DEVELOPMENTAL MEDICINE & CHILD NEUROLOGY
ORIGINAL ARTICLE
Intense imagery movements: a common and distinct paediatric subgroup of motor stereotypies SALLY ROBINSON 1
| MARTIN WOODS 1 | FRANCESCO CARDONA 2 | VALENTINA BAGLIONI 2 |
TAMMY HEDDERLY 1 1 Tic and Neurodevelopmental Movements (TANDeM), Children’s Neurosciences Centre, Evelina London Children’s Hospital, Guy’s and St Thomas’ NHS Foundation Trust, London, UK. 2 Department of Paediatrics and Child Neuropsychiatry, University La Sapienza of Rome, Rome, Italy. Correspondence to Tammy Hedderly at, TANDeM, Children’s Neurosciences Centre, St Thomas’ Hospital, Westminster Bridge Road, London, SE1 7EH, UK. E-mail: [email protected] This article is commented on by Freeman on pages 1139–1140 of this issue.
PUBLICATION DATA
Accepted for publication 24th April 2014. Published online 20th June 2014. ABBREVIATIONS
ASD IIM OCB
Autism spectrum disorder Intense imagery movements Obsessive–compulsive behaviour
AIM The aim of this article is to describe a subgroup of children who presented with stereotyped movements in the context of episodes of intense imagery. This is of relevance to current discussions regarding the clinical usefulness of diagnosing motor stereotypies during development. METHOD The sample consisted of 10 children (nine males, one female; mean age 8y 6mo [SD 2y 5mo], range 6–15y). Referrals were from acute paediatricians, neurologists, and tertiary epilepsy services. Children were assessed by multidisciplinary teams with expertise in paediatric movement disorders. RESULTS Stereotypies presented as paroxysmal complex movements involving upper and lower limbs. Imagery themes typically included computer games (60%), cartoons/films (40%), and fantasy scenes (30%). Comorbid developmental difficulties were reported for 80% of children. Brain imaging and electrophysiological investigations had been conducted for 50% of the children before referral to the clinic. INTERPRETATION The descriptive term ‘intense imagery movements’ (IIM) was applied if (after interview) the children reported engaging in acts of imagery while performing stereotyped movements. We believe these children may form a common and discrete stereotypy subgroup, with the concept of IIM being clinically useful to ensure the accurate diagnosis and clinical management of this paediatric movement disorder.
Movement disorders are typically classified by clinical descriptions of physical signs, with the term stereotypy referring to a wide range of movements, behaviours and/or vocalizations that are repetitive, seemingly driven, and lack clear function (for review see Barry et al.1 or Goldman et al.2). There are on-going discussions regarding the clinical usefulness of the concept3–5 and the need for a more clinically useful definition that describes a cohesive group of conditions linked pathophysiologically has been proposed by Edwards et al.3 Nevertheless, to date, there are no reports in the literature that consider the differential diagnosis of subtypes of stereotypies during development. To address this limitation, we report a group of 10 children who have presented at our clinics with movements that would currently be considered as motor stereotypies, but which are associated with engagement in episodes of intense imagery. Parents are often very anxious about the nature of the complex and unusual movements, and children typically have not disclosed their thoughts during the movements to them. Many children also have been extensively investigated 1212
DOI: 10.1111/dmcn.12518
with expensive and intrusive tests such as brain magnetic resonance imaging (MRI) and electroencephalography (EEG) including video telemetry, but the phenomena can be identified with careful history taking and specific questioning of the child. This phenomena has not yet been reported in the literature, however, we believe this movement disorder may be very common. Thus, the aim of this article is to contribute to current discussions regarding the clinical usefulness of diagnosing stereotypies during development by considering phenotypic presentation, classification, and behavioural management of this subgroup of children. Stereotypies form a normal part of development, with as many as 60% of children reported to present with stereotyped movements between 2 years and 5 years of age.6 They also commonly occur in the context of neurodevelopmental (e.g. autism spectrum disorder [ASD]) or neurological (e.g. blindness, stroke) disorders, with the prevalence of stereotypies varying in relation to underlying aetiology and condition severity. Stereotypies are described in relation to movement and may be defined as common © 2014 Mac Keith Press
(e.g. rocking, head banging, finger tapping), complex arm and hand movements (e.g. flapping, waving), or head nodding.7 For children with typical development, Harris et al.8 found that common triggers included being excited (80%), engrossed in activities (33%), anxious/stressed (26%), or fatigued (21%). Children may report multiple triggers, however, it is unknown whether the associated movements are consistent and influenced by co-occurring cognitive and/or emotional processes. Similarly, while children often use terms such as ‘bouncing’, ‘exercises’, ‘relaxation’, or ‘pacing’ to describe their stereotypies,9,10 what children mean by these terms and their underlying cognitions remains unclear. The accurate identification of stereotypies from other childhood disorders (e.g. tics, myoclonus, epilepsy) is essential to ensure appropriate clinical management. In contrast to other movement disorders, children subjectively ‘enjoy’ engaging with their movements and can be ‘called out of’ or distracted from bouts of stereotyped movements, though they are often reluctant to be interrupted and may even become angry or annoyed. As such, children often lack the motivation to disengage from stereotypies, leaving parents feeling anxious and frustrated as how best to manage their children’s unusual or ‘odd’ appearance. Generally, as children develop, increased social awareness may contribute to a process of privatization, with children choosing to only engage with their stereotypies in situations where it would be deemed acceptable (e.g. at home, in their bedroom9). In our clinical experience, however, the process of privatization can be drawn out across the childhood years and is more difficult for some children to achieve than others. As such, parental anxiety management may also be required. Further, these movements are often misdiagnosed as tics with parents often following advice available on the internet aimed at tic management. In our experience, these strategies seem to worsen stereotypies as they involve parents actively ‘ignoring’ them, which appears to contribute to an exacerbation of the stereotypies as children feel free to enjoy them. However, to what extent parental management strategies and peer pressures independently contribute to changes in stereotypies during development is unclear and an area for future research. Given the paucity of research in this area there is a lack of understanding regarding the neurological, cognitive, emotional, and social characteristics that may interact to contribute to individual differences in the expression and suppression of stereotypies during development. The developmental trajectory of stereotypies is also unclear, though a general ‘view’ is that children who present with stereotypies do not experience adverse long-term effects in adulthood. Interestingly, however, there is an emerging adult literature regarding individuals who engage in intense episodes of daydreaming or fantasizing that had a childhood onset and was typically associated with movements and activity (79%11–13). In the Bigelsen and Schupak study,11 the majority of individuals (88%) reported moderate to severe psychological distress, with many attempting
• • • • •
What this paper adds Describes a new subgroup of motor stereotypies. Features of children with intense imagery movements (IIM) are described. Provides clinical guidance to support the early identification of IIM and avoid unnecessary investigations. Suggests questions to identify children presenting with episodes of IIM. Outlines behavioural strategies to help children and families manage IIM.
