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Multidisciplinary inpatient rehabilitation for Functional Movement Disorders: A prospective study with long term follow up

      Highlights

      • Functional movement disorder severity declines after acute inpatient rehabilitation.
      • Anxiety, depression, and somatization decrease following acute rehabilitation.
      • Long-term recurrence of physical symptoms often follows successful rehabilitation.
      • Despite initial abatement, recurrence of psychiatric illness is common at one-year.

      Abstract

      Introduction

      Functional Movement Disorders (FMDs) are challenging to treat. We assessed the effect of multidisciplinary inpatient rehabilitation, involving motor retraining, psychotherapy and psychotropic medication on FMD patient function and maintenance of improvement after one year.

      Methods

      FMD patients in a movement disorders clinic were referred for inpatient rehabilitation. Baseline, discharge and one year follow-up measures included: Clinical Global Impression (CGI-severity, CGI-change); Depression and Somatic Symptom Scale (DSSS); Generalized Anxiety Disorder-7 (GAD-7); Patient Health Questionnaire-9 (PHQ-9); Post-traumatic stress disorder check-list for DSM-5 (PCL-5). Outcomes were analyzed with non-parametric models.

      Results

      Seventeen patients completed rehabilitation. Thirteen completed one-year follow-up. Median CGI-severity was “markedly ill.” At discharge, movement disorder improved in 93% (median CGI-change = 2, “much improved”) as assessed by neurologist and patient. Psychiatrist ratings showed improvement among 86.7%; physiatrist and psychologist ratings were 66.7% and 53.3%, respectively. Symptoms improved on DSSS (Wilcoxon Z = −2.914, p ≤ 0.004); GAD-7 (Z = −3.045, p ≤ 0.002); PHQ-9 (Z = −3.415, p ≤ 0.01) but not PCL-5 (Z = −1.506, p = 0.132). At 1 year, 54% maintained at least minimal improvement by neurologist rating and 77% by patient rating (median CGI-change = 3, “minimally improved”). Improvement was not maintained for DSSS (Wilcoxon Z = −0.385. p = 0.701), GAD-7 (Z = −0.943, p = 0.357) or PHQ-9 (Z = −0.55, p = 0.582).

      Conclusions

      Multidisciplinary inpatient rehabilitation improved FMD patient function, depression, anxiety and somatic symptoms. One-year follow-up demonstrated minimal sustained improvement and worsening psychopathology, reflecting chronic debility despite initial rehabilitative success.

