Dance and rehabilitation in cerebral palsy: a systematic search and review

DEVELOPMENTAL MEDICINE & CHILD NEUROLOGY SYSTEMATIC REVIEW Dance and rehabilitation in cerebral palsy: a systematic search and review CITLALI LOPEZ-ORTIZ1,2 | DEBORAH J GAEBLER-SPIRA3,4 | SARA N MCKEEMAN1 | REIKA N MCNISH1 | DIDO GREEN5,6,7 1 Neuroscience of Dance in Health and Disability Laboratory, Department of Kinesiology and Community Health, College of Applied Health Sciences, University of Illinois at Urbana–Champaign, Champaign, IL; 2 Joffrey Ballet Adacemy, The Official School of the Joffrey Ballet, Chicago, IL; 3 Feinberg School of Medicine, Northwestern University, Chicago, IL; 4 Shirley Ryan AbilityLab, Chicago, IL, USA. 5 Ealing Services for Children with Special Needs, London North West Healthcare NHS Trust, London, UK. 6 Department of Rehabilitation, School of Health and Welfare, J€onk€oping University, J€onk€oping, Sweden. 7 Centre for Rehabilitation, Oxford Brookes University, Oxford, UK. Correspondence to Citlali Lopez-Ortiz, 906 S Goodwin Avenue, Urbana, IL 61801, USA. E-mail: [email protected] This article is commented on by Herman on page 386 of this issue. PUBLICATION DATA AIM To conduct a review of research literature on the use of dance and movement with Accepted for publication 21st August 2018. music (rhythmic auditory stimulation [RAS]) in the neurorehabilitation of children and adults Published online 23 October 2018 with cerebral palsy (CP). METHOD We conducted a systematic search and quality appraisal of the research literature ABBREVIATIONS on dance and RAS in CP. Additionally, we linked the research outcomes to the International ICF International Classification of Classification of Functioning, Disability and Health (ICF) framework. RESULTS Studies showed preliminary evidence of the benefits of dance and RAS on body Functioning, Disability and functions, particularly balance, gait, walking, and cardiorespiratory fitness for individuals with Health CP. Research gaps are evident across all domains of the ICF, particularly in the participation ICF-CY International Classification of and environment domains. Functioning, Disability and INTERPRETATION To facilitate translation of quantitative research outcomes to the clinical Health – Children and Youth classification of the ICF, a table was constructed that links traditional areas of quantitative RAS Rhythmic auditory stimulation rehabilitation research with the ICF categories highlighting areas of research strengths and areas where increased rigor is desirable. The potential for dance and RAS to have positive impacts on body functions, emotional expression, social participation, and attitudinal change are indicated areas for consideration in future research. Individuals with cerebral palsy (CP) have limitations with individuals with CP opportunities to explore movement in environments that can address associated impairments. posture and movement that reduce participation in physical Dance – a creative and expressive art – generally involves and social activities.1 CP is diagnosed in childhood but is a the performance of movement to music. Incorporating dance as an art form into rehabilitation has capacity to lifelong condition; with increasing life expectancy, most peo- transcend traditional barriers in therapy that differentially focus on impairments and limitations. Dance enables ple with CP now live into adulthood. However, balance, opportunities for engaging in a social activity, while pro- viding therapeutic benefit.7 Rhythmic auditory stimulation strength, and endurance appear to decline with age, thereby (RAS), which also includes movement and rhythm, has a reducing activity.2 Along with motor deficits, coexisting con- greater emphasis on synchronization of gait to a steady external rhythm, which may include melody, and does not ditions commonly occur with CP, including sensory deficits, focus on the performative capacity of gait.8 Participation in artistic endeavours may not only provide a motivating envir- epilepsy, intellectual impairment, learning problems, atten- onment, but also contribute to enhanced recovery through opportunities for enjoyable creative expression.9 tion deficit, autism, and musculoskeletal misalignment. These deficits impact the child over time.3 With age, chil- For children with CP, emerging dance programmes offer adjuncts to physical and occupational therapy to enhance dren and adults with CP become increasingly sedentary, fur- enjoyment, motivation, and participation in therapy.10,11 ther reducing social engagement and participation.4 Evidence shows that dance has potential to impact proprio- ception, balance, sensorimotor function, posture, motor Activities and opportunities for therapeutic movement and rehabilitation, such as physical therapy, decline over time for children with CP.5 Community resources and accessibility vary, and often are limited within healthcare systems.6 Children with CP face shifts in priorities with increasing responsibility in school and greater importance placed on relationships and socializing as they become ado- lescents. Dance classes and moving to music provide © 2018 Mac Keith Press DOI: 10.1111/dmcn.14064 393