to control the fantasizing (79%) and in some seeking therapeutic or mental health care (23%), but all with limited success. Thus, despite enjoying the fantasy acts, these adults report a compulsive aspect that they find distressing, which has become established since the childhood years and negatively impacts upon daily functioning and emotional well-being. It is possible that these adults may present with the same phenomena that we are seeing in childhood. In this article we report our original clinical data for these children, whom we refer to as engaging in episodes of ‘intense imagery movements’ (IIM). We consider the clinical features that may help contribute to the differential diagnosis of IIM from other stereotypies, as well as the importance of effective clinical management during development.
METHOD Participants Children presenting with movement disorder in the context of episodes of intense imagery were originally identified by TH, consultant paediatric neurologist and clinical lead for the Tic and Neurodevelopmental Movement Disorders (TANDeM) service at Evelina London Children’s Hospital, UK. The current sample consists of 10 children who were identified following referral to TANDeM, UK (n=7) and after collaboration with FC and VB at the Department of Paediatrics and Child Neuropsychiatry at University La Sapienza of Rome, Italy (n=3). The children were aged between 6 year and 15 years when they presented at the clinics. Most referrals had been received from acute paediatrics, acute neurology, and tertiary epilepsy clinics. Data collection All children were seen by multidisciplinary teams that included a neurologist, psychiatrist, and clinical psychologist with expertise in childhood movement disorders. Movement abnormalities were observed directly during the clinical visit and on video recordings, with associated thought processes reported directly by the children after careful questioning, sometimes without parents present but with parental consent. A positive family history was reported if first degree relatives (i.e. parents and siblings) presented with clinical features that would be consistent with developmental, neurological, or mental health disorders. Diagnostic criteria Diagnoses were made in accordance with the DSM-5.4 Stereotypies were defined as repetitive, seemingly driven and non-functional motor behaviour, which interferes with normal activities or result in injury, is not better accounted for Intense Imagery Movements Sally Robinson et al.
1213
by a compulsion or pervasive developmental disorder, is not drug-induced, and persists for a minimum of 4 weeks. Episodes of intense imagery were recorded after careful open questioning if children described engaging in acts of imagery while performing their stereotyped movements. These were defined as internally experienced episodes where children reported forming thoughts or mental images of something not present to the senses that reflected novel (e.g. pretending to be a fairy) or elaborative (e.g. developing a computer game) acts of imagination. Associated disorders and behaviours were determined by a review of the child’s developmental history during the clinical assessment (e.g. diagnosis made on the basis of DSM-5 criteria or by other professionals).
Statistical analyses Summary statistics were generated for categorical variables. In relation to general background information this included age of presentation to the clinic, initial referral questions, comorbid diagnosis, previous neurology investigations, pharmacological intervention, and family history. In relation to episodes of intense imagery with movements this included age at onset of stereotyped movements, body parts involved in the movements, imagery themes, location where the episodes occurred and associated emotions. Subjective experiences are included to provide a more comprehensive understanding of these episodes of intense imagery with movements. RESULTS Table I provides an overview of participant characteristics, stereotyped movements, and intense imagery episodes. Participant characteristics Ten children (nine males, one female) are reported, of whom seven were white British (patients 1–7) and three were Italian (patients 8–10). All children were referred for the specialist assessment of movement disorders, with queries around possible stereotypies (n=6), tics (n=3), other unusual movements (n=4), autism spectrum traits (n=6), and psychopathological problems (n=10). At the time of the initial clinical assessment the mean age was 8 years 6 months (SD 2y 5mo; median age 8y; range 6– 15y). Comorbid diagnoses made before attendance at the clinics were reported for six children, which included ASD (n=2), attention-deficit–hyperactivity disorder (ADHD; n=2), delayed speech development (n=1), obsessive–compulsive behaviours (OCBs; n=3), and anxiety (n=3). Some patients had received MRI (n=2) and EEG (n=3) investigations before attendance at the clinics, all of which were reported as normal. Three children were on aripiprazole for the movements and one was on penicillin following a Paediatric Autoimmune Neuropsychiatric Disorder Associated with Streptococcal Infections (PANDAS) diagnosis. A positive family history of mental health disorders was reported for six children. Mental health disorders were characterized by maternal anxiety (n=2), maternal depres1214
Developmental Medicine & Child Neurology 2014, 56: 1212–1218
sion (n=3), and paternal depression (n=1). Neurodevelopmental disorders were reported for three fathers, and included ADHD (n=1), comorbid Tourette syndrome, stutter, dyslexia, OCBs (n=1), and a specific language disorder (n=1). The mother of one child reported fantasizing when younger and engaging in similar stereotyped movements, while the father of one child reported currently engaging in imaginary ‘rugby games’ that were associated with flicking finger movements.
Stereotyped movements The mean age at onset of motor stereotypies was 2 years 9 months (SD 2y 2mo; median age 2y 9mo; range 3mo– 8y), with three patients reporting the onset of motor stereotypies before 1 year of age and a further nine before 4 years of age. None of the children reported imaginary episodes spontaneously at presentation. Stereotypies associated with intense imagery episodes reflected complex movements that involved upper and lower limbs, with bouncing, pacing or skipping reported for six children and vocalizations for four. Body parts involved in movements included, but were not limited to, combinations of writhing of the hands and fingers, clicking of the fingers, hand flapping, arm waving, twisting of the arms across the body, leg kicking, jumping, head banging, tongue protruding, neck rolling, pacing, skipping, grunting, and screaming. Intense imagery episodes All children reported engaging in visual imagery while performing the movements, with some reporting multiple themes to their imagery episodes. The movements do not resemble any specific actions. The reported themes were computer games (n=6), cartoons and/or films (n=4), fantasy scenes (n=3), circus tricks/jokes (n=1), imaging items moving (n=1), and imaging film scenes that included family members (n=1). Specific information on the length of episodes was not recorded, although clinical discussions indicated that they ranged in duration from a few seconds to minutes. For eight children, these episodes were said to occur anywhere if bored or relaxed (e.g. at home, school classroom, playground, walking to school, brushing teeth) and ‘being in the back of the car’ was common. All children reported finding the episodes enjoyable, although half reported feeling some embarrassment. Subjective experiences Some children described engaging in elaborative imagery experiences during movements, while others described recalling memories of computer games or films: I can just be looking at things and thinking of nothing and then if I look for a bit too long the things will come alive and I can see pictures and things that are not there. . . I know it’s not real because I can see the real object and the imaginary figures at the same time. . . I enjoy looking at all the lovely amazing things happening around me and then “ahh” it’s all
Intense Imagery Movements Sally Robinson et al.
