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Botulinum Toxin as an Adjunct to Serial Casting Treatment in Children with Cerebral Palsy
Robert M. Kay, MD1; Susan A. Rethlefsen, PT1; Anna Fern-Buneo, MA, PT1; Tishya A.L. Wren, PhD1; David L. Skaggs, MD1
1 Childrens Orthopaedic Center, MS #69 (R.M.K., S.A.R., T.A.L.W., and D.L.S.) and Department of Rehabilitation Services, MS #56 (A.F.-B.), Childrens Hospital Los Angeles, 4650 Sunset Boulevard, Los Angeles, CA 90027. E-mail address for S.A. Rethlefsen: srethlefsen@chla.usc.edu
View Disclosures and Other Information
In support of their research or preparation of this manuscript, one or more of the authors received grants or outside funding from Allergan, Incorporated (Irvine, California). None of the authors received payments or other benefits or a commitment or agreement to provide such benefits from a commercial entity. No commercial entity paid or directed, or agreed to pay or direct, any benefits to any research fund, foundation, educational institution, or other charitable or nonprofit organization with which the authors are affiliated or associated.
Investigation performed at the Childrens Hospital Los Angeles, Los Angeles, California

The Journal of Bone and Joint Surgery, Incorporated
J Bone Joint Surg Am, 2004 Nov 01;86(11):2377-2384
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Abstract

Background: Although botulinum toxin A is frequently used to augment serial casting in the treatment of soft-tissue contractures in children with cerebral palsy, its effectiveness for this purpose has not been evaluated. The purpose of the present study was to determine whether botulinum toxin A injection increases the efficacy of serial casting.

Methods: A prospective, randomized trial was undertaken to compare serial casting only with serial casting combined with botulinum toxin A (Botox) injection for the treatment of ankle equinus contractures in twenty-three children with cerebral palsy. Range-of-motion testing, spasticity assessment, and computerized gait analysis were performed as long as twelve months after treatment.

Results: There was no difference between the groups with regard to the duration of casting required to correct the equinus contracture. Both groups maintained a significant improvement in passive ankle dorsiflexion throughout the follow-up period, although the group managed with casting and Botox had a significant loss of dorsiflexion when the values at six, nine, and twelve months were compared with the value at three months. Peak dorsiflexion during the stance and swing phases was significantly improved in both groups at three months but only in the group managed with casting alone at twelve months. Plantar flexor spasticity was significantly decreased at three months in both groups, but it was significantly decreased at six, nine, and twelve months only in the group managed with casting alone. Spasticity was significantly greater in the group managed with casting and Botox than it was in the group managed with casting only at six, nine, and twelve months.

Conclusions: The present study demonstrates the efficacy of serial casting in the treatment of equinus contractures in children with cerebral palsy who are able to walk. Contrary to our hypothesis, the addition of botulinum toxin A to a serial casting regimen led to earlier recurrence of spasticity, contracture, and equinus during gait. The results of the present study suggest that botulinum toxin combined with serial casting for the treatment of fixed contractures will lead to a recurrence of plantar flexor spasticity and equinus contracture by six months in this patient population. While previous research has indicated that the injection of botulinum toxin A is superior to casting for the treatment of dynamic equinus, the present study suggests that serial casting alone is preferable for the treatment of fixed equinus contractures in children with cerebral palsy.

Level of Evidence: Therapeutic study, Level I-1a (randomized controlled trial [significant difference]). See Instructions to Authors for a complete description of levels of evidence.

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    References

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    Robert M. Kay
    Posted on November 22, 2004
    Dr. Kay responds to Dr Graham and colleagues
    Keck School of Medicine, University of Southern California; Childrens Hospital Los Angeles

    To the Editor:

    We thank Drs. Graham, Baker, Boyd, and Flett for their letter in response to our article(1). We appreciate their letter emphasizing that the indication for Botulinum toxin injection is spasticity, not contracture. However, anecdotal evidence suggests that Botulinum toxin is used frequently in conjunction with serial casting for fixed deformities in children with cerebral palsy, and we have personally seen hundreds of children who have had this combination of treatments.

    Due to a lack of directly comparable studies, we compared our results to the studies by Drs. Corry (2) and Flett (3), which were the only other reports in the literature examining these two treatments in children with cerebral palsy. It is clearly stated in both the Introduction and Discussion sections of the paper that the Corry and Flett studies “excluded patients with fixed contractures, using serial casting only for spasticity reduction.” We also state that this primary difference between our patient populations (fixed vs. dynamic equinus) may account for the differences in our results.

    While we agree that dynamic deformity is a primary indication for Botulinum toxin injection in children with CP, we also recognize that it has other uses in current clinical practice. As Dr. Graham and colleagues acknowledge, our current paper addresses one of these other uses.

    1. Kay RM; Rethlefsen SA; Fern-Buneo A; Wren TAL; and Skaggs, DL. Botulinum toxin as an adjunct to serial casting treatment in children with cerebral palsy. J Bone Joint Surg Am, 2004, 86: 2377-84.

    2. Corry, I. S.; Cosgrove, A. P.; Duffy, C. M.; McNeill, S.; Taylor, T. C.; and Graham, H. K.: Botulinum toxin A compared with stretching casts in the treatment of spastic equinus: a randomised prospective trial. J Pediatr Orthop, 1998,18(3): 304-11.

    3. Flett, P. J.; Stern, L. M.; Waddy, H.; Connell, T. M.; Seeger, J. D.; and Gibson, S. K.: Botulinum toxin A versus fixed cast stretching for dynamic calf tightness in cerebral palsy. J Paediatr Child Health, 1999,35(1): 71-7.

