Loading…

The circling behavior of the deafblind LEW-ci2 rat is linked to a segment of RNO10 containing Myo15 and Kcnj12

The LEW/Ztm-ci2 rat is an autosomal recessive mutant that displays circling behavior, deafness, progressive retinopathy, locomotor hyperactivity, ataxia, and opisthotonus. We performed a genome-wide scan of a (LEW/Ztm-ci2 x BN/Ztm) F1 x LEW/Ztm-ci2 backcross population with anonymous microsatellite...

Full description

Saved in:
Bibliographic Details
Published in:Mammalian genome 2003-09, Vol.14 (9), p.620-627
Main Authors: Chwalisz, Wojciech T, Koelsch, Bernd U, Kindler-Röhrborn, Andrea, Hedrich, Hans J, Wedekind, Dirk
Format: Article
Language:English
Subjects:
Citations: Items that cite this one
Online Access:Get full text
Tags: Add Tag
No Tags, Be the first to tag this record!
cited_by cdi_FETCH-LOGICAL-c355t-dd241f8b78be0bb01e83d1524c84be326ecb1485b32760c62aaef908ede5f9663
cites
container_end_page 627
container_issue 9
container_start_page 620
container_title Mammalian genome
container_volume 14
creator Chwalisz, Wojciech T
Koelsch, Bernd U
Kindler-Röhrborn, Andrea
Hedrich, Hans J
Wedekind, Dirk
description The LEW/Ztm-ci2 rat is an autosomal recessive mutant that displays circling behavior, deafness, progressive retinopathy, locomotor hyperactivity, ataxia, and opisthotonus. We performed a genome-wide scan of a (LEW/Ztm-ci2 x BN/Ztm) F1 x LEW/Ztm-ci2 backcross population with anonymous microsatellite markers to analyze the genetics of this mutant rat. This linkage analysis demonstrated a very strong association of RNO10 SSLP markers to the phenotype with a core region in the central part of the chromosome. The knowledge of genes mapping to this part of the rat genome and their linkage to SSLP markers is still poor. We developed SSLP markers closely linked to genes, which might be responsible for the mutant phenotype by using the growing amount of rat-specific DNA sequences available at World Wide Web databases. Application of this method facilitated the search for candidate genes for the phenotype of the LEW-ci2 rat. We were able to map Myo15 and its neighboring genes, Znf179 and Aldh3a1, to the region of interest and Myo1c to a more distal location on RNO10. Further rat BAC clones were used to create a physical map of the region of interest. This map revealed the position of further genes. Among those is Kcnj12. Owing to their localization on RNO10 and their involvement in a similar pathology in human and mouse, Myo15 and Kcnj12 can be regarded as candidate genes for the deafblind phenotype of the LEW-ci2 rat.