1215
M
M
M
M
M
4
5
6
7
8
8y
4y
2y 6mo
2y 6mo
1y
4y
3y
9mo
3mo
3y
Age at onset
15y
6y
7y
8y
8y
9y
8y
7y
9y
8y
Age at referral
Complex stereotypies, psychopathological problems? Complex stereotypies, other movement disorders?
Stereotypies, OCB/OCD?
ASD traits, stereotypies?
Stereotypies, ASD traits?
ASD traits, emerging tic disorder?
Tics, mannerisms, other unusual movements?
Complex movements during a daze, ASD traits?
Stereotypies?
Tourettes, PANDAS?
Referral questions
OCBs
–
Delayed speech development, ADHD ASD (high functioning), OCBs, anxiety
EEG: normal
EEG: theta activity in central temporal region Repeat EEG: normal
ASD (high functioning)
EEG: normal
–
Tics
OCBs
–
MRI: normal
ADHD, worrier
Worrier
Developmental comorbidities
–
–
MRI: normal
Previous imaging
Upper body, facial, lower limbs, bouncing Upper body, neck rolling, facial, hand flapping, lower limbs, pacing Upper body, facial, lower limbs, pacing Upper body, arm writhing, hand twisting, facial, torso, lower limbs Upper body, hand flapping, lower limbs, pacing, coughing Shaking hands and objects, pacing, grumbling noises Hand flapping, skipping, screaming
Upper body, facial, lower limbs
Upper body, arm waving, facial, vocalizations Upper body, hand flapping/rotating
Body parts involved
Computer games, cartoons
Computer games, fantasy scenes
Cartoons
Fantasy and magic scenes of films with family members as characters
When bored, no specific places
When bored, no specific places Alone in his bedroom
Computer games
Computer games
Times of boredom, walking to school, brushing teeth School playground, at home playing computer games, in public when bored In the car, anywhere when bored When bored, no specific places
Grandmother’s conservatory, relaxed at home, bored at school
Imagines toys/ objects moving, fairies, cartoon characters He is a clown in a circus performing tricks/jokes (e.g. slipping on pancakes) Computer games
Computer games
Back of car
School playground, anywhere if bored
Imagery themes
Locations performed
PANDAS, Paediatric Autoimmune Neuropsychiatric Disorder Associated with Streptococcal Infections; ASD, autism spectrum disorder; ADHD, attention-deficit–hyperactivity disorder; OCB, obsessive–compulsive behaviour; OCD, obsessive–compulsive disorder; MRI, magnetic resonance imaging; EEG, electroencephalography.
M
M
3
10
F
2
M
M
1
9
Sex
Pt no.
Table I: Participant characteristics, features of movements, and imagery episodes
gone. . . I wish I could stay like that, it feels quite nice, it feels like it’s really happening but no one else can see it just me. (Patient 2)
I’m just thinking about Angry Birds [a computer game]. (Patient 6)
. . . I was just doing my movements. What were you thinking about? Rayman. Can you tell me what was happening? Rayman was being chased by a giant spiky man eating plant. . . and the rocks were collapsing on Rayman and he had to make it away from them and there’s this other boss, the final boss, the Momma of all Nightmares! So when you were doing the movements was it as if you were watching it on TV or playing it? Playing it [immediately continues]. . . and the Momma of all Nightmares has loads of tentacles and she has about 13 eyes and mouth with lots of teeth and she has spiky elbows and spiky bracelets and Rayman had to jump over the spiky bracelets and avoid her spiky elbow. (Patient 7) Children reported varying degrees of control and awareness of the episodes of imagery and movements, though all described the imagery episodes as occurring before the movements: Sometimes it happens without me realizing. . . I always know when I’m imaging things and sometimes I notice the twitching, but not always. . . it [the movements] just feels right. . . sometimes I hold my hands by my side to stop the movements but it doesn’t feel nice. (Patient 2) What comes first, the thinking about computer games or the movement? The thinking about computer games. Does the movement ever come first? No. (Patient 7)
DISCUSSION In this article we report our initial clinical observations of a subgroup of 10 children who present with episodes of IIM. These children form a cohesive group that provide new insights into the accurate classification and clinical management of so-called stereotyped movements, thereby contributing and extending current discussions regarding the usefulness of diagnosing stereotypies during development.3 The following section includes discussion and reflections, to help support the accurate classification and clinical management of children with IIM, as we have now identified further cases and consider this to be a common phenomenon. Table II provides examples of questions that we have found clinically useful to help identify and manage children with intense imagery movements. 1216
Developmental Medicine & Child Neurology 2014, 56: 1212–1218
Presenting features In this sample, all children presented with ‘a movement disorder’. Children initially presented to epilepsy/neurology clinics or in acute paediatric settings. In over half of the cases the referring clinicians had raised the possibility of ASD because of the unusual nature of the movements, although only two met diagnostic criteria when formally assessed. Similarly, half had undergone previous brain imaging with normal MRI, which was in contrast to a previous study where reductions in frontal white matter volume were reported for males with complex stereotypies.14 In line with the stereotypy literature,10 the onset of stereotyped movements was reported during infancy or early childhood, with clinical presentation delayed until around 7 years of age. It was not possible to determine the age at which IIM first occurred as children did not present with this degree of insight into the imagery experiences and for most parents our clinical interview was the first time they had heard their children describing the thoughts. Of interest, however, one of the children (Patient 2) was said to have described herself as a ‘butterfly’ if other children asked what she was doing when she was in nursery (around 4y of age). Furthermore, the parents of two children with high functioning ASD (Patients 8 and 10) reported a change in the presentation of stereotypies around 7 years of age, from the more simple ones to the complex ones. It is speculated that developmental changes in neural structures and associated cognitive processes (e.g. language, episodic memory, introspection) may contribute to the emergence of IIM during development. Of particular interest, when early videos of the movements have been retrospectively reviewed, we have noticed that some children with IIM have experienced probable infantile gratification movements in the first year of life. We have also noticed a higher prevalence of parental obsessive–compulsive disorder, so anxiety may, therefore be part of a pathophysiological association within the brain circuitry of possible
Table II: Key questions to ask children to help identify and manage intense imagery movements Identification questions What are you doing when you are having the movements? Are you thinking about anything when you are having the movements? When do you have the movements/when are the movements most likely to happen? Do you get a feeling before doing the movements? How do you feel when doing the movements? Management questions Do you know when you are thinking about things that might make you have the movements? Do you like doing the movements/having the thoughts that make the movements happen? Can you control the movements/thoughts that make the movements happen? Do the movements and/or thoughts get in the way of anything? Do you want to stop them? (. . . or do parents/carers want their child to stop them?)
