Validation of the Persistent Somatic Symptom Stigma scale for Healthcare Professionals (PSSS-HCP).

OBJECTIVE: Persistent somatic symptoms (PSS) describe recurrent or continuously occurring symptoms such as fatigue, dizziness, or pain that have persisted for at least several months. These include single symptoms such as chronic pain, combinations of symptoms, or functional disorders such as fibromyalgia or irritable bowel syndrome. While many studies have explored stigmatisation by healthcare professionals towards people with PSS, there is a lack of validated measurement instruments. We recently developed a stigma scale, the Persistent Somatic Symptom Stigma scale for Healthcare Professionals (PSSS-HCP). The aim of this study is to evaluate the measurement properties (validity and reliability) and factor structure of the PSSS-HCP. STUDY DESIGN AND SETTING: The PSSS-HCP was tested with 121 healthcare professionals across the United Kingdom to evaluate its measurement properties. Analysis of the factor structure was conducted using principal component analysis. We calculated Cronbach's alpha to determine the internal consistency of each (sub)scale. Test re-test reliability was conducted with a sub-sample of participants with a two-week interval. We evaluated convergent validity by testing the association between the PSSS-HCP and the Medical Condition Regard Scale (MCRS) and the influence of social desirability using the short form of the Marlowe Crowne Social Desirability Scale (MCSDS). RESULTS: The PSSS-HCP showed sufficient internal consistency (Cronbach's alpha = 0.84) and sufficient test-retest reliability, intraclass correlation = 0.97 (95% CI 0.94 to 0.99, p<0.001). Convergent validity was sufficient between the PSSS-HCP and the MCRS, and no relationship was found between the PSSS-HCP and the MCSDS. A three factor structure was identified (othering, uneasiness in interaction, non-disclosure) which accounted for 60.5% of the variance using 13 of the 19 tested items. CONCLUSION: The PSSS-HCP can be used to measure PSS stigmatisation by healthcare professionals. The PSSS-HCP has demonstrated sufficient internal consistency, test-retest reliability, convergent validity and minimal influence of social desirability bias. The PSSS-HCP has demonstrated potential to measure important aspects of stigma and provide a foundation for stigma reduction intervention evaluation.

Functional neurological disorder in Europe: regional differences in education and health policy.

BACKGROUND: Functional neurological disorder (FND) is a common cause of neurological disability. Despite recent advances in pathophysiological understanding and treatments, application of this knowledge to clinical practice is variable and limited. OBJECTIVE: Our aim was to provide an expert overview of the state of affairs of FND practice across Europe, focusing on education and training, access to specialized care, reimbursement and disability policies, and academic and patient-led representation of people with FND. METHODS: We conducted a survey across Europe, featuring one expert per country. We asked experts to compare training and services for people with FND to those provided to people with multiple sclerosis (MS). RESULTS: Responses from 25 countries revealed that only five included FND as a mandatory part of neurological training, while teaching about MS was uniformly included. FND was part of final neurology examinations in 3/17 countries, unlike MS that was included in all 17. Seventeen countries reported neurologists with an interest in FND but the estimated mean ratio of FND-interested neurologists to MS neurologists was 1:20. FND coding varied, with psychiatric coding for FND impacting treatment access and disability benefits in the majority of countries. Twenty countries reported services refusing to see FND patients. Eight countries reported an FND special interest group or network; 11 reported patient-led organizations. CONCLUSIONS: FND is largely a marginal topic within European neurology training and there is limited access to specialized care and disability benefits for people with FND across Europe. We discuss how this issue can be addressed at an academic, healthcare and patient organization level.

Reflections on the CODES trial for adults with dissociative seizures: what we found and considerations for future studies.

The COgnitive behavioural therapy versus standardised medical care for adults with Dissociative non-Epileptic Seizures multicentre randomised controlled trial is the largest, fully-powered study to test the clinical and cost-effectiveness of a psychotherapeutic intervention in this population. We also explored predictors or moderators of outcomes and investigated mechanisms of change in therapy. In this current review of findings, we discuss issues related to the design of the trial and consider the study's nested qualitative studies which were undertaken not only to shed light on the original research questions but to provide insights and recommendations for other researchers in the field of functional neurological disorder. Finally, we consider issues relating to the possible clinical application of our study findings.

Stigma in functional neurological disorder (FND) - A systematic review.

