Maria Oto

Psychogenic nonepileptic seizures in patients with learning disability: comparison with patients with no learning disability.

Among a consecutive series of patients with psychogenic nonepileptic seizures (PNES), we compared patients with learning disability (LD) (n=25) with patients with no LD (n=263), with respect to demographic and clinical variables. A higher proportion of the LD group had epilepsy as well as PNES (P<0.001) (uncorrected P values are quoted), and a higher proportion were taking antiepileptic drugs at the time of diagnosis of PNES (P=0.007). Fewer patients with LD had a history of antecedent sexual abuse (P=0.036). A higher proportion of the LD group had previous pseudostatus (P<0.001), and a higher proportion had immediate situational or emotional triggers for their attacks (P<0.001). There were trends toward a higher proportion of men in the LD group (P=0.056) and a longer delay between onset of PNES and diagnosis (P=0.072). Our data suggest potentially important clinical differences between PNES populations with and without LD, as well as possible differences in mechanism.

Do patients whose psychogenic non-epileptic seizures resolve, ‘replace’ them with other medically unexplained symptoms? Medically unexplained symptoms arising after a diagnosis of psychogenic non-epileptic seizures

Background In clinical practice, it is sometimes observed that patients in whom psychogenic non-epileptic seizures (PNES) cease, develop another medically unexplained symptom (MUS). Methods In order to determine how many patients develop new MUS post diagnosis and whether patients whose attacks cease are more likely to do so, new MUS were recorded 6–12 months after the diagnosis of PNES in 187 consecutive patients. Results Compared with baseline, the overall proportion of patients with MUS increased slightly, from 70.1% to 76.5%, with 44/187 patients (23.5%) developing new MUS. There were no significant differences between attack free and non-attack free patients. Binary logistic regression analysis showed that predictors of new MUS diverged between attack free and non-attack free patients. Among patients continuing to have attacks, those with previous health related psychological trauma were 18.00 times more likely to develop new MUS (p<0.0005). In patients who became attack free, patients drawing disability benefits were 5.04 times more likely to have new MUS (p=0.011). Conclusions The results suggest that almost 25% of patients develop new MUS following a diagnosis of PNES, although most of those have MUS pre-diagnosis. Patients with a history of health related psychological trauma whose attacks continue after diagnosis are at particularly high risk of developing new MUS. The data do not support the hypothesis that PNES that resolve are likely to be ‘replaced’ by other MUS.

An exploratory randomized controlled trial of immediate versus delayed withdrawal of antiepileptic drugs in patients with psychogenic nonepileptic attacks (PNEAs).

Purpose:  To determine whether withdrawal of antiepileptic drugs (AEDs) in patients with psychogenic nonepileptic attacks (PNEAs) improves outcome.

Primary and secondary care attendance, anticonvulsant and antidepressant use and psychiatric contact 5–10 years after diagnosis in 188 patients with psychogenic non-epileptic seizures

Background and objectives There have been few studies of long-term outcome in psychogenic non-epileptic seizures (PNES), and none of long-term healthcare utilization. Methods We studied attendance with seizures, healthcare use and employment over a 6-month period from the family doctors of 260 consecutive patients with psychogenic non-epileptic seizures (PNES), 5–10 years after diagnosis. Results We obtained clinical data in 188/260 patients (72.3%), of whom 60 (31.9%) had attended primary or secondary care with seizures in the previous 6 months. Predictors of attendance with seizures included a diagnosis of epilepsy+PNES (OR 5.7, p=0.009), work status (OR 3.9, p=0.027) and social security payments (OR 6.3, p=0.003). Latency to diagnosis was not predictive. Emergency admission data were available in 187 patients, of whom 25 (13.4%) had emergency hospital attendances. Prescription data were available for 172 patients, of whom 154 had ‘PNES only’. Of these, 17 (11.0%) remained on antiepileptic medication (AED). 68/172 patients (39.5%) were prescribed antidepressant (AD) drugs. We had psychiatric contact data in 185 patients, of whom 49 (26.5%) had accessed psychiatric services in the last 6 months. Conclusions Surprisingly few of our patients had presented with seizures during the study period. Early reductions in both AED use and healthcare use were sustained long term. Although psychiatric and employment outcomes were less encouraging, some aspects of PNES outcome may be better than previously thought.

