Document Detail


Cerebellar leukoencephalopathy: most likely histiocytosis-related.
MedLine Citation:
PMID:  18936429     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
BACKGROUND: Histiocytosis, both Langerhans and non-Langerhans cell type, can be associated with cerebellar white matter abnormalities, thought to be paraneoplastic. The associated clinical picture consists of ataxia, spasticity, and cognitive decline. Hormonal dysfunction is frequent. MRI shows cerebellar white matter abnormalities, as well as brainstem and basal ganglia abnormalities. This so-called "neurodegenerative syndrome" may occur years before or during manifest histiocytosis and also years after cure. We discovered similar MRI abnormalities in 13 patients and wondered whether they could have the same syndrome. METHODS: We reviewed the clinical and laboratory information of these 13 patients and evaluated their brain MRIs. Seven patients underwent spinal cord MRI. RESULTS: All patients were isolated cases; 10 were male. They had signs of cerebellar and pyramidal dysfunction, behavioral problems, and cognitive decline. MRI showed abnormalities of the cerebellar white matter, brainstem, basal ganglia, and, to a lesser extent, cerebral white matter. Three patients had spinal cord lesions. Three patients had laboratory evidence of hormonal dysfunction. No evidence was found of an underlying metabolic defect. In two patients biopsy of nodular brain lesions revealed histiocytic infiltrates. CONCLUSIONS: Considering the striking clinical and MRI similarities between our patients and the patients with this neurodegenerative syndrome in the context of proven histiocytosis, it is likely that they share the same paraneoplastic syndrome, although we cannot exclude a genetic disorder with certainty. The fact that we found histiocytic lesions in two patients substantiates our conclusion. Patients with cerebellar white matter abnormalities should be monitored for histiocytosis.
Authors:
M S van der Knaap; W F M Arts; J Y Garbern; G Hedlund; F Winkler; C Barbosa; M D King; A Bjørnstad; N Hussain; M K Beyer; C Gomez; M C Patterson; P Grattan-Smith; M Timmons; P van der Valk
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Publication Detail:
Type:  Comparative Study; Journal Article; Research Support, Non-U.S. Gov't    
Journal Detail:
Title:  Neurology     Volume:  71     ISSN:  1526-632X     ISO Abbreviation:  Neurology     Publication Date:  2008 Oct 
Date Detail:
Created Date:  2008-10-21     Completed Date:  2008-11-06     Revised Date:  2009-08-12    
Medline Journal Info:
Nlm Unique ID:  0401060     Medline TA:  Neurology     Country:  United States    
Other Details:
Languages:  eng     Pagination:  1361-7     Citation Subset:  AIM; IM    
Affiliation:
Department of Child Neurology, VU University Medical Center, Amsterdam, The Netherlands. ms.vanderknaap@vumc.nl
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MeSH Terms
Descriptor/Qualifier:
Adult
Cerebellar Ataxia / complications,  diagnosis,  pathology
Cerebellar Diseases / complications,  diagnosis*,  pathology
Child
Female
Histiocytosis / complications,  diagnosis*,  pathology
Humans
Magnetic Resonance Imaging / methods
Male
Posterior Leukoencephalopathy Syndrome / complications,  diagnosis*,  pathology
Retrospective Studies
Comments/Corrections
Comment In:
Neurology. 2009 Aug 11;73(6):488; author reply 488-9   [PMID:  19667329 ]

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


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