Effects of botulinum toxin A and/or bimanual task-oriented therapy on upper extremity activities in unilateral Cerebral Palsy: a clinical trial

Speth et al. BMC Neurology (2015) 15:143 DOI 10.1186/s12883-015-0404-3 RESEARCH ARTICLE Open Access Effects of botulinum toxin A and/or bimanual task-oriented therapy on upper extremity activities in unilateral Cerebral Palsy: a clinical trial Lucianne Speth1,2*, Yvonne Janssen-Potten2,3†, Eugene Rameckers2,3†, Anke Defesche1†, Bjorn Winkens4†, Jules Becher5†, Rob Smeets2,3,6† and Hans Vles7,8† Abstract Background: This study reports on the effects of botulinum toxin A (BoNT-A) injections in the upper extremity (UE) in children with unilateral Cerebral Palsy (uCP) combined with bimanual task-oriented therapy (BITT) or either treatment modality performed separately. Bimanual activities were measured with the Assisting Hand Assessment (AHA), the ABILHand-Kids questionnaire (AK), the Observational Skills Assessment Score (OSAS). Goal achievement was measured with Goal Attainment Scaling (GAS), using blind video assessment, and the Canadian Occupational Performance Measure (COPM). Methods: Thirty-five children, mean age 7.14 years (SD 2.63), 11 Manual Ability Classification Score (MACS) I, 15 MACS II and 9 MACS III, participated. The trial started with four study groups: BoNT-A-only (n = 5), BITT-only (n = 11), BoNT-A + BITT (n = 13), and control (n = 6). Twenty-two children were randomised, 13 children received their parents’ preferred treatment: BoNT-A + BITT or BITT-only. Three comparisons were analysed: BITT (BoNT-A + BITT and BITT-only; n = 24) versus no BITT (BoNT-A-only and control; n = 11), BoNT-A (BoNT-A-only and BoNT-A + BITT; n = 18) versus no BoNT-A (BITT-only and control; n = 17), and the additional effect of BoNT-A (BoNT-A + BITT versus BITT-only). Follow-up time: 24 weeks. Results: No significant differences between the groups were found on the AHA. The amount of use of both hands on the OSAS was significantly better in the BoNT-A group in the beading and sandwich-making task. The BoNT-A group also showed significant improvement in the quality scores of the OSAS: the wrist position during grasping and holding, especially in the younger children. The BITT group improved significantly on the AK and significantly more on the performance and satisfaction scores of the COPM at 12 and 24 weeks regarding several goals. BoNT-A showed a significant negative effect at 12 and 24 weeks in the most important goal. BITT, more than BoNT-A + BITT, showed positive effects on the GAS score at 12 (significant), 18 and 24 weeks. Conclusions: BoNT-A has a positive effect on quality of movement and amount of use of the affected UE during the 3 months’ working time. BoNT-A has no additional effect on bimanual performance and goal achievement. BITT has a positive effect on goal achievement and bimanual performance, even up to 6 weeks after therapy had stopped. Trial registration: Current Controlled Trials ISRCTN69541857. * Correspondence: † Equal contributors 1 Adelante, Paediatric Rehabilitation, Onderstestraat 29, 6301 KA, Valkenburg, The Netherlands 2 Adelante, Centre of Expertise in Rehabilitation and Audiology, Zandbergsweg 111, 6432 CC, Hoensbroek, The Netherlands Full list of author information is available at the end of the article © 2015 Speth et al. Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Speth et al. BMC Neurology (2015) 15:143 Background According to the Cochrane review [1], the combination of botulinum toxin A (BoNT-A) in the upper extremity (UE) and intensive physiotherapy(PT)/occupational therapy(OT) is more effective than intensive therapy alone in improving the activity level of the International Classification of Functioning (ICF, www.who.int/entity/classifications/icf/en/) and in goal achievement in children with unilateral spastic Cerebral Palsy (uCP). Assessments used to measure effects at the activity level in the studies in this review are the Melbourne Assessment of Unilateral Upper Limb Function (MUUL) [2], the Quality of Upper Extremities Skills Test (QUEST) [3] and the Pediatric Disability Inventory (PEDI) [4]. Goal achievement was measured with Goal Attainment Scaling (GAS) [5] and the Canadian Occupational Performance Measure (COPM) [6]. Although the studies themselves reported no significant differences on the MUUL and limited effects on the QUEST, a meta analysis of the pooled data in this review [1] showed slight positive effects on both tests 3 months after injection of BoNT-A, which had disappeared at 6 months. On the PEDI functional skills score almost no effect was found. There were clear positive effects on GAS and limited positive effects on the COPM concluded. Olesch et al. studied young children (mean age 3 years 8 months) with uCP and found no significant differences on the QUEST between repeated BoNT-A + OT and OT alone. However, they did find positive effects on GAS of BoNT-A + OT [7]. The way GAS was performed in these studies (scoring by parents or therapists) had subjective elements. This should be taken into account, while interpreting these results. Also, in a more recent review evaluating upper limb therapies for uCP there was a modest supplementary effect of BoNT-A as an adjunct to OT to improve unilateral capacity, quality of movement, with the MUUL and the QUEST, and a clear positive effect on achieving individualised treatment outcomes [8]. In this review in only one study looking at the effect of BoNT-A on strength in children receiving resistance training, the assisting hand assessment (AHA) was used to measure bimanual performance [9]. OT after BoNT-A, modified constraint induced movement therapy (mCIMT), bimanual intensive therapy (BIMT) and goal-directed and context-focused therapies are considered to be evidence-based effective improving UE activities [8, 10]. After BoNT-A, mCIMT and bimanual OT (BOT) each consisting of 16 one-hour therapy sessions during 6 weeks both with home therapy elements, were equally effective in young children with uCP, although in hindsight the home programme intensity in the BOT group was significantly less [11]. In this study and in several studies reporting effects of CIMT Page 2 of 15 and/or BIMT, they used the AHA, a reliable, responsive tool to measure the effective use of the assisting hand in bimanual performance [12, 13]. With the AHA, in several studies comparing the effects of two forms of intensive therapy currently often used in uCP, CIMT and BIMT, no difference between these treatments could be demonstrated [14–16]. At the start of our (...truncated)