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

Identifieur interne : 001840 ( Main/Merge ); précédent : 001839; suivant : 001841

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

Auteurs : Yan Rolland [France] ; Marc Vérin [France] ; Christine A. Payan [France] ; Simon Duchesne [Canada] ; Eduard Kraft [Allemagne] ; Till K. Hauser [Allemagne] ; Josef Jarosz [Royaume-Uni] ; Neil Deasy [Royaume-Uni] ; Luc Defevbre [France] ; Christine Delmaire [France] ; Didier Dormont [France] ; Albert C. Ludolph [Allemagne] ; Gilbert Bensimon [France] ; P Nigel Leigh [Royaume-Uni, France]

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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<name sortKey="Kraft, Eduard" sort="Kraft, Eduard" uniqKey="Kraft E" first="Eduard" last="Kraft">Eduard Kraft</name>
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<wicri:regionArea>Department of Neuroradiology, University of Tübingen, Tübingen</wicri:regionArea>
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<name sortKey="Jarosz, Josef" sort="Jarosz, Josef" uniqKey="Jarosz J" first="Josef" last="Jarosz">Josef Jarosz</name>
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<name sortKey="Dormont, Didier" sort="Dormont, Didier" uniqKey="Dormont D" first="Didier" last="Dormont">Didier Dormont</name>
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<wicri:regionArea>Département de Neuroradiologie, Hôpital de la Pitié-Salpétrière, Assistance Publique Hôpitaux de Paris and UPMC Univ, Paris</wicri:regionArea>
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<region type="region">Île-de-France</region>
<region type="old region">Île-de-France</region>
<settlement type="city">Paris</settlement>
</placeName>
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<name sortKey="Ludolph, Albert C" sort="Ludolph, Albert C" uniqKey="Ludolph A" first="Albert C" last="Ludolph">Albert C. Ludolph</name>
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<country xml:lang="fr">Allemagne</country>
<wicri:regionArea>Department of Neurology, University of Ulm, Baden-Wuerttemberg</wicri:regionArea>
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<settlement type="city">Ulm</settlement>
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<name sortKey="Bensimon, Gilbert" sort="Bensimon, Gilbert" uniqKey="Bensimon G" first="Gilbert" last="Bensimon">Gilbert Bensimon</name>
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<country xml:lang="fr">France</country>
<wicri:regionArea>Département de Pharmacologie Clinique, Centre Hospitalo-Universitaire de la Pitié-Salpétrière, Assistance publique hôpitaux de Paris and UPMC Univ, Paris</wicri:regionArea>
<placeName>
<region type="region">Île-de-France</region>
<region type="old region">Île-de-France</region>
<settlement type="city">Paris</settlement>
</placeName>
</affiliation>
</author>
<author>
<name sortKey="Leigh, P Nigel" sort="Leigh, P Nigel" uniqKey="Leigh P" first="P Nigel" last="Leigh">P Nigel Leigh</name>
<affiliation wicri:level="1">
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>Department of Neurology, Brighton and Sussex Medical School, Trafford Centre for Biomedical Research, University of Sussex Falmer, East Sussex</wicri:regionArea>
<wicri:noRegion>East Sussex</wicri:noRegion>
</affiliation>
<affiliation wicri:level="4">
<country wicri:rule="url">France</country>
<wicri:regionArea>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</wicri:regionArea>
<orgName type="university">Université du Sussex</orgName>
<placeName>
<settlement type="city">Brighton</settlement>
<settlement type="town">Falmer</settlement>
<region type="nation">Angleterre</region>
<region nuts="2" type="region">Sussex de l'Est</region>
