Literature DB >> 12925883

Phenotypic heterogeneity in the stargazin allelic series.

Verity A Letts1, Myoung-Goo Kang, Connie L Mahaffey, Barbara Beyer, Heather Tenbrink, Kevin P Campbell, Wayne N Frankel.   

Abstract

The stargazer mutant mouse is characterized by its ataxic gait, head tossing, and absence seizures. The mutation was identified in the gamma 2 subunit gene of the high voltage-dependent calcium channel, Cacng2. Subsequently, two allelic variants of stargazer have arisen, waggler and stargazer 3J. In this study, we have compared these new alleles to the original stargazer allele. All three mutations affect the Cacng2 mRNA levels as they all arise from disruptions within the introns of this gene. Our results show that the mutations cause reduced Cacng2 mRNA and protein levels. Stargazer and waggler mice have the least amount of mRNA and undetectable protein, whereas stargazer 3J appears to be the mildest allele, both in terms of the phenotype and protein expression. Electroencephalographic (EEG) analysis confirmed that stargazer has frequent spike-wave discharges (SWDs); the average duration of each discharge burst is 5 seconds and recurs every minute. The waggler allele causes a greater variation in SWD activity depending on the individual mouse, and the stargazer 3J mouse has no SWDs. The preliminary characterization of this heterogeneous allelic series provides a basis to explore more biochemical and physiological parameters relating to the role of the Cacng2 product in calcium channel activity, AMPA receptor localization, and cerebellar disturbances.

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Year:  2003        PMID: 12925883     DOI: 10.1007/s00335-003-2268-x

Source DB:  PubMed          Journal:  Mamm Genome        ISSN: 0938-8990            Impact factor:   2.957


  20 in total

1.  A family of gamma-like calcium channel subunits.

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Journal:  Nat Rev Neurosci       Date:  2002-05       Impact factor: 34.870

3.  Impaired eye-blink conditioning in waggler, a mutant mouse with cerebellar BDNF deficiency.

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Journal:  Learn Mem       Date:  1998 Sep-Oct       Impact factor: 2.460

4.  Calcium channel gamma subunits provide insights into the evolution of this gene family.

Authors:  P J Chu; H M Robertson; P M Best
Journal:  Gene       Date:  2001-12-12       Impact factor: 3.688

5.  Stargazin regulates synaptic targeting of AMPA receptors by two distinct mechanisms.

Authors:  L Chen; D M Chetkovich; R S Petralia; N T Sweeney; Y Kawasaki; R J Wenthold; D S Bredt; R A Nicoll
Journal:  Nature       Date:  2000 Dec 21-28       Impact factor: 49.962

6.  Biochemical and anatomical evidence for specialized voltage-dependent calcium channel gamma isoform expression in the epileptic and ataxic mouse, stargazer.

Authors:  A H Sharp; J L Black; S J Dubel; S Sundarraj; J P Shen; A M Yunker; T D Copeland; M W McEnery
Journal:  Neuroscience       Date:  2001       Impact factor: 3.590

7.  Aberrant expression of neuropeptide Y in hippocampal mossy fibers in the absence of local cell injury following the onset of spike-wave synchronization.

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8.  Neural network of structures in which GABAB receptors regulate absence seizures in the lethargic (lh/lh) mouse model.

Authors:  D A Hosford; F H Lin; D L Kraemer; Z Cao; Y Wang; J T Wilson
Journal:  J Neurosci       Date:  1995-11       Impact factor: 6.167

9.  Cerebellar brain-derived neurotrophic factor-TrkB defect associated with impairment of eyeblink conditioning in Stargazer mutant mice.

Authors:  X Qiao; L Chen; H Gao; S Bao; F Hefti; R F Thompson; B Knusel
Journal:  J Neurosci       Date:  1998-09-01       Impact factor: 6.167

10.  Cerebellar Golgi, Purkinje, and basket cells have reduced gamma-aminobutyric acid immunoreactivity in stargazer mutant mice.

Authors:  Christine A Richardson; Beulah Leitch
Journal:  J Comp Neurol       Date:  2002-11-04       Impact factor: 3.215

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  9 in total

1.  Stargazer--a mouse to seize!

Authors:  Verity A Letts
Journal:  Epilepsy Curr       Date:  2005 Sep-Oct       Impact factor: 7.500

2.  A targeted mutation in Cacng4 exacerbates spike-wave seizures in stargazer (Cacng2) mice.

Authors:  Verity A Letts; Connie L Mahaffey; Barbara Beyer; Wayne N Frankel
Journal:  Proc Natl Acad Sci U S A       Date:  2005-01-26       Impact factor: 11.205

3.  Evidence for association of bipolar disorder to haplotypes in the 22q12.3 region near the genes stargazin, IFT27 and parvalbumin.

Authors:  Stephanie Nissen; Sherri Liang; Tatyana Shehktman; John R Kelsoe; Tiffany A Greenwood; Caroline M Nievergelt; Rebecca McKinney; Paul D Shilling; Erin N Smith; Nicholas J Schork; Cinnamon S Bloss; John I Nurnberger; Howard J Edenberg; Tatiana Foroud; Daniel L Koller; Elliot S Gershon; Chunyu Liu; Judith A Badner; William A Scheftner; William B Lawson; Evaristus A Nwulia; Maria Hipolito; William Coryell; John Rice; William Byerley; Francis J McMahon; Wade H Berrettini; James B Potash; Peter P Zandi; Pamela B Mahon; Melvin G McInnis; Sebastian Zöllner; Peng Zhang; David W Craig; Szabolics Szelinger; Thomas B Barrett; Thomas G Schulze
Journal:  Am J Med Genet B Neuropsychiatr Genet       Date:  2012-10-04       Impact factor: 3.568

Review 4.  Role of voltage-gated calcium channels in epilepsy.

Authors:  Gerald W Zamponi; Philippe Lory; Edward Perez-Reyes
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Review 5.  The Role of Calcium Channels in Epilepsy.

Authors:  Sanjeev Rajakulendran; Michael G Hanna
Journal:  Cold Spring Harb Perspect Med       Date:  2016-01-04       Impact factor: 6.915

Review 6.  How voltage-gated calcium channels gate forms of homeostatic synaptic plasticity.

Authors:  C Andrew Frank
Journal:  Front Cell Neurosci       Date:  2014-02-14       Impact factor: 5.505

Review 7.  Trafficking of neuronal calcium channels.

Authors:  Norbert Weiss; Gerald W Zamponi
Journal:  Neuronal Signal       Date:  2017-02-20

8.  Home-cage behavior in the Stargazer mutant mouse.

Authors:  Catharina Schirmer; Mark A Abboud; Samuel C Lee; John S Bass; Arindam G Mazumder; Jessica L Kamen; Vaishnav Krishnan
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Review 9.  Comparative genetic analysis: the utility of mouse genetic systems for studying human monogenic disease.

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Journal:  Mamm Genome       Date:  2007-05-21       Impact factor: 2.957

  9 in total

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