Literature DB >> 19259385

Mouse models of Huntington disease: variations on a theme.

Dagmar E Ehrnhoefer1, Stefanie L Butland, Mahmoud A Pouladi, Michael R Hayden.   

Abstract

An accepted prerequisite for clinical trials of a compound in humans is the successful alleviation of the disease in animal models. For some diseases, however, successful translation of drug effects from mouse models to the bedside has been limited. One question is whether the current models accurately reproduce the human disease. Here, we examine the mouse models that are available for therapeutic testing in Huntington disease (HD), a late-onset neurodegenerative disorder for which there is no effective treatment. The current mouse models show different degrees of similarity to the human condition. Significant phenotypic differences are seen in mouse models that express either truncated or full-length human, or full-length mouse, mutant huntingtin (mHTT). These differences in phenotypic expression may be attributable to the influences of protein context, mouse strain and a difference in regulatory sequences between the mouse Htt and human HTT genes.

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Year:  2009        PMID: 19259385      PMCID: PMC2650190          DOI: 10.1242/dmm.002451

Source DB:  PubMed          Journal:  Dis Model Mech        ISSN: 1754-8403            Impact factor:   5.758


  75 in total

1.  Genetic modification of the phenotypes produced by amyloid precursor protein overexpression in transgenic mice.

Authors:  G A Carlson; D R Borchelt; A Dake; S Turner; V Danielson; J D Coffin; C Eckman; J Meiners; S P Nilsen; S G Younkin; K K Hsiao
Journal:  Hum Mol Genet       Date:  1997-10       Impact factor: 6.150

2.  Aggregation of huntingtin in neuronal intranuclear inclusions and dystrophic neurites in brain.

Authors:  M DiFiglia; E Sapp; K O Chase; S W Davies; G P Bates; J P Vonsattel; N Aronin
Journal:  Science       Date:  1997-09-26       Impact factor: 47.728

3.  The identification of a functional nuclear localization signal in the Huntington disease protein.

Authors:  D A Bessert; K L Gutridge; J C Dunbar; L R Carlock
Journal:  Brain Res Mol Brain Res       Date:  1995-10

4.  Human huntingtin derived from YAC transgenes compensates for loss of murine huntingtin by rescue of the embryonic lethal phenotype.

Authors:  J G Hodgson; D J Smith; K McCutcheon; H B Koide; K Nishiyama; M B Dinulos; M E Stevens; N Bissada; J Nasir; I Kanazawa; C M Disteche; E M Rubin; M R Hayden
Journal:  Hum Mol Genet       Date:  1996-12       Impact factor: 6.150

5.  Exon 1 of the HD gene with an expanded CAG repeat is sufficient to cause a progressive neurological phenotype in transgenic mice.

Authors:  L Mangiarini; K Sathasivam; M Seller; B Cozens; A Harper; C Hetherington; M Lawton; Y Trottier; H Lehrach; S W Davies; G P Bates
Journal:  Cell       Date:  1996-11-01       Impact factor: 41.582

Review 6.  Silencing is golden: negative regulation in the control of neuronal gene transcription.

Authors:  C J Schoenherr; D J Anderson
Journal:  Curr Opin Neurobiol       Date:  1995-10       Impact factor: 6.627

7.  Heterogeneous topographic and cellular distribution of huntingtin expression in the normal human neostriatum.

Authors:  R J Ferrante; C A Gutekunst; F Persichetti; S M McNeil; N W Kowall; J F Gusella; M E MacDonald; M F Beal; S M Hersch
Journal:  J Neurosci       Date:  1997-05-01       Impact factor: 6.167

8.  Expression of normal and mutant huntingtin in the developing brain.

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Journal:  J Neurosci       Date:  1996-09-01       Impact factor: 6.167

9.  Motor neuron degeneration in mice that express a human Cu,Zn superoxide dismutase mutation.

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Journal:  Science       Date:  1994-06-17       Impact factor: 47.728

10.  Intranuclear inclusions and neuritic aggregates in transgenic mice expressing a mutant N-terminal fragment of huntingtin.

Authors:  G Schilling; M W Becher; A H Sharp; H A Jinnah; K Duan; J A Kotzuk; H H Slunt; T Ratovitski; J K Cooper; N A Jenkins; N G Copeland; D L Price; C A Ross; D R Borchelt
Journal:  Hum Mol Genet       Date:  1999-03       Impact factor: 6.150

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  41 in total

1.  HACE1 is essential for astrocyte mitochondrial function and influences Huntington disease phenotypes in vivo.

Authors:  Dagmar E Ehrnhoefer; Amber L Southwell; Meenalochani Sivasubramanian; Xiaofan Qiu; Erika B Villanueva; Yuanyun Xie; Sabine Waltl; Lisa Anderson; Anita Fazeli; Lorenzo Casal; Boguslaw Felczak; Michelle Tsang; Michael R Hayden
Journal:  Hum Mol Genet       Date:  2018-01-15       Impact factor: 6.150

Review 2.  Advances in reprogramming-based study of neurologic disorders.

Authors:  Anjana Nityanandam; Kristin K Baldwin
Journal:  Stem Cells Dev       Date:  2015-04-06       Impact factor: 3.272

Review 3.  Choosing an animal model for the study of Huntington's disease.

Authors:  Mahmoud A Pouladi; A Jennifer Morton; Michael R Hayden
Journal:  Nat Rev Neurosci       Date:  2013-10       Impact factor: 34.870

Review 4.  Corticostriatal network dysfunction in Huntington's disease: Deficits in neural processing, glutamate transport, and ascorbate release.

Authors:  George V Rebec
Journal:  CNS Neurosci Ther       Date:  2018-02-21       Impact factor: 5.243

5.  Early or late-stage anti-N-terminal Huntingtin intrabody gene therapy reduces pathological features in B6.HDR6/1 mice.

Authors:  Abigail Snyder-Keller; Julie A McLear; Tyisha Hathorn; Anne Messer
Journal:  J Neuropathol Exp Neurol       Date:  2010-10       Impact factor: 3.685

Review 6.  Genetic LRRK2 models of Parkinson's disease: Dissecting the pathogenic pathway and exploring clinical applications.

Authors:  Zhenyu Yue; M Lenard Lachenmayer
Journal:  Mov Disord       Date:  2011-04-29       Impact factor: 10.338

7.  Detecting soluble polyQ oligomers and investigating their impact on living cells using split-GFP.

Authors:  Patrick Lajoie; Erik Lee Snapp
Journal:  Methods Mol Biol       Date:  2013

8.  Role of context in RNA structure: flanking sequences reconfigure CAG motif folding in huntingtin exon 1 transcripts.

Authors:  Steven Busan; Kevin M Weeks
Journal:  Biochemistry       Date:  2013-11-07       Impact factor: 3.162

9.  Intrabody gene therapy ameliorates motor, cognitive, and neuropathological symptoms in multiple mouse models of Huntington's disease.

Authors:  Amber L Southwell; Jan Ko; Paul H Patterson
Journal:  J Neurosci       Date:  2009-10-28       Impact factor: 6.167

10.  Adult neural progenitor cells from Huntington's disease mouse brain exhibit increased proliferation and migration due to enhanced calcium and ROS signals.

Authors:  Wenjuan Xie; Jiu-Qiang Wang; Qiao-Chu Wang; Yun Wang; Sheng Yao; Tie-Shan Tang
Journal:  Cell Prolif       Date:  2015-08-13       Impact factor: 6.831

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