Intense Imagery Movements May Lead to Maladaptive Daydreaming: A Case Series and Literature Review.

BACKGROUND: This case series highlights the connection between childhood intense imagery movements (IIM) and adult-reported maladaptive daydreaming (MD). Motor stereotypies occur in typically developing children and also with co-occurring neurodevelopmental differences. A subgroup with complex motor stereotypies reports accompanying intense imagery, often enhanced by the movements. This phenomenon can persist into adulthood and, in some cases, will need active management to prevent significant distress and impairment. CASES: Six adults, self-reporting maladaptive daydreaming associated with stereotypies, are presented to demonstrate the associations. LITERATURE REVIEW: The clinical significance and function of IIM and MD are unclear, but several hypotheses are discussed, including the mechanism of emotional regulation through sensory seeking, as a process for processing childhood psychological trauma, as intrusive thoughts or images as part of a subtype of Obsessive Compulsive Disorder, or as a result of diverse attentional networks seen in neurodevelopmental disorders. CONCLUSIONS: This paper highlights important connections between IIM and MD. Many adults with MD show a childhood origin of stereotypical movements. Whilst immersive daydreaming may provide creativity and emotional regulation, there is evidence of distress and impairment of function for some adults, leading to MD diagnoses. Recognizing this phenomenon is important for all neurologists and physicians working with stereotypical movements.

An investigation into mothers' experiences of their children's functional tic-like behaviour and tic attacks.

OBJECTIVE: This is the first study to systematically explore the lived experiences of sudden and new onset of severe functional tics from the perspective of the mother's experiences and describes their attempts to access support services in the United Kingdom. METHOD: Twenty-One mothers of young people aged between 12 to 17 years with functional tic-like behaviour (FTLB) took part in semi-structured interviews. Thematic analysis of the transcribed interviews revealed gaps and inconsistencies within the process of gaining access to professional services and a lack of support for the management of tics and functional tic-like movements, in addition to highlighting the impact it had on daily family life. RESULTS: The themes generated included the occurrence and development of tics, the severity and intensity of symptoms, the psychological impact on the family and the need to make recommendations for a clear care pathway. Managing the impact of the FTLB and co-occurring conditions such as suicidal ideation and self-harm, as well as the physical and emotional trauma, commonly contributed to feelings of isolation and helplessness, which impacted negatively on the family's ability to function and participate in society. CONCLUSIONS: The findings emphasize the urgent need to create a clear management pathway for those experiencing FTLB, including the need for more professionals with relevant knowledge, to improve the dialogue with families during the referral process, whilst prioritising the treatment of anxiety and other identified mental health concerns.

European Society for the Study of Tourette Syndrome 2022 criteria for clinical diagnosis of functional tic-like behaviours: International consensus from experts in tic disorders.

BACKGROUND AND PURPOSE: In 2020, health professionals witnessed a dramatic increase in referrals of young people with rapid onset of severe tic-like behaviours. We assembled a working group to develop criteria for the clinical diagnosis of functional tic-like behaviours (FTLBs) to help neurologists, pediatricians, psychiatrists, and psychologists recognize and diagnose this condition. METHODS: We used a formal consensus development process, using a multiround, web-based Delphi survey. The survey was based on an in-person discussion at the European Society for the Study of Tourette Syndrome (ESSTS) meeting in Lausanne in June 2022. Members of an invited group with extensive clinical experience working with patients with Tourette syndrome and FTLBs discussed potential clinical criteria for diagnosis of FTLBs. An initial set of criteria were developed based on common clinical experiences and review of the literature on FTLBs and revised through iterative discussions, resulting in the survey items for voting. RESULTS: In total, 24 members of the working group were invited to participate in the Delphi process. We propose that there are three major criteria and two minor criteria to support the clinical diagnosis of FTLBs. A clinically definite diagnosis of FTLBs can be confirmed by the presence of all three major criteria. A clinically probable diagnosis of FTLBs can be confirmed by the presence of two major criteria and one minor criterion. CONCLUSIONS: Distinguishing FTLBs from primary tics is important due to the distinct treatment paths required for these two conditions. A limitation of the ESSTS 2022 criteria is that they lack prospective testing of their sensitivity and specificity.

The spectrum of functional tic-like behaviours: Data from an international registry.

