Long-term adherence with psychiatric treatment among patients with psychogenic nonepileptic seizures

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1 Accepted: 6 November 2017 DOI: /epi BRIEF COMMUNICATION Long-term adherence with psychiatric treatment among patients with psychogenic nonepileptic seizures Benjamin Tolchin 1 Barbara A. Dworetzky 2 Gaston Baslet 3 1 Division of Epilepsy, Department of Neurology, Yale New Haven Hospital and VA Connecticut Healthcare System, Yale School of Medicine, New Haven, CT, USA 2 The Edward B. Bromfield Epilepsy Program, Department of Neurology, Brigham and Women s Hospital, Harvard Medical School, Boston, MA, USA 3 Department of Psychiatry, Brigham and Women s Hospital, Harvard Medical School, Boston, MA, USA Correspondence Benjamin Tolchin, Department of Neurology, Yale School of Medicine, New Haven, CT, USA. benjamin.tolchin@yale.edu Funding information American Academy of Neurology, Grant/ Award Number: Practice Research Training Fellowship Summary We conducted a prospective cohort study, examining long-term adherence with psychiatric treatment among patients with psychogenic nonepileptic seizures (PNES). Subjects diagnosed with documented PNES were scheduled for 4 psychiatric visits. Survival analysis was performed, and covariates were assessed for association with time to nonadherence using Cox proportional hazard regression analysis. One hundred twenty-three subjects were recruited and followed for up to 17 months. Eighty percent of subjects attended the first outpatient visit, 42% attended the second, 24% attended the third, and only 14% remained adherent through the fourth visit. Two covariates were associated with nonadherence: (1) a prior diagnosis of PNES (hazard ratio 1.57, P-value.046); (2) a lower score on the Brief Illness Perception Questionnaire (BIPQ), signifying lower concern about one s illness (hazard ratio 0.77 for every 10-point increment on the 80-point scale, P-value.008). Adherence with psychiatric treatment among patients with PNES is initially reasonably good but worsens rapidly over visits 2-4. Risk factors for nonadherence include a history of a prior diagnosis of PNES, and a lower level of concern about the illness as assessed by a lower score on the BIPQ. KEYWORDS compliance, conversion disorder, functional neurological symptom disorder, nonepileptic attack disorder, PNES 1 INTRODUCTION Psychogenic nonepileptic seizures (PNES) are common and highly disabling: one third of patients evaluated in epilepsy monitoring units are ultimately diagnosed with PNES, 1-4 and ratings of disability in PNES are as severe as those in epilepsy. 5 Patients with PNES experience long-term economic and social dependence, have frequent emergency department visits and hospital admissions, and incur high medical care costs. 6-8 The prognosis for the untreated illness is poor, with 71% of patients having ongoing psychogenic seizures and 56% of patients on disability at 1- to 10-year follow-up. 7 Psychotherapy has been shown to reduce psychogenic seizure frequency and improve quality of life. The strongest evidence basis, including two randomized trials, demonstrates the efficacy of cognitive behavioral therapy. 9,10 Yet 20%-30% of patients fail to attend their first appointment after the diagnosis of PNES, and small studies and clinical experience suggest that adherence may significantly deteriorate over longer periods Long-term adherence with behavioral health treatments of PNES has received limited attention, and longterm dropout rates are unknown. We therefore conducted a prospective cohort study, measuring adherence with psychiatric follow-up for up to 17 months after diagnosis of PNES. 2 METHODS The study was approved by the Brigham and Women s Hospital (BWH) Institutional Review Board. All enrollees provided written informed consent. e18 Wiley Periodicals, Inc International League Against Epilepsy wileyonlinelibrary.com/journal/epi Epilepsia. 2018;59:e18 e22.

