Vestibular function in patients with Niemann-Pick type C disease.
Identifieur interne : 000631 ( Main/Corpus ); précédent : 000630; suivant : 000632Vestibular function in patients with Niemann-Pick type C disease.
Auteurs : Tatiana Bremova ; Siegbert Krafczyk ; Stanislavs Bardins ; Jörg Reinke ; Michael StruppSource :
- Journal of neurology [ 1432-1459 ] ; 2016.
English descriptors
- KwdEn :
- Adolescent (MeSH), Adult (MeSH), Disability Evaluation (MeSH), Female (MeSH), Head Impulse Test (MeSH), Humans (MeSH), Male (MeSH), Middle Aged (MeSH), Neurologic Examination (MeSH), Niemann-Pick Disease, Type C (complications), Postural Balance (MeSH), Reflex, Vestibulo-Ocular (MeSH), Vestibular Diseases (etiology), Vestibular Evoked Myogenic Potentials (MeSH), Young Adult (MeSH).
- MESH :
Abstract
We investigated whether vestibular dysfunction may cause or contribute to postural imbalance and falls in patients with Niemann-Pick type C disease (NP-C). Eight patients with NP-C disease and 20 healthy controls were examined using the video-based head impulse test (vHIT) and caloric irrigation to investigate horizontal canal function as well as ocular- and cervical vestibular evoked myogenic potentials (o- and cVEMP), and binocular subjective visual vertical estimation (SVV) for otolith function, and static posturography. There were no significant differences in vestibulo-ocular gain, caloric excitability, o-/cVEMP measures or SVV between the two groups. Posturographic total sway path (tSP) and root mean square (RMS) were significantly higher in NP-C than in controls in 3 out of 4 conditions. The Romberg quotient (RQ) to assess the amount of visual stabilization was significantly lower in the NP-C than in the HC group. In contrast to other inherited metabolic disorders, such as Morbus Gaucher type 3, we did not find any evidence for an impairment of canal or otolith function in patients with NP-C as their cause of postural imbalance. Since RQ was low in NP-C patients, indicating proper sensory input, the observed increased postural sway is most likely due to a cerebellar dysfunction in NP-C, which may therefore, explain postural imbalance.
DOI: 10.1007/s00415-016-8247-4
PubMed: 27544496
Links to Exploration step
pubmed:27544496Le document en format XML
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Tatiana.Bremova@med.uni-muenchen.de.</nlm:affiliation></affiliation></author><author><name sortKey="Krafczyk, Siegbert" sort="Krafczyk, Siegbert" uniqKey="Krafczyk S" first="Siegbert" last="Krafczyk">Siegbert Krafczyk</name><affiliation><nlm:affiliation>German Center for Vertigo and Balance Disorders, Grosshadern Campus, University Hospital Munich, Marchioninistrasse 15, 81377, Munich, Germany.</nlm:affiliation></affiliation><affiliation><nlm:affiliation>Department of Neurology, Grosshadern Campus, University Hospital Munich, Munich, Germany.</nlm:affiliation></affiliation></author><author><name sortKey="Bardins, Stanislavs" sort="Bardins, Stanislavs" uniqKey="Bardins S" first="Stanislavs" last="Bardins">Stanislavs Bardins</name><affiliation><nlm:affiliation>German Center for Vertigo and Balance Disorders, Grosshadern Campus, University Hospital Munich, Marchioninistrasse 15, 81377, Munich, Germany.</nlm:affiliation></affiliation></author><author><name sortKey="Reinke, Jorg" sort="Reinke, Jorg" uniqKey="Reinke J" first="Jörg" last="Reinke">Jörg Reinke</name><affiliation><nlm:affiliation>Villa Metabolica, Center for Paediatric and Adolescent Medicine, University Medical Center of the Johannes Gutenberg University, Mainz, Germany.</nlm:affiliation></affiliation></author><author><name sortKey="Strupp, Michael" sort="Strupp, Michael" uniqKey="Strupp M" first="Michael" last="Strupp">Michael Strupp</name><affiliation><nlm:affiliation>German Center for Vertigo and Balance Disorders, Grosshadern Campus, University Hospital Munich, Marchioninistrasse 15, 81377, Munich, Germany.</nlm:affiliation></affiliation><affiliation><nlm:affiliation>Department of Neurology, Grosshadern Campus, University Hospital Munich, Munich, Germany.