Pallidopyramidal disease: A misnomer?
Identifieur interne : 001437 ( Istex/Corpus ); précédent : 001436; suivant : 001438Pallidopyramidal disease: A misnomer?
Auteurs : Martin W. I. M. Horstink ; Marieke C. Dekker ; Pasquale Montagna ; Vincenzo Bonifati ; Bart P. Van De WarrenburgSource :
- Movement Disorders [ 0885-3185 ] ; 2010-07-15.
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- KwdEn :
Abstract
The combination of recessive early‐onset parkinsonism and pyramidal tract signs caused by pallidopyramidal degeneration is known as pallidopyramidal disease or syndrome (PPD/S). We investigated whether patients diagnosed as Davison's PPD/S showed any definite proof of pyramidal and pallidal involvement, without findings suggestive of other nosological entities. Since Davison's original description, 15 other PPD/S cases have been reported, yet all lack proof of pyramidal or pallidal degeneration. Because of the dopa‐responsiveness in all patients subsequent to Davison's report, we argue that these patients probably suffered from early‐onset nigral parkinsonism or dopa‐responsive dsystonia, rather than pallidal parkinsonism; in such cases, the presumed pyramidal Babinski could be a pseudobabinski (“striatal toe”). Secondary pallidopyramidal syndromes do occur, for example, in multiple system atrophy or Wilson's disease, but in these patients additional findings indicate diseases other than Davison's PPD/S. We conclude that the existence of PPD/S as a distinct clinico‐pathological nosological entity, as proposed by Davison, is doubtful. In cases reported as Davison's PPD/S, the description “pallidopyramidal” seems to be a misnomer. © 2010 Movement Disorder Society
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DOI: 10.1002/mds.23118
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<front><div type="abstract" xml:lang="en">The combination of recessive early‐onset parkinsonism and pyramidal tract signs caused by pallidopyramidal degeneration is known as pallidopyramidal disease or syndrome (PPD/S). We investigated whether patients diagnosed as Davison's PPD/S showed any definite proof of pyramidal and pallidal involvement, without findings suggestive of other nosological entities. Since Davison's original description, 15 other PPD/S cases have been reported, yet all lack proof of pyramidal or pallidal degeneration. Because of the dopa‐responsiveness in all patients subsequent to Davison's report, we argue that these patients probably suffered from early‐onset nigral parkinsonism or dopa‐responsive dsystonia, rather than pallidal parkinsonism; in such cases, the presumed pyramidal Babinski could be a pseudobabinski (“striatal toe”). Secondary pallidopyramidal syndromes do occur, for example, in multiple system atrophy or Wilson's disease, but in these patients additional findings indicate diseases other than Davison's PPD/S. We conclude that the existence of PPD/S as a distinct clinico‐pathological nosological entity, as proposed by Davison, is doubtful. In cases reported as Davison's PPD/S, the description “pallidopyramidal” seems to be a misnomer. © 2010 Movement Disorder Society</div>
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<keyword xml:id="kwd5">juvenile parkinsonism</keyword>
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<abstractGroup><abstract type="main" xml:lang="en"><title type="main">Abstract</title>
<p>The combination of recessive early‐onset parkinsonism and pyramidal tract signs caused by pallidopyramidal degeneration is known as pallidopyramidal disease or syndrome (PPD/S). We investigated whether patients diagnosed as Davison's PPD/S showed any definite proof of pyramidal and pallidal involvement, without findings suggestive of other nosological entities. Since Davison's original description, 15 other PPD/S cases have been reported, yet all lack proof of pyramidal or pallidal degeneration. Because of the dopa‐responsiveness in all patients subsequent to Davison's report, we argue that these patients probably suffered from early‐onset nigral parkinsonism or dopa‐responsive dsystonia, rather than pallidal parkinsonism; in such cases, the presumed pyramidal Babinski could be a pseudobabinski (“striatal toe”). Secondary pallidopyramidal syndromes do occur, for example, in multiple system atrophy or Wilson's disease, but in these patients additional findings indicate diseases other than Davison's PPD/S. We conclude that the existence of PPD/S as a distinct clinico‐pathological nosological entity, as proposed by Davison, is doubtful. In cases reported as Davison's PPD/S, the description “pallidopyramidal” seems to be a misnomer. © 2010 Movement Disorder Society</p>
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<noteGroup><note xml:id="fn1"><p>Potential conflict of interest: None reported.</p>
</note>
<note xml:id="fn2"><p>Deceased</p>
