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OnabotulinumtoxinA Injection as an Adjunct in the Treatment of Posterior Shoulder Subluxation in Neonatal Brachial Plexus Palsy
Marybeth Ezaki, MD1; Kanchai Malungpaishrope, MD2; Richard J. Harrison, MD3; Janith K. Mills, MPAS1; Scott N. Oishi, MD1; Mauricio Delgado, MD1; Patricia A. Bush, MS1; Richard H. Browne, PhD1
1 Texas Scottish Rite Hospital for Children, 2222 Welborn Street, Dallas, TX 75219. E-mail address for M. Ezaki: Marybeth.Ezaki@tsrh.org
2 Lerdsin Hospital, Silom Road, Bangkok 10500, Thailand
3 Womack Army Medical Center at Fort Bragg, Fayetteville, NC 28314
View Disclosures and Other Information
Disclosure: The authors did not receive any outside funding or grants in support of their research for or preparation of this work. Neither they nor a member of their immediate families received payments or other benefits or a commitment or agreement to provide such benefits from a commercial entity.

A commentary by Michael L. Pearl, MD, is available at www.jbjs.org/commentary and as supplemental material to the online version of this article.
Investigation performed at Texas Scottish Rite Hospital for Children, Dallas, Texas

Copyright © 2010 by The Journal of Bone and Joint Surgery, Inc.
J Bone Joint Surg Am, 2010 Sep 15;92(12):2171-2177. doi: 10.2106/JBJS.I.00499
A commentary by Michael L. Pearl, MD, is available here
5 Recommendations (Recommend) | 3 Comments | Saved by 3 Users Save Case

Abstract

Background: 

Botulinum toxin A is used to treat contractures in children with spasticity by temporarily interfering with neural transmission at the motor end plate. In infants with brachial plexus palsy, posterior shoulder subluxation and dislocation are the result of muscle imbalance, in which neurologic recovery is evolving, and spasticity is not a deforming force. We postulated that temporary weakening of the shoulder internal rotator muscles with botulinum toxin A would facilitate reduction of the glenohumeral joint in such infants with early posterior shoulder subluxation or dislocation.

Methods: 

Thirty-five infants with posterior subluxation or dislocation of the shoulder due to brachial plexus palsy were treated with botulinum toxin A between January 1999 and December 2006, and were followed for a minimum period of one year. Records were reviewed for the severity of the palsy, age at time of treatment, recurrence of subluxation or dislocation, and the subsequent need for further treatment to reduce the glenohumeral joint.

Results: 

The average age at the time of shoulder reduction and botulinum toxin-A injection was 5.7 months. Six patients had a second injection. Reduction of the shoulder was maintained in twenty-four (69%) of the thirty-five patients. There were no complications related to the use of botulinum toxin A.

Conclusions: 

Although there may be specific risks associated with its use, botulinum toxin-A injection into the internal rotator muscles is a useful adjunct to the treatment of early posterior subluxation or dislocation of the shoulder in infants with neonatal brachial plexus palsy, and may help to avoid the need for open surgical procedures to restore or maintain shoulder reduction.

Level of Evidence: 

Therapeutic Level IV. See Instructions to Authors for a complete description of levels of evidence.

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    References

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    These activities have been planned and implemented in accordance with the Essential Areas and policies of the Accreditation Council for Continuing Medical Education (ACCME) through the joint sponsorship of the American Academy of Orthopaedic Surgeons and The Journal of Bone and Joint Surgery, Inc. The American Academy of Orthopaedic Surgeons is accredited by the ACCME to provide continuing medical education for physicians.
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    Marybeth Ezaki, MD
    Posted on October 19, 2010
    Dr. Ezaki responds to Drs. Duijnisveld and Nelissen
    Texas Scottish Rite Hospital for Children, Dallas, Texas

    Thank you for your comment on our article. We do not feel that this is aggressive treatment, but rather a closed treatment to try to prevent the need for open treatment should the deformity worsen.

    All of the patients in our series had both passive external rotation of the involved shoulder of less than neutral, as well as documented posterior subluxation of the humeral head that did not reduce during attempted external rotation seen during real-time ultrasonographic imaging.

    The measurement of external rotation of the shoulder while a child is awake takes experience. All examiners in our clinic do the examination in the same way, with the shoulder adducted, the scapula stabilized, and the elbow flexed and supinated. None of these infant shoulders in this series could be brought even to neutral. When the child was anesthestized, the position of the shoulder was again assessed, and then with passive manipulation over the course of the procedure, the contracture could be stretched out to a target of at least sixty degrees of external rotation.

    All of these patients had failed a pre-operative stretching protocol if they had been followed for a period of time in our clinic. If posterior subluxation and tight contracture was found on the first visit to our clinic, we did not do extensive stretching prior to the botullinumtoxin and casting. My prior experience with casting alone in these children, although anecdotal, was unsatisfactory, with rapid return to the internally rotated position because the internal rotators remained strong.

    While I agree with all of your comments about the benefits of a Level I study, and I concur that this is Level IV evidence, my goal in presenting this information is to share a technique that, for me, has given an additional option for closed treatment of a difficult problem in these shoulders. Early subluxation may occur during the period of plexus recovery. If the shoulder can be maintained in a reduced position during this time, I am hopeful that function will either improve spontaneously, or that it can be more easily augmented with rebalancing tendon transfers at a later date.

    We continue to follow these patients and are aware of the multitude of confounding variables in these very complex shoulders, including the degenerative muscle changes about the shoulder in these babies. Again, the purpose of this report is to share a closed method that has helped treat the infant with a brachial plexus palsy and a shoulder that is progressing toward a fixed posterior dysplasia. I hope to be able to report back with greater detail and long-term results.