Neurologist assessment of reactions to the diagnosis of psychogenic nonepileptic seizures: Relationship to short- and long-term outcomes

PURPOSE To determine the relationship between neurologist assessment of reactions to the diagnosis of PNESs and outcomes at 6-12 months and at 5-10 years. METHODS Two hundred thirty-eight patients with psychogenic nonepileptic seizures (PNES) were recruited into a long-term follow-up study. At diagnosis and 6-12 months post diagnosis, doctors recorded their assessments of patient and caregiver reactions to the diagnosis of PNESs. RESULTS At baseline, 92/238 patients (38.7%) and 73/106 caregivers (68.9%) were assessed as having understood and accepted the diagnosis, while 6.7% of patients and 10.4% of caregivers reacted with anger. At 6-12 months, patient acceptance rose to 57.7%, with caregiver acceptance static at 70.8%. Attendance at follow-up was predicted by the presence of a caregiver at baseline: only one patient who came with a caregiver at baseline did not attend at 6-12 months (OR: 123.80, p < 0.001). Outcome at 6-12 months was predicted by patient acceptance at baseline (OR: 2.85, p = 0.006) and at 6-12 months (OR: 13.83, p < 0.001) and by caregiver acceptance at 6-12 months (OR: 10.77, p < 0.001). Presentation to primary or secondary care with attacks at 5-10 years was predicted by caregiver acceptance at 6-12 months (OR: 3.50, p = 0.007). CONCLUSION Patient understanding and acceptance of the diagnosis of PNESs are linked to outcome at 6-12 months. The beliefs of caregivers may be important for outcome in the longer term, particularly with respect to health-care use.

The misdiagnosis of epilepsy: Appraising risks and managing uncertainty

PURPOSE To present evidence from the literature on the rates, underlying causes and consequences of the misdiagnosis of epilepsy and place these meaningfully within a practical framework of risk appraisal and managed diagnostic uncertainty towards informing a clinical practice that might make misdiagnosis less likely. METHOD Narrative review. RESULTS Misdiagnosis of epilepsy remains common and the consequences for the individual significant. Evidence and critical appraisal are presented as regards the absolute level of risk associated with the false positive diagnosis epilepsy, and reasons as to why those risks need to be appraised against the risks associated to false negative diagnosis. CONCLUSIONS Diagnostic error is not entirely avoidable and a degree of uncertainty, and perforce risk, is intrinsic to the diagnostic process of epilepsy. The risks of a false negative diagnosis of epilepsy must be appraised against the also significant risks of a false positive diagnosis.

Healthcare resource utilization after medium-term residential assessment for epilepsy and psychogenic nonepileptic seizures

INTRODUCTION Epilepsy and epilepsy mimics may lead to high healthcare resource utilization (HRU) including diagnostic resources. The William Quarrier Scottish Epilepsy Centre (SEC) provides medium-term residential assessment (MTRA; average length of stay: 28days) and treatment for complex presentations of epilepsy and related conditions (principally psychogenic nonepileptic seizures, PNES). We studied the effect of MTRA on HRU in a defined health board area in Scotland. METHODS A retrospective audit of individuals admitted to the SEC from a defined health board area using SEC and health board medical records. Neurological HRU assessed included emergency department visits, hospital admissions, outpatient clinic appointments, and brain imaging prior to and post-MTRA. Healthcare resource utilization was also compared with individuals referred but not admitted to the SEC because of individual circumstances and choice. RESULTS Seventy-three individuals (51 female, average age: 37.51; 22 men, average age: 43.72) were identified from three years of admissions (1st April 2010 to 31st March 2013). Final diagnosis was epilepsy (ES), 32; ES and psychogenic nonepileptic seizures (ES+PNES), 17; and PNES alone, 24. Twenty-two individuals were identified as a comparison group (8 men, 14 women; average age: 37.21 and 43.90, respectively). Total average contacts per patient per year (CPY) was significantly different pre- and post-MTRA (4.16 vs. 1.32; t(72)=6.11, p<.0001, d=.72). Comparison of HRU in the first year of baseline and last full year of follow-up showed a post-MTRA reduction in HRU for PNES of 92.28%, for ES of 46.81%, and for ES+PNES of 28.3%. During the course of follow-up, PNES CPY continued to drop (1.13 first year vs. 0.10 at 3years post-MTRA). For individuals with epilepsy (with or without PNES), HRU use dropped significantly in the year after admission, and these gains remained stable (total first vs. third postdischarge CPY, 1.74 vs. 1.29). The participants in the comparison group, who were not admitted, had no comparable drop across the study period and were using significantly more resources at each follow-up point than those in the admitted group (F (1, 48)=44.45, p<.01, ηp(2)=.49). CONCLUSION Medium-term residential assessment is associated with sustained reduction in HRU especially in patients with PNES. Overall HRU reduction was 68.27% following admission (d=.72). This suggests benefit from the MTRA model for people with complex presentations.

