillance for BTC should be considered for MLH1 carriers in Japan. |
| doi_str_mv | 10.21037/jgo-22-165 |
| format | Article |
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This study aimed to clarify the clinical features of BTC in individuals with LS and to discuss its management. MethodsWe obtained data from genetically verified Japanese individuals with LS who were diagnosed at a single institution, between January 2003 and April 2021. Moreover, 21 individuals with sporadic BTC (n=15) and LS associated BTC (n=6) underwent microsatellite instability (MSI) testing. ResultsAmong 92 individuals with LS, 6 individuals with MLH1 variants developed BTCs (10 lesions, male/female, 2:1). The median age at diagnosis of initial BTC was 69 years (range, 34-78 years). Histological examination revealed a predominance of differentiated adenocarcinoma (89%). Then, 2 individuals had multiple BTCs. All available 7 BTC lesions showed high-frequency of microsatellite instability (MSI-H). MLH1 carriers showed a 7.2% cumulative risk of BTC development at an age of 70 years. Five of the six individuals died of BTC. ConclusionsMSI analysis could facilitate LS identification in individuals with BTC. Surveillance for BTC should be considered for MLH1 carriers in Japan.</description><identifier>ISSN: 2078-6891</identifier><identifier>EISSN: 2219-679X</identifier><identifier>DOI: 10.21037/jgo-22-165</identifier><language>eng</language><publisher>AME Publishing Company</publisher><subject>Original</subject><ispartof>Journal of gastrointestinal oncology, 2022-10, Vol.13 (5), p.2532-2538</ispartof><rights>2022 Journal of Gastrointestinal Oncology. All rights reserved. 2022 Journal of Gastrointestinal Oncology.</rights><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC9660066/pdf/$$EPDF$$P50$$Gpubmedcentral$$Hfree_for_read</linktopdf><linktohtml>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC9660066/$$EHTML$$P50$$Gpubmedcentral$$Hfree_for_read</linktohtml><link.rule.ids>230,314,723,776,780,881,27901,27902,53766,53768</link.rule.ids></links><search><creatorcontrib>Kanaya, Nobuhiko</creatorcontrib><creatorcontrib>Aoki, Hideki</creatorcontrib><creatorcontrib>Morito, Toshiaki</creatorcontrib><creatorcontrib>Taniguchi, Fumitaka</creatorcontrib><creatorcontrib>Shigeyasu, Kunitoshi</creatorcontrib><creatorcontrib>Tamura, Chieko</creatorcontrib><creatorcontrib>Sugano, Kokichi</creatorcontrib><creatorcontrib>Akagi, Kiwamu</creatorcontrib><creatorcontrib>Ishida, Hideyuki</creatorcontrib><creatorcontrib>Tanakaya, Kohji</creatorcontrib><title>Clinical features of biliary tract cancer in Japanese individuals with Lynch syndrome</title><title>Journal of gastrointestinal oncology</title><description>BackgroundBiliary tract cancer (BTC) is a Lynch syndrome (LS)-associated cancer with a high mortality rate. This study aimed to clarify the clinical features of BTC in individuals with LS and to discuss its management. MethodsWe obtained data from genetically verified Japanese individuals with LS who were diagnosed at a single institution, between January 2003 and April 2021. Moreover, 21 individuals with sporadic BTC (n=15) and LS associated BTC (n=6) underwent microsatellite instability (MSI) testing. ResultsAmong 92 individuals with LS, 6 individuals with MLH1 variants developed BTCs (10 lesions, male/female, 2:1). The median age at diagnosis of initial BTC was 69 years (range, 34-78 years). Histological examination revealed a predominance of differentiated adenocarcinoma (89%). Then, 2 individuals had multiple BTCs. All available 7 BTC lesions showed high-frequency of microsatellite instability (MSI-H). MLH1 carriers showed a 7.2% cumulative risk of BTC development at an age of 70 years. Five of the six individuals died of BTC. ConclusionsMSI analysis could facilitate LS identification in individuals with BTC. Surveillance for BTC should be considered for MLH1 carriers in Japan.