Economic Cost of Functional Neurologic Disorders: A Systematic Review.

BACKGROUND AND OBJECTIVES: Functional neurologic disorder (FND) represents genuine involuntary neurologic symptoms and signs including seizures, weakness, and sensory disturbance, which have characteristic clinical features, and represent a problem of voluntary control and perception despite normal basic structure of the nervous system. The historical view of FND as a diagnosis of exclusion can lead to unnecessary health care resource utilization and high direct and indirect economic costs. A systematic review was performed using Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines to assess these economic costs and to assess for any cost-effective treatments. METHODS: We searched electronic databases (PubMed, PsycInfo, MEDLINE, EMBASE, and the National Health Service Economic Evaluations Database of the University of York) for original, primary research publications between inception of the databases and April 8, 2022. A hand search of conference abstracts was also conducted. Key search terms included "functional neurologic disorder," "conversion disorder," and "functional seizures." Reviews, case reports, case series, and qualitative studies were excluded. We performed a descriptive and qualitative thematic analysis of the resulting studies. RESULTS: The search resulted in a total of 3,244 studies. Sixteen studies were included after screening and exclusion of duplicates. These included the following: cost-of-illness (COI) studies that were conducted alongside cohort studies without intervention and those that included a comparator group, for example, another neurologic disorder (n = 4); COI studies that were conducted alongside cohort studies without intervention and those that did not include a comparator group (n = 4); economic evaluations of interventions that were either pre-post cohort studies (n = 6) or randomized controlled trials (n = 2). Of these, 5 studies assessed active interventions, and 3 studies assessed costs before and after a definitive diagnosis of FND. Studies showed an excess annual cost associated with FND (range $4,964-$86,722 2021 US dollars), which consisted of both direct and large indirect costs. Studies showed promise that interventions, including provision of a definitive diagnosis, could reduce this cost (range 9%-90.7%). No cost-effective treatments were identified. Study comparison was limited by study design and location heterogeneity. DISCUSSION: FND is associated with a significant use of health care resources, resulting in economic costs to both the patient and the taxpayer and intangible losses. Interventions, including accurate diagnosis, seem to offer an avenue toward reducing these costs.

Assessment of Emergency Department and Inpatient Use and Costs in Adult and Pediatric Functional Neurological Disorders.

Importance: There is limited information about health care use and costs in patients with functional neurological disorders (FNDs). Objective: To assess US emergency department (ED) and inpatient use and charges for FNDs. Design, Setting, and Participants: This economic evaluation used Healthcare Cost and Utilization Project data to assess all-payer (1) adult (age, ≥18 years) hospitalizations (2008-2017), (2) pediatric (age, 5-17 years) hospitalizations (2003, 2006, 2009, 2012, and 2016), and (3) adult and pediatric ED evaluations (2008-2017). International Classification of Diseases, Ninth Revision, Clinical Modification code 300.11 (conversion disorder) or 306.0 (musculoskeletal malfunction arising from mental factors) and International Statistical Classification of Diseases and Related Health Problems, Tenth Revision, Clinical Modification codes for conversion disorder/functional neurological symptom disorder (F44.4 to F44.7) were used to conservatively define FNDs and to compare them with other neurological disorders that are associated with high levels of health care use. Analysis was performed between January 2019 and July 2020. Main Outcomes and Measures: Admission traits (eg, demographic characteristics of patients, length of stay, and discharge disposition) and hospital charges. Results: Compared with other neurological disorders in 2017, emergency FND evaluations of 36 359 adults (25 807 women [71.0%] and 3800 children (2733 girls [71.9%]) more frequently resulted in inpatient admissions (22 895 adult admissions [69.2% female] and 1264 pediatric admissions [73.4% ]). These FND admissions had a shorter mean (SEM) hospital length of stay (5.21 [0.15] days vs 6.03 [0.03] days, P < .001) but higher workup rates than admissions for comparable neurological diagnoses. Admissions for FNDs had low rates of inpatient physical therapy, occupational therapy, speech and language pathology, and psychiatric consultation. The total annual costs (a proxy for total costs in 2017 US dollars) were $1066 million (95% CI, $971-$1160 million) for adult FND inpatient charges in 2017 compared with $1241 million (95% CI, $1132-$1351 million) for anterior horn cell disease; $75 million (95% CI, $57-$92 million) for pediatric FND inpatient charges in 2012 compared with $86 million (95% CI, $63-$108 million) for demyelinating diseases; and $163 million (95% CI, $144-$182 million) for adult and pediatric ED visits in 2017 compared with $135 million (95% CI $111-$159 million) for refractory epilepsy. Total charges per admission for ED care of FNDs were higher than the other comparison groups in adults. Total costs and costs per admission for FNDs increased from 2008 to 2017 at a higher rate than that of other neurological disorders. Conclusions and Relevance: This economic evaluation found that the more than $1.2 billion and increasing annual costs for ED and inpatient care of FNDs were similar to other investigation-intensive and pharmacologically demanding neurological disorders. Unnecessary investigations and iatrogenic harm inflate costs at the expense of necessary but neglected psychiatric and rehabilitative treatments.

