Functional Neurological Symptom Disorders Research Articles

From Hysteria to Functional Neurological Symptom Disorder: Developments in Clinical Diagnosis and Neurobiology

Changes in the nomenclature of functional neurological symptom disorders (FND) from the past to the present represent historical changes in understanding etiology. Today, there is still difficulty in excluding potential underlying neurological disorders. In addition, there is no consensus on the psychological mechanism leading to the disorder. As a result, diagnostic problems continue to exist. While functional neuroimaging studies show that suppression and conversion mechanisms, which are the concepts of the psychoanalytical theory, may have neural counterparts, neurobiological data suggests that the conversion model cannot be explanatory for every patient. The dorsolateral prefrontal cortex (dlPFC), amygdala, temporoparietal junction (TPJ), insula, anterior cingulate structures, and their connections come to the fore. The fact that the connections between the dlPFC and the hippocampus can prevent the recall of an unwanted memory, as well as the changes detected in the amygdala in these disorders and the increased connectivity between the amygdala and the motor areas, suggest an abnormal connection between emotions and the motor system. It is addressed how changes in the TPJ are related to the loss of the sense of agency. However, it is unclear whether the findings of these studies suggest a "predisposition", "onset of disorder", or "compensatory changes secondary to disorder". Exploring FND to learn how the brain and mind react to psychosocial stressors can be a turning point in understanding the brain-mind connection. The goal of this review is to present the history of the changes in terminology and perspective on this disorder that followed the establishment of psychoanalysis, as well as what kind of evidence has been presented regarding hysteria in light of advances in neuroscience

Agreement and disagreement in pediatric functional neurological symptom disorders: Comparing patient reported outcome measures (PROMs) and clinician assessments

Youth with functional neurological symptom disorder (FNSD) often perceive themselves as having limited capabilities, which may not align with clinical evaluations. This study assessed the disparities between clinician evaluations and patient-reported outcome measures (PROMs) regarding pain, motor function, and learning difficulties in youth with FNSD. Sixty-two youths with FNSD participated in this study, all of whom reported experiencing pain, motor problems, and/or learning difficulties. Clinicians also assessed these domains, resulting in a two-by-two categorization matrix: (1) agreement: child and clinician report "problems"; (2) agreement: child and clinician report "no problems"; (3) disagreement: child reports "problems" while the clinician does not; and (4) disagreement: clinician reports "problems" while the child does not. Agreement/disagreement differences were analyzed. No significant differences in prevalence were observed between the evaluators regarding pain (clinician-85%, child-88%), motor (clinician-98%, child-95%), or learning problems (clinician-69%, child-61%). More than 80% of the children and clinicians report pain and motor disorders. Instances in which children and clinicians reported learning problems (40.3%) exceeded cases in which both reported no problems (9.6%) or only the child reported problems (20.9%). Overall, the agreement between pain and motor function assessments was high (>90%), whereas that concerning learning difficulties was moderate (49.9%). Disagreement in pain/motor assessments was minimal (<5%), whereas for learning difficulties, disagreement rates were high (>20%). In conclusion, a significant concordance exists between PROMs and clinician assessments of pain and motor problems. However, the higher frequency of disagreements regarding learning difficulties emphasizes the importance of incorporating patient and clinician evaluations in pediatric FNSD treatment.

Misdiagnosis of functional neurological symptom disorders in paediatrics: Narrative review and relevant case report.

Functional neurological symptom disorders (FNSD) pose a common challenge in clinical practice, particularly in pediatric cases where the clinical phenotypes can be intricate and easily confused with structural disturbances. The frequent coexistence of FNSDs with other medical disorders often results in misdiagnosis. In this review, we highlight the distinctions between FNSD and various psychiatric and neurological conditions. Contrary to the misconception that FNSD is a diagnosis of exclusion, we underscore its nature as a diagnosis of inclusion, contingent upon recognizing specific clinical features. However, our focus is on a critical learning point illustrated by the case of a 14-year-old male initially diagnosed with FNSD, but subsequently found to have a rare primary monogenic movement disorder (paroxysmal kinesigenic dyskinesia, PKD). The crucial takeaway from this case is the importance of avoiding an FNSD diagnosis based solely on psychiatric comorbidity and suppressible symptoms. Instead, clinicians should diligently assess for specific features indicative of FNSD, which were absent in this case. This emphasizes the importance of making a diagnosis of inclusion. Extended follow-up and clinical-oriented genetic testing might help identify comorbidities, prevent misdiagnosis, and guide interventions in complex cases, which cannot be simply classified as "functional" solely because other conditions can be excluded.

