Abstract
BackgroundPrimary dystonia is a chronic neurological movement disorder that causes abnormal muscle movements. Pain and emotional distress may accompany these physical symptoms. Behavioural interventions are used to help people with long term conditions improve their quality of life. Little is known about behavioural interventions applied to Dystonia. We report a systematic review of studies reporting current evidence of behavioural interventions for people with primary dystonia.MethodsWe did systematic searches of Medline, PsycINFO, AHMED and CINAHL. We assessed the methodological quality of included studies using a risk of bias tool. Any disagreements were resolved by liaising with an independent rater. Physiological outcomes such as dystonia severity and psychological outcomes such as sleep and depression were selected on the basis that primary dystonia causes motor and non-motor symptoms. No time limit was placed on the searches. A narrative synthesis of the results is presented.ResultsOf 1798 titles and abstracts screened, 14 full articles were retrieved and inclusion and exclusion criteria applied. Of these a final nine were eligible for the review (N = 73). Only two were Randomised Controlled Trials (RCTs). Using the Movement Disorders Society (MDS) dystonia classification, that was published after this work started, all of the included studies were of idiopathic adult onset focal dystonia without associated features. These included: blepharospasm (eye dystonia) (N = 1), cervical dystonia (neck dystonia) (N = 2), writer’s cramp (hand dystonia) (N = 3) and the yips (N = 3). No studies reported on dystonia that affects two or more body regions. Studies reported good adherence and response rates to treatment. Physiological and psychological improvements were noted in all studies at weekly, monthly and yearly follow-ups. Caution should be taken when interpreting the results because of the scarcity of RCTs identified, use of small sample sizes, and inappropriate statistical methods.ConclusionWe identified few studies; mainly of poor methodological quality that all studied a focal dystonia. It is not possible to draw firm conclusions. Nevertheless, the data suggests that a combined behavioural therapy approach including relaxation practice for people with idiopathic adult onset focal dystonia merits further investigation.
Highlights
Primary dystonia is a chronic neurological movement disorder that causes abnormal muscle movements
When we developed the protocol for this review we used the terms current and set our parameter for study entry to be a primary dystonia; this was the terminology used in our PRoSEPRO registration
We found limited evidence of the following behavioural interventions for different idiopathic adult-onset focal dystonia types: Solution-Focused Guided Imagery (SFGI) for the yips [18, 21, 22], exercise and relaxation practice [23] and Cognitive Behavioural Therapy (CBT) [24] for cervical dystonia, relaxation, systematic desensitisation (SD), assertiveness training and/or Electromyography (EMG) feedback [25], habit reversal with in vivo exposure, awareness training and re-education [26] and relaxation or habit reversal therapy [27] for writer’s cramp and conditioning therapy and relaxation practice for blepharospasm [28]
Summary
Primary dystonia is a chronic neurological movement disorder that causes abnormal muscle movements. Little is known about behavioural interventions applied to Dystonia. We report a systematic review of studies reporting current evidence of behavioural interventions for people with primary dystonia. Dystonia is a neurological condition that can cause abnormal and repetitive muscle contractions and spasms [1]. It is the third most commonly diagnosed movement disorder after Parkinson’s disease and essential tremor [2] with a prevalence variously estimated to be in range of 30–7320 per million [2]. Generalised dystonia typically occurs during childhood and causes spasms and cramping in two or more affected body regions [1]. There may be genetic associations with some idiopathic adult onset dystonias; in a majority of cases the cause of disease is unknown [1]
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