fronto-striatal or cerebellar loops. This is a subject for future exploration. In terms of the association between the thought processes and movements, all children described voluntary engagement in acts of imagery with stereotyped movements occurring as a secondary response over which they had limited control. It appears plausible that engagement in episodes of intense imagery may serve the purpose of increasing cognitive stimulation, while the associated stereotyped movements may provide a sensory gain. In addition to concerns about the potential impact of these episodes on classroom learning (e.g. missing task instructions), all parents raised concerns regarding the social appropriateness of movements and potential bullying by peers. These worries were also shared by many of the children, and given their reticence in talking about their thought processes it is likely that they are also uncertain of how these mental acts might be perceived by both peers and adults. Despite this, none of the children expressed a desire to stop engaging with their IIM, which clearly supports the enjoyable and pleasurable nature associated with them. Taking into consideration all clinical features, it appears highly likely that some of these children go on to become the adult compulsive fantasizers who reported significant concerns regarding their engagement in extensive periods of highly-structured, immersive imaginary experiences, which for the majority included kinaesthetic activity.11 These adults reported experiencing psychological distress caused by difficulty controlling their compulsive desire to engage in enjoyable acts of imagination, concern regarding interference with actual relationships and endeavours, and overwhelming feelings of shame with exhaustive efforts to keep their fantasy behaviours hidden from others. There is a current lack of knowledge about the relationship between individuals who engage in excessive acts of imagery as children and adult fantasizers, with longitudinal follow-up planned for children with IIM to help address this issue. Nevertheless, it is our opinion that effective clinical management of children with IIM may be of importance to help minimize the potentially negative impact of engagement in imaginary acts and movements on cognitive, social and emotional development. Thus, the accurate description and identification of children with IIM is essential to avoid anxiety provoking and unnecessary investigation with EEG and brain imaging, as well as to ensure access to appropriate treatment strategies.
Behavioural management strategies First, to identify the types of behavioural strategies that are likely to be effective it is important to identify triggers and locations where the IIM occur. For example, there may be particular lessons or times of the day when the child typically engages with IIM (e.g. delivery of task instructions, watching films, and computer time), and if this is the case, increasing cognitive demands at these times may help keep the child engaged in the ‘real world’ (e.g. getting them to
write the instructions on the board). Visual displays in the classroom or birds flying past the window may trigger their imagination, and if this is the case then changing where the child is seated in the classroom may be effective at reducing IIM. Based on our experiences, providing a specific time and place at school, that is a private space, where children are allowed to engage with their IIM also appears to be an effective strategy to help them remain taskfocused in the classroom. Habit reversal strategies, such as awareness training (e.g. discrete gesture or sign when the child engages with IIM), providing an alternative response (e.g. a small toy, Blu-Tack to fiddle with), and positive reinforcement of the alternative response (e.g. behavioural reward charts) are also likely to help children modify their movements. Thus, in our experiences we have found these behavioural strategies to be useful in helping children gain ‘control’ over their episodes of imagery with movements.
Limitations The primary limitation of this study is that we have not yet systematically compared our findings to children who have motor stereotypies without episodes of intense imagery, as well as children who have a diagnosis of ASD with motor stereotypies. Clearly this would allow for more detailed observations regarding clinical features, which would enable the subtyping of IIM from stereotypies more generally. It is also important to recognize that, despite presenting as a movement disorder, several of the children in the current study presented with previously unreported ASD traits, therefore identifying associated neurodevelopmental difficulties is of importance to ensure potential social, emotional, and cognitive difficulties are appropriately supported. We plan to conduct future studies to address these limitations and hope that increased insight into IIM will enhance understanding of stereotypies and cognitive processes in children with ASD who also show these interesting motor patterns. CONCLUSIONS This article describes a group of children who present with what would previously be described as motor stereotypies, but in the context of episodes of intense imagery. We refer to these as IIM and believe they may form a common and discrete motor stereotypy subgroup. This has proved to be a very useful concept clinically and we believe it may be very common. In conclusion, the children reported in this article clearly highlight that although the diagnosis of stereotypies may be given on the basis of observable movements, it is the underlying thoughts and associated cognitive processes that need to be defined if we are to enhance the clinical usefulness of the concept of stereotypies during development. A CK N O W L E D G E M E N T S We would like to thank all the children and families reported in this article. We would also like to thank all the referrers, and Dr Penny Bunton (clinical psychologist), Professor Caterina D’Ardia Intense Imagery Movements Sally Robinson et al.
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(neuropsychiatrist), Claire Grose (clinical nurse specialist), Dr Peter Hindley (consultant paediatric neuropsychiatrist), and Dr Anya Kaushik (paediatric psychiatrist) for their contributions to clinical assessments and early discussions regarding the clinical presentation of some of the children reported in this article. We would also like to thank Professor Gillian Baird, Dr Keith Pohl, Dr Ann
Ozsivadijan, Dr Vicky Slonims, Dr Isabel Garrood, Dr Rebecca Martyn, Dr Michael Absoud, Professor Paul Gringras, Dr Elaine Hughes, Dr Ruth Williams, Dr Ming Lim, and Dr Karine Lascelles for their helpful comments. The authors have stated that they had no interests that might be perceived as posing a conflict or bias.