</nlm:affiliation></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PubMed</idno><date when="2016">2016</date><idno type="RBID">pubmed:27544496</idno><idno type="pmid">27544496</idno><idno type="doi">10.1007/s00415-016-8247-4</idno><idno type="wicri:Area/Main/Corpus">000631</idno><idno type="wicri:explorRef" wicri:stream="Main" wicri:step="Corpus" wicri:corpus="PubMed">000631</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en">Vestibular function in patients with Niemann-Pick type C disease.</title><author><name sortKey="Bremova, Tatiana" sort="Bremova, Tatiana" uniqKey="Bremova T" first="Tatiana" last="Bremova">Tatiana Bremova</name><affiliation><nlm:affiliation>German Center for Vertigo and Balance Disorders, Grosshadern Campus, University Hospital Munich, Marchioninistrasse 15, 81377, Munich, Germany. Tatiana.Bremova@med.uni-muenchen.de.</nlm:affiliation></affiliation><affiliation><nlm:affiliation>Department of Neurology, Grosshadern Campus, University Hospital Munich, Munich, Germany. Tatiana.Bremova@med.uni-muenchen.de.</nlm:affiliation></affiliation><affiliation><nlm:affiliation>Graduate School of Systemic Neurosciences, Ludwig-Maximilians University, Munich, Germany. Tatiana.Bremova@med.uni-muenchen.de.</nlm:affiliation></affiliation></author><author><name sortKey="Krafczyk, Siegbert" sort="Krafczyk, Siegbert" uniqKey="Krafczyk S" first="Siegbert" last="Krafczyk">Siegbert Krafczyk</name><affiliation><nlm:affiliation>German Center for Vertigo and Balance Disorders, Grosshadern Campus, University Hospital Munich, Marchioninistrasse 15, 81377, Munich, Germany.</nlm:affiliation></affiliation><affiliation><nlm:affiliation>Department of Neurology, Grosshadern Campus, University Hospital Munich, Munich, Germany.</nlm:affiliation></affiliation></author><author><name sortKey="Bardins, Stanislavs" sort="Bardins, Stanislavs" uniqKey="Bardins S" first="Stanislavs" last="Bardins">Stanislavs Bardins</name><affiliation><nlm:affiliation>German Center for Vertigo and Balance Disorders, Grosshadern Campus, University Hospital Munich, Marchioninistrasse 15, 81377, Munich, Germany.</nlm:affiliation></affiliation></author><author><name sortKey="Reinke, Jorg" sort="Reinke, Jorg" uniqKey="Reinke J" first="Jörg" last="Reinke">Jörg Reinke</name><affiliation><nlm:affiliation>Villa Metabolica, Center for Paediatric and Adolescent Medicine, University Medical Center of the Johannes Gutenberg University, Mainz, Germany.</nlm:affiliation></affiliation></author><author><name sortKey="Strupp, Michael" sort="Strupp, Michael" uniqKey="Strupp M" first="Michael" last="Strupp">Michael Strupp</name><affiliation><nlm:affiliation>German Center for Vertigo and Balance Disorders, Grosshadern Campus, University Hospital Munich, Marchioninistrasse 15, 81377, Munich, Germany.</nlm:affiliation></affiliation><affiliation><nlm:affiliation>Department of Neurology, Grosshadern Campus, University Hospital Munich, Munich, Germany.</nlm:affiliation></affiliation></author></analytic><series><title level="j">Journal of neurology</title><idno type="eISSN">1432-1459</idno><imprint><date when="2016" type="published">2016</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Adolescent (MeSH)</term><term>Adult (MeSH)</term><term>Disability Evaluation (MeSH)</term><term>Female (MeSH)</term><term>Head Impulse Test (MeSH)</term><term>Humans (MeSH)</term><term>Male (MeSH)</term><term>Middle Aged (MeSH)</term><term>Neurologic Examination (MeSH)</term><term>Niemann-Pick Disease, Type C (complications)</term><term>Postural Balance (MeSH)</term><term>Reflex, Vestibulo-Ocular (MeSH)</term><term>Vestibular Diseases (etiology)</term><term>Vestibular Evoked Myogenic Potentials (MeSH)</term><term>Young Adult (MeSH)</term></keywords><keywords scheme="MESH" qualifier="complications" xml:lang="en"><term>Niemann-Pick Disease, Type C</term></keywords><keywords scheme="MESH" qualifier="etiology" xml:lang="en"><term>Vestibular Diseases</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Adolescent</term><term>Adult</term><term>Disability Evaluation</term><term>Female</term><term>Head Impulse Test</term><term>Humans</term><term>Male</term><term>Middle Aged</term><term>Neurologic