COgnitive behavioural therapy vs standardised medical care for adults with Dissociative non-Epileptic Seizures (CODES): a multicentre randomised controlled trial protocol.

BACKGROUND: The evidence base for the effectiveness of psychological interventions for patients with dissociative non-epileptic seizures (DS) is currently extremely limited, although data from two small pilot randomised controlled trials (RCTs), including from our group, suggest that Cognitive Behavioural Therapy (CBT) may be effective in reducing DS occurrence and may improve aspects of psychological status and psychosocial functioning. METHODS/DESIGN: The study is a multicentre, pragmatic parallel group RCT to evaluate the clinical and cost-effectiveness of specifically-tailored CBT plus standardised medical care (SMC) vs SMC alone in reducing DS frequency and improving psychological and health-related outcomes. In the initial screening phase, patients with DS will receive their diagnosis from a neurologist/epilepsy specialist. If patients are eligible and interested following the provision of study information and a booklet about DS, they will consent to provide demographic information and fortnightly data about their seizures, and agree to see a psychiatrist three months later. We aim to recruit ~500 patients to this screening stage. After a review three months later by a psychiatrist, those patients who have continued to have DS in the previous eight weeks and who meet further eligibility criteria will be told about the trial comparing CBT + SMC vs SMC alone. If they are interested in participating, they will be given a further booklet on DS and study information. A research worker will see them to obtain their informed consent to take part in the RCT. We aim to randomise 298 people (149 to each arm). In addition to a baseline assessment, data will be collected at 6 and 12 months post randomisation. Our primary outcome is monthly seizure frequency in the preceding month. Secondary outcomes include seizure severity, measures of seizure freedom and reduction, psychological distress and psychosocial functioning, quality of life, health service use, cost effectiveness and adverse events. We will include a nested qualitative study to evaluate participants' views of the intervention and factors that acted as facilitators and barriers to participation. DISCUSSION: This study will be the first adequately powered evaluation of CBT for this patient group and offers the potential to provide an evidence base for treating this patient group. TRIAL REGISTRATION: Current Controlled Trials ISRCTN05681227 ClinicalTrials.gov NCT02325544.

Psychogenic nonepileptic seizures: diagnosis and initial management.

Psychogenic nonepileptic seizures (PNES; also known as pseudoseizures, nonepileptic attack disorder) are common. They continue to pose diagnostic difficulties, with mean delays from onset to diagnosis of several years, during which time they are often treated as epilepsy. The literature suggests that clinical diagnosis has limited reliability. However, it may be useful to regard the diagnosis of PNES as having two stages-- that of suspecting the diagnosis and that of confirming it. Clinical features of the history and spells allow the diagnosis of PNES to be suspected in the first place, so that the appropriate expertise and tests can be brought to bear. The diagnosis of PNES is usually confirmed by recording spells using video EEG. A minority of patients also have epilepsy. Once the diagnosis is made, initial management consists of communicating the diagnosis to the patient and carers in a clear and nonpejorative way, as well as withdrawing anticonvulsant medication with appropriate monitoring in patients with no evidence of epilepsy. In many patients, spells will cease without psychological intervention. Emergency healthcare utilization may drop sharply after explanation of the diagnosis, and this may occur even in patients whose spells continue. It is not clear to what degree these positive effects are maintained in the long term.