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ORIGINAL ARTICLE
Intense imagery movements: a common and distinct paediatric subgroup of motor stereotypies SALLY ROBINSON 1
| MARTIN WOODS 1 | FRANCESCO CARDONA 2 | VALENTINA BAGLIONI 2 |
TAMMY HEDDERLY 1 1 Tic and Neurodevelopmental Movements (TANDeM), Children’s Neurosciences Centre, Evelina London Children’s Hospital, Guy’s and St Thomas’ NHS Foundation Trust, London, UK. 2 Department of Paediatrics and Child Neuropsychiatry, University La Sapienza of Rome, Rome, Italy. Correspondence to Tammy Hedderly at, TANDeM, Children’s Neurosciences Centre, St Thomas’ Hospital, Westminster Bridge Road, London, SE1 7EH, UK. E-mail: [email protected] This article is commented on by Freeman on pages 1139–1140 of this issue.
PUBLICATION DATA
Accepted for publication 24th April 2014. Published online 20th June 2014. ABBREVIATIONS
ASD IIM OCB
Autism spectrum disorder Intense imagery movements Obsessive–compulsive behaviour
AIM The aim of this article is to describe a subgroup of children who presented with stereotyped movements in the context of episodes of intense imagery. This is of relevance to current discussions regarding the clinical usefulness of diagnosing motor stereotypies during development. METHOD The sample consisted of 10 children (nine males, one female; mean age 8y 6mo [SD 2y 5mo], range 6–15y). Referrals were from acute paediatricians, neurologists, and tertiary epilepsy services. Children were assessed by multidisciplinary teams with expertise in paediatric movement disorders. RESULTS Stereotypies presented as paroxysmal complex movements involving upper and lower limbs. Imagery themes typically included computer games (60%), cartoons/films (40%), and fantasy scenes (30%). Comorbid developmental difficulties were reported for 80% of children. Brain imaging and electrophysiological investigations had been conducted for 50% of the children before referral to the clinic. INTERPRETATION The descriptive term ‘intense imagery movements’ (IIM) was applied if (after interview) the children reported engaging in acts of imagery while performing stereotyped movements. We believe these children may form a common and discrete stereotypy subgroup, with the concept of IIM being clinically useful to ensure the accurate diagnosis and clinical management of this paediatric movement disorder.
Movement disorders are typically classified by clinical descriptions of physical signs, with the term stereotypy referring to a wide range of movements, behaviours and/or vocalizations that are repetitive, seemingly driven, and lack clear function (for review see Barry et al.1 or Goldman et al.2). There are on-going discussions regarding the clinical usefulness of the concept3–5 and the need for a more clinically useful definition that describes a cohesive group of conditions linked pathophysiologically has been proposed by Edwards et al.3 Nevertheless, to date, there are no reports in the literature that consider the differential diagnosis of subtypes of stereotypies during development. To address this limitation, we report a group of 10 children who have presented at our clinics with movements that would currently be considered as motor stereotypies, but which are associated with engagement in episodes of intense imagery. Parents are often very anxious about the nature of the complex and unusual movements, and children typically have not disclosed their thoughts during the movements to them. Many children also have been extensively investigated 1212
DOI: 10.1111/dmcn.12518
with expensive and intrusive tests such as brain magnetic resonance imaging (MRI) and electroencephalography (EEG) including video telemetry, but the phenomena can be identified with careful history taking and specific questioning of the child. This phenomena has not yet been reported in the literature, however, we believe this movement disorder may be very common. Thus, the aim of this article is to contribute to current discussions regarding the clinical usefulness of diagnosing stereotypies during development by considering phenotypic presentation, classification, and behavioural management of this subgroup of children. Stereotypies form a normal part of development, with as many as 60% of children reported to present with stereotyped movements between 2 years and 5 years of age.6 They also commonly occur in the context of neurodevelopmental (e.g. autism spectrum disorder [ASD]) or neurological (e.g. blindness, stroke) disorders, with the prevalence of stereotypies varying in relation to underlying aetiology and condition severity. Stereotypies are described in relation to movement and may be defined as common © 2014 Mac Keith Press
(e.g. rocking, head banging, finger tapping), complex arm and hand movements (e.g. flapping, waving), or head nodding.7 For children with typical development, Harris et al.8 found that common triggers included being excited (80%), engrossed in activities (33%), anxious/stressed (26%), or fatigued (21%). Children may report multiple triggers, however, it is unknown whether the associated movements are consistent and influenced by co-occurring cognitive and/or emotional processes. Similarly, while children often use terms such as ‘bouncing’, ‘exercises’, ‘relaxation’, or ‘pacing’ to describe their stereotypies,9,10 what children mean by these terms and their underlying cognitions remains unclear. The accurate identification of stereotypies from other childhood disorders (e.g. tics, myoclonus, epilepsy) is essential to ensure appropriate clinical management. In contrast to other movement disorders, children subjectively ‘enjoy’ engaging with their movements and can be ‘called out of’ or distracted from bouts of stereotyped movements, though they are often reluctant to be interrupted and may even become angry or annoyed. As such, children often lack the motivation to disengage from stereotypies, leaving parents feeling anxious and frustrated as how best to manage their children’s unusual or ‘odd’ appearance. Generally, as children develop, increased social awareness may contribute to a process of privatization, with children choosing to only engage with their stereotypies in situations where it would be deemed acceptable (e.g. at home, in their bedroom9). In our clinical experience, however, the process of privatization can be drawn out across the childhood years and is more difficult for some children to achieve than others. As such, parental anxiety management may also be required. Further, these movements are often misdiagnosed as tics with parents often following advice available on the internet aimed at tic management. In our experience, these strategies seem to worsen stereotypies as they involve parents actively ‘ignoring’ them, which appears to contribute to an exacerbation of the stereotypies as children feel free to enjoy them. However, to what extent parental management strategies and peer pressures independently contribute to changes in stereotypies during development is unclear and an area for future research. Given the paucity of research in this area there is a lack of understanding regarding the neurological, cognitive, emotional, and social characteristics that may interact to contribute to individual differences in the expression and suppression of stereotypies during development. The developmental trajectory of stereotypies is also unclear, though a general ‘view’ is that children who present with stereotypies do not experience adverse long-term effects in adulthood. Interestingly, however, there is an emerging adult literature regarding individuals who engage in intense episodes of daydreaming or fantasizing that had a childhood onset and was typically associated with movements and activity (79%11–13). In the Bigelsen and Schupak study,11 the majority of individuals (88%) reported moderate to severe psychological distress, with many attempting
• • • • •
What this paper adds Describes a new subgroup of motor stereotypies. Features of children with intense imagery movements (IIM) are described. Provides clinical guidance to support the early identification of IIM and avoid unnecessary investigations. Suggests questions to identify children presenting with episodes of IIM. Outlines behavioural strategies to help children and families manage IIM.