      Keywords

      1. Introduction

      Functional movement disorders (FMDs) are clinical syndromes characterized by abnormal involuntary movements that are incongruent with known neurological diseases and have specific clinical characteristics including, but not limited to, distractibility, variability, and entrainment [
      • Edwards M.J.
      • Bhatia K.P.
      Functional (psychogenic) movement disorders: merging mind and brain.
      ]. FMDs cause significant disability, with high utilization of health care resources and a large proportion of patients unable to sustain meaningful employment [
      • Lehn A.
      • Gelauff J.
      • Hoeritzauer I.
      • et al.
      Functional neurological disorders: mechanisms and treatment.
      ]. Long term follow-up studies of this condition are lacking, but evidence suggests that FMDs are chronic disorders, with as many as two thirds of patients not improving and even worsening in physical status [
      • Feinstein A.
      • Stergiopoulos V.
      • Fine J.
      • Lang A.E.
      Psychiatric outcome in patients with a psychogenic movement disorder: a prospective study.
      ,
      • Gelauff J.
      • Stone J.
      • Edwards M.
      • Carson A.
      The prognosis of functional (psychogenic) motor symptoms: a systematic review.
      ].
      Historically, FMD's – previously addressed as somatoform and conversion disorders – have been relegated to psychiatry both in terms of etiology and management. Abundant prior psychoanalytic theory espoused that negative affective states such as anger, guilt, and others could manifest as physical ailments [
      • Alexander F.
      Fundamental concepts of psychosomatic research: psychogenesis, conversion, specificity.
      ]. Dyadic distinctions between mental and physical states made treatment of FMDs challenging. Patients frequently were lost among different medical disciplines and failed to improve.
      Although psychiatric comorbidity, including anxiety and depression, is well-known in FMD, not all patients have psychological symptoms [
      • Kranick S.
      • Ekanayake V.
      • Martinez V.
      • Ameli R.
      • Hallett M.
      • Voon V.
      Psychopathology and psychogenic movement disorders.
      ,
      • van der Hoeven R.M.
      • Broersma M.
      • Pijnenborg G.H.
      • Koops E.A.
      • van Laar T.
      • et al.
      Functional (psychogenic) movement disorders associated with normal scores in psychological questionnaires: a case control study.
      ]. Psychiatric intervention largely focuses on these comorbid conditions when evident since depression and anxiety are known amplifiers of somatic symptoms [
      • Barsky A.J.
      • Borus J.F.
      Functional somatic syndromes.
      ]. However, directed treatment with antidepressants has demonstrated only modest improvement [
      • Voon V.
      • Lang A.E.
      Antidepressant treatment outcomes of psychogenic movement disorder.
      ]. Although cognitive behavioral therapy (CBT) is well established for somatoform disorders and paroxysmal nonepileptic seizures (PNES), limited evidence for efficacy in FMD exists [
      • Kroenke K.
      Efficacy of treatment for somatoform disorders: a review of randomized controlled trials.
      ,
      • Goldstein L.H.
      • Chalder T.
      • Chigwedere C.
      • Khondoker M.R.
      • Moriarty J.
      • et al.
      Cognitive-behavioral therapy for psychogenic nonepileptic seizures: a pilot RCT.
      ,
      • Sharpe M.
      • Walker J.
      • Williams C.
      • Stone J.
      • Cavanagh J.
      • et al.
      Guided self-help for functional (psychogenic) symptoms: a randomized controlled efficacy trial.
      ,
      • LaFrance Jr., W.C.
      • Friedman J.H.
      Cognitive behavioral therapy for psychogenic movement disorder.
      ]. Despite this lack of evidence, effectiveness of CBT in addressing other functional neurological symptoms has fostered the belief that it may be effective for FMDs. Other approaches, including brief psychodynamic psychotherapy and hypnosis, have produced modest benefits [
      • Hinson V.K.
      • Weinstein S.
      • Bernard B.
      • Leurgans S.E.
      • Goetz C.G.
      Single-blind clinical trial of psychotherapy for treatment of psychogenic movement disorders.
      ,
      • Kompoliti K.
      • Wilson B.
      • Stebbins G.
      • Bernard B.
      • Hinson V.
      Immediate vs. delayed treatment of psychogenic movement disorders with short term psychodynamic psychotherapy: randomized clinical trial.
      ,
      • Moene F.C.
      • Spinhoven P.
      • Hoogduin K.A.
      • van Dyck R.
      A randomized controlled clinical trial of a hypnosis-based treatment for patients with conversion disorder, motor type.
      ].
      While the above therapeutic approaches have merit in their own right, patients are unlikely to experience the greatest benefit with any as a sole treatment strategy. In recent years, several studies have reported positive outcomes from physical therapy-based interventions with most approaches focusing on motor retraining [
      • Czarnecki K.
      • Thompson J.M.
      • Seime R.
      • Geda Y.E.
      • Duffy J.R.
      • Ahlskog J.E.
      Functional movement disorders: successful treatment with a physical therapy rehabilitation protocol.
      ,
      • Jordbru A.A.
      • Smedstad L.M.
      • Klungsoyr O.
      • Martinsen E.W.
      Psychogenic gait disorder: a randomized controlled trial of physical rehabilitation with one-year follow-up.
      ,
      • Nielsen G.
      • Ricciardi L.
      • Demartini B.
      • Hunter R.
      • Joyce E.
      • Edwards M.J.
      Outcomes of a 5-day physiotherapy programme for functional (psychogenic) motor disorders.
      ]. However, few studies have examined the effects of multidisciplinary inpatient rehabilitation, involving both physical and psychological interventions [
      • Demartini B.
      • Batla A.
      • Petrochilos P.
      • Fisher L.
      • Edwards M.J.
      • Joyce E.
      Multidisciplinary treatment for functional neurological symptoms: a prospective study.
      ,
      • Jacob A.E.
      • Kaelin D.L.
      • Roach A.R.
      • Ziegler C.H.
      • LaFaver K.
      Motor retraining (MoRe) for functional movement disorders: outcomes from a 1-week multidisciplinary rehabilitation program.
      ]. Demartini et al. examined prospectively the short and long-term efficacy of an inpatient multidisciplinary program for patients with FMDs involving physiotherapy, occupational therapy, cognitive behavioral therapy, psychiatric and neurological care [
      • Demartini B.
      • Batla A.
      • Petrochilos P.
      • Fisher L.
      • Edwards M.J.
      • Joyce E.
      Multidisciplinary treatment for functional neurological symptoms: a prospective study.
      ]. Findings were promising with demonstrated functional gains after rehabilitation and at follow-up, as well as decreases in patient-reported depression and anxious distress.
      The course of FMD's requires further scrutiny. Apart from aforementioned studies, little is known about the trajectory of physical symptomatology in FMD patients who have completed inpatient multidisciplinary rehabilitation. The course of co-existing psychopathology (when evident) for patients after such programs is also unclear. In this study, we prospectively assessed the effect of a multimodal inpatient rehabilitation intervention involving motor retraining, psychotherapy, and psychotropic medication management, on the function of patients with FMDs and the maintenance of improvement on functional and psychological measures one year after discharge.