OBJECTIVE: The purpose of this systematic review was to evaluate stigma and Functional Neurological Disorder (FND) regarding: 1) prevalence and associated factors, 2) the nature and context of stigma in FND, and 3) stigma-reduction interventions. METHODS: We searched four relevant databases from inception to December 2023, using search terms relevant to FND and stigma themes. We employed the method of synthesis by "aggregation and configuration" to synthesise and analyse the data into emergent themes. RESULTS: We found 127 studies, spanning 148 countries, involving 18,886 participants. Of these, 4889 were patients, 13,123 were healthcare professionals, and 526 were caregivers. Quantitatively, stigma has been mainly studied in patients with functional seizures, and was higher than patients with epilepsy in three studies. Stigma experienced by patients is associated with poorer quality of life and caregiver burden. We found 10 themes and 29 subthemes revealing stigma as a systemic process, with intrapersonal, interpersonal and structural aspects. Few studies examined the perspective of caregivers, the public or online community. We identified six anti-stigma interventions. CONCLUSION: Stigma in FND is a layered process, and affects patient quality of life and provision of care. Stigma needs to be addressed from the top structures, at governmental level, so that appropriate care pathways can be created, giving patients with FND parity of esteem with other medical conditions.

Measuring persistent somatic symptom related stigmatisation: Development of the Persistent Somatic Symptom Stigma scale for Healthcare Professionals (PSSS-HCP).

OBJECTIVE: Persistent somatic symptoms (PSS) describe recurrent or continuously occurring symptoms such as fatigue, dizziness, or pain that have persisted for at least several months. These include single symptoms such as chronic pain, combinations of symptoms, or functional disorders such as fibromyalgia or irritable bowel syndrome. While stigmatisation by healthcare professionals is regularly reported, there are limited measurement instruments demonstrating content validity. This study develops a new instrument to measure stigmatisation by healthcare professionals, the Persistent Somatic Symptom Stigma scale for Healthcare Professionals (PSSS-HCP). METHODS: Development was an iterative process consisting of research team review, item generation and cognitive interviewing. We generated a longlist of 60 items from previous reviews and qualitative research. We conducted 18 cognitive interviews with healthcare professionals in the United Kingdom (UK). We analysed the relevance, comprehensibility and comprehensiveness of items, including the potential for social desirability bias. RESULTS: After research team consensus and initial feedback, we retained 40 items for cognitive interviewing. After our first round of interviews (n = 11), we removed 20 items, added three items and amended five items. After our second round of interviews (n = 7), we removed four items and amended three items. No major problems with relevance, comprehensibility, comprehensiveness or social desirability were found in remaining items. CONCLUSIONS: The provisional version of the PSSS-HCP contains 19 items across three domains (stereotypes, prejudice, discrimination), demonstrating sufficient content validity. Our next step will be to perform a validation study to finalise item selection and explore the structure of the PSSS-HCP.

How do I manage functional visual loss.

Functional visual loss is a subtype of functional neurological disorder (FND) and is a common cause of visual impairment seen in both general and neuro-ophthalmological practice. Ophthalmologists can generally diagnose functional visual loss reasonably confidently but often find it harder to know what to say to the patient, how to approach, or even whether to attempt, treatment. There is little evidence-based treatment despite studies showing up to 60% of adults having impactful symptoms on long-term follow-up. The last 20 years has seen large changes in how we understand, approach, and manage FND more widely. In this article, we set out our practical approach to managing functional visual loss which includes : 1) Make a positive diagnosis based on investigations that demonstrate normal vision in the presence of subjectively impaired vision, not just because tests or ocular exam is normal; 2) Explain and label the condition with an emphasis on these positive diagnostic features, not reassurance; 3) Consider eye or brain comorbidities such as migraine, idiopathic intracranial hypertension or amblyopia; 4) Consider working with an orthoptist using diagnostic tests in a positive way to highlight the possibility of better vision; 5) Develop simple treatment strategies for photophobia; 6) Consider psychological factors and comorbidity as part of assessment and therapy, but keep a broader view of aetiology and don't use this to make a diagnosis; 7) Other treatment modalities including hypnotherapy, transcranial magnetic stimulation and more advanced forms of visual feedback are promising candidates for functional visual loss treatment in the future.

Specialist physiotherapy for functional motor disorder in England and Scotland (Physio4FMD): a pragmatic, multicentre, phase 3 randomised controlled trial.

