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A new MRI rating scale for progressive supranuclear palsy and multiple system atrophy: validity and reliability

Identifieur interne : 001038 ( Main/Corpus ); précédent : 001037; suivant : 001039

A new MRI rating scale for progressive supranuclear palsy and multiple system atrophy: validity and reliability

Auteurs : Yan Rolland ; Marc Vérin ; Christine A. Payan ; Simon Duchesne ; Eduard Kraft ; Till K. Hauser ; Josef Jarosz ; Neil Deasy ; Luc Defevbre ; Christine Delmaire ; Didier Dormont ; Albert C. Ludolph ; Gilbert Bensimon ; P Nigel Leigh

Source :

RBID : ISTEX:EC1A86B2145155C772D9465608300E551C02AD9F

Abstract

Aim To evaluate a standardised MRI acquisition protocol and a new image rating scale for disease severity in patients with progressive supranuclear palsy (PSP) and multiple systems atrophy (MSA) in a large multicentre study. Methods The MRI protocol consisted of two-dimensional sagittal and axial T1, axial PD, and axial and coronal T2 weighted acquisitions. The 32 item ordinal scale evaluated abnormalities within the basal ganglia and posterior fossa, blind to diagnosis. Among 760 patients in the study population (PSP=362, MSA=398), 627 had per protocol images (PSP=297, MSA=330). Intra-rater (n=60) and inter-rater (n=555) reliability were assessed through Cohen's statistic, and scale structure through principal component analysis (PCA) (n=441). Internal consistency and reliability were checked. Discriminant and predictive validity of extracted factors and total scores were tested for disease severity as per clinical diagnosis. Results Intra-rater and inter-rater reliability were acceptable for 25 (78%) of the items scored (≥0.41). PCA revealed four meaningful clusters of covarying parameters (factor (F) F1: brainstem and cerebellum; F2: midbrain; F3: putamen; F4: other basal ganglia) with good to excellent internal consistency (Cronbach α 0.75–0.93) and moderate to excellent reliability (intraclass coefficient: F1: 0.92; F2: 0.79; F3: 0.71; F4: 0.49). The total score significantly discriminated for disease severity or diagnosis; factorial scores differentially discriminated for disease severity according to diagnosis (PSP: F1–F2; MSA: F2–F3). The total score was significantly related to survival in PSP (p<0.0007) or MSA (p<0.0005), indicating good predictive validity. Conclusions The scale is suitable for use in the context of multicentre studies and can reliably and consistently measure MRI abnormalities in PSP and MSA. Clinical Trial Registration Number The study protocol was filed in the open clinical trial registry (http://www.clinicaltrials.gov) with ID No NCT00211224.

