Document Detail


Fragile X dementia Parkinsonism Syndrome (FXDPS).
MedLine Citation:
PMID:  20299862     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
BACKGROUND/OBJECTIVE: The fragile X-associated tremor/ataxia syndrome is characterized by intention tremor and ataxia in people who are premutation carriers of the Fragile X gene. Patients with this disorder might also demonstrate signs of dementia with parkinsonian features. We report a patient with dementia and parkinsonian signs who did not demonstrate an intention tremor or gait ataxia. METHODS: A 58-year-old woman who had 2 sons with fragile X retardation syndrome and was a carrier, developed progressive dementia, including impaired memory, executive dysfunction, nonfluent speech, and parkinsonian signs, but had no action-intention tremor and no gait ataxia. Magnetic resonance imaging revealed extensive abnormalities of the white matter. RESULTS: On post-mortem examination, 7 years after this evaluation, she demonstrated extensive subcortical white matter pallor (spongiosis) and widespread ubiquitin-positive intranuclear inclusions in both neurons and in protoplasmic astrocytes characteristic of fragile X-associated tremor/ataxia syndrome, but no spongiosis in the cerebellar peduncles a defining feature of this tremor/ataxia syndrome. CONCLUSIONS: Patients who present with dementia and signs of Parkinson syndrome, even in the absence of ataxia or intention tremor should be evaluated for this fragile X dementia parkinsonism syndrome.
Authors:
Anthony T Yachnis; Heidi L Roth; Kenneth M Heilman
Publication Detail:
Type:  Case Reports; Journal Article; Research Support, Non-U.S. Gov't; Research Support, U.S. Gov't, Non-P.H.S.    
Journal Detail:
Title:  Cognitive and behavioral neurology : official journal of the Society for Behavioral and Cognitive Neurology     Volume:  23     ISSN:  1543-3641     ISO Abbreviation:  Cogn Behav Neurol     Publication Date:  2010 Mar 
Date Detail:
Created Date:  2010-03-19     Completed Date:  2010-06-23     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  101167278     Medline TA:  Cogn Behav Neurol     Country:  United States    
Other Details:
Languages:  eng     Pagination:  39-43     Citation Subset:  IM    
Affiliation:
Department of Neurology, Center for Neuropsychological Studies and Dementia, University of Florida College of Medicine, Gainesville, FL 32610, USA.
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MeSH Terms
Descriptor/Qualifier:
Brain / pathology
Dementia / complications*
Fatal Outcome
Female
Fragile X Syndrome / complications*,  diagnosis,  genetics*
Hallucinations / diagnosis,  etiology
Humans
Middle Aged
Parkinson Disease / complications*

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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