CASE REPORTS

Changes in Muscle Spasticityin Patients With Cerebral PalsyAfter Spinal Manipulation: Case Series

Oleh Kachmar, MD, PhD,aTaras Voloshyn, MD,band Mykhailo Hordiyevych, MDb

ABSTRACT

Objective: The purpose of this case series was to report quantitative changes in wrist muscle spasticity in childrenwith cerebral palsy after 1 spinal manipulation (SM) and a 2-week course of treatment.
Methods: Twenty-nine patients, aged 7 to 18 years, with spastic forms of cerebral palsy and without fixed contractureof the wrist, were evaluated before initiation of treatment, after 1 SM, and at the end of a 2-week course of treatment.Along with daily SM, the program included physical therapy,massage,reflexotherapy,extremity joint mobilization,mechanotherapy, and rehabilitation computer games for 3 to 4 hours’duration. Spasticity of the wrist flexor was measuredquantitatively using a Neuroflexor device, which calculates the neural component (NC) of muscle tone, representing truespasticity, and excluding nonneural components, caused by altered muscle properties: elasticity and viscosity.
Results: Substantial decrease in spasticity was noted in all patient groups after SM. The average NC values decreasedby 1.65 newtons (from 7.6 ± 6.2 to 5.9 ± 6.5) after 1 SM. Another slight decrease of 0.5 newtons was noted after a 2-weekcourse of treatment. In the group of patients with minimal spasticity, the decrease in NC after the first SM was almost twofold—from3.93 ± 2.9 to 2.01 ± 1.0. In cases of moderate spasticity, NC reduction was noted only after the 2-week course of intensive treatment.
Conclusions: In this sample of patients with cerebral palsy, a decrease in wrist muscle spasticity was noted after SM.Spasticity reduction was potentiated during the 2-week course of treatment. (J Chiropr Med 2016;15:299-304)Key Indexing Terms:Spinal Manipulation; Muscle Spasticity; Cerebral Palsy

INTRODUCTION

The termcerebral palsy(CP) refers to a group ofpermanent disorders of the development of movement andposture, which cause activity limitations and are attributedto nonprogressive disturbances of a developing brain.1Itis the most common motor disorder among children,affecting approximately 2 children per 1000 births. One in5 children with CP (20%) has a severe intellectual deficitand is unable to walk.2
Muscle spasticity is a clinical syndrome of CP resultingfrom upper motor neuron lesions, and the reduction of theselesions is an important therapeutic target for optimizingmotor performance. The treatment program for a child withspasticity may include different options: exercises, casting,constraint-induced therapy, oral medications, chemodenerva-tion, intrathecal baclofen, selective dorsal rhizotomy, andorthopedic surgery.3Because of the limited efficiency of“traditional”treatments, a wide range of complementary andalternative therapies are used for muscle tone management inpatients with CP, including spinal manipulation (SM).4,5
Spinal manipulation could possibly be used as aseparate intervention in CP treatment and as part of anintegrated treatment program called theintensive neuro-physiologic rehabilitation system, which includesdifferent treatment modalities: physical and occupationaltherapy, extremity joint mobilization, reflexotherapy,body massage, and mechanotherapy. This treatment maybe performed in intensive 2-week courses lasting 3 to4 hours daily.6
Descriptive studies of this rehabilitation approach havereported improvements in gross motor functions7and adecrease in muscle spasticity in 94% of the cases.8However, these studies had methodologic limitations, andspasticity was measured using the Modified AshworthScale,9whose validity and reliability have been questionedby many authors.10

a) Innovative Technologies Department, International Clinic of Rehabilitation, Truskavets, Ukraine.
b) Rehabilitation Department, International Clinic of Rehabili-tation, Truskavets, Ukraine.Corresponding author: Oleh Kachmar, MD, PhD, 37, Pomir-etska Street, Truskavets, 82200, Ukraine. Tel.: +38 067 3537 927.(e-mail:au.vivl.aher.ci%40ramhcako).
Paper submitted April 26, 2016; in revised form June 24,2016; accepted July 31, 2016.1556-3707© 2016 National University of Health Sciences.http://dx.doi.org/10.1016/j.jcm.2016.07.003