timing, procedural and working memory, rehearsing, copy- What this paper adds ing, mirroring, and aesthetic expression and appreciation.12 Discrete postures and gestures used in dance constitute the • The potential for dance and movement to music help balance, gait, and simplest elements of a movement vocabulary that can be used to compose sequences of meaningful movement. These walking in children and adults with cerebral palsy. movement sequences may transfer to everyday functional mobility, thereby providing potential rehabilitative benefit. • Research gaps are evident across International Classification of Functioning, Dance may offer therapeutic benefit to individuals with Disability and Health domains, particularly participation and environment CP across different therapeutic areas and health and educa- domains. tion disciplines, as well as personal artistic development, within multiple contexts for participation; each provides search process; we did not restrict the types of included different perspectives and terminologies. Thus, there is a studies.15 We aimed for an exhaustive, comprehensive need for a consistent language and framework to consider search to produce a best-evidence synthesis mapped against the mechanisms and processes of dance within rehabilita- the ICF. tion, as well as the outcomes that might be facilitated. The International Classification of Functioning, Disability and Search methods Health – Children and Youth (ICF-CY) is a useful con- We searched six academic databases: Academic Search struct to organize this review as it provides a common lan- Complete, PubMed, Scopus, CINAHL, Cochrane Data- guage that considers the characteristics of the individual, base of Systematic Reviews, and SPORTDiscus; limiting influences of the environment, and interactions with the searches to scholarly peer-reviewed academic journals. intervention.13 The International Classification of Func- Boolean search phrases were identified in consultation with tioning, Disability and Health (ICF) framework is recom- a health sciences librarian. Search terms were ‘art therapy’, mended to guide development of common data elements ‘cerebral palsy’, ‘danc*’, and ‘rhythmic auditory stimula- for clinical/research studies in CP. tion’. Medical subject heading terms were used in PubMed, when applicable. All available years were included; how- The ICF-CY is consistent with recent models of partici- ever, language restrictions were applied to include only pation as both a process and an outcome.14 A family of par- articles in English. All review articles were excluded. All ticipation-related constructs defines intrinsic person-focused searches were done electronically and the last day that processes in interaction with extrinsic environment-focused searches were run was June 9th, 2017. The search strategy processes to influence participation. Activity can then be is described in Table SI (online supporting information), measured as capacity and performance. Capacity reflects the along with Boolean search phrases, the number of relevant highest level of skill performance that can be achieved in a results, the citations of relevant articles, citations of supportive environment, performance reflects the level of excluded articles, and the reasons for exclusion. skill demonstrated in a typical environment. It is important to consider this distinction with respect to the opportunities Initially, two authors (SNM, RNM) screened titles, that dance has for not only developing skills, but also hav- abstracts, and full texts of the returned results to reject stud- ing the confidence to integrate and use these across daily ies that possessed any of the following characteristics: (1) activities and special events. Performance therefore takes on exercise studies with no performing arts aspects; (2) studies different dimensions; at one level having the confidence to of surgeries as treatment method; (3) a focus on the status of execute a more discrete performance reflecting skills used in accessibility to rehabilitation, appropriate class structure, cur- daily life, through to having the confidence to perform ricula, or methods of analysis of therapy sessions; (4) studies either solely or as part of a group in public. The ICF-CY on virtual reality; (5) video gaming; (6) studies focusing on has been applied to CP research, increasing understanding the experience of caregivers, parental competencies, or the of the impact of impairments on participation. interpersonal relationships of therapists and clients; and (7) art therapy studies with no dance aspect. All types of study This review