Unexpected symptoms after concussion: Potential links to functional neurological and somatic symptom disorders

ObjectiveReporting of unexpected symptoms after concussion might, in some people, reflect a Functional Neurological Disorder (FND), Somatic Symptom Disorder (SSD), or exaggeration (feigning).This study aimed to determine whether reporting unexpected symptoms after concussion was associated with risk factors for FND/SSD, exaggeration, or both. MethodAdults with persistent symptoms following concussion (N = 77; 61% women) rated the presence and severity of unexpected neurological symptoms (from the Screening for Somatoform Symptoms scale, e.g., paralysis) and somatic symptoms (from the Patient Health Questionnaire-15, e.g., stomach pain) that did not overlap with post-concussion symptom scale items. The independent variables were risk factors for exaggeration (neuropsychological performance validity test failure and personal injury litigation) and predisposing and perpetuating factors for developing FND and/or SSD (e.g., fear avoidance behavior). ResultsWhen adjusting for all covariates, fear avoidance behavior was most strongly related to unexpected neurological symptoms (B = 0.11, 95% confidence interval = 0.05–0.18, p < .001), while current anxiety scores were most strongly related to unexpected somatic symptoms (B = 0.34, 95% confidence interval = 0.15–0.52, p < .001). Performance validity test failure and litigation were not significant predictors in either model. ConclusionUnexpected neurological and other somatic symptoms after concussion should not be dismissed as exaggeration. Psychological factors thought to perpetuate FND and SSD (e.g., fear avoidance behavior) may contribute to unexpected symptoms following concussion. More research is needed at the intersection of FND, SSD, and persistent post-concussive symptoms.

Maladaptive schemas of patients with functional neurological symptom disorder.

The psychological underpinnings of functional neurological symptom disorders (FNSD) remain poorly understood. A disintegration of explicit and implicit information processing in patients with FNSD has previously been suggested; however, this suggestion has so far received little empirical support. Trauma and maladaptive schemas probably reinforce disintegration in FNSD. The present study explored the occurrence of maladaptive schemas and investigated the impact of trauma-related maladaptive schemas in patients with FNSD. Forty-eight FNSD patients were assessed at the start of treatment using the Young Schema Questionnaire (YSQ-2) to explore maladaptive schemas. The Life Event Checklist (LEC-5) and the PTSD Checklist for DSM-5 (PCL-5) were used to explore trauma states, and the Sickness Impact Profile (SIP-68) was used to measure health dysfunction. The self-sacrifice schema scored within a clinically relevant range where no increased scores were found in other maladaptive schemas. Linear regression models showed a positive association between mistrust/abuse and severity of health dysfunction. Results suggest that maladaptive schemas play a modest role in FNSD at the start of treatment. It is suggested to examine the occurrence of maladaptive schemas in FNS-disordered patients with a longitudinal design.

How do Physicians Manage Functional Neurological Symptom Disorder and Somatic Symptom Disorder in the Emergency Department? A Vignette Study.

Introduction:We aimed to explore how physicians from different specialties approach the management of functional neurological symptom (conversion) and somatic symptom disorders in the emergency department compared with pulmonary embolism and how physicians’ professional and personal characteristics influence their diagnostic preferences.Methods:Using a vignette methodology, and cross-sectional design, three emergency department case vignettes of possible functional neurological symptom, somatic symptom disorder, and pulmonary embolism were presented to physicians from internal medicine, emergency medicine, and psychiatry. A structured survey including questions on diagnosis and management of these cases, and physicians’ professional and personal characteristics (childhood trauma, attachment style) was conducted.Results:Physicians from internal medicine and emergency medicine tended to consider functional neurological symptom disorder as ‘malingering’ while psychiatrists tended to diagnose the pulmonary embolism case as a psychiatric condition. Emergency medicine physicians preferred to manage functional neurological symptom disorder themselves, while other doctors endorsed recommending a psychiatric consultation. In the univariable model, being a physician from psychiatry, emergency medicine, or internal medicine; being a specialist, history of childhood sexual abuse, dismissive, and fearful attachment styles of doctors were significant predictors of diagnosing functional neurological symptom disorder as malingering. Being a psychiatrist stayed as the only significant predictor in the multivariable model.Conclusion:Objectively-aberrant functional neurological symptoms and subjective somatic symptoms may be creating different reactions in physicians. In the emergency department, physicians’ diagnostic and treatment preferences of conversion disorder may be influenced by lack of training in conversion disorder, rather than their personal characteristics.