doi_str_mv 10.1007/s00335-003-3009-x
format article
fullrecord <record><control><sourceid>proquest_cross</sourceid><recordid>TN_cdi_proquest_miscellaneous_71386046</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><sourcerecordid>2190439521</sourcerecordid><originalsourceid>FETCH-LOGICAL-c355t-dd241f8b78be0bb01e83d1524c84be326ecb1485b32760c62aaef908ede5f9663</originalsourceid><addsrcrecordid>eNqFkdFrFDEQxoMo9lr9A3yR4INv0ZlkN8k-Smm1eLZQKj6GJDvb7nm3qcleaf97c9yB0BdfZmDm933M8DH2DuETApjPBUCpVtQqFEAnHl-wBTZKCjTGvGQL6JQVtuvgiB2XsgJAo9G8ZkfYaNkhygWbbu6IxzHH9Tjd8kB3_mFMmaeBz3XRkx9C3fR8efZLxFHy7Gc-Fl5nv6nnc-KeF7rd0DTvNNeXVwg8pmn247Qz_PGUsOW-GnyP0wrlG_Zq8OtCbw_9hP08P7s5_SaWV18vTr8sRVRtO4u-lw0ONhgbCEIAJKt6bGUTbRNISU0xYGPboKTRELX0noYOLPXUDp3W6oR93Pve5_RnS2V2m7FEWq_9RGlbnEFlNTT_B9Fa21rVVvDDM3CVtnmqTzjTQb1Calsh3EMxp1IyDe4-jxufnxyC20Xm9pG5Wt0uMvdYNe8Pxtuwof6f4pCR-gu9s48v</addsrcrecordid><sourcetype>Aggregation Database</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>790760268</pqid></control><display><type>article</type><title>The circling behavior of the deafblind LEW-ci2 rat is linked to a segment of RNO10 containing Myo15 and Kcnj12</title><source>Springer Nature</source><creator>Chwalisz, Wojciech T ; Koelsch, Bernd U ; Kindler-Röhrborn, Andrea ; Hedrich, Hans J ; Wedekind, Dirk</creator><creatorcontrib>Chwalisz, Wojciech T ; Koelsch, Bernd U ; Kindler-Röhrborn, Andrea ; Hedrich, Hans J ; Wedekind, Dirk</creatorcontrib><description>The LEW/Ztm-ci2 rat is an autosomal recessive mutant that displays circling behavior, deafness, progressive retinopathy, locomotor hyperactivity, ataxia, and opisthotonus. We performed a genome-wide scan of a (LEW/Ztm-ci2 x BN/Ztm) F1 x LEW/Ztm-ci2 backcross population with anonymous microsatellite markers to analyze the genetics of this mutant rat. This linkage analysis demonstrated a very strong association of RNO10 SSLP markers to the phenotype with a core region in the central part of the chromosome. The knowledge of genes mapping to this part of the rat genome and their linkage to SSLP markers is still poor. We developed SSLP markers closely linked to genes, which might be responsible for the mutant phenotype by using the growing amount of rat-specific DNA sequences available at World Wide Web databases. Application of this method facilitated the search for candidate genes for the phenotype of the LEW-ci2 rat. We were able to map Myo15 and its neighboring genes, Znf179 and Aldh3a1, to the region of interest and Myo1c to a more distal location on RNO10. Further rat BAC clones were used to create a physical map of the region of interest. This map revealed the position of further genes. Among those is Kcnj12. Owing to their localization on RNO10 and their involvement in a similar pathology in human and mouse, Myo15 and Kcnj12 can be regarded as candidate genes for the deafblind phenotype of the LEW-ci2 rat.</description><identifier>ISSN: 0938-8990</identifier><identifier>EISSN: 1432-1777</identifier><identifier>DOI: 10.1007/s00335-003-3009-x</identifier><identifier>PMID: 14629112</identifier><language>eng</language><publisher>United States: Springer Nature B.V</publisher><subject>Animals ; Base Sequence ; Behavior, Animal ; Blindness - genetics ; Chromosome Mapping ; Deafness - genetics ; DNA, Complementary - genetics ; Genetic Markers ; Humans ; Mice ; Myosins - genetics ; Phenotype ; Potassium Channels - genetics ; Rats ; Rats, Inbred BN ; Rats, Inbred Lew ; Rats, Mutant Strains</subject><ispartof>Mammalian genome, 2003-09, Vol.14 (9), p.620-627</ispartof><rights>Springer-Verlag New York Inc. 