A more precise quantitative evaluation of spasticity ispossible using the Neuroflexor device, developed by theSwedish company Aggero MedTech AB (Stockholm,Sweden) and validated by a research team from theKarolinska Institute (Solna, Sweden). 11 Recent studieshave indicated that Neuroflexor is a reliable measurementtool with high test–retest and interrater reliability, 12 and itssensitivity is good enough to measure changes in spasticityduring CP treatment.13
The purpose of this case series is to describe thequantitative changes in wrist muscle spasticity in childrenwith CP after 1 SM and after a 2-week course of treatment

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METHODS

Patient Selection

Patients were selected for this prospective case seriesaccording to the established inclusion criteria and evaluated3 times. Initial evaluation was followed by SM in 10 to 15minutes, and the second evaluation was carried out after 15minutes. The third evaluation was performed at the end ofthe 2-week course of treatment. All procedures wereperformed in accordance with the ethical standards of theinstitutional committee on human experimentation and theHelsinki Declaration of 1975, as revised in 2000; writteninformed consent was obtained from all patients included inthe study. Research work was approved by the MedicalEthics Commission of the International Clinic of Rehabilitation, located in Truskavets, Ukraine.

Illustration

A total of 30 children admitted to the RehabilitationClinic took part in the study. Inclusion criteria were asfollows: unilateral and bilateral forms of spastic CP, age 7to 18 years, and Manual Ability Classification Scale levelsI–IV. Exclusion criteria were as follows: ataxic ordyskinetic form of CP, fixed contractures of the wristwith less than 50° of passive wrist extension, and inabilityto understand and comply with instructions. The clinicaldiagnosis was confirmed by a child neurologist before thesubjects were included in the study.

One patient failed to participate in the final evaluationbecause of somatic disease and was excluded fromthe study; analysis was carried out in 29 children.

The demographic characteristics of the group are presentedin Table 1.Patients were divided into 3 groups according tothe spasticity level: minimal spasticity (“1” by the ModifiedAshworth scale), 10 children; mild spasticity (“1+” by theModified Ashworth scale), 10 children; moderate spasticity(“2” by the Modified Ashworth scale), 9 children.

Intervention

Spinal manipulation was performed by an orthopedicmedical doctor certified in Manual Therapy. After manualevaluation, high-velocity low-amplitude SM was carriedout in all regions of the spine, including thoracicadjustments in the prone position, lumbar manipulation inlateral recumbent position, and cervical manipulation insitting position.
Spinal manipulation was repeated every day, with a totalof 12 manipulations during the 2-week period. The programfor children with CP also included daily sessions ofphysical therapy, massage, reflexotherapy, extremity jointmobilization, mechanotherapy, and rehabilitation computergames with average daily duration of 3 to 4 hours. A detailed description of the treatment is provided in themanual. 6 No side effects were detected by the researcher and doctor in charge or reported by the patients ortheir parents.

Illustration

Fig 2. Neural component values before intervention (NC-1), after 1 spinal manipulation (NC-2), and after the 2-week course oftreatment (NC-3). Data presented for the whole group, for patients with minimal spasticity (“1” by the Modified Ashworth scale), forpatients with mild spasticity (“1+” by the Modified Ashworth scale), and for patients with moderate spasticity (“2” by the ModifiedAshworth scale). The NC values are given in newtons on the vertical axis.

Evaluation Procedure

aluation ProcedureMuscle tone was measured using the Neuroflexordevice. This instrument extends the wrist and stretchesthe muscles at 2 different constant velocities, whilethe force transducers measure resistance during movements(Fig 1).
Total movement resistance testifies to true spasticity,called the neural component (NC) of muscle tone, which isinduced by the stretch reflex, and nonneural components,caused by altered muscle properties: inertia, elasticity, andviscosity. One test session consisted of 5 slow movementsand 10 fast movements; dedicated software was used toseparate total resistance into its elastic, viscous, and neuralcomponents. Lower NC values correspond to lowerspasticity levels.
A Modified Ashworth Scale score of wrist spasticity wasobtained with the child seated with the elbow flexed to 90° andthe forearm pronated.9 The children’s gross motor functionswere evaluated according to the Gross Motor FunctionClassification System. 14 Hand function was evaluatedaccording to the Manual Ability Classification System.15

