Linkage of familial Wilms' tumor predisposition to chromosome 19 and a two-locus model for the etiology of familial tumors
Familial predisposition to Wilms' tumor (WT), a childhood kidney tumor, is inherited as an autosomal dominant trait. For most WT families studied, the 11p13 gene WT1 and genomic regions implicated in tumorigenesis in a subset of tumors can be ruled out as the site of the familial predisposition...
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| Veröffentlicht in: | Cancer research (Chicago, Ill.) Ill.), 1998-04, Vol.58 (7), p.1387-1390 |
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| container_title | Cancer research (Chicago, Ill.) |
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| creator | MCDONALD, J. M DOUGLASS, E. C FISHER, R GEISER, C. F KRILL, C. E STRONG, L. C VIRSHUP, D HUFF, V |
| description | Familial predisposition to Wilms' tumor (WT), a childhood kidney tumor, is inherited as an autosomal dominant trait. For most WT families studied, the 11p13 gene WT1 and genomic regions implicated in tumorigenesis in a subset of tumors can be ruled out as the site of the familial predisposition gene. Following a genome-wide genetic linkage scan, we have obtained strong evidence (log of the odds ratio = 4.0) in five families for an inherited WT predisposition gene (FWT2) at 19q13.3-q13.4. In addition, we observed loss of heterozygosity at 19q in tumors from individuals from two families in which 19q can be ruled out as the site of the inherited predisposing mutation. From these data, we hypothesize that alterations at two distinct loci are critical rate-limiting steps in the etiology of familial WTs. |
| format | Article |
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M ; DOUGLASS, E. C ; FISHER, R ; GEISER, C. F ; KRILL, C. E ; STRONG, L. C ; VIRSHUP, D ; HUFF, V</creator><creatorcontrib>MCDONALD, J. M ; DOUGLASS, E. C ; FISHER, R ; GEISER, C. F ; KRILL, C. E ; STRONG, L. C ; VIRSHUP, D ; HUFF, V</creatorcontrib><description>Familial predisposition to Wilms' tumor (WT), a childhood kidney tumor, is inherited as an autosomal dominant trait. For most WT families studied, the 11p13 gene WT1 and genomic regions implicated in tumorigenesis in a subset of tumors can be ruled out as the site of the familial predisposition gene. Following a genome-wide genetic linkage scan, we have obtained strong evidence (log of the odds ratio = 4.0) in five families for an inherited WT predisposition gene (FWT2) at 19q13.3-q13.4. In addition, we observed loss of heterozygosity at 19q in tumors from individuals from two families in which 19q can be ruled out as the site of the inherited predisposing mutation. From these data, we hypothesize that alterations at two distinct loci are critical rate-limiting steps in the etiology of familial WTs.</description><identifier>ISSN: 0008-5472</identifier><identifier>EISSN: 1538-7445</identifier><identifier>PMID: 9537236</identifier><identifier>CODEN: CNREA8</identifier><language>eng</language><publisher>Philadelphia, PA: American Association for Cancer Research</publisher><subject>Biological and medical sciences ; Child, Preschool ; Chromosomes, Human, Pair 19 ; Disease Susceptibility ; Family Health ; Female ; Genetic Linkage ; Humans ; Kidneys ; Loss of Heterozygosity ; Male ; Medical sciences ; Models, Genetic ; Mutation ; Nephrology. 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F</creatorcontrib><creatorcontrib>KRILL, C. E</creatorcontrib><creatorcontrib>STRONG, L. C</creatorcontrib><creatorcontrib>VIRSHUP, D</creatorcontrib><creatorcontrib>HUFF, V</creatorcontrib><title>Linkage of familial Wilms' tumor predisposition to chromosome 19 and a two-locus model for the etiology of familial tumors</title><title>Cancer research (Chicago, Ill.)</title><addtitle>Cancer Res</addtitle><description>Familial predisposition to Wilms' tumor (WT), a childhood kidney tumor, is inherited as an autosomal dominant trait. For most WT families studied, the 11p13 gene WT1 and genomic regions implicated in tumorigenesis in a subset of tumors can be ruled out as the site of the familial predisposition gene. Following a genome-wide genetic linkage scan, we have obtained strong evidence (log of the odds ratio = 4.0) in five families for an inherited WT predisposition gene (FWT2) at 19q13.3-q13.4. In addition, we observed loss of heterozygosity at 19q in tumors from individuals from two families in which 19q can be ruled out as the site of the inherited predisposing mutation. From these data, we hypothesize that alterations at two distinct loci are critical rate-limiting steps in the etiology of familial WTs.