</description><subject>Original</subject><issn>2078-6891</issn><issn>2219-679X</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2022</creationdate><recordtype>article</recordtype><recordid>eNpVUU1LAzEUDKJgqT35B3IUZDXJbrLJRZDiJwUvFryFbDZpU3aTmuxW-u-Ntgi-yxuYYd48BoBLjG4IRmV9u1mFgpACM3oCJoRgUbBafJxmjGpeMC7wOZiltEF5KkERJROwnHfOO606aI0axmgSDBY2rnMq7uEQlR6gVl6bCJ2Hr2qrvEkm49btXDuqLsEvN6zhYu_1Gqa9b2PozQU4s5kys-OeguXjw_v8uVi8Pb3M7xeFLjEbCq5aWlGtObaIcKIVryxVpKQI10KIqjLc6JZa2jCmsOV1o4xlnPGGCU5EVU7B3cF3Oza9abXxOXEnt9H1Ob4Mysn_jHdruQo7KRhDiLFscHU0iOFzNGmQvUvadF1-M4xJkrqsK0Y5-ZFeH6Q6hpSisX9nMJK_BchcgCRE5gLKb1Vqesk</recordid><startdate>202210</startdate><enddate>202210</enddate><creator>Kanaya, Nobuhiko</creator><creator>Aoki, Hideki</creator><creator>Morito, Toshiaki</creator><creator>Taniguchi, Fumitaka</creator><creator>Shigeyasu, Kunitoshi</creator><creator>Tamura, Chieko</creator><creator>Sugano, Kokichi</creator><creator>Akagi, Kiwamu</creator><creator>Ishida, Hideyuki</creator><creator>Tanakaya, Kohji</creator><general>AME Publishing Company</general><scope>AAYXX</scope><scope>CITATION</scope><scope>7X8</scope><scope>5PM</scope></search><sort><creationdate>202210</creationdate><title>Clinical features of biliary tract cancer in Japanese individuals with Lynch syndrome</title><author>Kanaya, Nobuhiko ; Aoki, Hideki ; Morito, Toshiaki ; Taniguchi, Fumitaka ; Shigeyasu, Kunitoshi ; Tamura, Chieko ; Sugano, Kokichi ; Akagi, Kiwamu ; Ishida, Hideyuki ; Tanakaya, Kohji</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c316t-8ad545cc81f0282ca84f5a23501799944e8ecd5f5b66a1f87baef6868b6982943</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2022</creationdate><topic>Original</topic><toplevel>online_resources</toplevel><creatorcontrib>Kanaya, Nobuhiko</creatorcontrib><creatorcontrib>Aoki, Hideki</creatorcontrib><creatorcontrib>Morito, Toshiaki</creatorcontrib><creatorcontrib>Taniguchi, Fumitaka</creatorcontrib><creatorcontrib>Shigeyasu, Kunitoshi</creatorcontrib><creatorcontrib>Tamura, Chieko</creatorcontrib><creatorcontrib>Sugano, Kokichi</creatorcontrib><creatorcontrib>Akagi, Kiwamu</creatorcontrib><creatorcontrib>Ishida, Hideyuki</creatorcontrib><creatorcontrib>Tanakaya, Kohji</creatorcontrib><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>Journal of gastrointestinal oncology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Kanaya, Nobuhiko</au><au>Aoki, Hideki</au><au>Morito, Toshiaki</au><au>Taniguchi, Fumitaka</au><au>Shigeyasu, Kunitoshi</au><au>Tamura, Chieko</au><au>Sugano, Kokichi</au><au>Akagi, Kiwamu</au><au>Ishida, Hideyuki</au><au>Tanakaya, Kohji</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Clinical features of biliary tract cancer in Japanese individuals with Lynch syndrome</atitle><jtitle>Journal of gastrointestinal oncology</jtitle><date>2022-10</date><risdate>2022</risdate><volume>13</volume><issue>5</issue><spage>2532</spage><epage>2538</epage><pages>2532-2538</pages><issn>2078-6891</issn><eissn>2219-679X</eissn><abstract>BackgroundBiliary tract cancer (BTC) is a Lynch syndrome (LS)-associated cancer with a high mortality rate. This study aimed to clarify the clinical features of BTC in individuals with LS and to discuss its management. MethodsWe obtained data from genetically verified Japanese individuals with LS who were diagnosed at a single institution, between January 2003 and April 2021. Moreover, 21 individuals with sporadic BTC (n=15) and LS associated BTC (n=6) underwent microsatellite instability (MSI) testing. ResultsAmong 92 individuals with LS, 6 individuals with MLH1 variants developed BTCs (10 lesions, male/female, 2:1). The median age at diagnosis of initial BTC was 69 years (range, 34-78 years). Histological examination revealed a predominance of differentiated adenocarcinoma (89%). Then, 2 individuals had multiple BTCs. All available 7 BTC lesions showed high-frequency of microsatellite instability (MSI-H). MLH1 carriers showed a 7.2% cumulative risk of BTC development at an age of 70 years. Five of the six individuals died of BTC. ConclusionsMSI analysis could facilitate LS identification in individuals with BTC. Surveillance for BTC should be considered for MLH1 carriers in Japan.</abstract><pub>AME Publishing Company</pub><doi>10.21037/jgo-22-165</doi><tpages>7</tpages><oa>free_for_read</oa></addata></record> |
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| subjects | Original |
| title | Clinical features of biliary tract cancer in Japanese individuals with Lynch syndrome |
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