TY - JOUR
T1 - Motor and psychiatric features in idiopathic blepharospasm
T2 - A data-driven cluster analysis
AU - Defazio, Giovanni
AU - Gigante, Angelo F.
AU - Hallett, Mark
AU - Berardelli, Alfredo
AU - Perlmutter, Joel S.
AU - Berman, Brian D.
AU - Jankovic, Joseph
AU - Bäumer, Tobias
AU - Comella, Cynthia
AU - Ercoli, Tommaso
AU - Ferrazzano, Gina
AU - Fox, Susan H.
AU - Kim, Han Joon
AU - Moukheiber, Emile Sami
AU - Richardson, Sarah Pirio
AU - Weissbach, Anne
AU - Jinnah, Hyder A.
N1 - Funding Information:
HJ Kim received travel grant support from the International Parkinson and Movement Disorder Society and Korean Movement Disorder Society and research grants support from the Institute for Information and Communications Technology Promotion , Seoul National University Hospital , New York University , and C-TRI.
Funding Information:
JS Perlmutter has received research grant support from NIH ( NCRR / NCATS , UNM CTSC KL21TR001448-01 and UL1TR001449 ) and Dystonia Coalition Projects ( NIH / NINDS / ORDR ) and has received publishing royalties from Springer; G. Berkmen reports no disclosure; B. Berman has received research grant support from the Dystonia Coalition (receives the majority of its support through NIH grant NS065701 from the Office of Rare Diseases Research in the National Center for Advancing Translational Science and National Institute of Neurological Disorders and Stroke ), Benign Essential Blepharospasm Research Foundation , Colorado Clinical & Translational Science Institute and Center for Neuroscience , Tools4Patient, Parkinson's Foundation , and Virginia Commonwealth University School of Medicine . He is on the medical advisory board of the Benign Essential Blepharospasm Research Foundation and the National Spasmodic Torticollis Association;
Funding Information:
S Pirio Richardson has received research support from the National Institutes of Health ( P20 GM109899 ; U54 NS116025 ), Department of Defense ( W81XWH-19-CTRR-CTA ); Pharma 2B; Addex and Aeon.
Funding Information:
C. Comella serves on the editorial board of Clinical Neuropharmacology and Sleep Medicine. She receives compensation/honoraria for services as a consultant or an advisory committee member: Acorda Therapeutics, Allergan, Inc; Lundbeck Ltd.; Merz Pharmaceuticals; Acadia Pharmaceuticals; Ipsen Pharmaceuticals, Jazz. Pharmaceuticals, Neurocrine Biosciences Inc., Revance Therapeutic, Sunovion., AEON Biopharma. She receives royalties from Cambridge University Press and Wolters Kluwer. She receives research support from the Parkinson's Disease Foundation .
Funding Information:
A Weissbach received funding from the Else Kröner-Fresenius Foundation ( EKFS, 2018_A55 ) and the German Research Foundation ( DFG, WE 5919/2-1 ).
Funding Information:
Susan Fox has received clinic support from the Edmond J Safra Foundation for Parkinson Research; Parkinson Foundation and the Toronto Western and General Foundation ; salary from UHN Dept of Medicine Practice Plan; research Funding from Michael J Fox Foundation for Parkinson Research, NIH (Dystonia Coalition); Parkinson Canada; honoraria from the International Parkinson and Movement Disorder Society; Consultancy/Speaker fees from Alexion; Bial, Merck; Pharma 2B; Sunovion; Teva; Paladin; royalties from Oxford University Press . She was site PI for Clinical Trials for Alexion, Biotie, Eisai, Pharma2B, Revance.
Funding Information:
J Jankovic has received research/training funding from AbbVie Inc ; Acadia Pharmaceuticals ; Allergan , Inc; Biotek; Cerevel Therapeutics; CHDI Foundation ; Dystonia Coalition; Emalex Biosciences, Inc; F. Hoffmann-La Roche Ltd; Huntington Study Group; Medtronic Neuromodulation; Merz Pharmaceuticals; Michael J Fox Foundation for Parkinson Research; National Institutes of Health; Neuraly, Inc.; Neurocrine Biosciences; Parkinson's Foundation; Parkinson Study Group; Prilenia Therapeutics; Revance Therapeutics, Inc; Teva Pharmaceutical Industries Ltd. Dr. Jankovic has served as a consultant for Aeon BioPharma; Nuvelution Pharma, Inc; Teva Pharmaceutical Industries Ltd. Dr. Jankovic has received royalties from Cambridge; Elsevier; Medlink: Neurology; Lippincott Williams and Wilkins; Wiley-Blackwell;
Funding Information:
This work was supported in part by NIH grant NS119831 and NIH grants to The Dystonia Coalition ( NS065701 , TR001456 , NS116025 ) which is part of the National Institutes of Health (NIH) Rare Disease Clinical Research Network (RDCRN) , supported by the Office of Rare Diseases Research (ORDR) at the Rare Disease Clinical Research Network (RDCRN) , and the National Institute of Neurological Diseases and Stroke (NINDS) .
