Loss of zebrafish lgi1b leads to hydrocephalus and sensitization to pentylenetetrazol induced seizure-like behavior.
Mutations in the LGI1 gene predispose to a hereditary epilepsy syndrome and is the first gene associated with this disease which does not encode an ion channel protein. In zebrafish, there are two paralogs of the LGI1 gene, lgi1a and lgi1b. Knockdown of lgi1a results in a seizure-like hyperactivity...
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| Format: | Article |
| Language: | English |
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Public Library of Science (PLoS)
2011-01-01
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| Series: | PLoS ONE |
| Online Access: | https://journals.plos.org/plosone/article/file?id=10.1371/journal.pone.0024596&type=printable |
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| author | Yong Teng Xiayang Xie Steven Walker Meera Saxena David J Kozlowski Jeff S Mumm John K Cowell |
| author_facet | Yong Teng Xiayang Xie Steven Walker Meera Saxena David J Kozlowski Jeff S Mumm John K Cowell |
| author_sort | Yong Teng |
| collection | DOAJ |
| description | Mutations in the LGI1 gene predispose to a hereditary epilepsy syndrome and is the first gene associated with this disease which does not encode an ion channel protein. In zebrafish, there are two paralogs of the LGI1 gene, lgi1a and lgi1b. Knockdown of lgi1a results in a seizure-like hyperactivity phenotype with associated developmental abnormalities characterized by cellular loss in the eyes and brain. We have now generated knockdown morphants for the lgi1b gene which also show developmental abnormalities but do not show a seizure-like behavior. Instead, the most striking phenotype involves significant enlargement of the ventricles (hydrocephalus). As shown for the lgi1a morphants, however, lgi1b morphants are also sensitized to PTZ-induced hyperactivity. The different phenotypes between the two lgi1 morphants support a subfunctionalization model for the two paralogs. |
| format | Article |
| id | doaj-art-14d97a1d2c2b4b6695cc6ee02723502f |
| institution | DOAJ |
| issn | 1932-6203 |
| language | English |
| publishDate | 2011-01-01 |
| publisher | Public Library of Science (PLoS) |
| record_format | Article |
| series | PLoS ONE |
| spelling | doaj-art-14d97a1d2c2b4b6695cc6ee02723502f2025-08-20T02:43:32ZengPublic Library of Science (PLoS)PLoS ONE1932-62032011-01-0169e2459610.1371/journal.pone.0024596Loss of zebrafish lgi1b leads to hydrocephalus and sensitization to pentylenetetrazol induced seizure-like behavior.Yong TengXiayang XieSteven WalkerMeera SaxenaDavid J KozlowskiJeff S MummJohn K CowellMutations in the LGI1 gene predispose to a hereditary epilepsy syndrome and is the first gene associated with this disease which does not encode an ion channel protein. In zebrafish, there are two paralogs of the LGI1 gene, lgi1a and lgi1b. Knockdown of lgi1a results in a seizure-like hyperactivity phenotype with associated developmental abnormalities characterized by cellular loss in the eyes and brain. We have now generated knockdown morphants for the lgi1b gene which also show developmental abnormalities but do not show a seizure-like behavior. Instead, the most striking phenotype involves significant enlargement of the ventricles (hydrocephalus). As shown for the lgi1a morphants, however, lgi1b morphants are also sensitized to PTZ-induced hyperactivity. The different phenotypes between the two lgi1 morphants support a subfunctionalization model for the two paralogs.https://journals.plos.org/plosone/article/file?id=10.1371/journal.pone.0024596&type=printable |
| spellingShingle | Yong Teng Xiayang Xie Steven Walker Meera Saxena David J Kozlowski Jeff S Mumm John K Cowell Loss of zebrafish lgi1b leads to hydrocephalus and sensitization to pentylenetetrazol induced seizure-like behavior. PLoS ONE |
| title | Loss of zebrafish lgi1b leads to hydrocephalus and sensitization to pentylenetetrazol induced seizure-like behavior. |
| title_full | Loss of zebrafish lgi1b leads to hydrocephalus and sensitization to pentylenetetrazol induced seizure-like behavior. |
| title_fullStr | Loss of zebrafish lgi1b leads to hydrocephalus and sensitization to pentylenetetrazol induced seizure-like behavior. |
| title_full_unstemmed | Loss of zebrafish lgi1b leads to hydrocephalus and sensitization to pentylenetetrazol induced seizure-like behavior. |
| title_short | Loss of zebrafish lgi1b leads to hydrocephalus and sensitization to pentylenetetrazol induced seizure-like behavior. |
| title_sort | loss of zebrafish lgi1b leads to hydrocephalus and sensitization to pentylenetetrazol induced seizure like behavior |
| url | https://journals.plos.org/plosone/article/file?id=10.1371/journal.pone.0024596&type=printable |
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