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Stereotypies in Adults: a Systematic Review CORE Metadata, citation and similar papers at core.ac.uk Provided by Via Medica Journals INVITED REVIEW ARTICLE Neurologia i Neurochirurgia Polska Polish Journal of Neurology and Neurosurgery 2020, Volume 54 DOI: 10.5603/PJNNS.a2020.0058 Copyright © 2020 Polish Neurological Society ISSN 0028–3843 Stereotypies in adults: a systematic review Tanvi Shukla, Sanjay Pandey Department of Neurology, Govind Ballabh Pant Postgraduate Institute of Medical Education and Research, New Delhi, India ABSTRACT Stereotypies are abnormal involuntary non-goal-directed movement patterns or vocalisations which repeat continuously in the same fashion over a period of time and on multiple occasions and are typically distractible. Stereotypies are common in both children and adults, but they are extensively reviewed only in children. There are very few studies, mainly in the form of case reports and case series, focusing on stereotypies occurring in adults as part of different neurological disorders. In adults, stereotypies can be both physiological and pathological. Common physiological stereotypies in adults are leg shaking, face touching, playing with pens or hair, nail biting, hand tapping, foot tapping, and body rocking. Pathological stereotypies in adults are associated with a variety of neuropsychiatric conditions like neurodegenerative disorders, viral encephalitis, autoim- mune encephalitis, stroke, psychiatric illness, and drug use. In this review, we focus on the various causes of stereotypic movements in adults, and their pathophysiology, clinical manife- stations, and treatment. Key words: stereotypy, physiological, autoimmune encephalitis, drugs, psychiatry, stroke Introduction thumb sucking or foot tapping, or as more complex stereotypies like hand waving, playing with hands or repeatedly opening and Stereotypy was defined by Edwards et al. as “a non–goal-di- closing hands, hand posturing, head nodding, headbanging, re- rected movement pattern or vocalisation that is repeated con- peatedly sitting down and getting up from a chair, finger wagging, tinuously for a period of time in the same form and on multiple pacing, lip smacking, chewing, mouth opening, self-biting and occasions, and which is typically distractible” [1]. other self-injurious behaviours. Apart from motor stereotypies, The word ‘stereotyped’ means something which is typical there are also vocal or phonic stereotypies like humming, grunt- or repeats in the same fashion. So, stereotypies are an abnormal ing, moaning, or repeating words and phrases [1, 3, 4]. involuntary movement which repeats rhythmically in the same Stereotypies can also be classified as primary or secondary fashion ‘in a loop’ for a long duration and at the expense of depending on the presence or absence of an underlying neu- other movements. Stereotypies are more common at times of rological or psychiatric disorder [5]. They are associated with anxiety, excitement, stress, focused concentration, or boredom, various genetically determined neurodegenerative disorders and in blind and deaf children they are a version of coping or can be acquired secondarily to infectious or non-infectious mechanism in both overstimulating and understimulating encephalitis, stroke, or drug exposure. The main pathophys- environments [2]. They can be differentiated from tics in that iological mechanism for the occurrence of stereotypies in stereotypy patients do not have an urge to do the movements, an individual is a dysfunction of the cortico-striatal-thala- but they however feel gratified and pleased while performing mo-cortical pathways leading to dopaminergic overactivity them [2, 3]. Stereotypies are distractible and can be easily and cholinergic/GABAergic underactivity causing abnormal suppressed by an external stimulus. However, these patients involuntary movements [2]. rarely make a conscious effort to quell movements [1, 4]. Stereotypies are extensively reviewed in children, espe- Stereotypies can be classified as simple stereotypies such as leg cially those with an autistic spectrum disorder or a pervasive shaking, hair twirling, nail biting, teeth grinding, body rocking, developmental disorder, but the literature on stereotypies in Address for correspondence: Sanjay Pandey, Department of Neurology, Academic Block, Room No 503, Govind Ballabh Pant Postgraduate Institute of Medical Education and Research; New Delhi, India 110002, e-mail: [email protected] www.journals.viamedica.pl/neurologia_neurochirurgia_polska 1 Neurologia i Neurochirurgia Polska 2020, vol. 54 adults is sparse. In this review, our focus will be on various [6-8]. In a study by Harris et al. on 100 non-autistic children conditions associated with stereotypies in adults. presenting with motor stereotypies, it