      2. Methods

      2.1 Participants

      Patients with FMD seen at the Movement Disorders clinic at Rush University Medical Center between March 2015 and September 2017 were invited to participate in the study. The study was approved by the Institutional Review Board and all patients signed informed consent. FMD was diagnosed by a movement disorders physician and confirmed by a panel of movement disorder specialists after review of the history and a protocolized video. Diagnostic criteria for FMD were adapted from Fahn and Williams [
      • Fahn S.
      • Williams D.T.
      Psychogenic dystonia.
      ]. Patients were deemed suitable candidates if they endorsed symptoms severe enough to cause functional disability in their personal, family or professional life. Participants were excluded if they were younger than 18 years of age, were psychiatrically unstable (defined as having a Diagnostic and Statistical Manual for Mental Disorders, Fifth Edition (DSM-5) diagnosis requiring frequent medication titration and/or high clinical acuity such as presence of suicidal ideation or psychosis), evidence of another co-existing movement disorder, or felt not to be appropriate candidates for inpatient rehabilitation. The latter were patients with brief and episodic spells of movement disorders, patients without gait involvement, and patients who had previously completed the Mayo Clinic inpatient rehabilitation protocol [
      • Czarnecki K.
      • Thompson J.M.
      • Seime R.
      • Geda Y.E.
      • Duffy J.R.
      • Ahlskog J.E.
      Functional movement disorders: successful treatment with a physical therapy rehabilitation protocol.
      ].

