Severe generalised dystonia associated with a mosaic pattern of striatal gliosis.
Identifieur interne : 001455 ( Ncbi/Curation ); précédent : 001454; suivant : 001456Severe generalised dystonia associated with a mosaic pattern of striatal gliosis.
Auteurs : W R Gibb [Royaume-Uni] ; L. Kilford ; C D MarsdenSource :
- Movement disorders : official journal of the Movement Disorder Society [ 0885-3185 ] ; 1992.
English descriptors
- KwdEn :
- MESH :
- chemical , analysis : Glial Fibrillary Acidic Protein.
- chemical , poisoning : Chlormethiazole.
- pathology : Astrocytes, Corpus Striatum, Drug Overdose, Dystonia Musculorum Deformans, Gliosis, Putamen.
- Adolescent, Humans, Male.
Abstract
A mosaic pattern of striatal pathology is described in a male who developed severe generalised dystonia from the age of 10 years, and died at the age of 18 years. There was no family history of dystonia, and extensive investigations during his life failed to identify a cause for the dystonia. The caudate nucleus and putamen showed a network of cell loss and gliosis surrounding islands of preserved striatum. Dorsal parts showed confluent gliosis, and ventral parts were spared. The pattern suggested a correlation with patch-matrix organisation, but there was no correlation with the distribution of calbindin immunoreactive cells, which are present in the matrix of the classical striosome-matrix organisation. The pathological findings were unlike those in status marmoratus, perinatal hypoxia-ischaemia, Huntington's disease, and neuroacanthocytosis, but similar to those reported in a 44-year-old man with predominantly cranial dystonia.
DOI: 10.1002/mds.870070305
PubMed: 1620138
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pubmed:1620138Le document en format XML
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<author><name sortKey="Gibb, W R" sort="Gibb, W R" uniqKey="Gibb W" first="W R" last="Gibb">W R Gibb</name>
<affiliation wicri:level="3"><nlm:affiliation>Parkinson's Disease Society Brain Bank, Institute of Neurology, London, United Kingdom.</nlm:affiliation>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>Parkinson's Disease Society Brain Bank, Institute of Neurology, London</wicri:regionArea>
<placeName><settlement type="city">Londres</settlement>
<region type="country">Angleterre</region>
<region type="région" nuts="1">Grand Londres</region>
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<author><name sortKey="Kilford, L" sort="Kilford, L" uniqKey="Kilford L" first="L" last="Kilford">L. Kilford</name>
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<author><name sortKey="Marsden, C D" sort="Marsden, C D" uniqKey="Marsden C" first="C D" last="Marsden">C D Marsden</name>
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<sourceDesc><biblStruct><analytic><title xml:lang="en">Severe generalised dystonia associated with a mosaic pattern of striatal gliosis.</title>
<author><name sortKey="Gibb, W R" sort="Gibb, W R" uniqKey="Gibb W" first="W R" last="Gibb">W R Gibb</name>
<affiliation wicri:level="3"><nlm:affiliation>Parkinson's Disease Society Brain Bank, Institute of Neurology, London, United Kingdom.</nlm:affiliation>
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<series><title level="j">Movement disorders : official journal of the Movement Disorder Society</title>
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<term>Astrocytes (pathology)</term>
<term>Chlormethiazole (poisoning)</term>
<term>Corpus Striatum (pathology)</term>
<term>Drug Overdose (pathology)</term>
<term>Dystonia Musculorum Deformans (pathology)</term>
<term>Glial Fibrillary Acidic Protein (analysis)</term>
<term>Gliosis (pathology)</term>
<term>Humans</term>
<term>Male</term>
<term>Putamen (pathology)</term>
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<keywords scheme="MESH" type="chemical" qualifier="analysis" xml:lang="en"><term>Glial Fibrillary Acidic Protein</term>
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<keywords scheme="MESH" type="chemical" qualifier="poisoning" xml:lang="en"><term>Chlormethiazole</term>
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<keywords scheme="MESH" qualifier="pathology" xml:lang="en"><term>Astrocytes</term>
<term>Corpus Striatum</term>
<term>Drug Overdose</term>
<term>Dystonia Musculorum Deformans</term>
<term>Gliosis</term>
<term>Putamen</term>
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<front><div type="abstract" xml:lang="en">A mosaic pattern of striatal pathology is described in a male who developed severe generalised dystonia from the age of 10 years, and died at the age of 18 years. There was no family history of dystonia, and extensive investigations during his life failed to identify a cause for the dystonia. The caudate nucleus and putamen showed a network of cell loss and gliosis surrounding islands of preserved striatum. Dorsal parts showed confluent gliosis, and ventral parts were spared. The pattern suggested a correlation with patch-matrix organisation, but there was no correlation with the distribution of calbindin immunoreactive cells, which are present in the matrix of the classical striosome-matrix organisation. The pathological findings were unlike those in status marmoratus, perinatal hypoxia-ischaemia, Huntington's disease, and neuroacanthocytosis, but similar to those reported in a 44-year-old man with predominantly cranial dystonia.</div>
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