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<!--Version 0.6 générée le 4-12-2015--><mods version="3.6"><titleInfo lang="en"><title>Pallidopyramidal disease: A misnomer?</title>
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<titleInfo type="abbreviated" lang="en"><title>Pallidopyramidal Disease: A Misnomer?</title>
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<titleInfo type="alternative" contentType="CDATA" lang="en"><title>Pallidopyramidal disease: A misnomer?</title>
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<name type="personal"><namePart type="given">Martin W.I.M.</namePart>
<namePart type="family">Horstink</namePart>
<namePart type="termsOfAddress">MD, PhD</namePart>
<affiliation>Department of Neurology, Donders Centre for Brain, Cognition, and Behaviour, Radboud University Nijmegen Medical Centre, Nijmegen, The Netherlands</affiliation>
<description>Deceased</description>
<role><roleTerm type="text">author</roleTerm>
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<name type="personal"><namePart type="given">Marieke C.</namePart>
<namePart type="family">Dekker</namePart>
<namePart type="termsOfAddress">MD, PhD</namePart>
<affiliation>Department of Neurology, Donders Centre for Brain, Cognition, and Behaviour, Radboud University Nijmegen Medical Centre, Nijmegen, The Netherlands</affiliation>
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<name type="personal"><namePart type="given">Pasquale</namePart>
<namePart type="family">Montagna</namePart>
<namePart type="termsOfAddress">MD</namePart>
<affiliation>Department of Neurological Sciences, University of Bologna, Bologna, Italy</affiliation>
<role><roleTerm type="text">author</roleTerm>
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</name>
<name type="personal"><namePart type="given">Vincenzo</namePart>
<namePart type="family">Bonifati</namePart>
<namePart type="termsOfAddress">MD, PhD</namePart>
<affiliation>Department of Clinical Genetics, Erasmus Medical Centre, Rotterdam, The Netherlands</affiliation>
<role><roleTerm type="text">author</roleTerm>
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<name type="personal"><namePart type="given">Bart P.</namePart>
<namePart type="family">van De Warrenburg</namePart>
<namePart type="termsOfAddress">MD, PhD</namePart>
<affiliation>Department of Neurology, Donders Centre for Brain, Cognition, and Behaviour, Radboud University Nijmegen Medical Centre, Nijmegen, The Netherlands</affiliation>
<description>Correspondence: Department of Neurology Donders Centre for Brain, Cognition, and Behaviour, Radboud University Nijmegen Medical Centre, PO Box 9101, Nijmegen 6500 HB, The Netherlands</description>
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<abstract lang="en">The combination of recessive early‐onset parkinsonism and pyramidal tract signs caused by pallidopyramidal degeneration is known as pallidopyramidal disease or syndrome (PPD/S). We investigated whether patients diagnosed as Davison's PPD/S showed any definite proof of pyramidal and pallidal involvement, without findings suggestive of other nosological entities. Since Davison's original description, 15 other PPD/S cases have been reported, yet all lack proof of pyramidal or pallidal degeneration. Because of the dopa‐responsiveness in all patients subsequent to Davison's report, we argue that these patients probably suffered from early‐onset nigral parkinsonism or dopa‐responsive dsystonia, rather than pallidal parkinsonism; in such cases, the presumed pyramidal Babinski could be a pseudobabinski (“striatal toe”). Secondary pallidopyramidal syndromes do occur, for example, in multiple system atrophy or Wilson's disease, but in these patients additional findings indicate diseases other than Davison's PPD/S. We conclude that the existence of PPD/S as a distinct clinico‐pathological nosological entity, as proposed by Davison, is doubtful. In cases reported as Davison's PPD/S, the description “pallidopyramidal” seems to be a misnomer. © 2010 Movement Disorder Society</abstract>
<note type="content">*Potential conflict of interest: None reported.</note>
<subject lang="en"><genre>Keywords</genre>
<topic>pallidopyramidal</topic>
<topic>pseudobabinski</topic>
<topic>pyramidal syndrome</topic>
<topic>pallidal atrophy</topic>
<topic>juvenile parkinsonism</topic>
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<topic>Historical Review</topic>
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<identifier type="ISSN">0885-3185</identifier>
<identifier type="eISSN">1531-8257</identifier>
<identifier type="DOI">10.1002/(ISSN)1531-8257</identifier>
<identifier type="PublisherID">MDS</identifier>
<part><date>2010</date>
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<number>25</number>
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<accessCondition type="use and reproduction" contentType="copyright">Copyright © 2010 Movement Disorder Society</accessCondition>
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