Results

Measurement results are summarized in Fig 2 andTable 2, which present NC values before intervention(NC-1), after 1 SM (NC-2), and after the 2-week course oftreatment (NC-3).
Data are presented for the whole group, for patients withminimal spasticity (“1” by the Modified Ashworth scale),for patients with mild spasticity (“1+” by the Modified Ashworth scale), and for patients with moderate spasticity(“2” by the Modified Ashworth scale).
Differences between NC-1 and NC-2 indicate changes inspasticity that occurred after 1 SM; differences betweenNC-1 and NC-3 show changes after the 2-week course oftreatment that included daily SM.Substantial decrease in spasticity was noted both after 1SM and after the 2-week course of treatment. The averagevalues of spasticity decreased by 1.65 newtons (from 7.6 ±6.2 to 5.9 ± 6.5) after 1 SM. After a 2-week course ofintensive treatment with daily SM, there was another slightdecrease in spasticity by 0.5 newtons.
In the group of patients with minimal spasticity, theNC decrease after the first SM was almost twofold—from3.93 ± 2.9 to 2.01 ± 1.0. During the course of treatment,there was a small “rebound” effect, with NC values returningto 2.27 ± 1.4.In cases of mild spasticity, changes in NC were alsonoted after the first SM (from 5.35 ± 3.4 to 3.64 ± 2.4) withsubsequent stabilization at 3.57 ± 2.0.In the moderate spasticity group, changes in NC after thefirst SM were not substantial (from 14.16 ± 3.3 to 12.88 ±7.6), but spasticity levels continued to decrease to 11.23 ±7.5 newtons during the course of treatment.

Discussion

Spinal manipulation is a common treatment modality formusculoskeletal problems, and in many cases, it is used fornonmusculoskeletal conditions.16 There is growing evidence from research studies of the effectiveness ofchiropractic and osteopathic manipulation for nonmusculoskeletal conditions, especially in patients with migraineand headache, 17,18 hypertension, 19,20 chronic obstructive pulmonary disease, 21 and different pediatric conditions,22including CP.23-2

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Our study was aimed at evaluating changes in wristmuscle spasticity in children with CP after 1 SM and a2-week intensive rehabilitation program with daily SMtogether with other treatment modalities: physical therapy,massage, reflexotherapy, extremity joint mobilization,mechanotherapy, and rehabilitation computer games.
In our case series, reduction in spasticity was noted afterthe first manipulation—the NC values of muscle tonedecreased from 7.6 ± 6.2 newtons to 5.9 ± 6.5. After the2-week course of intensive treatment with daily SM, therewas another small decrease in spasticity by 0.5 newtons.
The most pronounced decrease in spasticity after 1 SMwas observed in children with minimal spasticity. In casesof moderate spasticity, NC reduction after 1 SM was lesspronounced but became more prominent after the 2-weekcourse of treatment.
Because decrease in spasticity was noted after 1 SM andthis effect was potentiated by a multicomponent treatmentcourse, we can formulate the hypothesis that SM mighthave an impact on muscle tone regulation.
The influence of SM on muscle spasticity is not fullyunderstood at present. However, an experimental body ofevidence indicates that SM could impact primary afferentneurons from paraspinal tissues and influence musclespindle afferents and Golgi tendon organs,27,28 which aredirectly involved in muscle tone regulation.
The literature points to the influence of SM on spinalcord neural circuits 29,30 possibly modifying stretchreflexes. Interesting information about neural responses toSM has been included in reports of studies on animalmodels. 31,32 Studies have also indicated that SM has aninfluence on the H-reflex,33,34 which is a direct electrophysiologic equivalent for spasticity measurement.This explorative study describes decrease in spasticityafter SM in a group of children with CP.

