Exploring role-related dysphonia as a trigger in functional neurological voice disorder: a single case study in body psychotherapy

Interoception, Voice Symptom Reporting, and Voice Disorders

Twelve-Month Clinical Follow-Up Study of Voice Patients' Recovery Using the Voice Activity and Participation Profile (VAPP)

Stress system dysregulation is considered to have an important role in the aetiology of paediatric functional neurological (conversion) disorder. This study examined salivary cortisol and α-amylase awakening responses in children with functional neurological disorder to determine activation patterns of the hypothalamic-pituitary-adrenal axis and sympathetic system. A healthy cortisol awakening response involves a robust increase in cortisol within 30 minutes of awakening. Alpha-amylase awakening response is variable in children. Cortisol and α-amylase were measured in saliva from 32 patients with functional neurological disorder (26 girls and 6 boys, aged 11.3-16.1 years) and 31 healthy controls (23 girls and 8 boys, aged 8.6-17.7 years). Saliva samples were collected using a Salivette sampling device at two time points - upon awakening and 30 minutes after awakening. Patients with functional neurological disorder showed a decrease in cortisol awakening response (-4 nmol.min/L) and controls showed an increase (107 nmol.min/L), t(55) = -.4.6, p < 0.001. Within the functional neurological disorder group, 57% showed an attenuated cortisol awakening response and 43% showed an obliterated/reversed cortisol awakening response: Cortisol awakening response was negatively correlated with adverse childhood experiences, r(58) = -0.6, p = 0.002, and subjective distress (total Depression Anxiety and Stress Scales score), r(58) = -0.4, p = 0.050. In controls, cortisol awakening response showed no correlation with adverse childhood experiences and a positive correlation with subjective distress, r(56) = 0.4, p = 0.023. Total cortisol remained similar between the functional neurological disorder and control group. No significant differences were observed between the functional neurological disorder and control group in any of the α-amylase analyses. The results suggest dysregulation of the hypothalamic-pituitary-adrenal axis in children with functional neurological disorder. Hypothalamic-pituitary-adrenal dysregulation in children with functional neurological disorder may contribute to comorbid symptoms of fatigue, sleep disturbance and subjective loss of well-being because circadian rhythms and energy metabolism are disrupted. Hypothalamic-pituitary-adrenal dysregulation - and changes in glucocorticoid (cortisol) signalling at the molecular level - may also contribute to increased vulnerability for functional neurological disorder symptoms because of epigenetically mediated changes to neural networks implicated in functional neurological disorder.

Chapter 34 - Functional voice disorders: Clinical presentations and differential diagnosis

Evaluating the evidence of treatment efficacy allows clinicians to select the delivery of the intervention that is most likely to improve the patient outcomes for a given clinical condition. Collation and critical appraisal of this evidence highlights areas of strength but also areas of research weakness. This paper provides a collation and summary appraisal of the evidence of treatment efficacy for voice therapy for a number of voice disorders that are routinely treated by speech pathologists/therapists. Table 1 lists a hierarchy that is commonly used and is highly applicable to behavioural interventions (www.joannabriggs. edu.au). It is interesting to note that this hierarchy also includes level IV evidence which acknowledges opinion of respected authorities, clinical experience and reports of expert committees. Strictly speaking, this is not evidence at all. The summary of evidence in the following sections of this paper will refer to the hierarchical level detailed in Table 1. However, Robey1 argues that evaluation of treatment effectiveness must also be considered within a broader organising structure for conducting clinical-outcome research activities. His five-phase model (Table 2) describes a logically ordered sequence from initial ‘proof of concept’ via various stages of ‘treatment efficacy’ and then ‘treatment effectiveness’ through to complete adaptation into clinical practice. Treatment efficacy research (Phase 1–3) examines clinical outcomes in an environment that ‘minimizes all sources of extraneous or confounding variation’ and ‘indexes the maximum potential of a treatment protocol for bringing about change’ (pg 402). In contrast, treatment effectiveness research examines the benefit of an intervention ‘provided in a typical fashion by typical practitioners to typical patients in typical clinical settings’ (pg 402). Therefore, the ultimate goal is evidence of treatment effectiveness but effectiveness can only be tested when efficacy has been firmly established. However, as Pring2 highlights, these essential phases of building an evidence base can often be overlooked in the ‘rush’ to publish large group trials and make claims about clinical effectiveness. The five levels are summarised in Table 2. This paper addresses the literature and evidence base for speech/voice therapy intervention for five types of voice disorders which commonly utilise voice therapy intervention as a primary strategy. These are functional voice disorders, vocal nodules, organic voice disorders, unilateral vocal fold paralysis (UVFP) and Parkinson’s disease voice disorder. This evidence will be categorised according to Robey’s five-phase model. The study descriptions and levels consistent with the Joanna Briggs terminology listed in Table 1 will also be documented. Each section also includes a summary table of all recent relevant voice treatment efficacy published studies.