was observed that only six children had a complete resolution of their stereotypies. Methodology The remaining 94 children had persistent stereotypies even in adulthood. Children with arm/ hand stereotypies and whose We searched the PubMed database on 31 March 2020 using stereotypies initially persisted for more than a year had a great- the search terms “Adult-onset stereotypies”, “Drug induced er chance of developing persistent stereotypies throughout stereotypies”, “Stereotypies in psychiatry”, “Stereotypies in de- adulthood than children with head-nodding stereotypies [6]. mentia”, “Stereotypies in stroke”, “Stereotypies in autoimmune In children with persistent stereotypies, as the age advanced disorders” and “Stereotypies in viral encephalitis”. After exclud- the stereotypies remained the same, improved, or became ing animal studies, we identified 2,552 articles for screening. worse. In another study on 49 children (aged 9–20 years) with After removing duplicates and articles in languages other than primary complex motor stereotypies, Oakley et al. reported English, 110 articles were eligible for full text reading. Thir- that stereotypies were persistent in 98% of individuals in ty-one articles were further excluded as they did not describe a long term follow up; 18% reported the appearance of new a motor or phonic stereotypy in detail. Finally, 79 articles were stereotypic movements, and 45% reported a change in their included for our detailed review [Fig. 1, Tab. 1]. original stereotypic movements [7]. ‘Leg stereotypy disorder’ is another movement disorder re- Stereotypies in adults ported in adults and was first described by Jankovic in 2016 [9]. Stereotypic movements in this disorder are characterised by Physiological stereotypies in adults repetitive 1–4 Hz flexion-extension, abduction-adduction Physiological stereotypies are common in children, but movements of the hip while people are seated with their feet rarely seen in adults. However, various studies have demon- resting on the floor. Stereotypic movements can also occur at strated that paediatric stereotypies may persist into adulthood knee or ankle joints while sitting cross-legged or sometimes Records identified through database searching (n = 2,552) Identification Records excluded (n = 241). Reasons for Articles after duplicates exclusion — articles were not removed relevant to stereotypy or (n = 351) they were in languages other than English Screening Full text articles excluded Full-text articles assessed (n = 31) for eligibility Reason for exclusion — detailed (n = 110) description of motor and phonic stereotypy was missing Eligibility Full-text articles included in systematic review (n = 79) Included Clinical studies Case reports Case series and reviews (n = 20) (n = 7) (n = 52) Figure 1. Search strategy for the systematic review 2 www.journals.viamedica.pl/neurologia_neurochirurgia_polska Tanvi Shukla, Sanjay Pandey, Stereotypies in adults: a systematic review Table 1. PubMed search (on 31 March 2020) results, using different search terms for ‘Stereotypies in adults’ Search term Number of hits Relevant articles after remo- Articles finally selected ving duplication Adult-onset stereotypies 132 33 13 Drug induced stereotypies 1,408 197 21 Stereotypies in psychiatry 773 48 11 Stereotypies in dementia 167 36 13 Stereotypies in stroke 48 20 12 Stereotypies in autoimmune disorders 21 14 6 Stereotypies in viral encephalitis 3 3 3 Total 2,552 351 79 people may just tap their feet on the ground while sitting. the use of neuroleptics can be grouped under acute rea ctions, Also, there can be associated ‘fidgeting’ movements of hands for example acute dystonia, and chronic reactions such as and/or other body parts. These movements usually stop while tardive syndromes [14]. Although tardive dyskinesia (TD) standing or walking, but some patients may have a swaying or tardive stereotypies are more commonly associated with movement on standing. Patients also report anxiety with the first-generation antipsychotics, one series has demonstrated effort to stop the movements. Leg stereotypies are frequently that around 3.4% of patients developed TD with aripiprazole, familial, suggestive of a genetic origin. They may or may not which is a third-generation antipsychotic [15]. be associated with anxiety or Attention Deficit Disorder [9]. Stereotypic movements in ‘classic tardive dyskinesia’ typi- A screening questionnaire and rating scale have been proposed cally affect the orolingual region causing repetitive, rhythmic, for leg stereotypy disorder [9]. Although no definite treatment stereotyped movements like lip smacking, puckering, chewing, has been suggested, dopaminergic and GABAergic drugs have tongue protrusion, and tongue movements inside the mouth. been used in some
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