Url:
DOI: 10.1136/jnnp.2010.214890

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<div type="abstract">Aim To evaluate a standardised MRI acquisition protocol and a new image rating scale for disease severity in patients with progressive supranuclear palsy (PSP) and multiple systems atrophy (MSA) in a large multicentre study. Methods The MRI protocol consisted of two-dimensional sagittal and axial T1, axial PD, and axial and coronal T2 weighted acquisitions. The 32 item ordinal scale evaluated abnormalities within the basal ganglia and posterior fossa, blind to diagnosis. Among 760 patients in the study population (PSP=362, MSA=398), 627 had per protocol images (PSP=297, MSA=330). Intra-rater (n=60) and inter-rater (n=555) reliability were assessed through Cohen's statistic, and scale structure through principal component analysis (PCA) (n=441). Internal consistency and reliability were checked. Discriminant and predictive validity of extracted factors and total scores were tested for disease severity as per clinical diagnosis. Results Intra-rater and inter-rater reliability were acceptable for 25 (78%) of the items scored (≥0.41). PCA revealed four meaningful clusters of covarying parameters (factor (F) F1: brainstem and cerebellum; F2: midbrain; F3: putamen; F4: other basal ganglia) with good to excellent internal consistency (Cronbach α 0.75–0.93) and moderate to excellent reliability (intraclass coefficient: F1: 0.92; F2: 0.79; F3: 0.71; F4: 0.49). The total score significantly discriminated for disease severity or diagnosis; factorial scores differentially discriminated for disease severity according to diagnosis (PSP: F1–F2; MSA: F2–F3). The total score was significantly related to survival in PSP (p<0.0007) or MSA (p<0.0005), indicating good predictive validity. Conclusions The scale is suitable for use in the context of multicentre studies and can reliably and consistently measure MRI abnormalities in PSP and MSA. Clinical Trial Registration Number The study protocol was filed in the open clinical trial registry (http://www.clinicaltrials.gov) with ID No NCT00211224.</div>
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<forename type="first">Christine A</forename>
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<forename type="first">Luc</forename>
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<affiliation>Department of Neurology Movement Disorders, Lille University, Salengro Hospital, Cedex Lille, France</affiliation>
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<forename type="first">Christine</forename>
<surname>Delmaire</surname>
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<affiliation>Department of Neuroradiology, Lille University, Salengro Hospital, Cedex Lille, France</affiliation>
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<forename type="first">Didier</forename>
<surname>Dormont</surname>
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<affiliation>Correspondence to Professor P N Leigh, Professor of Neurology, Brighton and Sussex Medical School, Trafford Centre for Biomedical Research University of Sussex, Falmer, East Sussex BN1 9RY; bsms2899@bsms.ac.ukor Dr Gilbert Bensimon, Département de Pharmacologie Clinique, Hôpital Pitié-Salpêtrière, 47 Bd de L'Hôpital, 75651 Paris Cedex 13, France; gilbert.bensimon@psl.aphp.fr</affiliation>
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<p>Aim To evaluate a standardised MRI acquisition protocol and a new image rating scale for disease severity in patients with progressive supranuclear palsy (PSP) and multiple systems atrophy (MSA) in a large multicentre study. Methods The MRI protocol consisted of two-dimensional sagittal and axial T1, axial PD, and axial and coronal T2 weighted acquisitions. The 32 item ordinal scale evaluated abnormalities within the basal ganglia and posterior fossa, blind to diagnosis. Among 760 patients in the study population (PSP=362, MSA=398), 627 had per protocol images (PSP=297, MSA=330). Intra-rater (n=60) and inter-rater (n=555) reliability were assessed through Cohen's statistic, and scale structure through principal component analysis (PCA) (n=441). Internal consistency and reliability were checked. Discriminant and predictive validity of extracted factors and total scores were tested for disease severity as per clinical diagnosis. Results Intra-rater and inter-rater reliability were acceptable for 25 (78%) of the items scored (≥0.41). PCA revealed four meaningful clusters of covarying parameters (factor (F) F1: brainstem and cerebellum; F2: midbrain; F3: putamen; F4: other basal ganglia) with good to excellent internal consistency (Cronbach α 0.75–0.93) and moderate to excellent reliability (intraclass coefficient: F1: 0.92; F2: 0.79; F3: 0.71; F4: 0.49). The total score significantly discriminated for disease severity or diagnosis; factorial scores differentially discriminated for disease severity according to diagnosis (PSP: F1–F2; MSA: F2–F3). The total score was significantly related to survival in PSP (p<0.0007) or MSA (p<0.0005), indicating good predictive validity. Conclusions The scale is suitable for use in the context of multicentre studies and can reliably and consistently measure MRI abnormalities in PSP and MSA. Clinical Trial Registration Number The study protocol was filed in the open clinical trial registry (http://www.clinicaltrials.gov) with ID No NCT00211224.</p>
