Primary Complex Motor Stereotypies in Older Children and Adolescents: Clinical Features and Longitudinal Follow-Up

Oakley, Christopher; Mahone, E. Mark; Morris-Berry, Christina; Kline, Tina; Singer, Harvey S.

doi:10.1016/j.pediatrneurol.2014.11.002

Cited by 60 publications

(46 citation statements)

References 21 publications

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“…Phenomenologically, tics and rodent stereotypies have a number of characteristics in common. Like tics, spontaneous stereotypies in animals can be interrupted by distractors (Hill et al, 2012; Mason and Newsom, 1990; Oakley et al, 2015). Both tics and animal stereotypies can be precipitated by contextual triggers and particular psychological states, such as high or low sensory stimulation, stress, anxiety, frustration, and fatigue (Conelea and Woods, 2008; Godar and Bortolato, 2016; Kurlan, 2010; Leckman, 2002; Mason, 1991).…”

Section: Studying Tic-like Movements Using a Phenomenological Approachmentioning

confidence: 99%

Modeling tics in rodents: Conceptual challenges and paths forward

Bortolato

Pittenger

2017

Journal of Neuroscience Methods

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Background Recent advances in our understanding of the neurobiology of tics have led to the development of novel rodent models capturing different pathophysiological and phenotypic aspects of Tourette syndrome. The proliferation of these models, however, raises vexing questions on what standards should be adopted to assess their theoretical validity and empirical utility. Assessing the homology of a rodent motoric burst with a tic remains problematic, due to our incomplete knowledge of the underpinnings of tics, their high phenotypic complexity and variability, limitations in our ability test key aspects of tic phenomenology (such as premonitory sensory phenomena) in animals, and between-species differences in neuroanatomy and behavioral repertoire. These limitations underscore that any interpretation of behavioral output in an animal model cannot exclusively rely on the recognition of features that bear superficial resemblance with tics, but must be supported by other etiological and convergent phenomenological criteria. New method Here, we discuss two complementary approaches for the study and validation of tic-like manifestations in rodents, based respectively on the use of contextual modulators and accompanying features of repetitive motor manifestations and on the reproduction of pathogenic factors. Results Neither strategy can by itself provide convincing evidence that a model informatively recapitulates tic pathophysiology. Their combination holds promise to enhance the rigorous evaluation and translational relevance of rodent models of tic disorders. Conclusions This systematic consideration of different approaches to the validation and study of animal models of tic pathophysiology provides a framework for future work in this area.

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Section: Studying Tic-like Movements Using a Phenomenological Approachmentioning

confidence: 99%

Modeling tics in rodents: Conceptual challenges and paths forward

Bortolato

Pittenger

2017

Journal of Neuroscience Methods

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“…Repetitive movements are not specific to ASDs, as they are also observed in other neurodevelopmental and psychiatric disorders, such as intellectual disabilities and schizophrenia, and neurological conditions like Tourette syndrome and Parkinson's disease (Bodfish et al, 2000;Carcani-Rathwell et al, 2006;Frith and Done, 1990;Turner, 1997). Stereotypies can also sometimes occur in normal developing children (Freeman et al, 2010;Oakley et al, 2015).…”

Section: Restricted Repetitive and Stereotyped Patterns Of Behaviormentioning

confidence: 99%

Autism Spectrum Disorders: Translating human deficits into mouse behavior

Pasciuto

Borrie

Kanellopoulos

et al. 2015

Neurobiology of Learning and Memory

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“…On the other hand, major problems can be encountered in disentangling primary CMS from stereotypies occurring in high-functioning ASD. It should be mentioned that ASD was an exclusionary criterion in many studies on primary CMS: the children with a previous history of ASD diagnosis (based on a review of medical records) and those with overt risk of ASD (based on the Autism Spectrum Screening Questionnaire, carried out during a telephone screening) were excluded from the studies (20, 21). …”

Section: Introductionmentioning

confidence: 99%

Developmental Profile and Diagnoses in Children Presenting with Motor Stereotypies

et al. 2016

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IntroductionMotor stereotypies represent a typical example of the difficulty in distinguishing non-clinical behaviors (physiological and transient) from symptoms or among different disorders [“primary stereotypies,” associated with autistic spectrum disorder (ASD), intellectual disabilities, genetic syndromes, and sensory impairment]. The aim of this study was to obtain an accurate assessment on the relationship between stereotypies and neurodevelopmental disorders.MethodsWe studied 23 children (3 girls), aged 36–95 months, who requested a consultation due to the persistence or increased severity of motor stereotypies. None of the patients had a previous diagnosis of ASD. The assessment included the Motor Severity Stereotypy Scale (MSSS), the Repetitive Behavior Scale-Revised (RBS-R), the Raven’s Colored Progressive Matrices, the Child Behavior CheckList for ages 1½–5 or 4–18 (CBCL), the Social Responsiveness Scale (SRS), and the Autism Diagnostic Observation Schedule-second edition (ADOS 2).ResultsAll patients were showing motor stereotypies for periods of time varying from 6 to 77 months. The MSSS showed that each child had a limited number of stereotypies; their frequency and intensity were mild. The interference of stereotypies was variable; the impairment in daily life was mild. The RBS-R scores were positive for the subscale of “stereotypic behaviors” in all children. Moreover, several children presented other repetitive behaviors, mainly “ritualistic behavior” and “sameness behavior.” All patients showed a normal cognitive level. The CBCL evidenced behavioral problems in 22% of the children: internalizing problems, attention, and withdrawn were the main complaints. On the SRS, all but one of the tested patients obtained clinical scores in the clinical range for at least one area. On the ADOS 2, 4 patients obtained scores indicating a moderate level of ASD symptoms, 4 had a mild level, and 15 showed no or minimal signs of ASD.DiscussionMotor stereotypies in children with normal cognitive level represent a challenging diagnostic issue for which a finely tailored assessment is mandatory in order to define a precise developmental profile. Thus, careful and cautious use of standardized tests is warranted to avoid misdiagnosis. Furthermore, it is hard to consider motor stereotypies, even the primary ones, exclusively as a movement disorder.

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Primary Complex Motor Stereotypies in Older Children and Adolescents: Clinical Features and Longitudinal Follow-Up

Cited by 60 publications

References 21 publications

Modeling tics in rodents: Conceptual challenges and paths forward

Modeling tics in rodents: Conceptual challenges and paths forward

Autism Spectrum Disorders: Translating human deficits into mouse behavior

Developmental Profile and Diagnoses in Children Presenting with Motor Stereotypies

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