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<title level="j">Journal of Neurology, Neurosurgery & Psychiatry</title>
<title level="j" type="abbrev">J Neurol Neurosurg Psychiatry</title>
<idno type="ISSN">0022-3050</idno>
<idno type="eISSN">1468-330X</idno>
<imprint>
<publisher>BMJ Publishing Group Ltd</publisher>
<date type="published" when="2011-09">2011-09</date>
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<biblScope unit="issue">9</biblScope>
<biblScope unit="page" from="1025">1025</biblScope>
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<idno type="ISSN">0022-3050</idno>
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<idno type="DOI">10.1136/jnnp.2010.214890</idno>
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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>
</front>
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<name sortKey="Rolland, Yan" sort="Rolland, Yan" uniqKey="Rolland Y" first="Yan" last="Rolland">Yan Rolland</name>
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<name sortKey="Verin, Marc" sort="Verin, Marc" uniqKey="Verin M" first="Marc" last="Vérin">Marc Vérin</name>
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<nlm:aff id="aff2">Department of Neurology, Pontchaillou University Hospital, Rennes, France</nlm:aff>
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<wicri:regionArea>Department of Neurology, Pontchaillou University Hospital, Rennes</wicri:regionArea>
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<name sortKey="Duchesne, Simon" sort="Duchesne, Simon" uniqKey="Duchesne S" first="Simon" last="Duchesne">Simon Duchesne</name>
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<nlm:aff id="aff4">Department of Radiology and Robert Giffard Laval University Research Centre, Laval University, Quebec City, Canada</nlm:aff>
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<name sortKey="Jarosz, Josef" sort="Jarosz, Josef" uniqKey="Jarosz J" first="Josef" last="Jarosz">Josef Jarosz</name>
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<name sortKey="Deasy, Neil" sort="Deasy, Neil" uniqKey="Deasy N" first="Neil" last="Deasy">Neil Deasy</name>
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<nlm:aff id="aff10">Department of Neuroradiology, Lille University, Salengro Hospital, Cedex Lille, France</nlm:aff>
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<wicri:regionArea>Department of Neuroradiology, Lille University, Salengro Hospital, Cedex Lille</wicri:regionArea>
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<name sortKey="Ludolph, Albert C" sort="Ludolph, Albert C" uniqKey="Ludolph A" first="Albert C" last="Ludolph">Albert C. Ludolph</name>
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<region type="region">Île-de-France</region>
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<name sortKey="Leigh, P Nigel" sort="Leigh, P Nigel" uniqKey="Leigh P" first="P Nigel" last="Leigh">P Nigel Leigh</name>
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<nlm:aff id="aff7">Department of Neurology, Brighton and Sussex Medical School, Trafford Centre for Biomedical Research, University of Sussex Falmer, East Sussex, UK</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>Department of Neurology, Brighton and Sussex Medical School, Trafford Centre for Biomedical Research, University of Sussex Falmer, East Sussex</wicri:regionArea>
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<nlm:aff id="aff2">Department of Neurology, Pontchaillou University Hospital, Rennes, France</nlm:aff>
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</author>
<author>
<name sortKey="Duchesne, Simon" sort="Duchesne, Simon" uniqKey="Duchesne S" first="Simon" last="Duchesne">Simon Duchesne</name>
<affiliation wicri:level="1">