Subjective versus objective measures of distress, arousal and symptom burden in patients with functional seizures and other functional neurological symptom disorder presentations: A systematic review.

Symptoms and functioning can be measured subjectively using self-report measures or objectively, based on physiological changes. This raises the question whether subjective and objective measures are closely correlated and - if not - whether one is more accurate or meaningful than the other, especially in patients with Functional Seizures (FS) or other Functional Neurological Symptom Disorders (FND), where subjective and objective observations may be thought particularly likely to deviate. This systematic review explores these questions focussing on measures of distress, arousal and symptom burden. Eighteen studies (12 FS, 6 other FND) capturing 396 FND patients were included. Eleven reported no correlation between subjective and objective measures. Only four studies reported significant correlations (r's=-0.74-0.59, p's<0.05). The small number of studies and diverse methodologies do not provide conclusive answers to the questions posed. Given that subjective and objective measures capture different aspects of current state or function, a combination of measurement approaches is likely to provide optimal information about patients' health state. In view of the attentional and perceptual alterations implicated in FND, the difference between objective and subjective measures may represent an interesting observation in its own right.

Nocebo-Hypothesis Cognitive Behavioral Therapy (NH-CBT) for Persons With Functional Neurological Symptoms (Motor Type): Design and Implementation of a Randomized Active-Controlled Trial.

Introduction: Functional Neurological Symptom Disorders (FNSD) are associated with high levels of disability and immense direct and indirect health costs. An innovative interdisciplinary rehabilitation approach for individuals with functional neurological symptoms of motor type–Nocebo-Hypothesis Cognitive Behavioral Therapy (NH-CBT)—combines CBT and movement retraining with video feedback embedded in a comprehensive explanatory model of the etiology of FNSD.Methods: This protocol describes the development and implementation of a phase II, parallel group, randomized controlled trial with blinded outcome assessors to compare the efficacy of NH-CBT with an active control condition (supportive counseling and movement retraining). Individuals meeting diagnostic criteria of an FNSD or psychogenic movement disorder will be randomly assigned to one of the 8-week interventions. Self-report scales of motor and other physical symptoms, symptom-related psychological variables, and assessor ratings of participants' mobility will be administered at baseline, and at 8- and 16-week follow-up. Adverse events will be monitored across all sessions and therapeutic alliance will be measured at the end of therapy. The primary statistical analysis will test the hypothesis that NH-CBT is more effective than the control intervention at the 8-week follow-up.Discussion: The therapeutic strategies of NH-CBT are theory-driven by assumptions of the predictive coding model of the etiology of FNSD. Strengths and limitations of this trial will be discussed.Trial registration: Australian New Zealand Clinical Trials Registry (ANZCTR; identifier: ACTRN12620000550909).

Triggers and Clinical Presentations of Functional Neurological Disorders: Lessons from World War 1