2003</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c355t-dd241f8b78be0bb01e83d1524c84be326ecb1485b32760c62aaef908ede5f9663</citedby></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,780,784,27924,27925</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/14629112$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Chwalisz, Wojciech T</creatorcontrib><creatorcontrib>Koelsch, Bernd U</creatorcontrib><creatorcontrib>Kindler-Röhrborn, Andrea</creatorcontrib><creatorcontrib>Hedrich, Hans J</creatorcontrib><creatorcontrib>Wedekind, Dirk</creatorcontrib><title>The circling behavior of the deafblind LEW-ci2 rat is linked to a segment of RNO10 containing Myo15 and Kcnj12</title><title>Mammalian genome</title><addtitle>Mamm Genome</addtitle><description>The LEW/Ztm-ci2 rat is an autosomal recessive mutant that displays circling behavior, deafness, progressive retinopathy, locomotor hyperactivity, ataxia, and opisthotonus. We performed a genome-wide scan of a (LEW/Ztm-ci2 x BN/Ztm) F1 x LEW/Ztm-ci2 backcross population with anonymous microsatellite markers to analyze the genetics of this mutant rat. This linkage analysis demonstrated a very strong association of RNO10 SSLP markers to the phenotype with a core region in the central part of the chromosome. The knowledge of genes mapping to this part of the rat genome and their linkage to SSLP markers is still poor. We developed SSLP markers closely linked to genes, which might be responsible for the mutant phenotype by using the growing amount of rat-specific DNA sequences available at World Wide Web databases. Application of this method facilitated the search for candidate genes for the phenotype of the LEW-ci2 rat. We were able to map Myo15 and its neighboring genes, Znf179 and Aldh3a1, to the region of interest and Myo1c to a more distal location on RNO10. Further rat BAC clones were used to create a physical map of the region of interest. This map revealed the position of further genes. Among those is Kcnj12. Owing to their localization on RNO10 and their involvement in a similar pathology in human and mouse, Myo15 and Kcnj12 can be regarded as candidate genes for the deafblind phenotype of the LEW-ci2 rat.</description><subject>Animals</subject><subject>Base Sequence</subject><subject>Behavior, Animal</subject><subject>Blindness - genetics</subject><subject>Chromosome Mapping</subject><subject>Deafness - genetics</subject><subject>DNA, Complementary - genetics</subject><subject>Genetic Markers</subject><subject>Humans</subject><subject>Mice</subject><subject>Myosins - genetics</subject><subject>Phenotype</subject><subject>Potassium Channels - genetics</subject><subject>Rats</subject><subject>Rats, Inbred BN</subject><subject>Rats, Inbred Lew</subject><subject>Rats, Mutant Strains</subject><issn>0938-8990</issn><issn>1432-1777</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2003</creationdate><recordtype>article</recordtype><recordid>eNqFkdFrFDEQxoMo9lr9A3yR4INv0ZlkN8k-Smm1eLZQKj6GJDvb7nm3qcleaf97c9yB0BdfZmDm933M8DH2DuETApjPBUCpVtQqFEAnHl-wBTZKCjTGvGQL6JQVtuvgiB2XsgJAo9G8ZkfYaNkhygWbbu6IxzHH9Tjd8kB3_mFMmaeBz3XRkx9C3fR8efZLxFHy7Gc-Fl5nv6nnc-KeF7rd0DTvNNeXVwg8pmn247Qz_PGUsOW-GnyP0wrlG_Zq8OtCbw_9hP08P7s5_SaWV18vTr8sRVRtO4u-lw0ONhgbCEIAJKt6bGUTbRNISU0xYGPboKTRELX0noYOLPXUDp3W6oR93Pve5_RnS2V2m7FEWq_9RGlbnEFlNTT_B9Fa21rVVvDDM3CVtnmqTzjTQb1Calsh3EMxp1IyDe4-jxufnxyC20Xm9pG5Wt0uMvdYNe8Pxtuwof6f4pCR-gu9s48v</recordid><startdate>200309</startdate><enddate>200309</enddate><creator>Chwalisz, Wojciech T</creator><creator>Koelsch, Bernd U</creator><creator>Kindler-Röhrborn, Andrea</creator><creator>Hedrich, Hans J</creator><creator>Wedekind, Dirk</creator><general>Springer Nature B.V</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>3V.