<nlm:aff id="aff4">Department of Radiology and Robert Giffard Laval University Research Centre, Laval University, Quebec City, Canada</nlm:aff>
<country xml:lang="fr">Canada</country>
<wicri:regionArea>Department of Radiology and Robert Giffard Laval University Research Centre, Laval University, Quebec City</wicri:regionArea>
<wicri:noRegion>Quebec City</wicri:noRegion>
</affiliation>
</author>
<author>
<name sortKey="Kraft, Eduard" sort="Kraft, Eduard" uniqKey="Kraft E" first="Eduard" last="Kraft">Eduard Kraft</name>
<affiliation wicri:level="4">
<nlm:aff id="aff5">Department of Neurology, University of Ulm, Baden-Wuerttemberg, Germany</nlm:aff>
<country xml:lang="fr">Allemagne</country>
<wicri:regionArea>Department of Neurology, University of Ulm, Baden-Wuerttemberg</wicri:regionArea>
<wicri:noRegion>Baden-Wuerttemberg</wicri:noRegion>
<orgName type="university">Université d'Ulm</orgName>
<placeName>
<settlement type="city">Ulm</settlement>
<region type="land" nuts="1">Bade-Wurtemberg</region>
<region type="district" nuts="2">District de Tübingen</region>
</placeName>
</affiliation>
</author>
<author>
<name sortKey="Hauser, Till K" sort="Hauser, Till K" uniqKey="Hauser T" first="Till K" last="Hauser">Till K. Hauser</name>
<affiliation wicri:level="3">
<nlm:aff id="aff6">Department of Neuroradiology, University of Tübingen, Tübingen, Germany</nlm:aff>
<country xml:lang="fr">Allemagne</country>
<wicri:regionArea>Department of Neuroradiology, University of Tübingen, Tübingen</wicri:regionArea>
<placeName>
<region type="land" nuts="1">Bade-Wurtemberg</region>
<region type="district" nuts="2">District de Tübingen</region>
<settlement type="city">Tübingen</settlement>
</placeName>
</affiliation>
</author>
<author>
<name sortKey="Jarosz, Josef" sort="Jarosz, Josef" uniqKey="Jarosz J" first="Josef" last="Jarosz">Josef Jarosz</name>
<affiliation wicri:level="1">
<nlm:aff id="aff7">Department of Neurology, Brighton and Sussex Medical School, Trafford Centre for Biomedical Research, University of Sussex Falmer, East Sussex, UK</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>Department of Neurology, Brighton and Sussex Medical School, Trafford Centre for Biomedical Research, University of Sussex Falmer, East Sussex</wicri:regionArea>
<wicri:noRegion>East Sussex</wicri:noRegion>
</affiliation>
</author>
<author>
<name sortKey="Deasy, Neil" sort="Deasy, Neil" uniqKey="Deasy N" first="Neil" last="Deasy">Neil Deasy</name>
<affiliation wicri:level="3">
<nlm:aff id="aff8">MRC Centre for Neurodegeneration Research, King's College London, Institute of Psychiatry, Department of Clinical Neuroscience, London, UK</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>MRC Centre for Neurodegeneration Research, King's College London, Institute of Psychiatry, Department of Clinical Neuroscience, London</wicri:regionArea>
<placeName>
<settlement type="city">Londres</settlement>
<region type="country">Angleterre</region>
<region type="région" nuts="1">Grand Londres</region>
</placeName>
</affiliation>
</author>
<author>
<name sortKey="Defevbre, Luc" sort="Defevbre, Luc" uniqKey="Defevbre L" first="Luc" last="Defevbre">Luc Defevbre</name>
<affiliation wicri:level="1">
<nlm:aff id="aff9">Department of Neurology Movement Disorders, Lille University, Salengro Hospital, Cedex Lille, France</nlm:aff>
<country xml:lang="fr">France</country>
<wicri:regionArea>Department of Neurology Movement Disorders, Lille University, Salengro Hospital, Cedex Lille</wicri:regionArea>
<wicri:noRegion>Cedex Lille</wicri:noRegion>
<wicri:noRegion>Cedex Lille</wicri:noRegion>
</affiliation>
</author>
<author>
<name sortKey="Delmaire, Christine" sort="Delmaire, Christine" uniqKey="Delmaire C" first="Christine" last="Delmaire">Christine Delmaire</name>
<affiliation wicri:level="1">