BACKGROUND: Functional motor disorder-the motor variant of functional neurological disorder-is a disabling condition that is commonly associated with poor health outcomes. Pathophysiological models have inspired new treatment approaches such as specialist physiotherapy, although evidence from large randomised controlled trials is absent. We aimed to assess the clinical effectiveness of a specialist physiotherapy intervention for functional motor disorder compared with treatment as usual. METHODS: In this pragmatic, multicentre, phase 3 randomised controlled trial at 11 hospitals in England and Scotland, adults with a clinically definite diagnosis of functional motor disorder, diagnosed by a neurologist, were included. Participants were randomly assigned (1:1, stratified by site) using a remote web-based application to either specialist physiotherapy (a protocolised intervention of nine sessions plus follow-up) or treatment as usual (referral to local community neurological physiotherapy). Individuals working on data collection and analysis were masked to treatment allocation. The primary outcome was the physical functioning domain of the 36-item short form health questionnaire (SF36) at 12 months after randomisation. The primary analysis followed a modified intention-to-treat principle, using a complete case approach; participants who were unable to receive their randomised treatment due to the suspension of health-care services during the COVID-19 pandemic were excluded from the primary analysis. This trial is registered with the International Standard Randomised Controlled Trial registry, ISRCTN56136713, and is completed. FINDINGS: Recruitment occurred between Oct 19, 2018, and March 11, 2020, pausing during the COVID-19 lockdown, and resuming from Aug 3, 2021, to Jan 31, 2022. Of 355 participants who were enrolled, 179 were randomly assigned to specialist physiotherapy and 176 to treatment as usual. 89 participants were excluded from the primary analysis due to COVID-19 interruption to treatment (27 were assigned to specialist physiotherapy and 62 to treatment as usual). After accounting for withdrawals (n=11) and loss to follow-up (n=14), the primary analysis included data from 241 participants (138 [91%] assigned specialist physiotherapy and 103 [90%] assigned treatment as usual). Physical functioning, as assessed by SF36, did not differ significantly between groups (adjusted mean difference 3·5, 95% CI -2·3 to 9·3; p=0·23). There were no serious adverse events related to the trial interventions. 35 serious adverse events were recorded in the specialist physiotherapy group by 24 participants (17·0%), and 24 serious adverse events were recorded in the treatment as usual group by 18 participants (17·0%); one death occurred in the specialist physiotherapy group (cause of death was recorded as suicide). All were considered unrelated to specialist physiotherapy. INTERPRETATION: Although more participants who were assigned specialist physiotherapy self-rated their motor symptoms as improved and had better scores on subjective measures of mental health, the intervention did not result in better self-reported physical functioning at 12 months. Both the specialist and community neurological physiotherapy appeared to be a safe and a valued treatment for selected patients with functional motor disorder. Future research should continue to refine interventions for people with functional motor disorder and develop evidence-based methods to guide treatment triage decisions. FUNDING: National Institute for Health and Care Research and Health Technology Assessment Programme.

Perspectives on the diagnosis and management of functional cognitive disorder: An international Delphi study.

BACKGROUND: Current proposed criteria for functional cognitive disorder (FCD) have not been externally validated. We sought to analyse the current perspectives of cognitive specialists in the diagnosis and management of FCD in comparison with neurodegenerative conditions. METHODS: International experts in cognitive disorders were invited to assess seven illustrative clinical vignettes containing history and bedside characteristics alone. Participants assigned a probable diagnosis and selected the appropriate investigation and treatment. Qualitative, quantitative and inter-rater agreement analyses were undertaken. RESULTS: Eighteen diagnostic terminologies were assigned by 45 cognitive experts from 12 countries with a median of 13 years of experience, across the seven scenarios. Accurate discrimination between FCD and neurodegeneration was observed, independently of background and years of experience: 100% of the neurodegenerative vignettes were correctly classified and 75%-88% of the FCD diagnoses were attributed to non-neurodegenerative causes. There was <50% agreement in the terminology used for FCD, in comparison with 87%-92% agreement for neurodegenerative syndromes. Blood tests and neuropsychological evaluation were the leading diagnostic modalities for FCD. Diagnostic communication, psychotherapy and psychiatry referral were the main suggested management strategies in FCD. CONCLUSIONS: Our study demonstrates the feasibility of distinguishing between FCD and neurodegeneration based on relevant patient characteristics and history details. These characteristics need further validation and operationalisation. Heterogeneous labelling and framing pose clinical and research challenges reflecting a lack of agreement in the field. Careful consideration of FCD diagnosis is advised, particularly in the presence of comorbidities. This study informs future research on diagnostic tools and evidence-based interventions.

Chronic idiopathic urinary retention: Comorbidity and outcome in 102 individuals.