to control the fantasizing (79%) and in some seeking therapeutic or mental health care (23%), but all with limited success. Thus, despite enjoying the fantasy acts, these adults report a compulsive aspect that they find distressing, which has become established since the childhood years and negatively impacts upon daily functioning and emotional well-being. It is possible that these adults may present with the same phenomena that we are seeing in childhood. In this article we report our original clinical data for these children, whom we refer to as engaging in episodes of ‘intense imagery movements’ (IIM). We consider the clinical features that may help contribute to the differential diagnosis of IIM from other stereotypies, as well as the importance of effective clinical management during development.
METHOD Participants Children presenting with movement disorder in the context of episodes of intense imagery were originally identified by TH, consultant paediatric neurologist and clinical lead for the Tic and Neurodevelopmental Movement Disorders (TANDeM) service at Evelina London Children’s Hospital, UK. The current sample consists of 10 children who were identified following referral to TANDeM, UK (n=7) and after collaboration with FC and VB at the Department of Paediatrics and Child Neuropsychiatry at University La Sapienza of Rome, Italy (n=3). The children were aged between 6 year and 15 years when they presented at the clinics. Most referrals had been received from acute paediatrics, acute neurology, and tertiary epilepsy clinics. Data collection All children were seen by multidisciplinary teams that included a neurologist, psychiatrist, and clinical psychologist with expertise in childhood movement disorders. Movement abnormalities were observed directly during the clinical visit and on video recordings, with associated thought processes reported directly by the children after careful questioning, sometimes without parents present but with parental consent. A positive family history was reported if first degree relatives (i.e. parents and siblings) presented with clinical features that would be consistent with developmental, neurological, or mental health disorders. Diagnostic criteria Diagnoses were made in accordance with the DSM-5.4 Stereotypies were defined as repetitive, seemingly driven and non-functional motor behaviour, which interferes with normal activities or result in injury, is not better accounted for Intense Imagery Movements Sally Robinson et al.
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by a compulsion or pervasive developmental disorder, is not drug-induced, and persists for a minimum of 4 weeks. Episodes of intense imagery were recorded after careful open questioning if children described engaging in acts of imagery while performing their stereotyped movements. These were defined as internally experienced episodes where children reported forming thoughts or mental images of something not present to the senses that reflected novel (e.g. pretending to be a fairy) or elaborative (e.g. developing a computer game) acts of imagination. Associated disorders and behaviours were determined by a review of the child’s developmental history during the clinical assessment (e.g. diagnosis made on the basis of DSM-5 criteria or by other professionals).
Statistical analyses Summary statistics were generated for categorical variables. In relation to general background information this included age of presentation to the clinic, initial referral questions, comorbid diagnosis, previous neurology investigations, pharmacological intervention, and family history. In relation to episodes of intense imagery with movements this included age at onset of stereotyped movements, body parts involved in the movements, imagery themes, location where the episodes occurred and associated emotions. Subjective experiences are included to provide a more comprehensive understanding of these episodes of intense imagery with movements. RESULTS Table I provides an overview of participant characteristics, stereotyped movements, and intense imagery episodes. Participant characteristics Ten children (nine males, one female) are reported, of whom seven were white British (patients 1–7) and three were Italian (patients 8–10). All children were referred for the specialist assessment of movement disorders, with queries around possible stereotypies (n=6), tics (n=3), other unusual movements (n=4), autism spectrum traits (n=6), and psychopathological problems (n=10). At the time of the initial clinical assessment the mean age was 8 years 6 months (SD 2y 5mo; median age 8y; range 6– 15y). Comorbid diagnoses made before attendance at the clinics were reported for six children, which included ASD (n=2), attention-deficit–hyperactivity disorder (ADHD; n=2), delayed speech development (n=1), obsessive–compulsive behaviours (OCBs; n=3), and anxiety (n=3). Some patients had received MRI (n=2) and EEG (n=3) investigations before attendance at the clinics, all of which were reported as normal. Three children were on aripiprazole for the movements and one was on penicillin following a Paediatric Autoimmune Neuropsychiatric Disorder Associated with Streptococcal Infections (PANDAS) diagnosis. A positive family history of mental health disorders was reported for six children. Mental health disorders were characterized by maternal anxiety (n=2), maternal depres1214
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sion (n=3), and paternal depression (n=1). Neurodevelopmental disorders were reported for three fathers, and included ADHD (n=1), comorbid Tourette syndrome, stutter, dyslexia, OCBs (n=1), and a specific language disorder (n=1). The mother of one child reported fantasizing when younger and engaging in similar stereotyped movements, while the father of one child reported currently engaging in imaginary ‘rugby games’ that were associated with flicking finger movements.
Stereotyped movements The mean age at onset of motor stereotypies was 2 years 9 months (SD 2y 2mo; median age 2y 9mo; range 3mo– 8y), with three patients reporting the onset of motor stereotypies before 1 year of age and a further nine before 4 years of age. None of the children reported imaginary episodes spontaneously at presentation. Stereotypies associated with intense imagery episodes reflected complex movements that involved upper and lower limbs, with bouncing, pacing or skipping reported for six children and vocalizations for four. Body parts involved in movements included, but were not limited to, combinations of writhing of the hands and fingers, clicking of the fingers, hand flapping, arm waving, twisting of the arms across the body, leg kicking, jumping, head banging, tongue protruding, neck rolling, pacing, skipping, grunting, and screaming. Intense imagery episodes All children reported engaging in visual imagery while performing the movements, with some reporting multiple themes to their imagery episodes. The movements do not resemble any specific actions. The reported themes were computer games (n=6), cartoons and/or films (n=4), fantasy scenes (n=3), circus tricks/jokes (n=1), imaging items moving (n=1), and imaging film scenes that included family members (n=1). Specific information on the length of episodes was not recorded, although clinical discussions indicated that they ranged in duration from a few seconds to minutes. For eight children, these episodes were said to occur anywhere if bored or relaxed (e.g. at home, school classroom, playground, walking to school, brushing teeth) and ‘being in the back of the car’ was common. All children reported finding the episodes enjoyable, although half reported feeling some embarrassment. Subjective experiences Some children described engaging in elaborative imagery experiences during movements, while others described recalling memories of computer games or films: I can just be looking at things and thinking of nothing and then if I look for a bit too long the things will come alive and I can see pictures and things that are not there. . . I know it’s not real because I can see the real object and the imaginary figures at the same time. . . I enjoy looking at all the lovely amazing things happening around me and then “ahh” it’s all
Intense Imagery Movements Sally Robinson et al.