Introduction: The psychological contribution to functional neurological and somatic symptom disorders is a major topic in current medical debate. Objective: For an understanding of the processes leading to functional somatic symptoms, it is paramount to explore their relationship with stress and life events and to elucidate the contribution of cultural factors. Methods: A total of 937 case records of civilian and military patients with functional somatic disorders treated in London during World War 1 were analysed. Group differences in symptom profiles and contemporaneous diagnoses were tested with χ² tests. Results: Paralyses and speech disturbances were significantly more common in soldiers (43.3 and 17.2% of cases) than in civilian male (28.1 and 6.5%) and female patients (32.4 and 7.5%), whereas female patients had the highest rates of pain (48.6%) and somatic symptoms (67%). Triggers were identified in around two-thirds of cases and included accidents, physical illness, and work stress, in addition to the combat experience of the soldier patients. The nature of the trigger influenced symptom expression, with acute (combat and noncombat) events being particularly prone to trigger loss of motor function. Symptom profiles showed a great deal of multi-morbidity and overlap, although some symptom clusters were more (motor and speech disturbance) or less common (pain and loss of energy) in soldiers than civilians. Triggering life events in civilians were similar to those reported by patients with somatic symptom disorders today, with an important role of physical factors. Patterns of multi-morbidity and symptom clusters also resembled those of modern cohorts. Conclusions: Analysis of historical records, illness trajectories, and treatments can enhance the understanding of the presentation, mechanisms, and course of functional neurological and related disorders and their consistency over time.

Psychological comorbidities and functional neurological disorders in women with idiopathic urinary retention: International Consultation on Incontinence Research Society (ICI-RS) 2019.

Chronic urinary retention occurring in young women is poorly understood and a cause may not be found in a majority of cases. Different psychological comorbidities and functional neurological symptom disorders (FNDs) have been reported; however, these have been poorly explored. At the International Consultation on Incontinence Research Society meeting in 2019, a panel of clinicians generated a proposal to explore the relationship between psychological comorbidities, FNDs, and urinary retention in women with chronic idiopathic urinary retention. Psychological comorbidities such as depression and anxiety, and FNDs such as leg weakness and loss of consciousness, have been reported in women with idiopathic urinary retention. Individuals react differently to physical and emotional stressors, and experimental models have demonstrated a relationship between the stress response and developing urinary retention. Trauma, particularly sexual trauma, may be a shared risk factor for developing psychological comorbidities and urinary retention. Children with voiding postponement often suffer from psychological comorbidities and behavioral disturbances; however, there is no evidence to suggest that this progresses to urinary retention in adulthood. "Psychogenic urinary retention" has been described in the urology and psychiatry literature in the past, and anecdotal cases of successful voiding following psychotherapy have been reported, though the true pathophysiology of this entity is uncertain. Psychological and functional disorder comorbidities are reported in women with chronic urinary retention. The nature of the association between urinary retention and functional neurological disorder comorbidities needs to be further explored in terms of a disorder of bladder-brain interaction.

Signs of autonomic arousal precede tilt-induced psychogenic nonsyncopal collapse among youth

Characterizing the physiologic changes leading up to psychogenic nonsyncopal collapse (PNSC) may help to elucidate the processes that cause paroxysmal functional neurological symptom disorders and to clinically distinguish PNSC from syncope. Thus, we aimed to characterize preictal sweat rate, heart rate, and systolic blood pressure changes among patients with tilt-induced PNSC compared to patients with tilt-induced neurally mediated syncope. The presence of increased preictal sweating was compared between groups. Heart rates and systolic blood pressures were compared from the recumbent and tilted baselines to the periods 120 s and 30 s prior to PNSC and syncope. Patients with PNSC (n = 44) were more likely than patients with syncope (n = 44) to have preictal increases in sweating, n = 31 (70.5%) versus n = 21 (47.7%), p = 0.03, although all patients with syncope eventually developed a sweat response. Comparing the recumbent baseline to the period 30 s prior to PNSC, blood pressure (112 ± 9 versus 129 ± 13 mmHg, p < 0.001) and heart rate (76 ± 12 versus 119 ± 22 bpm, p < 0.001) increased. Similarly, comparing the tilted baseline to the period 30 s prior to PNSC, blood pressure (118 ± 12 versus 129 ± 13 mmHg, p < 0.001) and heart rate (95 ± 15 versus 119 ± 22 bpm, p < 0.001) increased. Preictal blood pressure and heart rate differed significantly between patients with PNSC and patients with syncope. In conclusion, signs of autonomic arousal (increased sweating, heart rate, and blood pressure) often precede tilt-induced PNSC. Sweating prior to fainting may not be useful in distinguishing PNSC from neurally mediated syncope.

Psychogenic Nonepileptic Seizures in Women.