      2.2 Design

      After neurological evaluation and diagnosis confirmation, patients were referred to the physiatrist (R.K.) for evaluation and inpatient admission. Patients were evaluated on admission by the physiatrist (R.K.), neurologist (K.K. or G.P.), psychiatrist (C·H.), psychologist (J.M.B.), physical therapist, occupational therapist, and speech therapist. The principles of physical therapy were adapted from the MoRe (Motor Reprogramming) program developed by the Frazier Rehabilitation Institute in Louisville, KY, based on a program developed at the Mayo Clinic in Rochester, MN [
      • Czarnecki K.
      • Thompson J.M.
      • Seime R.
      • Geda Y.E.
      • Duffy J.R.
      • Ahlskog J.E.
      Functional movement disorders: successful treatment with a physical therapy rehabilitation protocol.
      ,
      • Jacob A.E.
      • Kaelin D.L.
      • Roach A.R.
      • Ziegler C.H.
      • LaFaver K.
      Motor retraining (MoRe) for functional movement disorders: outcomes from a 1-week multidisciplinary rehabilitation program.
      ]. As described in their program, MoRe involves relearning of supportive and appropriate motor movements with the goal of neurological normalcy. Treatment aims to reestablish elementary movements in affected limbs or body regions while identifying triggers and other factors contributing to abnormal movements. Positive movements are actively reinforced and abnormal movements are generally ignored unless they significantly impinge upon a patient's performance. The physiatrist's role was to supervise all treatment efforts while the neurologist reinforced the diagnosis and administered assessment scales. The psychiatrist's role was to identify suboptimally treated psychiatric illness and make medication changes as clinically indicated. The psychologist performed a detailed diagnostic interview, identifying problem areas, introducing the principles of CBT and providing recommendations for continuation of psychotherapy upon discharge. Patients were reevaluated by the neurologist at 12 months.

      2.3 Assessments

      Patients were assessed at baseline (inpatient admission), at discharge, and at 12 months. Clinical Global Impression severity (CGI-s) was assessed at baseline by the patient (CGI-s-pat), neurologist (CGI-s-neuro), physiatrist (CGI-s-rehab), psychiatrist (CGI-s-psychi) and psychologist (CGI-s-psycho) within 24 h of the patient's admission. The investigators independently assigned CGI ratings incorporating findings from the bedside physical examination. To afford an appreciation for the multifaceted nature of FMD's, each scorer assigned ratings with respect to their individual field of expertise. CGI change (CGI-c) was assessed at discharge by the same parties. Twelve month follow-up CGI-c assessments were obtained from the neurologist and patient in clinic.
      Physical and occupational therapy measures at baseline and discharge included the Motor Assessment Scale (MAS), Functional Gait Assessment (FGA), Berg Balance Scale (BBS), Timed Up and Go (TUG) test, Function in Sitting Test (FIST), and aggregate Function Independence Measures (FIM), motor subscale. To examine the course of any comorbid psychopathology, patients were asked at study entry if they had a prior physical or psychological trigger preceding their symptom onset. Secondary outcomes at baseline, discharge and one year follow-up included the Depression and Somatic Symptom Scale (DSSS), Generalized Anxiety Disorder-7 (GAD-7), Patient Health Questionnaire-9 (PHQ-9), and Post-Traumatic Stress Disorder (PTSD) check-list for DSM-5 (PCL-5).

      2.4 Statistical Analysis

      Patient demographic and disease characteristics were examined using descriptive statistics. Distributions of selected variables (e.g., disease duration) were examined for outlier and extreme values. Given the heterogeneity of the enrolled participants, values are reported as median (range). Because of the small sample size and non-normality of data distributions, examination of primary and secondary outcomes was expected to follow a nonparametric model. Specifically, for the primary outcome we examined the probability of a CGI-c score of 3 or less (at least “minimally improved”) as opposed to a CGI-c score of 4 or greater (“no change” or worsened). For the continuous secondary outcomes (GAD-7, PCL-5, PHQ-9, and DSSS), we examined the probability that the “pre-post” (admission vs discharge) score was greater than 0 in both a positive and negative direction. Follow-up analyses compared change in symptoms (CGI-c) at one year versus admission rated by neurologist and patient as well as psychiatric inventories at one year versus baseline. Inferential comparisons employed Wilcoxon signed rank tests with statistical significance set at p < 0.05.