Examination</term><term>Postural Balance</term><term>Reflex, Vestibulo-Ocular</term><term>Vestibular Evoked Myogenic Potentials</term><term>Young Adult</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">We investigated whether vestibular dysfunction may cause or contribute to postural imbalance and falls in patients with Niemann-Pick type C disease (NP-C). Eight patients with NP-C disease and 20 healthy controls were examined using the video-based head impulse test (vHIT) and caloric irrigation to investigate horizontal canal function as well as ocular- and cervical vestibular evoked myogenic potentials (o- and cVEMP), and binocular subjective visual vertical estimation (SVV) for otolith function, and static posturography. There were no significant differences in vestibulo-ocular gain, caloric excitability, o-/cVEMP measures or SVV between the two groups. Posturographic total sway path (tSP) and root mean square (RMS) were significantly higher in NP-C than in controls in 3 out of 4 conditions. The Romberg quotient (RQ) to assess the amount of visual stabilization was significantly lower in the NP-C than in the HC group. In contrast to other inherited metabolic disorders, such as Morbus Gaucher type 3, we did not find any evidence for an impairment of canal or otolith function in patients with NP-C as their cause of postural imbalance. Since RQ was low in NP-C patients, indicating proper sensory input, the observed increased postural sway is most likely due to a cerebellar dysfunction in NP-C, which may therefore, explain postural imbalance.</div></front></TEI><pubmed><MedlineCitation Status="MEDLINE" Owner="NLM"><PMID Version="1">27544496</PMID><DateCompleted><Year>2017</Year><Month>04</Month><Day>07</Day></DateCompleted><DateRevised><Year>2018</Year><Month>11</Month><Day>13</Day></DateRevised><Article PubModel="Print-Electronic"><Journal><ISSN IssnType="Electronic">1432-1459</ISSN><JournalIssue CitedMedium="Internet"><Volume>263</Volume><Issue>11</Issue><PubDate><Year>2016</Year><Month>Nov</Month></PubDate></JournalIssue><Title>Journal of neurology</Title><ISOAbbreviation>J Neurol</ISOAbbreviation></Journal><ArticleTitle>Vestibular function in patients with Niemann-Pick type C disease.</ArticleTitle><Pagination><MedlinePgn>2260-2270</MedlinePgn></Pagination><Abstract><AbstractText>We investigated whether vestibular dysfunction may cause or contribute to postural imbalance and falls in patients with Niemann-Pick type C disease (NP-C). Eight patients with NP-C disease and 20 healthy controls were examined using the video-based head impulse test (vHIT) and caloric irrigation to investigate horizontal canal function as well as ocular- and cervical vestibular evoked myogenic potentials (o- and cVEMP), and binocular subjective visual vertical estimation (SVV) for otolith function, and static posturography. There were no significant differences in vestibulo-ocular gain, caloric excitability, o-/cVEMP measures or SVV between the two groups. Posturographic total sway path (tSP) and root mean square (RMS) were significantly higher in NP-C than in controls in 3 out of 4 conditions. The Romberg quotient (RQ) to assess the amount of visual stabilization was significantly lower in the NP-C than in the HC group. In contrast to other inherited metabolic disorders, such as Morbus Gaucher type 3, we did not find any evidence for an impairment of canal or otolith function in patients with NP-C as their cause of postural imbalance. Since RQ was low in NP-C patients, indicating proper sensory input, the observed increased postural sway is most likely due to a cerebellar dysfunction in NP-C, which may therefore, explain postural imbalance.</AbstractText></Abstract><AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Bremova</LastName><ForeName>Tatiana</ForeName><Initials>T</Initials><AffiliationInfo><Affiliation>German Center for Vertigo and Balance Disorders, Grosshadern Campus, University Hospital Munich, Marchioninistrasse 15, 81377, Munich, Germany. Tatiana.Bremova@med.uni-muenchen.de.