Functional speech and voice disorders represent a common subtype of functional movement disorder (FMD). This chapter reviews the epidemiology of functional speech and voice disorders, the general approach to their diagnosis, and the characteristics of specific functional speech and voice disorders that allow them to be distinguished from other neurological disorders affecting voice and speech production. While most commonly presenting as functional dysphonia, stuttering and prosodic abnormalities, functional speech and voice disorders can present with a variety of heterogeneous clinical features, and can be challenging to differentiate from motor speech disorders. As with other subtypes of FMD, a careful approach to the history and physical examination is required to arrive at the proper diagnosis. The potential for rapid reversibility of symptoms with appropriate treatment underscores the importance of prompt and accurate diagnosis of functional speech and voice disorders.KeywordsFunctional speech disorderFunctional voice disorderFunctional dysphoniaMuscle tension dysphoniaFunctional stutteringForeign accent syndrome

Stressful life events and maltreatment in conversion (functional neurological) disorder: systematic review and meta-analysis of case-control studies

With the creation of the Somatic Symptom and Related Disorders category of the Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition in 2013, the functional neurological (symptom) disorder diagnostic criteria underwent transformative changes. These included an emphasis on ‘rule-in’ physical examination signs/semiological features guiding diagnosis and the removal of a required proximal psychological stressor to be linked to symptoms. In addition, the Diagnostic and Statistical Manual of Mental Disorders, Fourth Edition somatization disorder, somatoform pain disorder and undifferentiated somatoform disorder conditions were eliminated and collapsed into the Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition somatic symptom disorder diagnosis. With somatic symptom disorder, emphasis was placed on a cognitive-behavioural (psychological) formulation as the basis for diagnosis in individuals reporting distressing bodily symptoms such as pain and/or fatigue; the need for bodily symptoms to be ‘medically unexplained’ was removed, and the overall utility of this diagnostic criteria remains debated. A consequence of the Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition restructuring is that the diagnosis of somatization disorder that encompassed individuals with functional neurological (sensorimotor) symptoms and prominent other bodily symptoms, including pain, was eliminated. This change negatively impacts clinical and research efforts because many patients with functional neurological disorder experience pain, supporting that the Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition would benefit from an integrated diagnosis at this intersection. We seek to revisit this with modifications, particularly since pain (and a Diagnostic and Statistical Manual of Mental Disorders, Fourth Edition somatization disorder comorbidity, more specifically) is associated with poor clinical prognosis in functional neurological disorder. As a first step, we systematically reviewed the Diagnostic and Statistical Manual of Mental Disorders, Fourth Edition somatization disorder literature to detail epidemiologic, healthcare utilization, demographic, diagnostic, medical and psychiatric comorbidity, psychosocial, neurobiological and treatment data. Thereafter, we propose a preliminary revision to the Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition allowing for the specifier functional neurological disorder ‘with prominent pain’. To meet this criterion, core functional neurological symptoms (e.g. limb weakness, gait difficulties, seizures, non-dermatomal sensory loss and/or blindness) would have ‘rule-in’ signs and pain (>6 months) impairing social and/or occupational functioning would also be present. Two optional secondary specifiers assist in characterizing individuals with cognitive-behavioural (psychological) features recognized to amplify or perpetuate pain and documenting if there is a pain-related comorbidity. The specifier of ‘with prominent pain’ is etiologically neutral, while secondary specifiers provide additional clarification. We advocate for a similar approach to contextualize fatigue and mixed somatic symptoms in functional neurological disorder. While this preliminary proposal requires prospective data and additional discussion, these revisions offer the potential benefit to readily identify important functional neurological disorder subgroups—resulting in diagnostic, treatment and pathophysiology implications.

The goal of this study was to assess and compare voice handicap in patients with benign organic and functional dysphonia. Voice handicap was assessed using a German version of the Voice Handicap Index. A total of 108 German speaking patients, 64 women and 44 men aged 45.3+/-15.1 years with benign organic ( n=69) or functional ( n=39) voice disorders completed the questionnaire immediately after clinical examination and without knowing their individual diagnosis. Fifty individuals without voice complaints and normal voice status, 28 women and 22 men aged 47.1+/-15.5 years, served as controls. Significant differences ( P<0.001) were found between all dysphonic patients and the control group. When comparing dysphonic patients according to their gender and diagnosis group (organic or functional) no significant differences were detected. It can no longer be assumed that patients with functional voice disorders over emphasize their disease. The data obtained in this study prove that there are no gender specific differences in the way a voice disorder is experienced. There are also no differences in the way patients with a different language background experience their voice handicap.