OBJECTIVES: Chronic Idiopathic urinary retention is poorly understood. One small study suggests higher than expected rates of functional neurological disorder and pain comorbidity which may have implications for understanding the disorder. We investigated the frequency of functional neurological disorder, chronic pain other medical and psychiatric comorbidity, triggers of urinary retention, results of urodynamic assessment, medication history, management, and outcome in patients with chronic idiopathic urinary retention. METHODS: A consecutive retrospective electronic notes analysis was undertaken of patients with chronic idiopathic urinary retention presenting to a secondary care urology clinic between Jan 2018-Jan 2021, with follow-up to their most recent urological appointment. RESULTS: 102 patients were identified (mean age of 41.9 years, 98% female). 25% had functional neurological disorder (n = 26), most commonly limb weakness (n = 19, 19%) and functional seizures (n = 16, 16%). Chronic pain (n = 58, 57%) was a common comorbidity. Surgical and medical riggers to urinary retention were found in almost half of patients (n = 49, 48%). 81% of patients underwent urodynamic assessment (n = 83). Most frequently no specific abnormality was reported (n = 30, 29%). Hypertonic urethral sphincter was the most identified urodynamic abnormality (n = 17, 17%). We noted high levels of opioid (n = 50, 49%) and benzodiazepine (n = 27, 26%) use. Urinary retention resolved in only a small number of patients (n = 6, 6%, median follow up 54 months), in three cases spontaneously. CONCLUSION: This preliminary data suggests idiopathic urinary retention is commonly comorbid with functional neurological disorder, and chronic pain, suggesting shared mechanisms.

How stigma unfolds for patients with Functional Neurological Disorder.

OBJECTIVE: The aim of this study was to explore experiences of stigma in Functional Neurological Disorder (FND) from the perspective of the patient as it manifests from the onset of symptoms, up to diagnosis and subsequently. BACKGROUND: The existing literature clearly shows that stigma exists for many patients with FND, and is associated with poorer quality of life. However, it is less clear how stigma unfolds, and how it can be alleviated. METHODS: We performed a qualitative interview study with patients who were diagnosed with FND, using data based on semi-structured interviews. Participants were recruited purposively via outpatient clinics. We analysed the data using a reflexive thematic analytic approach, through the lens of recognised stigma frameworks. RESULTS: 15 participants were included in the study, aged between 19 and 68 years, with varying presentations of FND. We identified six themes and 16 subthemes relevant to their stigma trajectory. We found that stigma unfolds through four main domains: 1) through their symptom experience; 2) through "othering" by the healthcare system; 3) through everyday interactions; and 4) from within the self. Across these four domains was a central theme of 5) stages of knowledge, which both fuelled and countered stigma. Lastly, 6) validation of the patient experience emerged as a theme that alleviated stigma. CONCLUSIONS: Stigma did not unfold as a linear process, rather it came from multiple interacting sources. Interventions to target stigma could take the form of improved clinician training, communication, especially around point of diagnosis, and public interventions, co-produced with patients with FND.

Pain and functional neurological disorder: a systematic review and meta-analysis.

BACKGROUND: Functional neurological disorder (FND) is characterised by neurological symptoms, such as seizures and abnormal movements. Despite its significance to patients, the clinical features of chronic pain in people with FND, and of FND in people with chronic pain, have not been comprehensively studied. METHODS: We systematically reviewed PubMed, Embase and PsycINFO for studies of chronic pain in adults with FND and FND in patients with chronic pain. We described the proportions of patients reporting pain, pain rating and timing, pain-related diagnoses and responsiveness to treatment. We performed random effects meta-analyses of the proportions of patients with FND who reported pain or were diagnosed with pain-related disorders. RESULTS: Seven hundred and fifteen articles were screened and 64 were included in the analysis. Eight case-control studies of 3476 patients described pain symptoms in a higher proportion of patients with FND than controls with other neurological disorders. A random effects model of 30 cohorts found that an estimated 55% (95% CI 46% to 64%) of 4272 patients with FND reported pain. Random effects models estimated diagnoses of complex regional pain syndrome in 22% (95% CI 6% to 39%) of patients, irritable bowel syndrome in 16% (95% CI 9% to 24%) and fibromyalgia in 10% (95% CI 8% to 13%). Five studies of FND diagnoses among 361 patients with chronic pain were identified. Most interventions for FND did not ameliorate pain, even when other symptoms improved. CONCLUSIONS: Pain symptoms and pain-related diagnoses are common in FND. Classification systems and treatments should routinely consider pain as a comorbidity in patients with FND.

