Functional dystonia: A case‐control study and risk prediction algorithm

Annals of Clinical and Translational Neurology - Tập 8 Số 4 - Trang 732-748 - 2021
Christopher D. Stephen1,2, David L. Perez3,4,2, Lori B. Chibnik5,6, Nutan Sharma1,2
1Dystonia Center and Movement Disorders Unit, Department of Neurology Massachusetts General Hospital and Harvard Medical School Boston Massachusetts USA
2Functional Neurological Disorder Research Program, Department of Neurology Massachusetts General Hospital and Harvard Medical School Boston Massachusetts USA
3Cognitive Behavioral Neurology Division, Department of Neurology Massachusetts General Hospital and Harvard Medical School Boston Massachusetts USA
4Division of Neuropsychiatry, Department of Psychiatry Massachusetts General Hospital and Harvard Medical School Boston Massachusetts USA
5Biostatistics Center, Department of Neurology Massachusetts General Hospital and Harvard Medical School Boston Massachusetts USA
6Department of Epidemiology Harvard T. H. Chan School of Public Health Boston Massachusetts USA

Tóm tắt

AbstractObjectiveFunctional dystonia (FD) is a disabling and diagnostically challenging functional movement disorder (FMD). We sought to identify historical predictors of FD vs. other primary dystonias (ODs) and develop a practical prediction algorithm to guide neurologists.Methods1475 consecutive new patient medical records were reviewed at an adult/pediatric tertiary‐referral dystonia clinic from 2005 to 2017. Ninety‐nine met criteria for clinically established FD (85 adults and 14 pediatric), paired with 99 age/dystonia distribution‐matched OD. Univariate and multivariate regression analyses were performed to identify predictors of FD and disability. We formed a prediction algorithm, assessed using the area under the receiver operating curve (AUC).ResultsMultivariate logistic regression analysis investigating independent predictors of FD (P < 0.001) followed by development of a prediction algorithm showed that the most robust predictors included abrupt onset, spontaneous resolution/recurrence, pain, cognitive complaints, being on or pursuing disability, lifetime mood/anxiety disorder, comorbid functional somatic disorders, and having ≥3 medication allergies. The prediction algorithm had utility for both adult and pediatric FD, with excellent sensitivity/specificity (89%/92%) and an area under the curve (AUC) 0.95 (0.92‐0.98). Greater disability (modified Rankin Scale) independently correlated with a number of functional examination features, unemployment/not attending school, number of medication allergies, and younger age of presentation. FD patients were high health‐care utilizers and were more frequently prescribed opiates/opioids and benzodiazepines (P < 0.003).InterpretationThis case‐control study provides an algorithm to guide clinicians in gauging their index of suspicion for a FD, with diagnostic confirmation subsequently informed by neurological examination. While this algorithm requires prospective validation, health‐care utilization data underscore the importance and need for more research in FD.

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