considers the potential of dance and RAS design were included and all ages were considered to ensure a within rehabilitation for infants, children, adolescents, and broad review scope. A table was created summarizing study adults with CP. In view of the clinical and non-clinical type, number of participants, age of participants, primary and contexts for inclusion of dance in rehabilitation, the evi- secondary outcomes, measurement techniques, and results of dence is mapped across the domains of the ICF-CY. We each article. This table was independently reviewed by CLO, aim to provide a consistent language to consider the mech- DJGS, and DG to determine final eligibility for each article anisms by which dance and RAS may influence skill acqui- (Table SII, online supporting information). sition and participation, with participation considered as both process (e.g. engagement, motivational influences) Quality appraisal and outcome. We employed rating criteria for quality appraisal of the arti- cles. The Grading of Recommendations, Assessment, Devel- METHOD opment and Evaluation system, Physiotherapy Evidence Database Scale, and Downs and Black Quality Checklist were A systematic search and review was carried out to combine not considered appropriate owing to difficulties in determin- the strengths of a critical review with a comprehensive ing ‘equivalence’ between different methodologies, or limits to randomized or non-randomized studies respectively. 394 Developmental Medicine & Child Neurology 2019, 61: 393–398

One author (DG), created a modified scale based on var- RESULTS ious Critical Appraisal Skills Programme checklists.16 The Search results resultant Quality Appraisal Scale (Appendix S1, online sup- The search yielded 66 articles. Once 25 duplicates were porting information) ensured sufficient representation of removed and articles were reviewed for inclusion or exclu- items across case control, cohort, and randomized con- sion criteria, 17 remained. Six articles were excluded for trolled trials, and relevant variations from the Critical the following reasons: the central topic of discussion was Appraisal Skills Programme checklists: theoretical rational not truly dance or RAS-related but addressed components for the study; appropriate methodological design; recruit- such as psychological measures or school counselling, or ment information; description and representativeness of the RAS included no dance component, only stair-step- participants; robustness of research, including control for ping. Eleven articles were left for review (Figure S1, online bias; sufficiently appropriate and rigorous data analysis (in- supporting information).8,10,11,20–27 cluding qualitative analyses where appropriate); control for confounders; and clear discussion of implications of Characteristics of the studies findings. Six studies involved dance and five used RAS. Seven studies included adults with CP, two included children and adults Articles were assessed on each quality listed in with CP, and two included only children with CP. The Appendix S1 with ‘1’ indicating the article possessed the number of participants ranged from 1 to 44. Study design assessed quality, ‘0’ indicating it did not, and ‘–1’ indicat- types included 1 case study, 10 clinical trials (of which ing that the given quality was not applicable. The extent to three were randomized controlled studies), and 3 were which the assessed quality (1) was good, adequate, or less pilot studies. For details on methods and results of each than adequate, was subdivided into ‘1+’, ‘1’, and ‘1–’ study see Table SII. The results are shown in Figure 1. reflecting good, adequate, and less than adequate attributes respectively. CLO, DJGS, and DG rated each included All studies included appropriate theoretical considera- article independently. Meetings ensued to discuss disagree- tions, and only one did not have a clearly designed ments until a consensus was reached. In order to mitigate research methodology,20 representing a qualitative phe- against a possible bias of authors assessing their own stud- nomenological case study. This paper constitutes a posteri- ies, in cases of disagreement, the rating by the author inde- ori presentation of mobility improvement in an adult pendent of the study was accepted as valid. professional actor with CP after dance training with a dance performance (public engagement) objective. The ICF-CY coding and linking rules remainder of the studies used validated and or reliable methods for outcome measures. Data analysis was adequate The outcomes were coded using the ICF-CY to better in 10 studies and not applicable in one. Eight studies understand and discuss dance within the rehabilitation and included clear discussions of the clinical implications of the CP research community, as well as provide a common lan- results. Half of the studies did not include a priori power guage