Literature DB >> 11045680

Alpha-synuclein accumulation in a case of neurodegeneration with brain iron accumulation type 1 (NBIA-1, formerly Hallervorden-Spatz syndrome) with widespread cortical and brainstem-type Lewy bodies.

M Neumann1, S Adler, O Schlüter, E Kremmer, R Benecke, H A Kretzschmar.   

Abstract

We studied a 27-year-old woman who died after a 6-year history of progressive dementia, dystonia, ataxia, apraxia, spasticity, choreoathetosis, visual and auditory hallucinations, and optic atrophy. Magnetic resonance imaging showed decreased intensity in the globus pallidus, substantia nigra, and dentate nuclei in T2-weighted images, supporting the clinical diagnosis of neurodegeneration with brain iron accumulation type 1 (NBIA-1; formerly known as Hallervorden-Spatz syndrome). At autopsy the brain showed mild frontotemporal atrophy and discoloration of the globus pallidus and the substantia nigra pars reticularis. Histologically, features typical of NBIA-1 were found including widespread axonal spheroids and large deposits of iron pigment in the discolored regions. Additionally, excessive numbers of Lewy bodies (LBs) were found throughout all examined brain stem and cortical regions. LBs of both types, as well as Lewy neurites in this case of NBIA-1, were strongly labeled by antibodies against alpha-synuclein. These findings give further evidence that accumulation of alpha-synuclein is generally associated with LB formation, i.e., in Parkinson's disease, dementia with Lewy bodies and NBIA-1. The case presented here is particularly notable for its high number of LBs in all areas of the cerebral cortex.

Entities:  

Mesh:

Substances:

Year:  2000        PMID: 11045680     DOI: 10.1007/s004010000224

Source DB:  PubMed          Journal:  Acta Neuropathol        ISSN: 0001-6322            Impact factor:   17.088


  28 in total

1.  Selective insolubility of alpha-synuclein in human Lewy body diseases is recapitulated in a transgenic mouse model.

Authors:  P J Kahle; M Neumann; L Ozmen; V Müller; S Odoy; N Okamoto; H Jacobsen; T Iwatsubo; J Q Trojanowski; H Takahashi; K Wakabayashi; N Bogdanovic; P Riederer; H A Kretzschmar; C Haass
Journal:  Am J Pathol       Date:  2001-12       Impact factor: 4.307

2.  The PARK8 locus in autosomal dominant parkinsonism: confirmation of linkage and further delineation of the disease-containing interval.

Authors:  Alexander Zimprich; Bertram Müller-Myhsok; Matthew Farrer; Petra Leitner; Manu Sharma; Mary Hulihan; Paul Lockhart; Audrey Strongosky; Jennifer Kachergus; Donald B Calne; Jon Stoessl; Ryan J Uitti; Ronald F Pfeiffer; Claudia Trenkwalder; Nikolaus Homann; Erwin Ott; Karoline Wenzel; Friedrich Asmus; John Hardy; Zbigniew Wszolek; Thomas Gasser
Journal:  Am J Hum Genet       Date:  2003-12-19       Impact factor: 11.025

Review 3.  Neurodegeneration with brain iron accumulation.

Authors:  Allison Gregory; Susan J Hayflick
Journal:  Folia Neuropathol       Date:  2005       Impact factor: 2.038

Review 4.  Metals in Alzheimer's and Parkinson's Disease: Relevance to Dementia with Lewy Bodies.

Authors:  Erin J McAllum; David I Finkelstein
Journal:  J Mol Neurosci       Date:  2016-08-08       Impact factor: 3.444

5.  Altered neuronal mitochondrial coenzyme A synthesis in neurodegeneration with brain iron accumulation caused by abnormal processing, stability, and catalytic activity of mutant pantothenate kinase 2.

Authors:  Paul T Kotzbauer; Adam C Truax; John Q Trojanowski; Virginia M-Y Lee
Journal:  J Neurosci       Date:  2005-01-19       Impact factor: 6.167

6.  Disrupted membrane homeostasis and accumulation of ubiquitinated proteins in a mouse model of infantile neuroaxonal dystrophy caused by PLA2G6 mutations.

Authors:  Ibrahim Malik; John Turk; David J Mancuso; Laura Montier; Mary Wohltmann; David F Wozniak; Robert E Schmidt; Richard W Gross; Paul T Kotzbauer
Journal:  Am J Pathol       Date:  2008-01-17       Impact factor: 4.307

Review 7.  Clinical and genetic delineation of neurodegeneration with brain iron accumulation.

Authors:  A Gregory; B J Polster; S J Hayflick
Journal:  J Med Genet       Date:  2008-11-03       Impact factor: 6.318

8.  Sleep in genetically confirmed pantothenate kinase-associated neurodegeneration: a video-polysomnographic study.

Authors:  Maria Livia Fantini; Giovanni Cossu; Andrea Molari; Monia Cabinio; Ozlem Uyanik; Roberto Cilia; Maurizio Melis; Angelo Antonini; Luigi Ferini-Strambi
Journal:  Parkinsons Dis       Date:  2010-06-29

Review 9.  Excess iron harms the brain: the syndromes of neurodegeneration with brain iron accumulation (NBIA).

Authors:  Susanne A Schneider; Kailash P Bhatia
Journal:  J Neural Transm (Vienna)       Date:  2012-12-02       Impact factor: 3.575

Review 10.  Biophysics of Parkinson's disease: structure and aggregation of alpha-synuclein.

Authors:  Vladimir N Uversky; David Eliezer
Journal:  Curr Protein Pept Sci       Date:  2009-10       Impact factor: 3.272

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.