Loss of Zebrafish lgi1b Leads to Hydrocephalus and Sensitization to Pentylenetetrazol Induced Seizure-Like Behavior

Hdl Handle:
http://hdl.handle.net/10675.2/764
Title:
Loss of Zebrafish lgi1b Leads to Hydrocephalus and Sensitization to Pentylenetetrazol Induced Seizure-Like Behavior
Authors:
Teng, Yong; Xie, Xiayang; Walker, Steven L.; Saxena, Meera T.; Kozlowski, David J.; Mumm, Jeff S.; Cowell, John K. ( 0000-0002-2079-5950 )
Abstract:
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.
Citation:
PLoS One. 2011 Sep 16; 6(9):e24596
Issue Date:
16-Sep-2011
URI:
http://hdl.handle.net/10675.2/764
DOI:
10.1371/journal.pone.0024596
PubMed ID:
22053218
PubMed Central ID:
PMC3203530
Type:
Article
ISSN:
1932-6203
Appears in Collections:
Georgia Cancer Center: Faculty Research and Presentations

Full metadata record

DC FieldValue Language
dc.contributor.authorTeng, Yongen_US
dc.contributor.authorXie, Xiayangen_US
dc.contributor.authorWalker, Steven L.en_US
dc.contributor.authorSaxena, Meera T.en_US
dc.contributor.authorKozlowski, David J.en_US
dc.contributor.authorMumm, Jeff S.en_US
dc.contributor.authorCowell, John K.en_US
dc.date.accessioned2012-10-26T20:30:41Z-
dc.date.available2012-10-26T20:30:41Z-
dc.date.issued2011-09-16en_US
dc.identifier.citationPLoS One. 2011 Sep 16; 6(9):e24596en_US
dc.identifier.issn1932-6203en_US
dc.identifier.pmid22053218en_US
dc.identifier.doi10.1371/journal.pone.0024596en_US
dc.identifier.urihttp://hdl.handle.net/10675.2/764-
dc.description.abstractMutations 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.en_US
dc.rightsTeng et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.en_US
dc.subjectResearch Articleen_US
dc.subjectBiologyen_US
dc.subjectGeneticsen_US
dc.subjectGene Functionen_US
dc.subjectGenetics of Diseaseen_US
dc.subjectModel Organismsen_US
dc.subjectAnimal Modelsen_US
dc.subjectZebrafishen_US
dc.subjectNeuroscienceen_US
dc.subjectDevelopmental Neuroscienceen_US
dc.subjectMedicineen_US
dc.subjectNeurologyen_US
dc.subjectHydrocephalusen_US
dc.titleLoss of Zebrafish lgi1b Leads to Hydrocephalus and Sensitization to Pentylenetetrazol Induced Seizure-Like Behavioren_US
dc.typeArticleen_US
dc.identifier.pmcidPMC3203530en_US
dc.contributor.corporatenameGHSU Cancer Center-
dc.contributor.corporatenameDepartment of Cellular Biology and Anatomy-
dc.contributor.corporatenameVision Discovery Institute-
dc.contributor.corporatenameInstitute of Molecular Medicine and Genetics-

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