with which to communicate with other professionals calculations. This might be explained by the lack of exist- of the dance community. The linking processes involved ing data in dance interventions applicable to studies. assigning codes of the ICF domains of ‘Body Structure and Function’, ‘Activities and Participation’, and ‘Environmen- Most studies did not directly address confounding fac- tal Factors’. Codes were further divided into chapters, and tors resulting from the absence of blinding. Studies on second, third, and fourth levels within the ICF-CY. The physical interventions are unavoidably characterized by the two dimensions of participation, and presence and involve- impossibility of blinding the participant to the interven- ment, were separated in the linking process according to tion. However, it is possible to blind assessors and no stud- the recommendations of Augustine et al.,17 to capture ies reported assessor blinding. mental health constructs involving interpersonal relations and subjective experiences. DG coded the outcomes ini- ICF-CY coding and linking results tially. CLO and SNM, and DJGS then independently veri- The reported outcomes were linked to ICF codes to pro- fied ICF-CY assigned codes. Disagreements in coding vide a consistent language to discuss the articles. The allocation were re-examined by CLO, DG, and DJGS and results are shown in Table SIII (online supporting infor- the most appropriate codes were agreed following the mation). recently updated refinements to ICF linking rules.18,19 This allowed each meaningful concept to be linked to the All papers directly measured Body Functions, most most precise ICF category and the methods for assigning prominently: b1 (mental functions); b4 (functions of the concepts as either personal or health factors (or not-assign- cardiovascular, haematological, immunological, and respi- able) in an objective manner.18 This process resulted in ratory systems); and b7 (neuromusculoskeletal and move- several coding rules for responses that were initially ment-related functions). Additional areas were indicated as ambiguous (e.g. ‘cadence’ was coded as b770-Gait pattern significant, including categories within: b1 (mental func- functions rather than d450-Walking because it reflects the tions); b4 (functions of the cardiovascular, haematological, rhythm associated with pattern of walking rather than the immunological, and respiratory systems); and b5 (functions activity of walking) and/or contained latent constructs.17 of the digestive, metabolic, and endocrine systems [Table SIII]). Review 395

Study Modified CASP categories 1 2 3 4a 4b 5a 5b 5c 5d 6 7a 7b 7c 8 9 10 Chaitow et al.20 –– – Efraimidou et al.21 – –– – Kim et al.22 – –– –– Kim et al.8 –– ––– Kwak 23 –– – López-Ortiz et al.10 – –– López-Ortiz et al.24 Shin et al.25 –– Teixeira-Machado et al.11 – – Terada et al.26 – Terada et al.27 – Figure 1: Representation of the agreed-upon ratings for the modified Critical Appraisal Skills Programme (CASP) Quality Appraisal Scale. Black boxes represent a rating of 0, indicating the article did not address the domain assessed; grey boxes represent a rating of –1, indicating that the domain assessed was not applicable to the given article; and white boxes represent a rating of 1, indicating that the article did address the domain assessed. Within white boxes, a ‘+’ or a ‘–’ represent a ‘1+’ or ‘1–’ respectively, which are subsets of the ‘1’ category, with ‘1+’ being an excellent example of the given domain and ‘1–’ being a less adequate example. Subsections of the Body Functions domain were related to muscles and movement functions) and b1643 distributed as follows: 80% of papers reflected mental (cognitive flexibility). Terada et al.27 indicated attendance functions; 70% reflected neuromusculoskeletal and move- to the cardiovascular system through the subsections of ment-related functions; 20% reflected cardiovascular and b429 (blood pressure) and b455 (exercise tolerance func- respiratory system functions. Aside from the papers by tions), and to metabolic and digestive functions through Terada et al.,26,27 all papers included b1471 (quality of b540 (general metabolic functions) and b530 (weight main- psychomotor function under specific mental functions tenance functions). [b140–b189]) of section b1 (mental functions). Under neuro- musculoskeletal and movement-related functions, several DISCUSSION subsections were shared across papers: control of b760 This systematic search and review shows the need to inves- (voluntary) and b765 (involuntary movement functions), tigate further the effects of dance-based interventions for b770 (gait pattern functions), and b720 (mobility of bone rehabilitation in CP. Most importantly, detailed reporting functions). Finally, cardiovascular system and respiratory on study protocols is recommended for effective replication system functions are only explored