2 TOLCHIN ET AL. e19 We prospectively recruited consecutive subjects, aged 18 and older, diagnosed with documented PNES via video electroencephalography (EEG) capture of typical seizure events. Exclusion criteria included static encephalopathy with estimated IQ <70 (sufficiently low to preclude participation in psychotherapy); active substance use disorder; pregnancy; or severe medical illness (such as cancer), which might be expected to prevent regular participation in psychotherapy. Diagnoses were made by board-certified epileptologists based on video-eeg review of typical events showing semiology consistent with psychogenic etiology and no epileptiform correlate before, during, or after events, consistent with a documented level of diagnostic certainty, the highest level of certainty as per International League Against Epilepsy (ILAE) criteria. 14 During the epilepsy monitoring unit (EMU) admission, all subjects were evaluated for risk factors for PNES and for psychiatric comorbidities by a neuropsychiatrist (GB) with fellowship training, board certification, and clinical experience in the evaluation and treatment of emotional and behavioral difficulties arising in the presence of neurologic illness. Baseline demographic, medical, and psychiatric data were collected in a semistructured psychiatric interview with a secondary goal of identifying potential risk factors for nonadherence. Illness perception was assessed at the time of the initial neuropsychiatric assessment using the Brief Illness Perception Questionnaire (BIPQ), an 80-point scale, with 80 indicating the greatest possible concern about one s illness and 0 indicating the least possible concern. An epileptologist (BAD) and neuropsychiatrist (GB) jointly delivered the diagnosis of PNES to patients and when present their families, reviewed the nature and treatment of PNES, and made an appointment for an interdisciplinary epilepsy-neuropsychiatry outpatient clinic visit within 1 month of the diagnosis. At the initial clinic visit, an epileptologist, neuropsychiatrist, and social worker again jointly reviewed the diagnosis and treatment, answered questions, and arranged the initiation of psychotherapy, with a licensed social worker psychotherapist at BWH, or with a local therapist if the subject already had an existing therapeutic relationship or lived sufficiently far from BWH to make weekly visits logistically difficult. Treatment at BWH consisted of a manualized regimen of 12 weekly hour-long mindfulness-based psychotherapy sessions. 15 The neuropsychiatrist called all local therapists to review the diagnosis and principles of treatment for all patients who were treated outside of BWH, and to give recommendations for treatment. All patients were scheduled for 3 additional appointments with the neuropsychiatrist at BWH, who would monitor PNES and psychotherapy, maintain communication with the treating psychotherapist, and provide psychopharmacologic treatment for any psychiatric comorbidities. Psychiatric appointments (including one initial multidisciplinary appointment) were scheduled at approximately 1 month following the time of diagnosis, at approximately 1.5 months following visit 1, at approximately 1.5 months following visit 2, and at approximately 6 months following visit 3. Subjects were considered to have dropped out of psychiatric treatment and become nonadherent if they failed to attend the first clinic visit within 2 months of diagnosis, if they failed to attend the second psychiatric visit within 3 months of visit 1, if they failed to attend the third psychiatric visit within 3 months of visit 2, or if they failed to attend the fourth visit within 9 months of visit 3, a maximum of 17 months after diagnosis. Survival analysis was performed, and covariates were assessed for association with time to nonadherence using Cox proportional hazard regression analysis. Forty variables were included in a univariate screen, including demographics, quality of life, illness perception, acceptance of the diagnosis of PNES, delay to diagnosis, psychiatric comorbidities, and history of abuse (Table 1). All covariates showing an association with time to nonadherence with a P-value of <.15 in the univariate screen were included in a multivariate model, with age and gender as potential confounders (Table S1). All statistical analyses were performed using Stata IC v14.1 (StataCorp, College Station, TX, USA). 