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<xref ref-type="aff" rid="aff5">5</xref>
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<xref ref-type="aff" rid="aff8">8</xref>
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<xref ref-type="aff" rid="aff9">9</xref>
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<given-names>Christine</given-names>
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<xref ref-type="aff" rid="aff10">10</xref>
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<xref ref-type="aff" rid="aff5">5</xref>
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<collab>on behalf of the NNIPPS Study Group</collab>
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<aff id="aff1">
<label>1</label>
Medical Imaging Department, Eugène Marquis Centre, Rennes, France</aff>
<aff id="aff2">
<label>2</label>
Department of Neurology, Pontchaillou University Hospital, Rennes, France</aff>
<aff id="aff3">
<label>3</label>
Département de Pharmacologie Clinique, Centre Hospitalo-Universitaire de la Pitié-Salpétrière, Assistance publique hôpitaux de Paris and UPMC Univ, Paris, France</aff>
<aff id="aff4">
<label>4</label>
Department of Radiology and Robert Giffard Laval University Research Centre, Laval University, Quebec City, Canada</aff>
<aff id="aff5">
<label>5</label>
Department of Neurology, University of Ulm, Baden-Wuerttemberg, Germany</aff>
<aff id="aff6">
<label>6</label>
Department of Neuroradiology, University of Tübingen, Tübingen, Germany</aff>
<aff id="aff7">
<label>7</label>
Department of Neurology, Brighton and Sussex Medical School, Trafford Centre for Biomedical Research, University of Sussex Falmer, East Sussex, UK</aff>
<aff id="aff8">
<label>8</label>
MRC Centre for Neurodegeneration Research, King's College London, Institute of Psychiatry, Department of Clinical Neuroscience, London, UK</aff>
<aff id="aff9">
<label>9</label>
Department of Neurology Movement Disorders, Lille University, Salengro Hospital, Cedex Lille, France</aff>
<aff id="aff10">
<label>10</label>
Department of Neuroradiology, Lille University, Salengro Hospital, Cedex Lille, France</aff>
<aff id="aff11">
<label>11</label>
Département de Neuroradiologie, Hôpital de la Pitié-Salpétrière, Assistance Publique Hôpitaux de Paris and UPMC Univ, Paris, France</aff>
<author-notes>
<corresp>
<label>Correspondence to</label>
Professor P N Leigh, Professor of Neurology, Brighton and Sussex Medical School, Trafford Centre for Biomedical Research University of Sussex, Falmer, East Sussex BN1 9RY;
<email>bsms2899@bsms.ac.uk</email>
or Dr Gilbert Bensimon, Département de Pharmacologie Clinique, Hôpital Pitié-Salpêtrière, 47 Bd de L'Hôpital, 75651 Paris Cedex 13, France;
<email>gilbert.bensimon@psl.aphp.fr</email>
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<p>GB and PNL contributed equally to this paper.</p>
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<title>Aim</title>
<p>To evaluate a standardised MRI acquisition protocol and a new image rating scale for disease severity in patients with progressive supranuclear palsy (PSP) and multiple systems atrophy (MSA) in a large multicentre study.</p>
</sec>
<sec>
<title>Methods</title>
<p>The MRI protocol consisted of two-dimensional sagittal and axial T1, axial PD, and axial and coronal T2 weighted acquisitions. The 32 item ordinal scale evaluated abnormalities within the basal ganglia and posterior fossa, blind to diagnosis. Among 760 patients in the study population (PSP=362, MSA=398), 627 had per protocol images (PSP=297, MSA=330). Intra-rater (n=60) and inter-rater (n=555) reliability were assessed through Cohen's statistic, and scale structure through principal component analysis (PCA) (n=441). Internal consistency and reliability were checked. Discriminant and predictive validity of extracted factors and total scores were tested for disease severity as per clinical diagnosis.</p>
</sec>
<sec>
<title>Results</title>
<p>Intra-rater and inter-rater reliability were acceptable for 25 (78%) of the items scored (≥0.41). PCA revealed four meaningful clusters of covarying parameters (factor (F) F1: brainstem and cerebellum; F2: midbrain; F3: putamen; F4: other basal ganglia) with good to excellent internal consistency (Cronbach α 0.75–0.93) and moderate to excellent reliability (intraclass coefficient: F1: 0.92; F2: 0.79; F3: 0.71; F4: 0.49). The total score significantly discriminated for disease severity or diagnosis; factorial scores differentially discriminated for disease severity according to diagnosis (PSP: F1–F2; MSA: F2–F3). The total score was significantly related to survival in PSP (p<0.0007) or MSA (p<0.0005), indicating good predictive validity.</p>
</sec>
<sec>
<title>Conclusions</title>
<p>The scale is suitable for use in the context of multicentre studies and can reliably and consistently measure MRI abnormalities in PSP and MSA.</p>
</sec>
<sec>
<title>Clinical Trial Registration Number</title>
<p>The study protocol was filed in the open clinical trial registry (
<ext-link ext-link-type="uri" xlink:href="http://www.clinicaltrials.gov">http://www.clinicaltrials.gov</ext-link>
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<ext-link ext-link-type="clintrialgov" xlink:href="NCT00211224">NCT00211224</ext-link>
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<namePart type="given">Albert C</namePart>