by Terada et al.26,27 and implementation of the interventions, especially: (1) Detailed specifications of each paper with respect to their blinding; (2) relationship between the experimenter, dance measured categories are included in Table SII. instructors, and participants; (3) training level of individu- als delivering the dance interventions; (4) recruitment Body Functions that were indicated within studies with- methods and population; (5) dance teaching methodology; out being directly measured were apparent in 2 of the 10 and (6) intervention location and environment. Regarding papers,20,27 and are listed in Table SIII. Chaitow et al.20 research methods, there is a need to bridge the results of indicated attendance to a subsections of movement func- rigorous quantitative techniques to clinical outcomes, to tions and specific mental functions: b1643 (sensations 396 Developmental Medicine & Child Neurology 2019, 61: 393–398

facilitate translation of research and adoption of successful their feelings regarding their dance programme. Given the interventions. potential of dance and creative movement to impact on a number of personal factors, including self-concept and self- The mapping of dance research outcomes to ICF classi- efficacy, as well as social participation and environmental fications bridges research to daily contexts.18 This endeav- domains, it is noteworthy how few of the studies explored our is challenging, as researchers have not typically these aspects. The RAS studies of walking to prescribed designed experiments based on the ICF. To promote such music rhythms did not consider social communication, envi- transition in further research on rehabilitation using dance, ronmental, or personal factors as outcomes. we created Table SIV (online supporting information), which lists the main attributes of dance skill training from There are limitations to this review, particularly with the neurophysiological point of view and maps them into respect to the difficulty in extracting data across such var- the most salient items of the ICF. ied methodologies and the small number of studies which have explored the role of dance and movement to music. Considering the theoretical potential of dance to modu- However, over half of these studies used validated and late health status, relatively few of the potential body struc- objective methods to assess outcomes. Additionally, it was tures and functions categories were addressed in the unclear from the studies what prior exposure and experi- studies, and even fewer ICF activity and participatory ence participants may have had with dance. Further, there domains were directly addressed. Additionally, internal and may be some inherent bias due to the professional interests external factors, which modify participation, were not mea- and backgrounds of the authors. A strength of this review sured. In contrast to the other articles, Chaitow et al.20 is the mapping of these outcomes against the ICF domains, reference change in attitudes towards performance and dis- which provides a framework for considering the mecha- ability. Attitudinal factors concerning disability are cultur- nisms and processes of the performing arts within rehabili- ally complex and deeply engrained. Understanding tation. attitudes is an important part of the biosocial framework of the ICF and has the potential to catalyse change in public CONCLUSION policy.28 The literature reports greater participation when This systematic review identified 11 studies providing pre- disability stigma decreases and attitudes toward disability liminary evidence of the benefits of dance and RAS on improve, with dance supporting social integration for chil- body functions, particularly those associated with balance, dren with intellectual disability.7 gait, and walking for individuals with CP, with some indi- cation for benefits to cardiorespiratory fitness for individ- The role of dance, music, and arts more generally, in uals with more movement restrictions. Using the ICF as a supporting emotional expression and facilitating well-being common language to define research outcomes across has been demonstrated across a number of public health interdisciplinary studies highlighted research gaps, particu- projects.29 The ICF-CY is limited in its capacity to capture larly in the domains of participation and environment. The personal aspects related to self-esteem and mood, which potential for dance and movement with music to have posi- may form an important component of a dance pro- tive impacts on emotional expression, social participation, gramme.21 There is an increased reported risk of mental and attitudinal change are indicated areas for consideration health problems in young people with CP.30 It thus seems in future research. imperative to consider additional therapeutic interventions that target broader concepts of participation, health, and