BACKGROUND AND PURPOSE: Between 2019 and 2022, there was a marked rise in adolescents/young adults seeking urgent help for functional tic-like behaviours (FTLBs). Given the global scale of this phenomenon, we aimed to pool cases from different institutions in an international registry to better characterize this spectrum and facilitate future longitudinal observation. METHODS: An international collaborative group from 10 tertiary referral centres for tic disorders collected retrospective data on FTLB patients who sought specialists' attention between the last quarter of 2019 and June 2022. An audit procedure was used for collection of data, which comprised demographics, course of presentation and duration, precipitating and predisposing factors, phenomenology, comorbidities, and pharmacological treatment outcome. RESULTS: During the study period, we collected data on 294 patients with FTLBs, 97% of whom were adolescents and young adults and 87% of whom were female. FTLBs were found to have a peak of severity within 1 month in 70% of patients, with spontaneous remissions in 20%, and a very high frequency of complex movements (85%) and vocalizations (81%). Less than one-fifth of patients had pre-existing primary tic disorder, 66% had comorbid anxiety disorders, 28% comorbid depressive disorders, 24% autism spectrum disorder and 23% attention deficit/hyperactivity disorder. Almost 60% explicitly reported exposure to tic-related social media content. The vast majority of pharmacologically treated patients did not report benefit with tic-suppressing medications. CONCLUSIONS: Our data from the largest multicentre registry of FTLBs to date confirm substantial clinical differences from primary tic disorders. Social modelling was the most relevant contributing factor during the pandemic. Future longitudinal analyses from this database may help understand treatment approaches and responsiveness.

Case report: Advice for schools on managing functional tic-like behaviours.

There has been an increase in the occurrence of sudden onset functional tic-like behaviours in adolescents during the COVID-19 pandemic, which has had a significant impact on the affected individual's ability to engage with education. The aim of this article is to generate discussion and inform practice within schools with regard to the management of functional tic-like behaviours. An advice sheet for schools has been produced based on clinical expertise and experience of consulting with schools around the management within education settings. Case examples are presented highlighting the importance and impact of these strategies. We also highlight the need for further evaluation of the effectiveness of the advice sheet in collaboration with schools and families.

Paediatric tic-like presentations during the COVID-19 pandemic.

BACKGROUND AND AIM: Clinical centres have seen an increase in tic-like movements during the COVID-19 pandemic. A series of children and adolescents are described. METHODS: A retrospective chart review of 34 consecutive paediatric patients presenting with sudden onset tic-like movements, seen over 6 months. RESULTS: 94% of patients were female, with an average age of sudden onset or increase of tic-like movements of 13.7 years. 44% had a previous diagnosis of tics, and 47% initially presented to an emergency department. Comorbid psychiatric and neurodevelopmental disorders were reported in 91% with 68% reporting anxiety. CONCLUSION: We highlight a dramatic presentation of sudden onset functional tic-like movements in predominantly female adolescents to help inform identification and management. There is need to research the neurobiological underpinnings and environmental exacerbating factors leading to these presentations and to explore effective therapeutic strategies.

Functional neurological movements in children: Management with a psychological approach.

AIM: To develop a cognitive behavioural treatment (CBT) approach that included novel attention training components to support symptom management in children with a primary diagnosis of Functional Movement Disorder (FMD). METHOD: Eighteen children (9 male and 9 female) with a mean age of 13 years (sd = 2.46, range 10-18 years) were assessed and completed CBT with novel attention training components. Treatment outcomes were measured using the Child Global Assessment Scale (CGAS) which was administered at baseline and post-treatment. RESULTS: Scores on the CGAS improved significantly post-treatment (p < 0.001) with all participants showing significant change in functioning on the basis of the Reliable Change Index (RCI), with clinically significant change across classification boundaries. INTERPRETATION: This case series provides support for the use of CBT with attention training components for the management of FMD. Larger trials are necessary to identify which individual treatment components are most effective and to better understand and quantify response to treatment. Future clinical treatment studies would benefit from the inclusion of objective measures of interoception and attentional focus.

Anxiety in children attending a specialist inherited cardiac arrhythmia clinic: a questionnaire study.

OBJECTIVES: Inherited cardiac arrhythmia syndromes are life-threatening conditions. There is a paucity of research examining the psychological impact of these conditions in children. This study had three main aims. The first was to explore how the Cardiac Anxiety Questionnaire (CAQ) performs in a child population. The second aim was to compare the level of anxiety of children with an inherited cardiac arrhythmia syndrome and children being screened due to a family history of an inherited cardiac arrhythmia syndrome to control children. The third aim was to examine associations between a sudden cardiac death in the immediate family and levels of anxiety. METHOD: 47 children with an inherited cardiac arrhythmia syndrome, 78 children with a family history and 75 control children completed the Revised Child Anxiety and Depression Scale (RCADS), the Cardiac Anxiety Questionnaire for Children (CAQ-C) and the Childhood Anxiety Sensitivity Index. Children were between the age of 8 and 16 years. RESULTS: The study found the CAQ-C had promising psychometric properties. There were no significant differences in total anxiety scores (as measured by the RCADS) between the three groups. There were significant differences in cardiac-focused anxiety scores between the three groups. CONCLUSIONS: The CAQ has promising psychometric properties in a child population. However, further research is needed. Children attending specialist inherited cardiac arrhythmia clinics should be targeted for routine psychological screening and offered psychological intervention where necessary.