</description><subject>Biological and medical sciences</subject><subject>Child, Preschool</subject><subject>Chromosomes, Human, Pair 19</subject><subject>Disease Susceptibility</subject><subject>Family Health</subject><subject>Female</subject><subject>Genetic Linkage</subject><subject>Humans</subject><subject>Kidneys</subject><subject>Loss of Heterozygosity</subject><subject>Male</subject><subject>Medical sciences</subject><subject>Models, Genetic</subject><subject>Mutation</subject><subject>Nephrology. Urinary tract diseases</subject><subject>Pedigree</subject><subject>Tumors of the urinary system</subject><subject>Wilms Tumor - genetics</subject><issn>0008-5472</issn><issn>1538-7445</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>1998</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNpVkE1LxDAQhoMo67r6E4QcRE-FJE2a9iiLX7DgRfFYps1kN5o0a9Mi-ustWgRPw_A-78MwB2TJVV5mWkp1SJaMsTJTUotjcpLS67QqztSCLCqVa5EXS_K1cd0bbJFGSy0E5x14-uJ8SFd0GEPs6b5H49I-Jje42NEh0nbXxxBTDEh5RaEzFOjwETMf2zHREA16aqfmsEOKU8nH7ec__484nZIjCz7h2TxX5Pn25ml9n20e7x7W15tsJxgbsjaXWlVMlAqlMoDQCKM4tsi5LMAYbppSC14ZkfMK0WrWSGCltSgtFmDzFbn89e77-D5iGurgUoveQ4dxTLWudCk45xN4PoNjE9DU-94F6D_r-VlTfjHnkFrwtoeudekPm26oCsHyb6CmdYQ</recordid><startdate>19980401</startdate><enddate>19980401</enddate><creator>MCDONALD, J. M</creator><creator>DOUGLASS, E. C</creator><creator>FISHER, R</creator><creator>GEISER, C. F</creator><creator>KRILL, C. E</creator><creator>STRONG, L. C</creator><creator>VIRSHUP, D</creator><creator>HUFF, V</creator><general>American Association for Cancer Research</general><scope>IQODW</scope><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>7X8</scope></search><sort><creationdate>19980401</creationdate><title>Linkage of familial Wilms' tumor predisposition to chromosome 19 and a two-locus model for the etiology of familial tumors</title><author>MCDONALD, J. M ; DOUGLASS, E. C ; FISHER, R ; GEISER, C. F ; KRILL, C. E ; STRONG, L. C ; VIRSHUP, D ; HUFF, V</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-h200t-c347590285e45daeab2d51ece1146add1db87219d2319eef70b4a08ffe4fe6af3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>1998</creationdate><topic>Biological and medical sciences</topic><topic>Child, Preschool</topic><topic>Chromosomes, Human, Pair 19</topic><topic>Disease Susceptibility</topic><topic>Family Health</topic><topic>Female</topic><topic>Genetic Linkage</topic><topic>Humans</topic><topic>Kidneys</topic><topic>Loss of Heterozygosity</topic><topic>Male</topic><topic>Medical sciences</topic><topic>Models, Genetic</topic><topic>Mutation</topic><topic>Nephrology. Urinary tract diseases</topic><topic>Pedigree</topic><topic>Tumors of the urinary system</topic><topic>Wilms Tumor - genetics</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>MCDONALD, J. M</creatorcontrib><creatorcontrib>DOUGLASS, E. C</creatorcontrib><creatorcontrib>FISHER, R</creatorcontrib><creatorcontrib>GEISER, C. F</creatorcontrib><creatorcontrib>KRILL, C. E</creatorcontrib><creatorcontrib>STRONG, L. C</creatorcontrib><creatorcontrib>VIRSHUP, D</creatorcontrib><creatorcontrib>HUFF, V</creatorcontrib><collection>Pascal-Francis</collection><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>MEDLINE - Academic</collection><jtitle>Cancer research (Chicago, Ill.)</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>MCDONALD, J. M</au><au>DOUGLASS, E. C</au><au>FISHER, R</au><au>GEISER, C. F</au><au>KRILL, C. E</au><au>STRONG, L. C</au><au>VIRSHUP, D</au><au>HUFF, V</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Linkage of familial Wilms' tumor predisposition to chromosome 19 and a two-locus model for the etiology of familial tumors</atitle><jtitle>Cancer research (Chicago, Ill.)</jtitle><addtitle>Cancer Res</addtitle><date>1998-04-01</date><risdate>1998</risdate><volume>58</volume><issue>7</issue><spage>1387</spage><epage>1390</epage><pages>1387-1390</pages><issn>0008-5472</issn><eissn>1538-7445</eissn><coden>CNREA8</coden><abstract>Familial predisposition to Wilms' tumor (WT), a childhood kidney tumor, is inherited as an autosomal dominant trait. For most WT families studied, the 11p13 gene WT1 and genomic regions implicated in tumorigenesis in a subset of tumors can be ruled out as the site of the familial predisposition gene. Following a genome-wide genetic linkage scan, we have obtained strong evidence (log of the odds ratio = 4.0) in five families for an inherited WT predisposition gene (FWT2) at 19q13.3-q13.4. In addition, we observed loss of heterozygosity at 19q in tumors from individuals from two families in which 19q can be ruled out as the site of the inherited predisposing mutation. From these data, we hypothesize that alterations at two distinct loci are critical rate-limiting steps in the etiology of familial WTs.</abstract><cop>Philadelphia, PA</cop><pub>American Association for Cancer Research</pub><pmid>9537236</pmid><tpages>4</tpages></addata></record> |
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| ispartof | Cancer research (Chicago, Ill.), 1998-04, Vol.58 (7), p.1387-1390 |
| issn | 0008-5472 1538-7445 |
| language | eng |
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| source | MEDLINE; American Association for Cancer Research; EZB-FREE-00999 freely available EZB journals |
| subjects | Biological and medical sciences Child, Preschool Chromosomes, Human, Pair 19 Disease Susceptibility Family Health Female Genetic Linkage Humans Kidneys Loss of Heterozygosity Male Medical sciences Models, Genetic Mutation Nephrology. Urinary tract diseases Pedigree Tumors of the urinary system Wilms Tumor - genetics |
| title | Linkage of familial Wilms' tumor predisposition to chromosome 19 and a two-locus model for the etiology of familial tumors |
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