ACKNOWLEDGMENTS well-being than are currently evidenced in the literature. The authors have stated that they had no interests that might be Engagement in creative movement, coupled with music perceived as posing a conflict or bias. and rhythm, provides opportunities for non-verbal and emotional expression, activity competence, and participa- SUPPORTING INFORMATION tion engagement. The opportunity to use dance to influ- The following additional material may be found online: ence societal attitudes about and towards disability has been hinted at by the work of Chaitow et al.20 These areas Figure S1: Preferred Reporting Items for Systematic Reviews of transdisciplinary research to understand the potential of and Meta-Analyses flow chart. dance have yet to be explored. Table SI: Search strategy Dance with music and rhythm may be integrated into reha- Table SII: Summary of studies bilitation, physical, and occupational therapy to increase par- Table SIII: Categories and linked codes for directly measured ticipation in therapy and enjoyment.10,11 Music may outcomes across all studies determine the dramatic or expressive quality of the dance Table SIV: Main attributes of dance skill training mapped to and, reciprocally, music may be written to match the require- the most salient items of the International Classification of Func- ments of the movement and/or narrative in a dance and pro- tioning, Disability and Health vide a temporal template for movement composition. Appendix S1: Modified Critical Appraisals Skills Programme Quality Appraisal Scale. Considering engagement in dance as both a process and outcome, only Lopez-Ortiz et al.10 asked participants about Review 397

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DEVELOPMENTAL MEDICINE & CHILD NEUROLOGY SYSTEMATIC REVIEW RESUMEN LA DANZA Y LA REHABILITACION EN LA PARALISIS CEREBRAL: UNA BUSQUEDA SISTEMATICA Y REVISION OBJETIVO Realizar una revision de la literatura de investigacion sobre el uso de la danza y el movimiento con la musica (estimulacion auditiva rıtmica [RAS]) en la neurorrehabilitacion de nin~os y adultos con paralisis cerebral (PC). METODO Llevamos a cabo una busqueda sistematica y una evaluacion de calidad de la literatura de investigacion sobre danza y RAS en PC. Ademas, vinculamos los resultados de la investigacion al marco teorico de la Clasificacion Internacional del Funcionamiento, de la Discapacidad y de la Salud (CIF), RESULTADOS Los estudios mostraron evidencia preliminar de los beneficios de la danza y el RAS en las funciones corporales, particularmente el equilibrio, la marcha, la marcha y la capacidad cardiorrespiratoria para las personas con PC. Las brechas de investigacion son evidentes en todos los dominios de la CIF, particularmente en los ambitos de participacion y medio ambiente. INTERPRETACION Para facilitar la traduccion de los resultados de la investigacion cuantitativa a la clasificacion clınica de la CIF, se construyo una tabla que vincula las areas tradicionales de investigacion de rehabilitacion cuantitativa con las categorıas CIF. destacando areas de fortalezas de investigacion y areas donde es deseable un mayor rigor. El potencial de la danza y el RAS para tener un impacto positivo en las funciones corporales, la expresion emocional, la participacion social y el cambio de actitud son areas identificadas para tener en consideracion en investigaciones futuras. RESUMO DANCßA E REABILITACßA~O EM PARALISIA CEREBRAL: UMA PESQUISA E REVISA~O SISTEMATICA OBJETIVO Conduzir uma revisa~o da literaturs sobre o uso da dancßa e movimento com musica (estimulacßa~o auditiva rıtmica [EAR]) na neuroreabilitacßa~o de criancßas e adolescentes com paralisia cerebral (PC). METODO Conduzimos uma busca sistematica e avaliacßa~o de qualidade da literatura sobre dancßa e EAR em PC. Adicionalmente, relacionamos os resultados da pesquisa com a estrutura da Classificacßa~o Internacional de Funcionalidade, Incapacidade e Saude (CIF). RESULTADOS Os estudos mostraram evide^ncia preliminar dos benefıcios e EAR nas funcßo~es do corpo, particularmente equilıbrio, marcha, caminhar e aptida~o cardio-respiratoria em indivıduos com PC. Lacunas nas pesquisas sa~o evidentes em todos os domınios da Classificacßa~o Internacional de Funcionalidade, Incapacidade e Saude , particularmente nos domınios de participacßa~o e ambiente. INTERPRETACßA~O Para facilitar a transicßa~o de resultados de pesquisas quantitativas para a classificacßa~o clınica da CIF, uma tabela foi elaborada para relacionar areas tradicionais de pesquisas quantitativas em reabilitacßa~o com as categorias da CIF. Foram realcßadas areas de em que pesquisas esta~o fortes e areas onde maior rigor e necessario. O potencial da dancßa e EAR para proporcionar impactos positivos nas funcßo~es do corpo, expressa~o emocional, participacßa~o social, e mudancßa atitudinal sa~o indicados como areas para consideracßa~o em futuras pesquisas.