</scope><scope>7TK</scope><scope>7X7</scope><scope>7XB</scope><scope>88A</scope><scope>88E</scope><scope>8AO</scope><scope>8FD</scope><scope>8FE</scope><scope>8FH</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>AZQEC</scope><scope>BBNVY</scope><scope>BENPR</scope><scope>BHPHI</scope><scope>CCPQU</scope><scope>DWQXO</scope><scope>FR3</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>GNUQQ</scope><scope>HCIFZ</scope><scope>K9.</scope><scope>LK8</scope><scope>M0S</scope><scope>M1P</scope><scope>M7P</scope><scope>P64</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>RC3</scope><scope>7X8</scope></search><sort><creationdate>200309</creationdate><title>The circling behavior of the deafblind LEW-ci2 rat is linked to a segment of RNO10 containing Myo15 and Kcnj12</title><author>Chwalisz, Wojciech T ; Koelsch, Bernd U ; Kindler-Röhrborn, Andrea ; Hedrich, Hans J ; Wedekind, Dirk</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c355t-dd241f8b78be0bb01e83d1524c84be326ecb1485b32760c62aaef908ede5f9663</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2003</creationdate><topic>Animals</topic><topic>Base Sequence</topic><topic>Behavior, Animal</topic><topic>Blindness - genetics</topic><topic>Chromosome Mapping</topic><topic>Deafness - genetics</topic><topic>DNA, Complementary - genetics</topic><topic>Genetic Markers</topic><topic>Humans</topic><topic>Mice</topic><topic>Myosins - genetics</topic><topic>Phenotype</topic><topic>Potassium Channels - genetics</topic><topic>Rats</topic><topic>Rats, Inbred BN</topic><topic>Rats, Inbred Lew</topic><topic>Rats, Mutant Strains</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Chwalisz, Wojciech T</creatorcontrib><creatorcontrib>Koelsch, Bernd U</creatorcontrib><creatorcontrib>Kindler-Röhrborn, Andrea</creatorcontrib><creatorcontrib>Hedrich, Hans J</creatorcontrib><creatorcontrib>Wedekind, Dirk</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Central (Corporate)</collection><collection>Neurosciences Abstracts</collection><collection>Health &amp; Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Biology Database (Alumni Edition)</collection><collection>Medical Database (Alumni Edition)</collection><collection>ProQuest Pharma Collection</collection><collection>Technology Research Database</collection><collection>ProQuest SciTech Collection</collection><collection>ProQuest Natural Science Collection</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>ProQuest Central (Alumni)</collection><collection>ProQuest Central</collection><collection>ProQuest Central Essentials</collection><collection>Biological Science Collection</collection><collection>AUTh Library subscriptions: ProQuest Central</collection><collection>Natural Science Collection</collection><collection>ProQuest One Community College</collection><collection>ProQuest Central Korea</collection><collection>Engineering Research Database</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Central Student</collection><collection>SciTech Premium Collection</collection><collection>ProQuest Health &amp; Medical Complete (Alumni)</collection><collection>ProQuest Biological Science Collection</collection><collection>Health &amp; Medical Collection (Alumni Edition)</collection><collection>Medical Database</collection><collection>ProQuest Biological Science Journals</collection><collection>Biotechnology and BioEngineering Abstracts</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest Central China</collection><collection>Genetics Abstracts</collection><collection>MEDLINE - Academic</collection><jtitle>Mammalian genome</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Chwalisz, Wojciech T</au><au>Koelsch, Bernd