Use of suggestion methods to induce psychogenic non-epileptic seizures

We welcome the comprehensive and timely systematic review of suggestive seizure induction for psychogenic non-epileptic seizures (PNES) by Popkirov and colleagues [1]. As the authors of two of the studies cited in this review we would like comment on two issues. The first concerns the degree of honesty or deceptiveness of the communication strategies employed. We agree that avoiding deception is crucial for ethical reasons, and that an honest approach tends to reinforce a positive therapeutic relationship [2]. In our published studies, we told patients explicitly that a psychological cause for their attacks was being considered and that we wished to record attacks to confirm the diagnosis and thus facilitate appropriate treatment [3,4]. This is clearly stated in our first article [3], in the fifth paragraph of the discussion section, where we discuss the ethical issue: ‘‘. . . we told patients that psychological attacks were being considered, and that recording of these was necessary for diagnosis.’’ The fact that two thirds of our patients went on to have a habitual attack during video-EEG might be taken to indicate openness to a psychological diagnosis, as well as providing strong evidence against malingering [3]. The same approach continued to be used in our clinical practice [5] with a similar yield of recorded attacks and good diagnostic accuracy [6,7]; no episodes of ‘‘pseudostatus’’ have occurred in over 1000 recordings (unpublished observation). We would consider that our method involves honest communication without omission and would therefore suggest that, in the present paper by Popkirov et al., our studies should be included in the ‘‘explicitly open’’ rather than the ‘‘truthful but omissive’’ category. The second aspect relates to possible psychobiological mechanisms underlying the efficacy of suggestion in eliciting PNES, clearly a difficult and interesting question. As well as interactions with stress [8,9], arousal [10], hypnotizability [11] and dissociative tendencies [12], another possible framework for thinking about suggestion in PNES relates to the placebo effect [13], which has been increasingly well-characterised over the last decade [14,15]. This is described as a ‘‘psychobiological phenomenon attributable to the overall psychosocial therapeutic context’’ [15], and hinges upon expectation of a certain outcome. In studies of PNES, the environmental setting is the video-EEG recording and the expectation (of both patient and doctor) is that there is a greater chance than usual that a seizure will occur, especially in the context of specific provocation methods that are judged likely to trigger an attack. It is interesting that, in patients in our previous study in whom suggestion during video-EEG provoked an event, there was a prior history of events occurring in medical settings [3], implying an effect of situational context for this subgroup. The likelihood of placebo response is multi-factorial, depending on neurobiological, environmental and psychosocial factors [16]. It

The William Quarrier Scottish Epilepsy Centre, Glasgow, Scotland.

TheWilliam Quarrier Scottish Epilepsy Centre (SEC) opened its new purpose-built, 12-bedded assessment unit for adults and young adults with complex epilepsy in April 2013. This transformation reflects the philanthropic activities of William Quarrier, a mid-nineteenth century philanthropist whose vision was to provide a protective and productive environment for vulnerable young people including those with epilepsy. Raised in poverty— his initial concernwas for the plight of homeless children who roamed the streets of Glasgow, one of the poorest, most deprived, and overcrowded cities in Europe at the time. His first venture was to bring young people into employment in his bootand shoemaking business— the Shoe Black Brigade. In return, they received an education. Later, he opened two homes for destitute children in Glasgow and then raised further funds to establish a childrens village away from the city center. The Orphan Homes of Scotland was opened in 1878 — comprising of cottage homes along the lines of Dr. Barnado in England and the Rauhes Haus model of Johann Wichern in Germany. Quarrier then turned his attention to those incapacitated by incurable illness, particularly tuberculosis — opening Scotlands only sanatorium for tuberculosis. He was also the first in Scotland to show concern for people with epilepsy — considered a much stigmatized untreatable condition where many afflicted ended up in the poorhouse or asylum. After Quarrier visited the newly established Chalfont ‘Colony for Epileptics’, he raised £20,000 for his ‘Colony of Mercy’ for epileptics, which opened in 1906, 3 years following his death, at Bridge of Weir, a small village 20 miles from Glasgow. The ethos of the new colony was more a caring environment than medical; and patients were expected to work if able in the workshop and gardens. An extract from the medical officers report from 1916 reads:

Medically unexplained symptoms in patients with PNES: Do they explain poor employment outcome in patients with good seizure outcomes?

BACKGROUND Many patients with psychogenic nonepileptic seizures (PNES) have other medically unexplained symptoms (MUS). In order to assess their long-term contribution to ill-health and unemployment, we recorded MUS, employment, healthcare utilization, and seizure outcomes in a cohort of patients with PNES. METHODS We had complete computerized healthcare records and employment information in 120 patients with PNES, 5-10years postdiagnosis. We analyzed these data to assess the contribution of MUS to ill-health and to determine whether MUS could explain differences among seizure, employment, and healthcare utilization outcomes in PNES. RESULTS At 5-10years, 41/120 patients (34.2%) were attending primary or secondary care for seizures, 42/120 patients (35.0%) had MUS other than PNES, and 36/120 patients (30.0%) were employed. Unemployment was predicted by age at presentation (OR: 0.90 (0.86-0.94), p<0.001), presenting to primary or secondary care with seizures (OR: 0.16 (0.05-0.52), p=0.003), and contact with psychiatric services at 5-10years (OR: 0.16 (0.05-0.58), p=0.005). No outcome measures were predicted by additional MUS, though MUS themselves were predicted by psychiatric contact (OR: 2.27 (1.01-5.01), p=0.048). CONCLUSIONS Our data suggest that MUS do not contribute independently to unemployment in the population with PNES, whereas psychiatric morbidity appears to do so. Nonetheless, MUS and psychiatric morbidity persist in the long term in a substantial minority of patients with PNES. We found no evidence that seizures in patients whose PNES have resolved are replaced with other MUS.