      3. Results

      3.1 Patient characteristics and Study Flow

      Eighty patients were assessed between March 2015 and September 2017. Twenty met inclusion criteria and were enrolled in the inpatient rehabilitation protocol whereas sixty did not qualify. The majority who were excluded were deemed not to qualify for inpatient rehabilitation either because of episodic or brief symptoms or absence of gait involvement. Twelve patients were denied insurance coverage for inpatient rehabilitation and nine were psychiatrically unstable. Thirteen did not accept the diagnosis of FMD or were uninterested in the recommended treatment. Attrition occurred in three (15%) patients, leaving seventeen patients (85%) who completed the protocol and were available for analysis (Fig. 1).
      Table 1 lists demographic and clinical characteristics of the seventeen patients who completed the inpatient protocol. The majority of the sample was female (82%). Physical or psychological triggers preceding onset of FMD were self-reported in 41% and 12%, respectively. Disease duration ranged from less than one month to 264 months with a median of two years. The median CGI-severity rating on admission was 5, “markedly ill”. Self-reported depression and anxiety were common, with 65% and 76% of patients endorsing these, respectively. Median baseline DSSS score was 27.0 and the median baseline PCL-5 score was 33. Median values for the GAD-7 and PHQ-9 at study entry were 11 and 13, respectively.
      Table 1Demographic and clinical characteristics of patients completing inpatient rehabilitation (n = 17).
      Patient Age/SexMotor PhenotypeDisease DurationPhysical trigger preceding onset?Psychological trigger preceding onset?Depression (self-report)Anxiety (self-report)Trauma (self-report)
      77/FTremor, gait4 yearsYesNoNoYesYes
      42/FTremor, gait, weakness3.5 yearsYesNoYesYesYes
      33/FTremor, gait0.7 yearsYesNoYesYesYes
      22/FGait7 yearsNoNoNoYesYes
      56/MTremor, gait2 yearsNoNoNoNoNo
      48/FTremor, gait6 yearsNoNoYesYesYes
      21/MTremor, gait1 yearNoNoYesYesYes
      48/FTremor, gait6 monthsYesNoYesYesNo
      43/FGait, weakness, slurred speech9 monthsNoYesYesYesNo
      43/FGait, tremor, myoclonus4 yearsNoNoYesYesNo
      56/FDystonia, gait22 yearsNoNoYesYesNo
      26/MGait, myoclonus, dystonia11 monthsYesNoYesNoNo
      55/FTremor, gait, speech7 daysYesNoNoNoYes
      51/FTremor, gait2 yearsYesNoYesYesNo
      33/FGait, hemiparesis1 monthNoYesYesNoYes
      37/FTremor, gait, freezing2 yearsNoYesYesYesYes
      19/FTremor, gait, slurred speech3 daysNoNoNoNoNo

      3.2 Efficacy of inpatient rehabilitation

      Fig. 2 shows the change in severity of symptoms (CGI-c) between admission and discharge for the 17 patients who completed the protocol as individually scored by all parties (neurologist, physiatrist, psychiatrist, psychologist, and patient). Median duration of stay in the inpatient rehabilitation protocol was 7.5 days (range 3–14). Improvement in physical symptoms at discharge (defined as a CGI-c rating by examiner of “very much improved,” “much improved”, or “minimally improved”) was achieved in 93.3% of patients by both neurologist and patient ratings (median CGI-c = 2, “much improved”; range 1–4). Although scores differed across assessors (see Fig. 2) the majority demonstrated improvement at discharge across all raters.
      Fig. 2
      Fig. 2Median CGI ratings of FMD severity at admission (n = 17) and change at discharge and one year follow-up (n = 13).
      Patients completing inpatient rehabilitation showed gains in functional assessments as well. The median Berg Balance Scale score increased from 24 on admission to 51.5 at the end of the inpatient protocol (Wilcoxon Z = −3.464, p < 0.001). Timed Up and Go testing (TUG) improved from a median of 49 s to 9.6 s between admission and discharge (Wilcoxon Z = −3.621, p < 0.0005). One subject had an extreme value for the baseline TUG measure (359 s). However, the comparison between baseline TUG and TUG at discharge from inpatient rehabilitation remained significant when this subject was removed from analysis. (Wilcoxon Z = −3.52, p < 0.0005). Statistically significant improvements were also noted in the median FIM-motor subscale between admission and discharge (52 vs 70.5, Wilcoxon Z = −3.638, p < 0.0005) and median MAS (18 vs 19, Wilcoxon Z = −2.533, p = 0.011). Although improvement was noted on the FGA at discharge, this finding was not statistically significant (4 vs 22, Wilcoxon Z = −0.314, p = 0.754).
      We sought to examine changes in comorbid psychopathology and follow its course during the inpatient protocol. Significant improvements in the DSSS (23.5 vs 15, Wilcoxon Z = −2.914, p = 0.004), GAD-7 (11 vs 5, Wilcoxon Z = −3.045, p = 0.002), and PHQ-9 (13 vs 7, Wilcoxon Z = −3.415, p = 0.01) were noted between admission and discharge. No significant changes were observed in the PCL-5 between admission and discharge (31 vs 29, Wilcoxon Z = −1.506, p = 0.132).

