Diagnostic criteria for blepharospasm: A multicenter international study

被引:22
作者
Defazio, Giovanni [1 ]
Jinnah, Hyder A. [2 ]
Berardelli, Alfredo [3 ,4 ]
Perlmutter, Joel S. [5 ]
Berkmen, Gamze Kilic [2 ]
Berman, Brian D. [6 ]
Jankovic, Joseph [7 ,8 ]
Baeumer, Tobias [9 ]
Comella, Cynthia [10 ]
Cotton, Adam C. [2 ]
Ercoli, Tommaso [1 ]
Ferrazzano, Gina [3 ,4 ]
Fox, Susan [11 ]
Kim, Han-Joon [12 ,13 ]
Moukheiber, Emile Sami [14 ]
Richardson, Sarah Pirio [15 ]
Weissbach, Anne [9 ,16 ]
Wrigth, Laura J. [5 ]
Hallett, Mark [17 ]
机构
[1] Univ Cagliari, Dept Med Sci & Publ Hlth, Cagliari, Italy
[2] Emory Univ, Dept Neurol & Human Genet, Atlanta, GA 30322 USA
[3] Sapienza Univ Rome, Rome, Italy
[4] IRCSS NEUROMED, Pozzilli, IS, Italy
[5] Washington Univ, St Louis, MO 63110 USA
[6] Virginia Commonwealth Univ, Richmond, VA USA
[7] Baylor Coll Med, Parkinsons Dis Ctr, Houston, TX 77030 USA
[8] Baylor Coll Med, Dept Neurol, Movement Disorders Clin, Houston, TX 77030 USA
[9] Univ Lubeck, Inst Syst Motor Sci, Lubeck, Germany
[10] Rush Univ, Med Ctr, New Philadelphia, OH USA
[11] Univ Toronto, Toronto Western Hosp, Toronto, ON, Canada
[12] Seoul Natl Univ Hosp, Dept Neurol, Seoul, South Korea
[13] Seoul Natl Univ Hosp, Movement Disorder Ctr, Seoul, South Korea
[14] Johns Hopkins Univ, Dept Neurol, Baltimore, MD 21218 USA
[15] Univ New Mexico, Dept Neurol, Albuquerque, NM USA
[16] Univ Lubeck, Inst Neurogenet, Lubeck, Germany
[17] NINDS, Human Motor Control Sect, NIH, Bethesda, MD USA
关键词
Blepharospasm; Dystonia; Diagnosis; DYSTONIA; PHENOMENOLOGY; AGREEMENT; TRICKS;
D O I
10.1016/j.parkreldis.2021.09.004
中图分类号
R74 [神经病学与精神病学];
学科分类号
摘要
Background: There are no widely accepted criteria to aid the physician in diagnosing BSP. Objective: To validate recently proposed diagnostic criteria for blepharospasm in a larger and geographically diverse population and to develop a screening system for blepharospasm. Methods: Video-recordings from 211 blepharospasm patients and 166 healthy/disease controls were examined by 8 raters. Agreement for presence of orbicularis oculi spasms, sensory trick, and increased blinking was measured by k statistics. Inability to voluntarily suppress the spasms was asked by the examiner but not captured in the video. Patients/controls were also requested to fill a self-administered questionnaire addressing relevant blepharospasm clinical aspects. The diagnosis at each site was the gold standard for sensitivity/specificity. Results: All the study items yielded satisfactory inter/intra-observer agreement. Combination of items rather than each item alone reached satisfactory sensitivity/specificity. The combined algorithm started with recognition of spasms followed by sensory trick. In the absence of a sensory trick, including "increased blinking" or "inability to voluntarily suppress the spasms" or both items yielded 88-92% sensitivity and 79-83% specificity. No single question of the questionnaire yielded high sensitivity/specificity. Serial application of the questionnaire to our blepharospasm and control subjects and subsequent clinical examination of subjects screening positive by the validated diagnostic algorithms yielded 78-81% sensitivity and 83-91% specificity. Conclusion: These results support the use of proposed diagnostic criteria in multi-ethnic, multi-center cohorts. We also propose a case-finding procedure to screen blepharospasm in a given population with less effort than would be required by examination of all subjects.
引用
收藏
页码:109 / 114
页数:6
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