Psychogenic nonepileptic seizures (PNES) are the most common type of functional neurological symptom disorders and are frequently diagnosed in tertiary care epilepsy monitoring units. These are associated with significant decline in social functioning and quality of life. The majority of patients with PNES are women, outnumbering men by a ratio of 3:1. Female sex preponderance occurs after puberty and usually before the age of 55 years. Many of the psychiatric risk factors in PNES (depression, anxiety, history of traumatic experiences, other somatic symptom disorders) are more common in women and may partially account for the difference in sex prevalence. Neurobiological and neurohumoral mechanisms may also play a role, but our understanding is limited at this point. In this review, we present information on epidemiology and risk factors, neurobiological and psychological mechanisms, clinical approach to diagnosis, evidence-based treatment, and long-term outcomes. We highlight findings related to differences between women and men in PNES. Most of these data are not decisive and require further corroboration. While the disorder may be more frequently suspected in women, all patients with suspected PNES deserve an objective and thorough investigation of their symptoms. Early and accurate identification of this disorder should be a priority, especially as evidence-based treatments, which may lead to improved outcomes, are increasingly available.

Youth With Psychogenic Non-Syncopal Collapse Have More Somatic and Psychiatric Symptoms and Lower Perceptions of Peer Relationships Than Youth With Syncope

BackgroundLittle is known about somatic and psychiatric symptoms and perceived peer relationships of patients with psychogenic nonsyncopal collapse. ObjectiveThis study aimed to compare somatic and psychiatric symptoms and other elements potentially related to functional neurological symptom disorders between youth with psychogenic nonsyncopal collapse and those with neurally mediated syncope. MethodsBefore testing, patients completed a structured interview and questionnaire addressing current symptoms, previous psychiatric diagnoses, referrals, diagnostic testing, prescribed medications, and patient self-ratings of anxiety, depression, and perceived peer relationships. ResultsCompared with patients with syncope (n = 60), patients with psychogenic nonsyncopal collapse (n = 60) had higher ratings for lightheadedness and vertigo, more abdominal pain, more chronic headaches, more fatigue, more sleep disturbances, more prescriptions for antidepressant medicines, more encephalograms performed, more referrals to psychiatry, and more psychiatric diagnoses including anxiety, depression, posttraumatic stress disorder, previous nonfainting conversion disorders, and eating disorders (all p < 0.05). Patients with psychogenic nonsyncopal collapse rated their anxiety (10.5 ± 7.7 versus 5.9 ± 5.8, p < 0.001) and depression (8.7 ± 8.3 versus 3.1 ± 5, p < 0.001) symptoms higher and their peer relationships (37 ± 12.3 versus 47.6 ± 7.9, p < 0.001) lower than patients with syncope. Peer relationships remained significantly lower (p = 0.001) when analyzed with anxiety and depression. ConclusionPatients with psychogenic nonsyncopal collapse have more symptom complaints and perceptions of poorer peer social interactions than patients with syncope. These results broaden our understanding of the biopsychosocial profile that increases an individual's vulnerability to psychogenic nonsyncopal collapse specifically and to functional neurological symptom disorders in general.

DIFFERENCES IN RELATIVES' AND PATIENTS' ILLNESS PERCEPTIONS IN FUNCTIONAL NEUROLOGICAL SYMPTOM DISORDERS COMPARED TO NEUROLOGICAL DISEASE

ObjectiveThe illness perceptions of the relatives of patients with functional neurological symptom disorders (FNSD) and the relation to patients' illness perceptions have been little studied. We aimed to compare illness...

Differences in relatives' and patients' illness perceptions in functional neurological symptom disorders compared with neurological diseases