      3.3 Results of one-year follow-up

      Thirteen patients were re-assessed at one year. The remainder were lost to follow-up despite significant retention efforts. Because the one year follow-up evaluation was performed in the outpatient movement disorders clinic, CGI-c ratings were only available from the neurologist and patient at this time point. Table 2 reflects the CGI-c scores for the cohort and findings from repeat evaluation at one year follow-up relative to admission. Although maintenance of improvement in FMD symptoms was not statistically significant, seven of thirteen patients (54%) were rated by the neurologist as “improved” – defined as “very much improved,” “much improved,” or “minimally improved” (CGI-c ratings of 1, 2, or 3) (p = 1.0). Patient-rated improvement was noted in ten of thirteen patients (77%, p = 0.09). Analysis of the DSSS, GAD-7, PHQ-9 and PCL-5 at one-year follow-up showed no statistically significant improvement when compared to time of study entry (DSSS = 30 vs 23.5, Wilcoxon Z = −0.385, p = 0.701; GAD-7 = 10 vs 11, Wilcoxon Z = −0.943, p = 0.345; PHQ-9 = 15 vs 13, Wilcoxon Z = −0.55, p = 0.582; PCL-5 = 37 vs 31, Wilcoxon Z = −1.05, p = 0.294).
      Table 2CGI-change in FMD severity at one year follow-up (n = 13).
      OutcomeFrequency (N, %)p (2-tailed)
      CGI-c neurologyImproved (CGI-c = 1,2,3)7 (54%)1.00
      No change or Worse (CGI-c > 3)6 (46%)
      CGI-c patientImproved (CGI-c = 1,2,3)10 (77%)0.09
      No change or Worse (CGI-c > 3)3 (23%)