    Kerr Graham
    Posted on November 09, 2004
    Indications for Use of Botulinum Toxin in Children with Cerebral Palsy
    Royal Children's Hospital, Melbourne, Australia

    As investigators on a number of previous randomized clinical trials evaluating the use of Botulinum toxin in spasticity associated with cerebral palsy, we read with interest the article by Kay et al, (1). Botulinum toxin A has been widely investigated in a range of animal and clinical studies and certain broad generalizations can be made from the literature:

    1. Botulinum neurotoxin A has a high affinity for presynaptic cholinergic nerve terminals and has a predictable dose dependent consistent clinical effect when injected into skeletal muscles affected by spasticity (2,3).

    2. The harnessing of this predictable biochemical effect has been established in a wide range of clinical studies such that the evidence for the efficacy of the intervention is greater than for any other medical or surgical procedure currently used in the management of cerebral palsy (4).

    3. The principal indication for the clinical use of Botulinum neurotoxin A in children with spasticity associated with cerebral palsy is for the temporary, focal or regional reduction in spasticity to facilitate more normal patterns of motion and on occasions to improve function. In previous work we defined the indication for injection of Botulinum toxin in cerebral palsy to be “dynamic spasticity interfering with function in the absence of fixed contracture” (2,3).

    The principal indication for use of Botulinum neurotoxin A in cerebral palsy was and remains reduction of spasticity. However we recognize that many “niche applications” have been explored by other investigators including Kay et al (1,5).

    Although we have used Botulinum toxin as an adjunct to serial casting and our anecdotal evidence suggests that it is effective, it is very important to stress that this is not the primary application of this therapy. It was therefore somewhat of a surprise for us to see that Kay et al made a direct comparison between their findings and our studies in which we made a concerted effort to exclude children with fixed contractures. The purpose of the study by Corry et al(6) and Flett et al (7) were clearly to compare two methods of spasticity management for equinus gait not contracture management. In addition, the recommendations for the use of serial casting after Botulinum toxin are to change the casts at weekly intervals and to restrict casting to a period of two weeks in most children or a maximum of three weeks8.

    Given that this is the first clinical trial to be published in the Journal of Bone and Joint Surgery addressing the use of Botulinum toxin in cerebral palsy, it is very important for the readers of the journal to understand that the use of Botulinum toxin in the management of dynamic spasticity is supported by a high level of evidence (3,4). The use of Botulinum toxin in this niche application, as an adjunct to serial casting for the management of fixed contractures, is not.

    RICHARD BAKER Gait Analysis Service Manager The Hugh Williamson Gait Laboratory The Royal Children’s Hospital Flemington Road Parkville Victoria 3052 Australia E-mail: richard.baker@rch.org.au

    ROSLYN BOYD Senior Physiotherapist Department of Orthopaedics The Royal Children’s Hospital Flemington Road Parkville Victoria 3052 Australia E-mail: roslyn.boyd@rch.org.au

    PETER J FLETT Director Paediatric Rehabilitation Department of Child and Adolescent Development, Neurology and Rehabilitation Women’s and Children’s Hospital Adelaide South Australia Australia

    E-mail: flettp@wch.sa.gov.au

    H. KERR GRAHAM Professor of Orthopaedic Surgery Orthopaedic Department The Royal Children’s Hospital Flemington Road Parkville Victoria 3052 Australia

    E-mail: kerr.graham@rch.org.au

    References

    1. Kay RM, Rethlefsen SA, Fern-Buneo A, Wren TAL, Skaggs DL. Botulinum toxin as an adjunct to serial casting treatment in children with cerebral palsy. J Bone Joint Surg Am 2004; 86:2377-84

    2. Cosgrove AP, Corry IS, Graham HK. Botulinum toxin in the management of the lower limb in cerebral palsy. Dev Med Child Neurol 1994:36:386-96

    3. Baker R, Jasinski M, Maciag-Tymecka I, Michalowska-Mrozek J, Bonikowski M, Carr L, MacLean J, Lin J-P, Lynch B, Theologis T, Wendorff J, Eunson P, Cosgrove A. Botulinum toxin treatment of spasticity in diplegic cerebral palsy: a randomized, double-blind, placebo-controlled, dose-ranging study. Dev Med Child Neurol 2002;44:666-75

    4. Boyd RN, Hays RM. Current evidence for the use of botulinum toxin type A in the management of children with cerebral palsy: a systematic review. Eur J Neurol 2001;8: Supp 5: 1-20

    5. Graham HK, Aoki KR, Autti-Ramo I, Boyd RN, Delgado MR, Gaebler- Spira DJ, Gormley ME, Guiyer BM, Heinen F, Holton AF, Matthews D, Molenaers G, Motta F, Garcia Ruiz PJ, Wissel J. Recommendations for the use of botulinum toxin type A in the management of cerebral palsy. Gait and Posture 2000;11:67-79

    6. Corry IS, Cosgrove AP, Duffy CM, McNeill S, Taylor TC, Graham HK. Botulinum toxin A compared with stretching casts in the treatment of spastic equinus: a randomised prospective trial. J Pediatr Orthop 1998;18:304-11

    7. Flett PJ, Stern LM, Waddy H, Connell TM, Seeger JD, Gibson SK. Botulinum toxin A versus fixed cast stretching for dynamic tightness in cerebral palsy. J Paediatr Child Health 1999;35:71-7

    8. Boyd RN, Graham HK. Botulinum toxin A in the management of children with cerebral palsy indications and outcome. Euro J Neurol 1997; 4 (supple 2):15-22

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