U</au><au>Kindler-Röhrborn, Andrea</au><au>Hedrich, Hans J</au><au>Wedekind, Dirk</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>The circling behavior of the deafblind LEW-ci2 rat is linked to a segment of RNO10 containing Myo15 and Kcnj12</atitle><jtitle>Mammalian genome</jtitle><addtitle>Mamm Genome</addtitle><date>2003-09</date><risdate>2003</risdate><volume>14</volume><issue>9</issue><spage>620</spage><epage>627</epage><pages>620-627</pages><issn>0938-8990</issn><eissn>1432-1777</eissn><abstract>The LEW/Ztm-ci2 rat is an autosomal recessive mutant that displays circling behavior, deafness, progressive retinopathy, locomotor hyperactivity, ataxia, and opisthotonus. We performed a genome-wide scan of a (LEW/Ztm-ci2 x BN/Ztm) F1 x LEW/Ztm-ci2 backcross population with anonymous microsatellite markers to analyze the genetics of this mutant rat. This linkage analysis demonstrated a very strong association of RNO10 SSLP markers to the phenotype with a core region in the central part of the chromosome. The knowledge of genes mapping to this part of the rat genome and their linkage to SSLP markers is still poor. We developed SSLP markers closely linked to genes, which might be responsible for the mutant phenotype by using the growing amount of rat-specific DNA sequences available at World Wide Web databases. Application of this method facilitated the search for candidate genes for the phenotype of the LEW-ci2 rat. We were able to map Myo15 and its neighboring genes, Znf179 and Aldh3a1, to the region of interest and Myo1c to a more distal location on RNO10. Further rat BAC clones were used to create a physical map of the region of interest. This map revealed the position of further genes. Among those is Kcnj12. Owing to their localization on RNO10 and their involvement in a similar pathology in human and mouse, Myo15 and Kcnj12 can be regarded as candidate genes for the deafblind phenotype of the LEW-ci2 rat.</abstract><cop>United States</cop><pub>Springer Nature B.V</pub><pmid>14629112</pmid><doi>10.1007/s00335-003-3009-x</doi><tpages>8</tpages></addata></record>
fulltext fulltext
identifier ISSN: 0938-8990
ispartof Mammalian genome, 2003-09, Vol.14 (9), p.620-627
issn 0938-8990
1432-1777
language eng
recordid cdi_proquest_miscellaneous_71386046
source Springer Nature
subjects Animals
Base Sequence
Behavior, Animal
Blindness - genetics
Chromosome Mapping
Deafness - genetics
DNA, Complementary - genetics
Genetic Markers
Humans
Mice
Myosins - genetics
Phenotype
Potassium Channels - genetics
Rats
Rats, Inbred BN
Rats, Inbred Lew
Rats, Mutant Strains
title The circling behavior of the deafblind LEW-ci2 rat is linked to a segment of RNO10 containing Myo15 and Kcnj12
url http://sfxeu10.hosted.exlibrisgroup.com/loughborough?ctx_ver=Z39.88-2004&ctx_enc=info:ofi/enc:UTF-8&ctx_tim=2024-12-25T22%3A38%3A52IST&url_ver=Z39.88-2004&url_ctx_fmt=infofi/fmt:kev:mtx:ctx&rfr_id=info:sid/primo.exlibrisgroup.com:primo3-Article-proquest_cross&rft_val_fmt=info:ofi/fmt:kev:mtx:journal&rft.genre=article&rft.atitle=The%20circling%20behavior%20of%20the%20deafblind%20LEW-ci2%20rat%20is%20linked%20to%20a%20segment%20of%20RNO10%20containing%20Myo15%20and%20Kcnj12&rft.jtitle=Mammalian%20genome&rft.au=Chwalisz,%20Wojciech%20T&rft.date=2003-09&rft.volume=14&rft.issue=9&rft.spage=620&rft.epage=627&rft.pages=620-627&rft.issn=0938-8990&rft.eissn=1432-1777&rft_id=info:doi/10.1007/s00335-003-3009-x&rft_dat=%3Cproquest_cross%3E2190439521%3C/proquest_cross%3E%3Cgrp_id%3Ecdi_FETCH-LOGICAL-c355t-dd241f8b78be0bb01e83d1524c84be326ecb1485b32760c62aaef908ede5f9663%3C/grp_id%3E%3Coa%3E%3C/oa%3E%3Curl%3E%3C/url%3E&rft_id=info:oai/&rft_pqid=790760268&rft_id=info:pmid/14629112&rfr_iscdi=true