Luminations

As this was a case series, there was no control group withrandomized allocation or blind testing of participants orexaminers, and the sample size was small. Therefore, wecan only note observed phenomena and cannot calculateinferential statistics or draw conclusions on causation. Thefindings of this study may not necessarily be replicable forother patients with CP or spasticity. Future randomizedcontrolled trials are required to evaluate this effect. Theauthors aim to conduct double-blind randomized clinicaltrials comparing SM and “sham” manipulation to investigatethe possible influence of SM on spasticity.

Conclusions

Decrease in wrist muscle spasticity after SM in patientswith CP was reported in this sample of young patients.Reduction in spasticity was further potentiated during the2-week course of treatment.

Funding sources and conflicts of interest

No funding sources or conflicts of interest were reportedfor this study.

Contributorship information

Concept development (provided idea for the research):O.K., T.V., M.H.
Design (planned the methods to generate the results): O.K.,T.V., M.H.Supervision (provided oversight, responsible fororganization and implementation, writing of themanuscript):O.K.Data collection/processing (responsible for experiments,patient management, organization, or reporting data):T.V., M.H.
Analysis/interpretation (responsible for statisticalanalysis, evaluation, and presentation of the results):O.K., T.V.
Literature search (performed the literature search):O.K., T.V.
Writing (responsible for writing a substantive part of themanuscript):O.K.
Critical review (revised manuscript for intellectual content,this does not relate to spelling and grammar checking):O.K., T.V., M.H.