The purpose of this study was to compare professional (Pro) and non-professional (Non-pro) voice users with voice disorders in self-reporting voice evaluation using Korean-Voice Handicap Index (K-VHI) and Korean-Voice Related Quality of Life (K-VRQOL). In addition, those were compared by voice quality and voice disorder type. 94 Pro and 106 Non-pro were asked to fill out the K-VHI and K-VRQOL, perceptually evaluated on GRBAS scales, and divided into three types of voice disorders (functional, organic and neurologic) by an experienced speech-language pathologist and an otolaryngologist. The results showed that the functional (F) and physical (P) scores of K-VHI in Pro group were significantly higher than those in Non-pro group. As the voice quality evaluated by G scale got worse, the scores of all aspects except emotional (E) of K-VHI and social-emotional (SE) of K-VRQOL were higher. All scores of K-VHI and K-VRQOL in neurologic voice disorders were significantly higher than those in functional and organic voice disorders. In conclusion, professional voice users are more sensitive to their functional and physical handicap resulted by their voice problems and that goes double for the patients with severe and neurologic voice disorders.

Functional Neurologic Disorders

Functional neurological disorders (FND), a subtype of functional disorders (FD), are a frequent motive for neurology referrals. The various presentations and the unknown physiopathology of FD have led to the multiplication of terms describing these disorders over the years. We examined the FD-related articles published from 1960 to 2020 in PubMed and PsycINFO databases. We searched for: psychogenic, somatization, somatoform, medically unexplained symptoms, hysteria, conversion disorder, dissociative, functional neurological disorder, and functional disorder. Use rates in the title, abstract, keyword, or MeSH fields were collected over successive 5-year periods. After correcting for off-topic results, we examined proportional distribution over time, term associations, and disciplinary fields (neurologyandpsychiatry). Term impact was estimated via H-index and number of citations. We found that none of the terms is prevailing in the recent medical literature. We observed three trends in the use rates: stability, increase, and decrease of use over time. While most of the terms were present in a stable proportion of the publications, hysteria and psychogenic lost popularity over time. We found a differential preference for terminology between disciplines. Functional neurological disorder showed the highest citation impact, yielding 10% of highly cited publications. We found a dynamic and evolving use of the different terms describing FD in the last 60years. Despite the tendency to use the term functional in the recent highly cited publications, its low prevalence and coexistence with several other terms suggest that a precise, explanatory and non-offensive term remains yet to be found.

Successful Treatment of Functional Neurologic Symptom Disorder (Conversion Disorder) With Subdissociative Dose Ketamine in the Emergency Department.

Published best-practice guidelines and standardized protocols for voice assessment recommend multidisciplinary evaluation utilizing a comprehensive range of clinical measures. Previous studies report variations in assessment practices when compared with these guidelines. To provide an up-to-date evaluation of current global multidisciplinary practice patterns and the opinions of otolaryngologist, ear, nose and throat (ENT) and speech-language pathology (SLP) clinicians on initial assessment and differential diagnosis of adults with voice disorders (VDs). ENTs and SLPs worldwide who had worked with VDs within the last 10 years completed an anonymous online survey. Themes explored demographic information about the clinical practice, information about diagnostic assessment pathways, clinical assessments routinely used for initial voice evaluation and clinician perceived value of clinical assessments important for diagnosis. Patterns in the clinical practice of 88 SLPs and 21 ENTs from 18 countries with 1 to more than 25 years' experience were analysed. Clinicians provided services across a range of locations, and a range of assessment pathways was available for initial evaluation. Case history, laryngoscopy and auditory-perceptual measures were the most frequently selected assessments. Most clinicians favoured formal assessment measures for auditory-perceptual evaluation. Clinicians placed equal weighting on ENT and SLP assessment to aid diagnosis for muscle tension VDs and functional neurological voice disorders (FVDs). Practice patterns for initial diagnostic voice assessment are largely consistent with the currently published guidelines. Decisions for the selection of assessment tools vary according to VD classification, and assessment decisions appear to be guided by case history. Clinicians are not always following established protocols for obtaining reliable standardized measures. Further research is needed to understand the barriers to adhering to standardized protocols and to develop evidence for the use of case history in the process of VD diagnosis. What is already known on the subject Best-practice guidelines recommend a multidisciplinary and multidimensional assessment of adults with vocal symptoms. Prior uni-disciplinary survey studies have reported a divergence in clinical practice with the recommended guidelines. No previous studies have examined otolaryngologists and SLPs concurrently to investigate the multidisciplinary approach clinicians' use in a diagnostic voice assessment. What this paper adds to the existing knowledge This study highlights new insights into multidisciplinary voice evaluation practice patterns with an emphasis on diagnostic assessment from a global perspective. The findings build on prior research exploring clinical assessment pathways, service utilization and clinicians' preferences when selecting clinical tools to inform a differential diagnosis. What are the potential or actual clinical implications of this work? This paper provides insights to inform future service and resource planning to ensure the delivery of evidenced-based diagnostic assessment pathways. This study also makes recommendations for areas of future research to understand barriers to clinicians following recommended best-practice guidelines.

Relationships Among Smoking, Organic, and Functional Voice Disorders in Korean General Population