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M
M
M
M
M
4
5
6
7
8
8y
4y
2y 6mo
2y 6mo
1y
4y
3y
9mo
3mo
3y
Age at onset
15y
6y
7y
8y
8y
9y
8y
7y
9y
8y
Age at referral
Complex stereotypies, psychopathological problems? Complex stereotypies, other movement disorders?
Stereotypies, OCB/OCD?
ASD traits, stereotypies?
Stereotypies, ASD traits?
ASD traits, emerging tic disorder?
Tics, mannerisms, other unusual movements?
Complex movements during a daze, ASD traits?
Stereotypies?
Tourettes, PANDAS?
Referral questions
OCBs
–
Delayed speech development, ADHD ASD (high functioning), OCBs, anxiety
EEG: normal
EEG: theta activity in central temporal region Repeat EEG: normal
ASD (high functioning)
EEG: normal
–
Tics
OCBs
–
MRI: normal
ADHD, worrier
Worrier
Developmental comorbidities
–
–
MRI: normal
Previous imaging
Upper body, facial, lower limbs, bouncing Upper body, neck rolling, facial, hand flapping, lower limbs, pacing Upper body, facial, lower limbs, pacing Upper body, arm writhing, hand twisting, facial, torso, lower limbs Upper body, hand flapping, lower limbs, pacing, coughing Shaking hands and objects, pacing, grumbling noises Hand flapping, skipping, screaming
Upper body, facial, lower limbs
Upper body, arm waving, facial, vocalizations Upper body, hand flapping/rotating
Body parts involved
Computer games, cartoons
Computer games, fantasy scenes
Cartoons
Fantasy and magic scenes of films with family members as characters
When bored, no specific places
When bored, no specific places Alone in his bedroom
Computer games
Computer games
Times of boredom, walking to school, brushing teeth School playground, at home playing computer games, in public when bored In the car, anywhere when bored When bored, no specific places
Grandmother’s conservatory, relaxed at home, bored at school
Imagines toys/ objects moving, fairies, cartoon characters He is a clown in a circus performing tricks/jokes (e.g. slipping on pancakes) Computer games
Computer games
Back of car
School playground, anywhere if bored
Imagery themes
Locations performed
PANDAS, Paediatric Autoimmune Neuropsychiatric Disorder Associated with Streptococcal Infections; ASD, autism spectrum disorder; ADHD, attention-deficit–hyperactivity disorder; OCB, obsessive–compulsive behaviour; OCD, obsessive–compulsive disorder; MRI, magnetic resonance imaging; EEG, electroencephalography.
M
M
3
10
F
2
M
M
1
9
Sex
Pt no.
Table I: Participant characteristics, features of movements, and imagery episodes
gone. . . I wish I could stay like that, it feels quite nice, it feels like it’s really happening but no one else can see it just me. (Patient 2)
I’m just thinking about Angry Birds [a computer game]. (Patient 6)
. . . I was just doing my movements. What were you thinking about? Rayman. Can you tell me what was happening? Rayman was being chased by a giant spiky man eating plant. . . and the rocks were collapsing on Rayman and he had to make it away from them and there’s this other boss, the final boss, the Momma of all Nightmares! So when you were doing the movements was it as if you were watching it on TV or playing it? Playing it [immediately continues]. . . and the Momma of all Nightmares has loads of tentacles and she has about 13 eyes and mouth with lots of teeth and she has spiky elbows and spiky bracelets and Rayman had to jump over the spiky bracelets and avoid her spiky elbow. (Patient 7) Children reported varying degrees of control and awareness of the episodes of imagery and movements, though all described the imagery episodes as occurring before the movements: Sometimes it happens without me realizing. . . I always know when I’m imaging things and sometimes I notice the twitching, but not always. . . it [the movements] just feels right. . . sometimes I hold my hands by my side to stop the movements but it doesn’t feel nice. (Patient 2) What comes first, the thinking about computer games or the movement? The thinking about computer games. Does the movement ever come first? No. (Patient 7)
DISCUSSION In this article we report our initial clinical observations of a subgroup of 10 children who present with episodes of IIM. These children form a cohesive group that provide new insights into the accurate classification and clinical management of so-called stereotyped movements, thereby contributing and extending current discussions regarding the usefulness of diagnosing stereotypies during development.3 The following section includes discussion and reflections, to help support the accurate classification and clinical management of children with IIM, as we have now identified further cases and consider this to be a common phenomenon. Table II provides examples of questions that we have found clinically useful to help identify and manage children with intense imagery movements. 1216
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Presenting features In this sample, all children presented with ‘a movement disorder’. Children initially presented to epilepsy/neurology clinics or in acute paediatric settings. In over half of the cases the referring clinicians had raised the possibility of ASD because of the unusual nature of the movements, although only two met diagnostic criteria when formally assessed. Similarly, half had undergone previous brain imaging with normal MRI, which was in contrast to a previous study where reductions in frontal white matter volume were reported for males with complex stereotypies.14 In line with the stereotypy literature,10 the onset of stereotyped movements was reported during infancy or early childhood, with clinical presentation delayed until around 7 years of age. It was not possible to determine the age at which IIM first occurred as children did not present with this degree of insight into the imagery experiences and for most parents our clinical interview was the first time they had heard their children describing the thoughts. Of interest, however, one of the children (Patient 2) was said to have described herself as a ‘butterfly’ if other children asked what she was doing when she was in nursery (around 4y of age). Furthermore, the parents of two children with high functioning ASD (Patients 8 and 10) reported a change in the presentation of stereotypies around 7 years of age, from the more simple ones to the complex ones. It is speculated that developmental changes in neural structures and associated cognitive processes (e.g. language, episodic memory, introspection) may contribute to the emergence of IIM during development. Of particular interest, when early videos of the movements have been retrospectively reviewed, we have noticed that some children with IIM have experienced probable infantile gratification movements in the first year of life. We have also noticed a higher prevalence of parental obsessive–compulsive disorder, so anxiety may, therefore be part of a pathophysiological association within the brain circuitry of possible
Table II: Key questions to ask children to help identify and manage intense imagery movements Identification questions What are you doing when you are having the movements? Are you thinking about anything when you are having the movements? When do you have the movements/when are the movements most likely to happen? Do you get a feeling before doing the movements? How do you feel when doing the movements? Management questions Do you know when you are thinking about things that might make you have the movements? Do you like doing the movements/having the thoughts that make the movements happen? Can you control the movements/thoughts that make the movements happen? Do the movements and/or thoughts get in the way of anything? Do you want to stop them? (. . . or do parents/carers want their child to stop them?)