Pre- and postsurgical cognitive trajectories and quantitative MRI changes in Rasmussen syndrome.

OBJECTIVE: To quantify the longitudinal cognitive trajectory, before and after surgery, of Rasmussen syndrome (RS), a rare disease characterized by focal epilepsy and progressive atrophy of one cerebral hemisphere. METHOD: Thirty-two patients (mean age = 6.7 years; 17 male, 16 left hemispheres affected) were identified from hospital records. The changes in intelligence scores during 2 important phases in the patients' journey to treatment were investigated: (1) during the preoperative period (n = 28, mean follow-up 3.4 years) and (2) from before to after surgery (n = 21 patients, mean time to follow-up 1.5 years). A volumetric magnetic resonance imaging (MRI) analysis of longitudinal changes in gray matter volume was conducted in a subsample of 18 patients. RESULTS: (1) IQ during the preoperative period: At baseline assessment (on average 2.4 years after seizure onset), the left RS group had lower verbal than nonverbal intellectual abilities, whereas the right group exhibited more difficulties in nonverbal than verbal intellect. Verbal and nonverbal scores declined during the follow-up in both groups, irrespective of the affected side. Hemispheric gray matter volumes declined over time in both groups in affected as well as unaffected hemispheres. (2) Postoperative IQ change: The left surgery group declined further in verbal and nonverbal intellect. The right group's nonverbal intellect declined after surgery, whereas verbal abilities did not. Patients with higher abilities preoperatively experienced large declines, whereas those with poorer abilities showed little change. Postoperative seizures negatively impacted on cognitive abilities. SIGNIFICANCE: During the chronic phase of the disease, parallel decline of verbal and nonverbal abilities suggest progressive bilateral hemispheric involvement, supported by evidence from MRI morphometry. Postsurgical cognitive losses are predicted by greater presurgical ability and continuing seizures. A shorter duration from seizure onset to surgery could reduce the postoperative cognitive burden by minimizing the decline in functions supported by the unaffected hemisphere.

Theory of mind, emotional and social functioning, and motor severity in children and adolescents with dystonic cerebral palsy.

AIMS: This cross-sectional study aimed to investigate whether children and adolescents with dystonic cerebral palsy (CP) present with emotional and social difficulties along side motor limitations. PARTICIPANTS/MEASURES: Twenty-two verbal and nonverbal children and adolescents with dystonic CP were compared with a normative sample of twenty children and adolescents on measures of theory of mind (ToM), emotion regulation (ER), and social difficulties (SD). RESULTS: Higher social and emotional difficulties were found in the dystonic CP group compared to the control group. Nonverbal participants with dystonic CP were found to present with greater social impairment and lower ToM ability than their verbal counterparts. Emotional regulation and hyperactivity and attentional difficulties (HAD) significantly predicted ToM ability and social difficulties. Lower Gross Motor Function Classification System (GMFCS) level and IQ also contributed to differences in ToM ability. INTERPRETATION: Findings support the need for greater attention to the emotional health and social development of children/adolescents with dystonic CP, along with assessments of motor difficulties in the planning and implementation of interventions and individual care plans. Further research is needed to explore links between motor disorder and mental state understanding in this clinical group.

The development of Attitudes of People from Ethnic Minorities to Help-Seeking for Dementia (APEND): a questionnaire to measure attitudes to help-seeking for dementia in people from South Asian backgrounds in the UK.

BACKGROUND: People from South Asian backgrounds present to dementia services relatively late, often responding to crises. We aimed to devise and validate a theory of planned behaviour questionnaire to measure attitudes that predict medical help-seeking for UK-based South Asian people, to assess the effectiveness of future interventions promoting earlier help-seeking. METHODS: We used focus groups to establish the content validity of culturally relevant questionnaire items, then asked participants to complete the questionnaire. We analysed reliability and validity and established the concurrent validity of questionnaire attitudes through correlation with willingness to seek help from a doctor for memory problems. We also correlated the scale with knowledge of dementia. RESULTS: The strongest predictor of willingness to seek help was perceived social pressure from significant others around help-seeking; these attitudes were associated with beliefs about the views of family members and embarrassment around help-seeking. Willingness to seek help was also strongly associated with attitudes about the benefits of seeing a doctor for memory problems, attitudes that were related to specific beliefs about what doctors can do to help. Attitudes in the questionnaire predicted 77% of variance in willingness to seek help, but no relationship was found with dementia knowledge. CONCLUSIONS: We present the Attitudes of People from Ethnic Minorities to Help-Seeking for Dementia (APEND) questionnaire, a valid and reliable measure of attitudes that influence help-seeking for dementia in people from South Asian backgrounds, which could assess the impact of intervention studies. We suggest that interventions target attitudes specified here, rather than dementia knowledge. Copyright © 2016 John Wiley & Sons, Ltd.