ObjectiveThe illness perceptions of the relatives of patients with functional neurological symptom disorders (FNSDs) and their relation to the illness perceptions of the patients have been little studied. We aimed to compare illness perceptions of relatives of patients with FNSDs with those held by patients themselves. We used control pairs with neurological diseases (NDs) to examine the specificity of the findings to FNSDs. Material and methodsPatients with FNSDs (functional limb weakness and psychogenic nonepileptic seizures) and patients with NDs causing limb weakness and epilepsy, as well as their relatives, completed adapted versions of the Illness Perception Questionnaire — Revised (IPQ-R). ResultsWe included 112 pairs of patients with FNSDs and their relatives and 60 pairs of patients with NDs and their relatives. Relatives of patients with FNSDs were more likely to endorse psychological explanations and, in particular, stress as causal factors than patients with FNSDs (p<.001). Relatives of patients with FNSDs were also more pessimistic about the expected duration of the disorder and perceived a greater emotional impact compared with patients themselves (p<.001). However, the latter two differences between patients and relatives were also found in pairs of patients with NDs and their relatives. ConclusionThe main difference in illness perceptions between relatives and patients that appeared specific to FNSDs was a tendency for relatives to see psychological factors as more relevant compared with patients. Some other differences were observed between pairs of patients with FNSDs and their relatives, but the same differences were also seen in pairs of patients with NDs and their relatives. These other differences were, therefore, not specific to FNSDs. Discussion about possibly relevant psychological factors with patients suffering from FNSDs may be helped by including relatives.

Increased Pediatric Functional Neurological Symptom Disorders After the Boston Marathon Bombings: A Case Series

BackgroundFunctional neurological symptom disorders are frequently the basis for acute neurological consultation. In children, they are often precipitated by high-frequency everyday stressors. The extent to which a severe traumatic experience may also precipitate functional neurological abnormalities is unknown. MethodsFor the 2-week period after the Boston Marathon bombings, we prospectively collected data on patients whose presentation suggested a functional neurological symptom disorder. We assessed clinical and demographic variables, duration of symptoms, extent of educational impact, and degree of connection to the Marathon bombing. We contacted all patients at 6 months after presentation to determine the outcome and accuracy of the diagnosis. ResultsIn a parallel study, we reported a baseline of 2.6 functional neurological presentations per week in our emergency room. In the week after the Marathon bombings, this frequency tripled. Ninety-one percent of presentations were delayed by 1 week, with onset around the first school day after a city-wide lockdown. Seventy-three percent had a history of a prior psychiatric diagnosis. At the 6 months follow-up, no functional neurological symptom disorder diagnoses were overturned and no new organic diagnosis was made. ConclusionsPediatric functional neurological symptom disorder may be precipitated by both casual and high-intensity stressors. The 3.4-fold increase in incidence after the Boston Marathon bombings and city-wide lockdown demonstrates the marked effect that a community-wide tragedy can have on the mental health of children. Care providers must be aware of functional neurological symptom disorders after stressful community events in vulnerable patient populations, particularly those with prior psychiatric diagnoses.

Functional Neurological Symptom Disorders in a Pediatric Emergency Room: Diagnostic Accuracy, Features, and Outcome

BackgroundIn children, functional neurological symptom disorders are frequently the basis for presentation for emergency care. Pediatric epidemiological and outcome data remain scarce. ObjectiveAssess diagnostic accuracy of trainee's first impression in our pediatric emergency room; describe manner of presentation, demographic data, socioeconomic impact, and clinical outcomes, including parental satisfaction. Methods(1) More than 1 year, psychiatry consultations for neurology patients with a functional neurological symptom disorder were retrospectively reviewed. (2) For 3 months, all children whose emergency room presentation suggested the diagnosis were prospectively collected. (3) Three to six months after prospective collection, families completed a structured telephone interview on outcome measures. ResultsTwenty-seven patients were retrospectively assessed; 31 patients were prospectively collected. Trainees' accurately predicted the diagnosis in 93% (retrospective) and 94% (prospective) cohorts. Mixed presentations were most common (usually sensory-motor changes, e.g. weakness and/or paresthesias). Associated stressors were mundane and ubiquitous, rarely severe. Families were substantially affected, reporting mean symptom duration 7.4 (standard error of the mean ± 1.33) weeks, missing 22.4 (standard error of the mean ± 5.47) days of school, and 8.3 (standard error of the mean ± 2.88) of parental workdays (prospective cohort). At follow-up, 78% were symptom free. Parental dissatisfaction was rare, attributed to poor rapport and/or insufficient information conveyed. ConclusionsTrainees' clinical impression was accurate in predicting a later diagnosis of functional neurological symptom disorder. Extraordinary life stressors are not required to trigger the disorder in children. Although prognosis is favorable, families incur substantial economic burden and negative educational impact. Improving recognition and appropriately communicating the diagnosis may speed access to treatment and potentially reduce the disability and cost of this disorder.