      4. Discussion

      In our study of 17 patients with disabling FMD symptoms (median CGI-s = 5, “markedly ill”) completing an inpatient rehabilitation protocol, 93.3% of patients demonstrated improvement on the CGI-c at discharge by neurologist rating, reflecting the benefit of this approach. The majority of patients were rated as improved by all raters, representing different disciplines. We also observed noteworthy improvements in key functional assessment measures, including the BBS, TUG, FIM-motor subscale, and MAS. Importantly, 93.3% of patients endorsed improvement, achieving a CGI-c score of 1, 2, or 3, consistent with at least minimal improvement in symptoms. Similarly, we noted decreases in psychiatric illness severity with statistically significant improvement in the GAD-7, PHQ-9, and DSSS between admission and discharge. Long-term follow-up at 12 months was available in 13 out of 17 patients completing the inpatient protocol. Among this group, 77% of participants reported continued improvement of their motor movements, but physician-rated assessment by a neurologist differed with only 54% of patients showing sustained improvement. Our experience is consistent with other studies of multidisciplinary inpatient rehabilitative approaches, including Jacob et al. [
      • Jacob A.E.
      • Kaelin D.L.
      • Roach A.R.
      • Ziegler C.H.
      • LaFaver K.
      Motor retraining (MoRe) for functional movement disorders: outcomes from a 1-week multidisciplinary rehabilitation program.
      ]. In their study utilizing motor retraining therapy for FMD patients of comparable severity to our cohort (mean CGI-s = 5.2 for Jacob et al.), 86.7% of patients reported that their abnormal movements were either “much improved” or “markedly improved” following rehabilitation based on CGI-c ratings. These findings support success for the multidisciplinary inpatient rehabilitation model. It is important to note that although remission or near remission of symptoms has been noted in some studies, many FMD patients often retain some abnormal motor movements even after completion of successful rehabilitation but report improvement. This positive self-report of patients after rehabilitation suggests that it is not essential to extinguish abnormal movements entirely in order to enhance patient function [
      • Jacob A.E.
      • Kaelin D.L.
      • Roach A.R.
      • Ziegler C.H.
      • LaFaver K.
      Motor retraining (MoRe) for functional movement disorders: outcomes from a 1-week multidisciplinary rehabilitation program.
      ].
      We noted that the percentage of patients who self-reported a physical trigger preceding onset of abnormal movements (41%) was higher than among patients with an antecedent psychological trigger (12%). Historical canon regarding the origins of conversion disorder and its correlates has upheld a psychiatric etiology, but our findings are more consistent with recent research in which physical precipitants were also reported more frequently than psychological precipitants [
      • Jacob A.E.
      • Kaelin D.L.
      • Roach A.R.
      • Ziegler C.H.
      • LaFaver K.
      Motor retraining (MoRe) for functional movement disorders: outcomes from a 1-week multidisciplinary rehabilitation program.
      ]. These findings support current DSM-5 psychiatric nosology, in which a psychological trigger is not required for the diagnosis of functional neurological disorder [
      ,
      • McKee K.
      • Glass S.
      • Adams C.
      • et al.
      The inpatient assessment and management of motor functional neurological disorders: an interdisciplinary perspective.
      ]. In our protocol, common physical precipitants included motor vehicle accidents and spinal surgery. One patient endorsed onset of symptoms after an antibiotic exposure.
      Maintenance of gains at one year following multidisciplinary rehabilitation was more modest, with only 54% of patients showing sustained improvement in their FMD based on neurologist rating. The lower physician rating is discouraging, although patient self-appraisals were more positive with 77% reporting continued improvement after one year and given the chronic debility associated with FMDs, it should not be dismissed that any incremental change in function observable to the patient living with the diagnosis may be valuable. All patients who completed inpatient rehabilitation were provided with follow-up with a psychiatrist and we hypothesize that some patients who remained invested in psychiatric aftercare retained some of their functional symptomatology but may have self-reported their symptoms as less severe at one year due to adoption of new coping strategies and other psychiatric intervention, leading to the above-noted disparity between neurologist and patient assessments.
      Initial improvements in the DSSS, PHQ-9, and GAD-7 were not sustained at the one year follow-up mark and can be attributed to patient and design factors. Although severity of depression, anxiety, and somatization were measured via the PHQ-9, GAD-7, and DSSS, respectively, we could not account for differences in treatment history (eg, number of prior major depressive episodes, number of prior trials of antidepressants, etc). Differences in chronicity of psychiatric illness and degree of treatment-refractoriness to past interventions, where evident, might have contributed to the lack of sustained improvement at one year follow-up. Additionally, although every effort was made to assure psychiatric follow up after discharge, we were not able to track engagement with psychiatric aftercare following inpatient rehabilitation; nor could we account for the quality and scope of psychiatric intervention rendered for those patients who maintained subsequent aftercare.