</Affiliation></AffiliationInfo><AffiliationInfo><Affiliation>Department of Neurology, Grosshadern Campus, University Hospital Munich, Munich, Germany. Tatiana.Bremova@med.uni-muenchen.de.</Affiliation></AffiliationInfo><AffiliationInfo><Affiliation>Graduate School of Systemic Neurosciences, Ludwig-Maximilians University, Munich, Germany. Tatiana.Bremova@med.uni-muenchen.de.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Krafczyk</LastName><ForeName>Siegbert</ForeName><Initials>S</Initials><AffiliationInfo><Affiliation>German Center for Vertigo and Balance Disorders, Grosshadern Campus, University Hospital Munich, Marchioninistrasse 15, 81377, Munich, Germany.</Affiliation></AffiliationInfo><AffiliationInfo><Affiliation>Department of Neurology, Grosshadern Campus, University Hospital Munich, Munich, Germany.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Bardins</LastName><ForeName>Stanislavs</ForeName><Initials>S</Initials><AffiliationInfo><Affiliation>German Center for Vertigo and Balance Disorders, Grosshadern Campus, University Hospital Munich, Marchioninistrasse 15, 81377, Munich, Germany.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Reinke</LastName><ForeName>Jörg</ForeName><Initials>J</Initials><AffiliationInfo><Affiliation>Villa Metabolica, Center for Paediatric and Adolescent Medicine, University Medical Center of the Johannes Gutenberg University, Mainz, Germany.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Strupp</LastName><ForeName>Michael</ForeName><Initials>M</Initials><AffiliationInfo><Affiliation>German Center for Vertigo and Balance Disorders, Grosshadern Campus, University Hospital Munich, Marchioninistrasse 15, 81377, Munich, Germany.</Affiliation></AffiliationInfo><AffiliationInfo><Affiliation>Department of Neurology, Grosshadern Campus, University Hospital Munich, Munich, Germany.</Affiliation></AffiliationInfo></Author></AuthorList><Language>eng</Language><PublicationTypeList><PublicationType UI="D016428">Journal Article</PublicationType></PublicationTypeList><ArticleDate DateType="Electronic"><Year>2016</Year><Month>08</Month><Day>20</Day></ArticleDate></Article><MedlineJournalInfo><Country>Germany</Country><MedlineTA>J Neurol</MedlineTA><NlmUniqueID>0423161</NlmUniqueID><ISSNLinking>0340-5354</ISSNLinking></MedlineJournalInfo><CitationSubset>IM</CitationSubset><MeshHeadingList><MeshHeading><DescriptorName UI="D000293" MajorTopicYN="N">Adolescent</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D000328" MajorTopicYN="N">Adult</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D004185" MajorTopicYN="N">Disability Evaluation</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D005260" MajorTopicYN="N">Female</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D064087" MajorTopicYN="N">Head Impulse Test</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D006801" MajorTopicYN="N">Humans</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D008297" MajorTopicYN="N">Male</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D008875" MajorTopicYN="N">Middle Aged</DescriptorName></MeshHeading><MeshHeading><DescriptorName 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disorders/ataxia</Keyword><Keyword MajorTopicYN="Y">Lipidoses</Keyword><Keyword MajorTopicYN="Y">Metabolic disease (inherited)</Keyword><Keyword MajorTopicYN="Y">Niemann-Pick type C</Keyword><Keyword MajorTopicYN="Y">Vertigo</Keyword><Keyword MajorTopicYN="Y">Vestibular evoked myogenic potentials</Keyword><Keyword MajorTopicYN="Y">Vestibular function</Keyword></KeywordList></MedlineCitation><PubmedData><History><PubMedPubDate PubStatus="received"><Year>2016</Year><Month>03</Month><Day>17</Day></PubMedPubDate><PubMedPubDate PubStatus="accepted"><Year>2016</Year><Month>07</Month><Day>27</Day></PubMedPubDate><PubMedPubDate PubStatus="revised"><Year>2016</Year><Month>06</Month><Day>30</Day></PubMedPubDate><PubMedPubDate PubStatus="pubmed"><Year>2016</Year><Month>8</Month><Day>22</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate PubStatus="medline"><Year>2017</Year><Month>4</Month><Day>8</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate 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