Funding Information:
Brian Berman received research grant support within last 12 months from the Dystonia Coalition (through NIH grant NS065701 from the Office of Rare Diseases Research in the National Center for Advancing Translational Science and National Institute of Neurological Disorders and Stroke ), Parkinson's Foundation , VCU School of Medicine , the Administration for Community Living and Dystonia Medical Research Foundation ; honoraria from the MedLink Corporation and the International Parkinson and Movement Disorder Society ; and consultancies from AbbVie Inc.
Funding Information:
HA Jinnah has active or recent grant support from the US government ( National Institutes of Health ), private philanthropic organizations (Cure Dystonia Now), and industry ( Revance Therapeutics, Inc. ). Dr. Jinnah has also served on advisory boards or as a consultant for Addex, Allergan, CoA Therapeutics, Cavion Therapeutics, EnePharmaceuticals, Ipsen, Retrophin, Revance, and Takaha Pharmaceuticals. He has received honoraria or stipends for lectures or administrative work from the International Parkinson's Disease and Movement Disorders Society. Dr. Jinnah serves on the Scientific Advisory Boards for several private foundations including the Benign Essential Blepharospasm Research Foundation, Cure Dystonia Now, the Dystonia Medical Research Foundation, the Tourette Association of America, and Tyler's Hope for a Cure. He also is principal investigator for the Dystonia Coalition , which has received the majority of its support through the NIH (grants NS116025, NS065701 from the National Institutes of Neurological Disorders and Stroke TR 001456 from the Office of Rare Diseases Research at the National Center for Advancing Translational Sciences ). The Dystonia Coalition has received additional material or administrative support from industry sponsors ( Allergan Inc. and Merz Pharmaceuticals ) as well as private foundations ( The Benign Essential Blepharospasm Foundation , Cure Dystonia Now, The Dystonia Medical Research Foundation , and The National Spasmodic Dysphonia Association ).
Publisher Copyright:
© 2022 Elsevier Ltd
PY - 2022/11
Y1 - 2022/11
N2 - Introduction: Idiopathic blepharospasm is a clinically heterogeneous dystonia also characterized by non motor symptoms. Methods: We used a k-means cluster analysis to assess 188 patients with idiopathic blepharospasm in order to identify relatively homogeneous subpopulations of patients, using a set of motor and psychiatric variables to generate the cluster solution. Results: Blepharospasm patients reached higher scores on scales assessing depressive- and anxiety-related disorders than healthy/disease controls. Cluster analysis suggested the existence of three groups of patients that differed by type of spasms, overall motor severity, and presence/severity of psychiatric problems. The greater severity of motor symptoms was observed in Group 1, the least severity in Group 3, while the severity of blepharospasm in Group 2 was between that observed in Groups 1 and 3. The three motor subtypes also differed by psychiatric features: the lowest severity of psychiatric symptoms was observed in the group with least severe motor symptoms (group 3), while the highest psychiatric severity scores were observed in group 2 that carried intermediate motor severity rather than in the group with more severe motor symptoms (group 1). The three groups did not differ by disease duration, age of onset, sex or other clinical features. Conclusions: The present study suggests that blepharospasm patients may be classified in different subtypes according to the type of spasms, overall motor severity and presence/severity of depressive symptoms and anxiety.
AB - Introduction: Idiopathic blepharospasm is a clinically heterogeneous dystonia also characterized by non motor symptoms. Methods: We used a k-means cluster analysis to assess 188 patients with idiopathic blepharospasm in order to identify relatively homogeneous subpopulations of patients, using a set of motor and psychiatric variables to generate the cluster solution. Results: Blepharospasm patients reached higher scores on scales assessing depressive- and anxiety-related disorders than healthy/disease controls. Cluster analysis suggested the existence of three groups of patients that differed by type of spasms, overall motor severity, and presence/severity of psychiatric problems. The greater severity of motor symptoms was observed in Group 1, the least severity in Group 3, while the severity of blepharospasm in Group 2 was between that observed in Groups 1 and 3. The three motor subtypes also differed by psychiatric features: the lowest severity of psychiatric symptoms was observed in the group with least severe motor symptoms (group 3), while the highest psychiatric severity scores were observed in group 2 that carried intermediate motor severity rather than in the group with more severe motor symptoms (group 1). The three groups did not differ by disease duration, age of onset, sex or other clinical features. Conclusions: The present study suggests that blepharospasm patients may be classified in different subtypes according to the type of spasms, overall motor severity and presence/severity of depressive symptoms and anxiety.
KW - Anxiety
KW - Blepharospasm
KW - Cluster analysis
KW - Depression
UR - http://www.scopus.com/inward/record.url?scp=85140304319&partnerID=8YFLogxK
U2 - 10.1016/j.parkreldis.2022.10.008
DO - 10.1016/j.parkreldis.2022.10.008
M3 - Article
C2 - 36306537
AN - SCOPUS:85140304319
SN - 1353-8020
VL - 104
SP - 94
EP - 98
JO - Parkinsonism and Related Disorders
JF - Parkinsonism and Related Disorders
ER -