3 RESULTS One hundred twenty-three consecutive subjects meeting inclusion criteria were enrolled. Three subjects refused consent, two were excluded for static encephalopathy, and two were excluded for active substance use disorder. Subjects were aged 18-80, with a mean age of 38, and were 85% female (Table 1). The majority (61%) were unemployed, and 37% were on disability. Only 18% worked full-time. Ninety-four percent of subjects had at least one psychiatric comorbidity (with depression, anxiety, and posttraumatic stress disorder [PTSD] being most common). Thirty-nine percent had a prior history of psychiatric hospitalization and 23% had made a prior suicide attempt. Seventy-four percent had some prior history of abuse. Thirty-seven percent reported receiving a prior diagnosis of PNES and were seeking a second opinion. Survival analysis showed that 80% of patients remained adherent at the first outpatient psychiatric visit (Figure 1). However, only 42% attended the second visit, 24% continued through visit 3, and only 14% were still adherent at appointment 4. In univariate analysis, the following covariates were associated with nonadherence: a prior diagnosis of PNES, lower score on the BIPQ, a history of physical abuse, and a history of substance abuse (Table S1). In the multivariate model, two covariates were associated with nonadherence:

3 e20 TOLCHIN ET AL. TABLE 1 Demographics and baseline characteristics N = 123 (%) Demographics Female gender 104 (85%) Race/ethnicity White, non-hispanic 95 (77%) African American 15 (12%) Hispanic 11 (9%) Other 2 (2%) Marital status Single 43 (35%) Married 40 (33%) Live-in partner 19 (15%) Separated/divorced 18 (15%) Widowed 3 (2%) Employment Unemployed, 46 (37%) collecting disability Unemployed, not on disability 29 (24%) Employed full-time 22 (18%) Employed part-time 14 (11%) Student 10 (8%) Years of education Mean 13.7 (SD 2.8, range 6-24) Psychiatric history Quality of life Mean 28.3 (SD 8.4, range 12-44) (QOLIE-10) Beck Depression Inventory II Illness perception score (BIPQ) Mean 18.5 (SD 11.6, range 0-48) Mean 49.3 (SD 11.8, range 10-76) Acceptance of PNES diagnosis (5-point Likert scale) Strongly agree 21 (28%) Agree 21 (28%) Neutral 12 (16%) Disagree 10 (13%) Strongly disagree 12 (16%) Weekly PNES frequency Median 2 (IQR , range 0-350) Delay to diagnosis (mo) Median 12 (IQR 4-72, range 0-612) Prior diagnosis of PNES 46 (37%) History of suicide attempt 28 (23%) Prior psychiatric 48 (39%) hospitalization History of substance abuse 28 (23%) Psychiatric comorbidities Depression 96 (78%) (Continues) TABLE 1 (Continued) N = 123 (%) Anxiety disorder 75 (61%) PTSD 66 (54%) Panic disorder 49 (40%) Personality disorder 38 (31%) Eating disorder 33 (27%) Psychotic disorder 17 (14%) OCD 16 (13%) Bipolar disorder 13 (11%) Any psychiatric 115 (94%) comorbidity Abuse history Emotional abuse 70 (57%) Physical abuse 55 (45%) Sexual abuse 52 (42%) Any abuse 91 (74%) PNES, psychogenic nonepileptic seizure; IQR, interquartile range; BIPQ, Brief Illness Perception Questionnaire; SD, standard deviation; QOLIE-10, Quality of Life in Epilepsy Inventory-10; PTSD, posttraumatic stress disorder; OCD, obsessive compulsive disorder. (1) a prior diagnosis of PNES was associated with increased odds of dropout, with a hazard ratio of 1.57 (confidence interval [CI] ) and a P-value of.046; (2) a higher score in the BIPQ, signifying greater concern about one s illness, correlated with decreased odds of dropout, with a hazard ratio of 0.77 (CI ) for every 10-point increment on the 80-point scale, and a P-value of.008. There was also a trend toward nonadherence among those subjects with a history of substance abuse (hazard ratio 1.80, P-value.078). 4 DISCUSSION This is the first study to look at long-term treatment adherence rates among patients with PNES over more than a year of follow-up, and shows a substantial decline from early adherence rates measured here and elsewhere in the literature. 12,13 Our cohort was similar to those in prior studies of PNES, which were also disproportionately female (85% vs 75%-91%), with high rates of unemployment (61% vs 65%-90%), psychiatric comorbidities, and history of sexual abuse (42% vs 33%-76%). 9,11,12 Adherence at the first outpatient visit was similar to that seen in on the initial visit in prior studies (80% vs 72%-80%) but worsened significantly at each subsequent visit to a nadir of 14% adherence by the fourth visit. In addition, our results suggest that patients previously diagnosed with PNES, returning for a second opinion or

