Loss of zebrafish lgi1b leads to hydrocephalus and sensitization to pentylenetetrazol induced seizure-like behavior

Yong Teng, Xiayang Xie, Steven Walker, Meera Saxena, David J Kozlowski, Jeff S. Mumm, John Kenneth Cowell

Research output: Contribution to journalArticle

27 Citations (Scopus)

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.

Original languageEnglish (US)
Article numbere24596
JournalPLoS One
Volume6
Issue number9
DOIs
StatePublished - Sep 16 2011

Fingerprint

hydrocephalus
Pentylenetetrazole
Zebrafish
Hydrocephalus
seizures
Danio rerio
Seizures
Genes
abnormal development
Phenotype
phenotype
genes
epilepsy
ion channels
Ion Channels
Epilepsy
Brain
eyes
mutation
brain

ASJC Scopus subject areas

  • Biochemistry, Genetics and Molecular Biology(all)
  • Agricultural and Biological Sciences(all)

Cite this

Loss of zebrafish lgi1b leads to hydrocephalus and sensitization to pentylenetetrazol induced seizure-like behavior. / Teng, Yong; Xie, Xiayang; Walker, Steven; Saxena, Meera; Kozlowski, David J; Mumm, Jeff S.; Cowell, John Kenneth.

In: PLoS One, Vol. 6, No. 9, e24596, 16.09.2011.

Research output: Contribution to journalArticle

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