The Impact of Depression, Anxiety and Personality Disorders on the Outcome of Patients with Functional Limb Weakness - Individual Patient Data Meta-Analysis.

OBJECTIVE: Psychiatric comorbidities such as depression, anxiety, and personality disorders are common in patients with functional limb weakness/paresis (FND-par). The impact of these conditions on the prognosis of FND-par has not been systematically reviewed. The aim of this study was to identify a potential prognostic effect of comorbid depression, anxiety, and/or personality disorder on prognosis in patients with FND-par. METHODS: A systematic review was performed to identify studies that reported measures of baseline depression, anxiety, and/or personality disorder, and physical disability. An individual patient data meta-analysis was subsequently performed. RESULTS: Eight studies comprising 348 individuals were included (7 prospective cohorts; 1 case-control study). There was heterogeneity in sample size, follow-up duration, and treatment modality. Depression and anxiety were present in 51.4% and 53.0% of FND-par patients, respectively. In individuals whose FND-par improved, there was no significant difference between those with versus without depression (52.6% vs 47.4%, p = 0.69) or those with versus without anxiety (50.3% vs 49.7%, p = 0.38). Meta-analysis showed no clear impact of baseline depression or anxiety per se [pooled OR for depression 0.85 (95%CI 0.50-1.45; p = 0.40) and anxiety 0.84 (95%CI 0.51-1.38; p = 0.91)]; and of depression or anxiety severity [pooled OR for depression 1.23 (95%CI 0.63-2.39; p = 0.91) and anxiety 1.40 (95%CI 0.70-2.78; p = 0.58)] on FND-par outcome. Insufficient data were available to assess the impact of personality disorders. CONCLUSION: We found no evidence that depression or anxiety influenced outcome in FND-par. Large-scale, prospective studies in FND-par, and other FND subtypes, are needed to fully contextualize the impact of concurrent mental health concerns on outcomes.

Lessons from a Neurologist After 25 Years of Functional Neurological Disorder Subspeciality Practice.

The idea that a neurologist would do an 'FND clinic' or that functional neurological disorder (FND) could be a subspecialty of neurology would have been outlandish even 20 years ago but has become a reality in many places around the world. In this personal review, I reflect on 25 years of being a neurologist with an interest in FND, initially as a research fellow and later as a consultant/attending. I review lessons from diagnosis and management in the hope that they may assist other neurologists embarking on a similar career as well as other health professionals whose roles overlap.

Setting up Functional Neurological Disorder Treatment Services: Questions and Answers.

Functional neurologic disorder (FND) is commonly encountered across outpatient and inpatient medical settings. Given the potential for a high burden of disability in some patients and mounting evidence for the efficacy of FND-specific multidisciplinary treatment services, expanding clinical services for this population is a necessity. In this perspective article, we discuss considerations for creating FND services, including the types of services that exist, how to start, how to identify appropriate referrals, and how to develop and monitor individualized treatment plans. In addition, we discuss how this effort can be done sustainably - balancing patient needs with limited healthcare resources.

Clinical signs in functional cognitive disorders: A systematic review and diagnostic meta-analysis.

OBJECTIVE: Functional cognitive disorder (FCD) accounts for around a third of patients attending specialized memory clinics. It is also overrepresented in patients with other functional and somatic diagnoses. So far, no long-term diagnostic validity studies were conducted, and a positive diagnostic profile is yet to be identified. We aimed to review the literature on diagnostic signs and symptoms that allow for a discrimination between FCD and neurodegeneration. METHODS: Systematic review of Ovid-Medline®, Embase and PsycINFO databases. Relevant clinical features were extracted including demographics, symptom history, comorbidities, language and interaction profiles and cognitive assessments. Studies with quantifiable diagnostic accuracy data were included in a diagnostic meta-analysis. RESULTS: Thirty studies (N = 8602) were included. FCD patients were younger, more educated, and more likely to have a family history of older onset dementia, abrupt symptom onset, and higher rates of anxiety, depression and sleep disturbance. Promising language profiles include longer duration of spoken answer, elaborated examples of memory failures, ability to answer compound and personal questions, and demonstration of working memory during interaction. The pooled analysis of clinical accuracy of different signs revealed that attending alone and bringing a handwritten list of problems particularly increase the odds of a FCD diagnosis. Current evidence from neuropsychometric studies in FCD is scarce. CONCLUSIONS: Our systematic review reinforces that positive signs contribute for an early differentiation between FCD and neurodegeneration in patients presenting with memory complaints. It is the first to attain quantitative value to clinical observations. These results will inform future diagnostic decision tools and intervention testing.