<nlm:aff id="aff10">Department of Neuroradiology, Lille University, Salengro Hospital, Cedex Lille, France</nlm:aff>
<country xml:lang="fr">France</country>
<wicri:regionArea>Department of Neuroradiology, Lille University, Salengro Hospital, Cedex Lille</wicri:regionArea>
<wicri:noRegion>Cedex Lille</wicri:noRegion>
<wicri:noRegion>Cedex Lille</wicri:noRegion>
</affiliation>
</author>
<author>
<name sortKey="Dormont, Didier" sort="Dormont, Didier" uniqKey="Dormont D" first="Didier" last="Dormont">Didier Dormont</name>
<affiliation wicri:level="3">
<nlm:aff id="aff11">Département de Neuroradiologie, Hôpital de la Pitié-Salpétrière, Assistance Publique Hôpitaux de Paris and UPMC Univ, Paris, France</nlm:aff>
<country xml:lang="fr">France</country>
<wicri:regionArea>Département de Neuroradiologie, Hôpital de la Pitié-Salpétrière, Assistance Publique Hôpitaux de Paris and UPMC Univ, Paris</wicri:regionArea>
<placeName>
<region type="region">Île-de-France</region>
<region type="old region">Île-de-France</region>
<settlement type="city">Paris</settlement>
</placeName>
</affiliation>
</author>
<author>
<name sortKey="Ludolph, Albert C" sort="Ludolph, Albert C" uniqKey="Ludolph A" first="Albert C" last="Ludolph">Albert C. Ludolph</name>
<affiliation wicri:level="4">
<nlm:aff id="aff5">Department of Neurology, University of Ulm, Baden-Wuerttemberg, Germany</nlm:aff>
<country xml:lang="fr">Allemagne</country>
<wicri:regionArea>Department of Neurology, University of Ulm, Baden-Wuerttemberg</wicri:regionArea>
<wicri:noRegion>Baden-Wuerttemberg</wicri:noRegion>
<orgName type="university">Université d'Ulm</orgName>
<placeName>
<settlement type="city">Ulm</settlement>
<region type="land" nuts="1">Bade-Wurtemberg</region>
<region type="district" nuts="2">District de Tübingen</region>
</placeName>
</affiliation>
</author>
<author>
<name sortKey="Bensimon, Gilbert" sort="Bensimon, Gilbert" uniqKey="Bensimon G" first="Gilbert" last="Bensimon">Gilbert Bensimon</name>
<affiliation wicri:level="3">
<nlm:aff id="aff3">Département de Pharmacologie Clinique, Centre Hospitalo-Universitaire de la Pitié-Salpétrière, Assistance publique hôpitaux de Paris and UPMC Univ, Paris, France</nlm:aff>
<country xml:lang="fr">France</country>
<wicri:regionArea>Département de Pharmacologie Clinique, Centre Hospitalo-Universitaire de la Pitié-Salpétrière, Assistance publique hôpitaux de Paris and UPMC Univ, Paris</wicri:regionArea>
<placeName>
<region type="region">Île-de-France</region>
<region type="old region">Île-de-France</region>
<settlement type="city">Paris</settlement>
</placeName>
</affiliation>
</author>
<author>
<name sortKey="Leigh, P Nigel" sort="Leigh, P Nigel" uniqKey="Leigh P" first="P Nigel" last="Leigh">P Nigel Leigh</name>
<affiliation wicri:level="1">
<nlm:aff id="aff7">Department of Neurology, Brighton and Sussex Medical School, Trafford Centre for Biomedical Research, University of Sussex Falmer, East Sussex, UK</nlm:aff>
<country xml:lang="fr">Royaume-Uni</country>
<wicri:regionArea>Department of Neurology, Brighton and Sussex Medical School, Trafford Centre for Biomedical Research, University of Sussex Falmer, East Sussex</wicri:regionArea>
<wicri:noRegion>East Sussex</wicri:noRegion>
</affiliation>
</author>
</analytic>
<series>
<title level="j">Journal of Neurology, Neurosurgery, and Psychiatry</title>
<idno type="ISSN">0022-3050</idno>
<idno type="eISSN">1468-330X</idno>
<imprint>
<date when="2011">2011</date>
</imprint>
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<textClass></textClass>
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</teiHeader>
<front>
<div type="abstract" xml:lang="en">
<sec>
<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>
) with ID No NCT00211224.</p>
</sec>
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