fronto-striatal or cerebellar loops. This is a subject for future exploration. In terms of the association between the thought processes and movements, all children described voluntary engagement in acts of imagery with stereotyped movements occurring as a secondary response over which they had limited control. It appears plausible that engagement in episodes of intense imagery may serve the purpose of increasing cognitive stimulation, while the associated stereotyped movements may provide a sensory gain. In addition to concerns about the potential impact of these episodes on classroom learning (e.g. missing task instructions), all parents raised concerns regarding the social appropriateness of movements and potential bullying by peers. These worries were also shared by many of the children, and given their reticence in talking about their thought processes it is likely that they are also uncertain of how these mental acts might be perceived by both peers and adults. Despite this, none of the children expressed a desire to stop engaging with their IIM, which clearly supports the enjoyable and pleasurable nature associated with them. Taking into consideration all clinical features, it appears highly likely that some of these children go on to become the adult compulsive fantasizers who reported significant concerns regarding their engagement in extensive periods of highly-structured, immersive imaginary experiences, which for the majority included kinaesthetic activity.11 These adults reported experiencing psychological distress caused by difficulty controlling their compulsive desire to engage in enjoyable acts of imagination, concern regarding interference with actual relationships and endeavours, and overwhelming feelings of shame with exhaustive efforts to keep their fantasy behaviours hidden from others. There is a current lack of knowledge about the relationship between individuals who engage in excessive acts of imagery as children and adult fantasizers, with longitudinal follow-up planned for children with IIM to help address this issue. Nevertheless, it is our opinion that effective clinical management of children with IIM may be of importance to help minimize the potentially negative impact of engagement in imaginary acts and movements on cognitive, social and emotional development. Thus, the accurate description and identification of children with IIM is essential to avoid anxiety provoking and unnecessary investigation with EEG and brain imaging, as well as to ensure access to appropriate treatment strategies.
Behavioural management strategies First, to identify the types of behavioural strategies that are likely to be effective it is important to identify triggers and locations where the IIM occur. For example, there may be particular lessons or times of the day when the child typically engages with IIM (e.g. delivery of task instructions, watching films, and computer time), and if this is the case, increasing cognitive demands at these times may help keep the child engaged in the ‘real world’ (e.g. getting them to
write the instructions on the board). Visual displays in the classroom or birds flying past the window may trigger their imagination, and if this is the case then changing where the child is seated in the classroom may be effective at reducing IIM. Based on our experiences, providing a specific time and place at school, that is a private space, where children are allowed to engage with their IIM also appears to be an effective strategy to help them remain taskfocused in the classroom. Habit reversal strategies, such as awareness training (e.g. discrete gesture or sign when the child engages with IIM), providing an alternative response (e.g. a small toy, Blu-Tack to fiddle with), and positive reinforcement of the alternative response (e.g. behavioural reward charts) are also likely to help children modify their movements. Thus, in our experiences we have found these behavioural strategies to be useful in helping children gain ‘control’ over their episodes of imagery with movements.
Limitations The primary limitation of this study is that we have not yet systematically compared our findings to children who have motor stereotypies without episodes of intense imagery, as well as children who have a diagnosis of ASD with motor stereotypies. Clearly this would allow for more detailed observations regarding clinical features, which would enable the subtyping of IIM from stereotypies more generally. It is also important to recognize that, despite presenting as a movement disorder, several of the children in the current study presented with previously unreported ASD traits, therefore identifying associated neurodevelopmental difficulties is of importance to ensure potential social, emotional, and cognitive difficulties are appropriately supported. We plan to conduct future studies to address these limitations and hope that increased insight into IIM will enhance understanding of stereotypies and cognitive processes in children with ASD who also show these interesting motor patterns. CONCLUSIONS This article describes a group of children who present with what would previously be described as motor stereotypies, but in the context of episodes of intense imagery. We refer to these as IIM and believe they may form a common and discrete motor stereotypy subgroup. This has proved to be a very useful concept clinically and we believe it may be very common. In conclusion, the children reported in this article clearly highlight that although the diagnosis of stereotypies may be given on the basis of observable movements, it is the underlying thoughts and associated cognitive processes that need to be defined if we are to enhance the clinical usefulness of the concept of stereotypies during development. A CK N O W L E D G E M E N T S We would like to thank all the children and families reported in this article. We would also like to thank all the referrers, and Dr Penny Bunton (clinical psychologist), Professor Caterina D’Ardia Intense Imagery Movements Sally Robinson et al.
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(neuropsychiatrist), Claire Grose (clinical nurse specialist), Dr Peter Hindley (consultant paediatric neuropsychiatrist), and Dr Anya Kaushik (paediatric psychiatrist) for their contributions to clinical assessments and early discussions regarding the clinical presentation of some of the children reported in this article. We would also like to thank Professor Gillian Baird, Dr Keith Pohl, Dr Ann
Ozsivadijan, Dr Vicky Slonims, Dr Isabel Garrood, Dr Rebecca Martyn, Dr Michael Absoud, Professor Paul Gringras, Dr Elaine Hughes, Dr Ruth Williams, Dr Ming Lim, and Dr Karine Lascelles for their helpful comments. The authors have stated that they had no interests that might be perceived as posing a conflict or bias.
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