Abstract

We aimed to evaluate the efficacy and safety of injecting botulinum toxin A (BoNT-A) into the neck muscles to treat cervical dystonia (CD) in patients with dyskinetic cerebral palsy (CP). This was a randomized, double-blinded, placebo-controlled trial with cross-over design. We prospectively enrolled adults with dyskinetic CP who were over 20 years old and had been clinically diagnosed with CD for more than one year. The primary outcome measure was the change in Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS) at four weeks from the baseline TWSTRS. Seventeen patients were initially enrolled, but one patient was excluded after the final evaluation because of a violation of the study protocol. At four weeks, the BoNT-A injections showed significant improvement in TWSTRS total scores compared to the saline injections (p = 0.0286 for ANCOVA). At 12 weeks, the BoNT-A injections resulted in greater improvements in TWSTRS total scores than the saline injections without statistical significance (p = 0.0783 for ANCOVA). Dysphagia occurred in three out of 16 patients: two after BoNT-A and one after saline. The dysphagia was transient and improved naturally within two weeks without any special treatment. BoNT-A injection for CD in adults with dyskinetic CP is relatively safe and improves pain and disability.

Highlights

  • In adults with dyskinetic cerebral palsy (CP), cervical dystonia (CD) may result in neurologic sequelae such as pain, dysfunction, and cervical myelopathy [1,2,3]

  • Our study revealed that Botulinum toxin A (BoNT-A) injections used to treat CD in patients with dyskinetic CP result in greater improvement of Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS) scores at four weeks compared with saline injections, especially on the pain and disability subscales

  • Previous work concerning botulinum toxin (BoNT)-A injections in CD reported that TWSTRS scores improved in 29 of 37 (78%) patients and were more effective than placebo [11]

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Summary

Introduction

In adults with dyskinetic cerebral palsy (CP), cervical dystonia (CD) may result in neurologic sequelae such as pain, dysfunction, and cervical myelopathy [1,2,3]. Surgery is often required to ameliorate these symptoms. Surgical outcomes are not satisfactory because the CD remains untreated even after the surgery. Anticholinergic, antidopaminergic, dopaminergic, and GABAergic medications have been proposed to treat CD in patients with dyskinetic CP, but none have proven to be effective [4]. Previous studies have suggested the botulinum toxin (BoNT) as a primary treatment for idiopathic CD [5,6]. Botulinum toxin A (BoNT-A) has been successfully used for many years in idiopathic CD and is considered the first line of therapy [7]. According to the Cochrane review [7], CD patients with an average baseline Toronto Western Spasmodic Torticollis Rating Scale

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