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REFERENCES

1. Rosenbaum P, Paneth N, Leviton A, et al. A report: the definition and classification of cerebral palsy. Dev Med Child Neurol. 2007;109:8-14.2. Prevalence and characteristics of children with cerebral palsy in Europe.Dev Med Child Neurol. 2002;44(9):633-640.3. Tilton A. Management of spasticity in children with cerebral palsy. Semin Pediatr Neurol. 2009;16:82-89.4. Oppenheim WL. Complementary and alternative methods in cerebral palsy. Dev Med Child Neurol. 2009;51(4):122-129.5. Liptak GS. Complementary and alternative therapies for cerebral palsy. Ment Retard Dev Disabil Res Rev. 2005;11(2): 156-163.6. Kozyavkin VI, Babadagly MO, Lun GP, et al. Intensive Neurophysiological Rehabilitation System—the Kozyavkin Method. A Manual for Rehabilitation Specialists. Lviv, Ukraine: Design studio Papuga; 2012.7. Koziavkin VI, Voloshin TB, Hordievich MS, Kachmar OA. Changes of motor function in patients with cerebral palsy during the treatment using the intensive neurophysiological rehabilitation system. Zh Nevrol Psikhiatr Im S S Korsakova. 2012;112(7 Pt 2):14-17 [in Russian].8. Kozyavkin VI, Kachmar OO. Rehabilitation outcome assessment methods in Intensive neurophysiological rehabilitation system. Ukrayinskyj Medychnyj Chasopys. 2003;3(35):61-66 [in Ukrainian]. 9. Bohannon RW, Smith MB. Inter-rater reliability of a modified Ashworth scale of muscle spasticity. Pys Ther. 1987;67(2): 206-207.10. Fleuren JF, Voerman GE, Erren-Wolters CV, et al. Stop using the Ashworth Scale for the assessment of spasticity. J Neurol Neurosurg Psychiatry. 2010;81(1):46-52.11. Lindberg PG, Gäverth J, Islam M, Fagergren A, Borg J, Forssberg H. Validation of a new biomechanical model to measure muscle tone in spastic muscles. Neurorehabil Neural Repair. 2011;25(7):617-625.12. Gäverth J, Sandgren M, Lindberg PG, Forssberg H, Eliasson AC. Test-retest and inter-rater reliability of a method to measure wrist and finger spasticity. J Rehabil Med. 2013; 45(7):630-636.13. Gäverth J, Eliasson AC, Kullander K, Borg J, Lindberg PG, Forssberg H. Sensitivity of the NeuroFlexor method to measure change in spasticity after treatment with botulinum toxin A in wrist and finger muscles. J Rehabil Med. 2014; 46(7):629-634.14. Palisano RJ, Rosenbaum P, Bartlett D, Livingston MH. Content validity of the expanded and revised Gross Motor15. Manual Ability Classification System for children with cerebral palsy. [cited 2016 March 30]. Available at: http:// www.macs.nu/.16. Clar C, Tsertsvadze A, Court R, Hundt GL, Clarke A, Sutcliffe P. Clinical effectiveness of manual therapy for the management of musculoskeletal and non-musculoskeletal conditions: systematic review and update of UK evidence report. Chiropr Man Therap. 2014;22(1):12.17. Ohlsen BA. Combination of acupuncture and spinal manipulative therapy: management of a 32-year-old patient with chronic tension-type headache and migraine. J Chiropr Med. 2012;11(3):192-201.18. Espí-López GV, Gómez-Conesa A. Efficacy of manual and manipulative therapy in the perception of pain and cervical motion in patients with tension-type headache: a randomized, controlled clinical trial. J Chiropr Med. 2014;13(1):4-13.19. Win NN, Jorgensen AM, Chen YS, Haneline MT. Effects of upper and lower cervical spinal manipulative therapy on blood pressure and heart rate variability in volunteers and patients with neck pain: a randomized controlled, cross-over, preliminary study. J Chiropr Med. 2015;14(1):1-9.20. Yu X, Wang X, Zhang J, Wang Y. Changes in pressure pain thresholds and basal electromyographic activity after instrument-assisted spinal manipulative therapy in asymptomatic participants: a randomized, controlled trial. J Manipulative Physiol Ther. 2012;35(6):437-445.21. Wearing J, Beaumont S, Forbes D, Brown B, Engel R. The use of spinal manipulative therapy in the management of chronic obstructive pulmonary disease: a systematic review. J Altern Complement Med. 2016;22(2):108-114.22. Hawk C, Schneider MJ, Vallone S, Hewitt EG. Best practices for chiropractic care of children: a consensus update. J Manipulative Physiol Ther. 2016;39(3):158-168.23. Davis MF, Worden K, Clawson D, Meaney FJ, Duncan B. Confirmatory factor analysis in osteopathic medicine: fascial and spinal motion restrictions as correlates of muscle spasticity in children with cerebral palsy. J Am Osteopath Assoc. 2007;107(6):226-232.24. Duncan B, McDonough-Means S, Worden K, Schnyer R, Andrews J, Meaney FJ. Effectiveness of osteopathy in the cranial field and myofascial release versus acupuncture as complementary treatment for children with spastic cerebral palsy: a pilot study. J Am Osteopath Assoc. 2008;108(10): 559-570.25. Wyatt K, Edwards V, Franck L, et al. Cranial osteopathy for children with cerebral palsy: a randomised controlled trial. Arch Dis Child. 2011;96(6):505-512.26. Posadzki P, Lee MS, Ernst E. Osteopathic manipulative treatment for pediatric conditions: a systematic review. Pediatrics. 2013;132(1):140-152.27. Pickar JG. Neurophysiological effects of spinal manipulation. Spine J. 2002;2(5):357-371.28. Clark BC, Thomas JS, Walkowski SA, Howell JN. The biology of manual therapies. J Am Osteopath Assoc. 2012; 112(9):617-629.29. Pickar JG, Bolton PS. Spinal manipulative therapy and somatosensory activation. J Electromyogr Kinesiol. 2012; 22(5):785-794.30. Chu J, Allen DD, Pawlowsky S, Smoot B. Peripheral response to cervical or thoracic spinal manual therapy: an evidencebased review with meta-analysis. J Man Manip Ther. 2014; 22(4):220-229.31. Reed WR, Long CR, Kawchuk GN, Pickar JG. Neural responses to the mechanical characteristics of high velocity32. Reed WR, Liebschner MA, Sozio RS, Pickar JG, Gudavalli MR. Neural response during a mechanically assisted spinal manipulation in an animal model: a pilot study. J Nov Physiother Phys Rehabil. 2015;2(2):20-27.33. Niazi IK, Türker KS, Flavel S, Kinget M, Duehr J, Haavik H. Changes in H-reflex and V-waves following spinal manipulation. Exp Brain Res. 2015;233(4):1165-1173.34. Dishman JD, Bulbulian R. Spinal reflex attenuation associated with spinal manipulation. Spine. 2000;25(19): 2519-252