Respondek, Gesine, Kurz, Carolin, Arzberger, Thomas, Compta, Yaroslau ORCID: 0000-0001-6443-0104, Englund, Elisabet ORCID: 0000-0002-2708-2443, Ferguson, Leslie W., Gelpi, Ellen, Giese, Armin ORCID: 0000-0002-8238-4102, Irwin, David J., Meissner, Wassilios G., Nilsson, Christer, Pantelyat, Alexander, Rajput, Alex, van Swieten, John C., Troakes, Claire ORCID: 0000-0002-1790-7376, Josephs, Keith A., Lang, Anthony E., Mollenhauer, Brit, Mueller, Ulrich, Whitwell, Jennifer L., Antonini, Angelo ORCID: 0000-0003-1040-2807, Bhatia, Kailash P., Bordelon, Yvette, Corvol, Jean-Christophe ORCID: 0000-0001-7325-0199, Colosimo, Carlo ORCID: 0000-0002-2216-3973, Dodel, Richard, Grossman, Murray, Kassubek, Jan ORCID: 0000-0002-7106-9270, Krismer, Florian ORCID: 0000-0002-4493-5073, Levin, Johannes ORCID: 0000-0001-5092-4306, Lorenzl, Stefan, Morris, Huw, Nestor, Peter ORCID: 0000-0002-5860-5921, Oertel, Wolfgang H., Rabinovici, Gil D., Rowe, James B., van Eimeren, Thilo, Wenning, Gregor K., Boxer, Adam, Golbe, Lawrence I., Litvan, Irene ORCID: 0000-0002-3485-3445, Stamelou, Maria and Hoeglinger, Guenter U. (2017). Which Ante Mortem Clinical Features Predict Progressive Supranuclear Palsy Pathology? Mov. Disord., 32 (7). S. 995 - 1006. HOBOKEN: WILEY. ISSN 1531-8257

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Abstract

Background: Progressive supranuclear palsy (PSP) is a neuropathologically defined disease presenting with a broad spectrum of clinical phenotypes. Objective: To identify clinical features and investigations that predict or exclude PSP pathology during life, aiming at an optimization of the clinical diagnostic criteria for PSP. Methods: We performed a systematic review of the literature published since 1996 to identify clinical features and investigations that may predict or exclude PSP pathology. We then extracted standardized data from clinical charts of patients with pathologically diagnosed PSP and relevant disease controls and calculated the sensitivity, specificity, and positive predictive value of key clinical features for PSP in this cohort. Results: Of 4166 articles identified by the database inquiry, 269 met predefined standards. The literature review identified clinical features predictive of PSP, including features of the following 4 functional domains: ocular motor dysfunction, postural instability, akinesia, and cognitive dysfunction. No biomarker or genetic feature was found reliably validated to predict definite PSP. High-quality original natural history data were available from 206 patients with pathologically diagnosed PSP and from 231 pathologically diagnosed disease controls (54 corticobasal degeneration, 51 multiple system atrophy with predominant parkinsonism, 53 Parkinson's disease, 73 behavioral variant frontotemporal dementia). We identified clinical features that predicted PSP pathology, including phenotypes other than Richardson's syndrome, with varying sensitivity and specificity. Conclusions: Our results highlight the clinical variability of PSP and the high prevalence of phenotypes other than Richardson's syndrome. The features of variant phenotypes with high specificity and sensitivity should serve to optimize clinical diagnosis of PSP. (C) 2017 International Parkinson and Movement Disorder Society

Item Type: Journal Article
Creators:
CreatorsEmailORCIDORCID Put Code
Respondek, GesineUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Kurz, CarolinUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Arzberger, ThomasUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Compta, YaroslauUNSPECIFIEDorcid.org/0000-0001-6443-0104UNSPECIFIED
Englund, ElisabetUNSPECIFIEDorcid.org/0000-0002-2708-2443UNSPECIFIED
Ferguson, Leslie W.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Gelpi, EllenUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Giese, ArminUNSPECIFIEDorcid.org/0000-0002-8238-4102UNSPECIFIED
Irwin, David J.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Meissner, Wassilios G.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Nilsson, ChristerUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Pantelyat, AlexanderUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Rajput, AlexUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
van Swieten, John C.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Troakes, ClaireUNSPECIFIEDorcid.org/0000-0002-1790-7376UNSPECIFIED
Josephs, Keith A.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Lang, Anthony E.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Mollenhauer, BritUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Mueller, UlrichUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Whitwell, Jennifer L.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Antonini, AngeloUNSPECIFIEDorcid.org/0000-0003-1040-2807UNSPECIFIED
Bhatia, Kailash P.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Bordelon, YvetteUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Corvol, Jean-ChristopheUNSPECIFIEDorcid.org/0000-0001-7325-0199UNSPECIFIED
Colosimo, CarloUNSPECIFIEDorcid.org/0000-0002-2216-3973UNSPECIFIED
Dodel, RichardUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Grossman, MurrayUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Kassubek, JanUNSPECIFIEDorcid.org/0000-0002-7106-9270UNSPECIFIED
Krismer, FlorianUNSPECIFIEDorcid.org/0000-0002-4493-5073UNSPECIFIED
Levin, JohannesUNSPECIFIEDorcid.org/0000-0001-5092-4306UNSPECIFIED
Lorenzl, StefanUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Morris, HuwUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Nestor, PeterUNSPECIFIEDorcid.org/0000-0002-5860-5921UNSPECIFIED
Oertel, Wolfgang H.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Rabinovici, Gil D.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Rowe, James B.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
van Eimeren, ThiloUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Wenning, Gregor K.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Boxer, AdamUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Golbe, Lawrence I.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Litvan, IreneUNSPECIFIEDorcid.org/0000-0002-3485-3445UNSPECIFIED
Stamelou, MariaUNSPECIFIEDUNSPECIFIEDUNSPECIFIED
Hoeglinger, Guenter U.UNSPECIFIEDUNSPECIFIEDUNSPECIFIED
URN: urn:nbn:de:hbz:38-226777
DOI: 10.1002/mds.27034
Journal or Publication Title: Mov. Disord.
Volume: 32
Number: 7
Page Range: S. 995 - 1006
Date: 2017
Publisher: WILEY
Place of Publication: HOBOKEN
ISSN: 1531-8257
Language: English
Faculty: Unspecified
Divisions: Unspecified
Subjects: no entry
Uncontrolled Keywords:
KeywordsLanguage
RICHARDSON-OLSZEWSKI SYNDROME; FRONTOTEMPORAL LOBAR DEGENERATION; NINDS NEUROPATHOLOGIC CRITERIA; MULTIPLE SYSTEM ATROPHY; CORTICOBASAL DEGENERATION; NATURAL-HISTORY; DIAGNOSTIC-CRITERIA; FAMILIAL AGGREGATION; NONFLUENT APHASIA; PURE AKINESIAMultiple languages
Clinical NeurologyMultiple languages
Refereed: Yes
URI: http://kups.ub.uni-koeln.de/id/eprint/22677

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