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<affiliation>Department of Neurology, University of Ulm, Baden-Wuerttemberg, Germany</affiliation>
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<namePart type="given">Gilbert</namePart>
<namePart type="family">Bensimon</namePart>
<affiliation>Département de Pharmacologie Clinique, Centre Hospitalo-Universitaire de la Pitié-Salpétrière, Assistance publique hôpitaux de Paris and UPMC Univ, Paris, France</affiliation>
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<name type="personal">
<namePart type="given">P Nigel</namePart>
<namePart type="family">Leigh</namePart>
<affiliation>Department of Neurology, Brighton and Sussex Medical School, Trafford Centre for Biomedical Research, University of Sussex Falmer, East Sussex, UK</affiliation>
<affiliation>Correspondence to Professor P N Leigh, Professor of Neurology, Brighton and Sussex Medical School, Trafford Centre for Biomedical Research University of Sussex, Falmer, East Sussex BN1 9RY; bsms2899@bsms.ac.ukor Dr Gilbert Bensimon, Département de Pharmacologie Clinique, Hôpital Pitié-Salpêtrière, 47 Bd de L'Hôpital, 75651 Paris Cedex 13, France; gilbert.bensimon@psl.aphp.fr</affiliation>
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<dateIssued encoding="w3cdtf">2011-09</dateIssued>
<dateCreated encoding="w3cdtf">2011-03-08</dateCreated>
<copyrightDate encoding="w3cdtf">2011</copyrightDate>
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<abstract>Aim To evaluate a standardised MRI acquisition protocol and a new image rating scale for disease severity in patients with progressive supranuclear palsy (PSP) and multiple systems atrophy (MSA) in a large multicentre study. Methods The MRI protocol consisted of two-dimensional sagittal and axial T1, axial PD, and axial and coronal T2 weighted acquisitions. The 32 item ordinal scale evaluated abnormalities within the basal ganglia and posterior fossa, blind to diagnosis. Among 760 patients in the study population (PSP=362, MSA=398), 627 had per protocol images (PSP=297, MSA=330). Intra-rater (n=60) and inter-rater (n=555) reliability were assessed through Cohen's statistic, and scale structure through principal component analysis (PCA) (n=441). Internal consistency and reliability were checked. Discriminant and predictive validity of extracted factors and total scores were tested for disease severity as per clinical diagnosis. Results Intra-rater and inter-rater reliability were acceptable for 25 (78%) of the items scored (≥0.41). PCA revealed four meaningful clusters of covarying parameters (factor (F) F1: brainstem and cerebellum; F2: midbrain; F3: putamen; F4: other basal ganglia) with good to excellent internal consistency (Cronbach α 0.75–0.93) and moderate to excellent reliability (intraclass coefficient: F1: 0.92; F2: 0.79; F3: 0.71; F4: 0.49). The total score significantly discriminated for disease severity or diagnosis; factorial scores differentially discriminated for disease severity according to diagnosis (PSP: F1–F2; MSA: F2–F3). The total score was significantly related to survival in PSP (p<0.0007) or MSA (p<0.0005), indicating good predictive validity. Conclusions The scale is suitable for use in the context of multicentre studies and can reliably and consistently measure MRI abnormalities in PSP and MSA. Clinical Trial Registration Number The study protocol was filed in the open clinical trial registry (http://www.clinicaltrials.gov) with ID No NCT00211224.</abstract>
<note type="footnotes">GB and PNL contributed equally to this paper.</note>
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<title>Journal of Neurology, Neurosurgery & Psychiatry</title>
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<title>J Neurol Neurosurg Psychiatry</title>
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<genre type="Journal">journal</genre>
<identifier type="ISSN">0022-3050</identifier>
<identifier type="eISSN">1468-330X</identifier>
<identifier type="PublisherID">jnnp</identifier>
<identifier type="PublisherID-hwp">jnnp</identifier>
<identifier type="PublisherID-nlm-ta">J Neurol Neurosurg Psychiatry</identifier>
<part>
<date>2011</date>
<detail type="volume">
<caption>vol.</caption>
<number>82</number>
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<detail type="issue">
<caption>no.</caption>
<number>9</number>
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<start>1025</start>
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<identifier type="istex">EC1A86B2145155C772D9465608300E551C02AD9F</identifier>
<identifier type="DOI">10.1136/jnnp.2010.214890</identifier>
<identifier type="href">jnnp-82-1025.pdf</identifier>
<identifier type="ArticleID">jnnp214890</identifier>
<identifier type="PMID">21386111</identifier>
<identifier type="local">jnnp;82/9/1025</identifier>
<accessCondition type="use and reproduction" contentType="open-access">This is an open-access article distributed under the terms of the Creative Commons Attribution Non-commercial License, which permits use, distribution, and reproduction in any medium, provided the original work is properly cited, the use is non commercial and is otherwise in compliance with the license. See: http://creativecommons.org/licenses/by-nc/2.0/ and http://creativecommons.org/licenses/by-nc/2.0/legalcode.</accessCondition>
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