What parents think and feel about deep brain stimulation in paediatric secondary dystonia including cerebral palsy: A qualitative study of parental decision-making.

BACKGROUND: Dystonia is characterised by involuntary movements and postures. Deep Brain Stimulation (DBS) is effective in reducing dystonic symptoms in primary dystonia in childhood and to lesser extent in secondary dystonia. How families and children decide to choose DBS surgery has never been explored. AIMS: To explore parental decision-making for DBS in paediatric secondary dystonia. METHODS: Data was gathered using semi-structured interviews with eight parents of children with secondary dystonia who had undergone DBS. Interviews were analysed using Interpretative Phenomenological Analysis. RESULTS: For all parents the decision was viewed as significant, with life altering consequences for the child. These results suggested that parents were motivated by a hope for a better life and parental duty. This was weighed against consideration of risks, what the child had to lose, and uncertainty of DBS outcome. Decisions were also influenced by the perspectives of their child and professionals. CONCLUSIONS: The decision to undergo DBS was an ongoing process for parents, who ultimately were struggling in the face of uncertainty whilst trying to do their best as parents for their children. These findings have important clinical implications given the growing referrals for consideration of DBS childhood dystonia, and highlights the importance of further quantitative research to fully establish the efficacy of DBS in secondary dystonia to enhance informed decision-making.

Stable cognitive functioning with improved perceptual reasoning in children with dyskinetic cerebral palsy and other secondary dystonias after deep brain stimulation.

BACKGROUND: Dystonia is characterised by involuntary movements (twisting, writhing and jerking) and postures. Secondary dystonias are described as a heterogeneous group of disorders with both exogenous and endogenous causes. There is a growing body of literature on the effects of deep brain stimulation (DBS) surgery on the motor function in childhood secondary dystonias, however research on cognitive function after DBS is scarce. METHODS: Cognitive function was measured in a cohort of 40 children with secondary dystonia following DBS surgery using a retrospective repeated measures design. Baseline pre-DBS neuropsychological measures were compared to scores obtained at least one year following DBS. Cognitive function was assessed using standardised measures of intellectual ability and memory. RESULTS: There was no significant change in the assessed domains of cognitive function following DBS surgery. A significant improvement across the group was found on the Picture Completion subtest, measuring perceptual reasoning ability, following DBS. CONCLUSION: Cognition remained stable in children with secondary dystonia following DBS surgery, with some improvements noted in a domain of perceptual reasoning. Further research with a larger sample is necessary to further explore this, in particular to further subdivide this group to account for its heterogeneity. This preliminary data has potentially positive implications for the impact of DBS on cognitive functioning within the childhood secondary dystonia population.

Cognitive function in children with primary dystonia before and after deep brain stimulation.

BACKGROUND: Dystonia is characterised by involuntary movements (twisting, writhing and jerking) and postures. The effects of deep brain stimulation (DBS) surgery on the motor aspect of primary dystonias have been well reported, however, there is a paucity of research investigating its impact on cognitive function, particularly in childhood dystonia. We performed a follow-up of cognitive function in children with primary dystonia following DBS pallidal surgery. METHODS: Cognitive function was measured in a cohort of 13 children with primary or primary plus dystonia who had undergone DBS surgery using a retrospective case series design. Baseline pre-DBS neuropsychological measures were compared to scores obtained at least one year following DBS. Cognitive function was assessed using standardised measures of intellectual ability and memory. RESULTS: All children demonstrated improvements with regard to dystonia reduction, as measured by the Burke-Fahn-Marsden Dystonia Rating Scale (BFMDRS). Overall, cognition remained stable following DBS in the majority of the cohort. Individual case analysis revealed improvements in some domains of cognitive function in eight members of the cohort and a deterioration of certain domains in four. CONCLUSION: Cognition largely remained stable in children with primary/primary plus dystonia following DBS surgery, although further research with a larger sample is necessary to explore this statistically. Notwithstanding the limitations of a small size, this preliminary data has potentially positive implications for the impact of DBS on cognitive functioning within a paediatric population.