      We noted significant disparities between raters' scores at the end of the inpatient rehabilitation protocol. For neurologist and patients' ratings, 93.3% reported a CGI-c score of “minimally improved” or better. Psychiatrist, psychologist, and physiatrist ratings were more varied, however. FMDs are multifaceted in their etiology, manifestations, and course; therefore, the investigators assigned CGI-s and CGI-c scores from the vantage point of their clinical specialty and area of expertise. Our approach likely contributes to the observed variation in the assigned ratings between evaluators. For example, a patient with less severe physical symptoms of tremor or dystonia but significant psychopathology may be assigned a lower CGI-s score by a physiatrist but have a correspondingly higher score from a psychologist. Despite such differences in scores across raters, the majority of patients were rated as improved regardless of evaluator, and we feel our approach represents a more naturalistic means of showing how FMD's influence different domains of patient functioning and experience.
      Patients who participated in this study were seen in a tertiary center movement disorders clinic. The majority of those not enrolled in the study were excluded either because they did not qualify based on movement disorder severity (e.g., insufficient severity of disability for inpatient rehabilitation or only intermittent symptomatology) or because they required psychiatric stabilization first to maintain their safety. Another significant reason was that the investigators could not obtain insurance approval for inpatient treatment. Even among patients who were deemed suitable candidates by our physiatrist, insurance approval was often difficult to attain. Denials were most often due to out-of-network status, lack of significant functional impairment to warrant inpatient care (ie, outpatient intervention was felt most appropriate), or insurance's assertion that multidisciplinary rehabilitation was not an indicated and approved intervention for FMD and therefore unlikely to be of benefit. Although this may appear to be one of the major limitations of this study, it is also a major conclusion: past successes with the inpatient rehabilitation model for FMDs are encouraging, but granular implementation in a natural setting poses many real-world challenges. Comprehensive multidisciplinary inpatient rehabilitation may be viable for only a minority of FMD patients.
      All patients who completed the inpatient rehabilitation protocol received resources for ongoing physical therapy and occupational therapy in the outpatient setting. These services were often community-based rather than at the primary research site. As a result, the frequency of services could not be easily tracked by investigators. Likewise, it was not possible to determine if community therapists employed strategies commensurate with the motor retraining approach utilized in the inpatient setting or if different therapy techniques were used, which may have influenced findings at one year follow-up.
      Other limitations of our approach include the lack of a control group and the lack of blinded ratings by investigators. Generalizability of our findings is also limited by disease duration. The median duration of illness in our population was two years. Disease duration is a known prognostic factor in FMD patients, with shorter duration of symptoms and early diagnosis predictive of a more positive course [
      • Gelauff J.
      • Stone J.
      • Edwards M.
      • Carson A.
      The prognosis of functional (psychogenic) motor symptoms: a systematic review.
      ]. Patients with shorter symptom duration may have been more likely to maintain long-term improvement. We also did not collect data regarding other secondary outcomes, such as changes in frequency of use of assistive devices from baseline after protocol completion, and we endorse examination of such outcomes in future studies of the longitudinal course of FMD's.
      Patients who elected to participate in our inpatient rehabilitation protocol were likely more accepting of the FMD diagnosis, but common clinical experience is that many patients are not. In our sample, thirteen patients among those initially assessed did not agree to participate because they disagreed with the FMD diagnosis even after extensive discussion. As such, clinician demeanor when delivering the diagnosis is critical so patients do not “close the door” to a potentially beneficial treatment strategy [
      • McKee K.
      • Glass S.
      • Adams C.
      • et al.
      The inpatient assessment and management of motor functional neurological disorders: an interdisciplinary perspective.
      ].

      5. Conclusion

      Multidisciplinary inpatient rehabilitation is effective for patients with FMD. In our sample, multidisciplinary intervention from neurology, physiatry, psychiatry, and psychology resulted in improvements in 93% of participants with corresponding decreases in anxiety, depression, and somatization. However, maintenance of these gains at one year follow-up was limited, and recurrence of psychiatric symptomatology was common despite self-reported maintenance of improvement among 77% of patients. Multidisciplinary inpatient rehabilitation is a viable acute treatment strategy for FMD's although further investigations regarding the influence of other factors, including comorbid psychopathology, on long-term success are needed.

      Funding sources

      This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

      Declaration of competing interest

      None.

      Acknowledgements

      We acknowledge Glenn T. Stebbins, PhD, Professor of Neurological Sciences for guidance with statistical analysis of data. We recognize the tremendous effort and care delivered by the multidisciplinary teams from physical therapy and occupational therapy in the service of the patients participating in this protocol.

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