4 TOLCHIN ET AL. e21 FIGURE 1 Kaplan-Meier curve showing subjects adherence to psychiatric follow-up over 4 psychiatric visits scheduled over a maximum of 17 months [Color figure can be viewed at wileyonlinelibrary.com] after the failure of a first treatment regimen, have significantly increased odds of long-term nonadherence. Our results also suggest that patients with a greater degree of concern about their illness, as measured by a higher score on the BIPQ, have significantly better adherence. This implies that it is important not to minimize the seriousness of PNES when delivering the diagnosis and treating the illness, but rather to acknowledge the very real disability and suffering that the illness can cause. Of note, our analysis did not detect a correlation between acceptance of the diagnosis of PNES and nonadherence, or between various psychiatric comorbidities and nonadherence, suggesting that psychiatric treatment of PNES is possible even for patients who are skeptical of the diagnosis, or have personality disorders or other psychiatric comorbidities. Although the high levels of nonadherence shown in our cohort raise concerns about the long-term efficacy of psychiatric treatments for PNES, this study is limited in that we do not yet have long-term outcome data for adherent and nonadherent subjects. We therefore cannot establish whether nonadherence causes or is even associated with worse clinical outcomes. It could be the case that patients drop out of treatment because their psychogenic seizures have improved, or that nonadherent patients would not improve even if they did participate in psychiatric treatment. The lack of outcome data makes it difficult to interpret predictive and nonpredictive covariates. A future direction for research will be to measure long-term outcomes such as change in seizure frequency, change in quality of life, and change in emergency department utilization, and to evaluate whether these outcomes correlate with adherence to treatment. Another direction of future research will be to perform randomized trials of interventions, such as motivational interviewing, to improve adherence with treatment, to better understand reasons for nonadherence, and to potentially improve outcomes. Such findings could help to establish a causal relationship between treatment adherence and outcomes. Our study is also limited in that it tracks adherence to 4 psychiatric appointments over more than a year but not adherence with psychotherapy. It is possible that some subjects may continue psychotherapy even as they become nonadherent to psychiatric follow-up. A future direction for research will be to measure adherence with psychotherapy, as this may more closely correlate with outcomes like seizure frequency and quality of life. Finally, the study is limited in that it was performed in a single quaternary care center with special interest in PNES, which utilizes a treatment regimen that is not available in many settings. This may limit the generalizability of our findings; we suspect that treatment adherence may be even worse in healthcare settings with fewer resources and less integration of care devoted to patients with PNES. A future direction for research may include a multicenter study of treatment adherence among patients with PNES in multiple care settings. In conclusion, our results show that treatment adherence among patients with PNES declines steadily over time, with only 14% of patients continuing to follow through the fourth psychiatric visit. Risk factors for nonadherence include a prior diagnosis of PNES and a lower level of concern about the illness. Neurologists diagnosing PNES must work closely with behavioral health specialists to improve long-term treatment adherence and outcomes. ACKNOWLEDGMENTS Tolchin is indebted to the American Academy of Neurology and the American Brain Foundation for a Practice Research Training Fellowship, which supported this work. DISCLOSURE None of the authors has any conflict of interest to disclose. We confirm that we have read the Journal s position on issues involved in ethical publication and affirm that this report is consistent with those guidelines. ORCID Benjamin Tolchin

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A GUIDE FOR PATIENTS & FAMILIES

A GUIDE FOR PATIENTS & FAMILIES WHAT ARE PSYCHOGENIC NON-EPILEPTIC SEIZURES? A seizure is a temporary loss of control, often with abnormal movements, loss of consciousness, or both. Epileptic seizures