Literature DB >> 9382472

Huntingtin localization in brains of normal and Huntington's disease patients.

E Sapp1, C Schwarz, K Chase, P G Bhide, A B Young, J Penney, J P Vonsattel, N Aronin, M DiFiglia.   

Abstract

The immunohistochemical localization of huntingtin was examined in the Huntington's disease (HD) brain with an antibody that recognizes the wild-type and mutant proteins. Neuronal staining was reduced in areas of the HD striatum depleted of medium-sized neurons; large striatal neurons, which are spared in HD, retained normal levels of huntingtin expression. Neuronal labeling was markedly reduced in both segments of the globus pallidus including in brains with minimal loss of pallidal neurons. In some HD cortical and striatal neurons with normal looking morphology, huntingtin was associated with punctate cytoplasmic granules that at the ultrastructural level resembled the multivesicular body, an organelle involved in retrograde transport and protein degradation. Some immunoreactive processes showed blebbing and segmentation similar to that induced experimentally by hypoxic-ischemic or excitotoxic injury. Huntingtin staining was more concentrated in the perinuclear cytoplasm and reduced or absent in processes of atrophic cortical neurons. Nuclear staining was also evident. Fibers in the subcortical white matter of HD patients had significantly increased huntingtin immunoreactivity compared with those of controls. Results suggest that there may be changes in the neuronal expression and transport of wild-type and/or mutant huntingtin at early and late stages of neuronal degeneration in affected areas of the HD brain.

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Year:  1997        PMID: 9382472     DOI: 10.1002/ana.410420411

Source DB:  PubMed          Journal:  Ann Neurol        ISSN: 0364-5134            Impact factor:   10.422


  101 in total

Review 1.  Polyglutamine pathogenesis.

Authors:  C A Ross; J D Wood; G Schilling; M F Peters; F C Nucifora; J K Cooper; A H Sharp; R L Margolis; D R Borchelt
Journal:  Philos Trans R Soc Lond B Biol Sci       Date:  1999-06-29       Impact factor: 6.237

2.  From neuronal inclusions to neurodegeneration: neuropathological investigation of a transgenic mouse model of Huntington's disease.

Authors:  S W Davies; M Turmaine; B A Cozens; A S Raza; A Mahal; L Mangiarini; G P Bates
Journal:  Philos Trans R Soc Lond B Biol Sci       Date:  1999-06-29       Impact factor: 6.237

Review 3.  Evidence for both the nucleus and cytoplasm as subcellular sites of pathogenesis in Huntington's disease in cell culture and in transgenic mice expressing mutant huntingtin.

Authors:  A S Hackam; J G Hodgson; R Singaraja; T Zhang; L Gan; C A Gutekunst; S M Hersch; M R Hayden
Journal:  Philos Trans R Soc Lond B Biol Sci       Date:  1999-06-29       Impact factor: 6.237

Review 4.  Altered neurotransmitter receptor expression in transgenic mouse models of Huntington's disease.

Authors:  J H Cha; A S Frey; S A Alsdorf; J A Kerner; C M Kosinski; L Mangiarini; J B Penney; S W Davies; G P Bates; A B Young
Journal:  Philos Trans R Soc Lond B Biol Sci       Date:  1999-06-29       Impact factor: 6.237

Review 5.  The localization and interactions of huntingtin.

Authors:  A L Jones
Journal:  Philos Trans R Soc Lond B Biol Sci       Date:  1999-06-29       Impact factor: 6.237

Review 6.  Modifiers and mechanisms of multi-system polyglutamine neurodegenerative disorders: lessons from fly models.

Authors:  Moushami Mallik; Subhash C Lakhotia
Journal:  J Genet       Date:  2010-12       Impact factor: 1.166

Review 7.  Huntingtin in health and disease.

Authors:  Anne B Young
Journal:  J Clin Invest       Date:  2003-02       Impact factor: 14.808

Review 8.  Engineered antibody therapies to counteract mutant huntingtin and related toxic intracellular proteins.

Authors:  David C Butler; Julie A McLear; Anne Messer
Journal:  Prog Neurobiol       Date:  2011-11-18       Impact factor: 11.685

9.  Early autophagic response in a novel knock-in model of Huntington disease.

Authors:  Mary Y Heng; Duy K Duong; Roger L Albin; Sara J Tallaksen-Greene; Jesse M Hunter; Mathieu J Lesort; Alex Osmand; Henry L Paulson; Peter J Detloff
Journal:  Hum Mol Genet       Date:  2010-07-08       Impact factor: 6.150

Review 10.  Multivesicular bodies in neurons: distribution, protein content, and trafficking functions.

Authors:  Christopher S Von Bartheld; Amy L Altick
Journal:  Prog Neurobiol       Date:  2011-01-07       Impact factor: 11.685

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