    Marybeth Ezaki, MD
    Posted on October 18, 2010
    Dr. Ezaki and colleagues respond to Dr. Soldado
    Texas Scottish Rite Hospital for Children, Dallas, Texas

    We thank Dr. Soldado for his comments. While we agree that muscle imbalance is the primary contributor to the shoulder subluxation in the very young infant, our use of botulinum toxin-A is not meant to restore muscle balance, but rather to cause a short period of chemodenervation and subsequent weakness of the shoulder internal rotator muscles. We suggest that this allows the humeral head to be repositioned more easily and with less force in the cast. We agree that the casting and stretching of the tightened internal rotator muscles is needed. Our experience in a small number of children who underwent casting alone was disappointing because of rapid recurrence of the deformity.

    Our casting protocol differs from Dr. Soldado's in positioning the shoulder in adduction to gain maximum stretch of the subscapularis muscle, and also to try to prevent the abduction contracture that contributes to the Putti sign that is so common in these children.

    While the case Dr. Soldado shared in the letter showed no evidence of remodeling after six months, we are hopeful that the use of botulinum toxin-A and short term casting will decrease the compressive and shear forces on the glenohumeral joint that are generated by casting alone, and that we will see evidence of remodeling as we follow our very young patients.

    We agree that long-term follow-up and sequential imaging studies are needed.

    Bouke J. Duijnisveld, MD, MSc
    Posted on October 13, 2010
    Does Botox in Brachial Plexus Injuries Improve Glenohumeral Range of Motion?
    Leiden University Medical Center, Leiden, The Netherlands

    To the Editor:

    In their article, "OnabotulinumtoxinA Injection as an Adjunct in the Treatment of Posterior Shoulder Subluxation in Neonatal Brachial Plexus Palsy" (2010;92:2171-7), Dr. Ezaki and coauthors have addressed an interesting treatment possibility for patients with neonatal brachial plexus palsy (NBPP) who develop internal rotation contractures with prevalence up to 39% (1, 2). They injected OnabotulinumtoxinA as an adjunct to closed manipulative shoulder reduction and cast immobilization in NBPP patients in whom a passive external rotation in adduction of at least 60º could be achieved. Although there is little consensus regarding the indication for treating internal rotation contractures, the indication performed by Ezaki and coauthors seems very aggressive because treatment of internal rotation contractures is usually performed when the passive external rotation in adduction is below 30º (3-6).

    Although MRI was not performed, ultrasonography was used to evaluate glenohumeral deformity. However, Ezaki et al. give no description on the glenoid version, the percentage of posterior displacement of the humeral head, and the extent of muscle atrophy and fatty degeneration, all of which are major confounding factors on outcome and also interpretation of the reported results (7-9). Thus the reader remains puzzled on which (pheno)types of patients were actually treated.

    Botulinum toxin injection in the internal rotators could result in muscle relaxation thereby improving the passive external rotation. However, the passive external rotation range of motion in the treated patients was not reported by the authors, neither before, nor after treatment. Instead, the authors describe whether or not initial reduction was maintained or additional treatment was performed including repeated botulinum injection, open reduction, tendon transfer or external rotation osteotomy. Even more, no information is present on which outcome parameter was used to determine which of these treatments had to be used.

    Presenting case series is valuable to orthopaedic literature, however prerequisites are: 1. a well defined study population, 2. the exact indications for treatment and 3. the formulation of well defined and validated outcome parameters. If these prerequisites are not present, interpretation of results will be cumbersome. An option would be, to analyze the results with a regression model to be able determine the influence of age, gender, type of injury, the extend of glenohumeral deformity, muscle atrophy, fatty degeneration, and pre-treatment clinical values on the outcome parameters.

    The authors did not receive any outside funding or grants in support of their research for or preparation of this work. Neither they nor a member of their immediate families received payments or other benefits or a commitment or agreement to provide such benefits from a commercial entity.

    References

    1. Hoeksma AF, Ter Steeg AM, Dijkstra P, Nelissen RG, Beelen A, de Jong BA. Shoulder contracture and osseous deformity in obstetrical brachial plexus injuries. J Bone Joint Surg Am. 2003;85:316-22.

    2. Pondaag W, de Boer R, van Wijlen-Hempel MS, Hofstede-Buitenhuis SM, Malessy MJ. External rotation as a result of suprascapular nerve neurotization in obstetric brachial plexus lesions. Neurosurgery. 2005;57:530-7.

    3. van der Sluijs JA, van Ouwerkerk WJ, de Gast A, Nollet F, Winters H, Wuisman PI. Treatment of internal rotation contracture of the shoulder in obstetric brachial plexus lesions by subscapular tendon lengthening and open reduction: early results and complications. J Pediatr Orthop B. 2004;13:218-24.

    4. Pearl ML, Edgerton BW, Kazimiroff PA, Burchette RJ, Wong K. Arthroscopic release and latissimus dorsi transfer for shoulder internal rotation contractures and glenohumeral deformity secondary to brachial plexus birth palsy. J Bone Joint Surg Am. 2006;88:564-74.

    5. Waters PM, Bae DS. The early effects of tendon transfers and open capsulorrhaphy on glenohumeral deformity in brachial plexus birth palsy. J Bone Joint Surg Am. 2008;90:2171-9.

    6. Kozin SH, Boardman MJ, Chafetz RS, Williams GR, Hanlon A. Arthroscopic treatment of internal rotation contracture and glenohumeral dysplasia in children with brachial plexus birth palsy. J Shoulder Elbow Surg. 2010;19:102-10.

    7. Waters PM, Monica JT, Earp BE, Zurakowski D, Bae DS. Correlation of radiographic muscle cross-sectional area with glenohumeral deformity in children with brachial plexus birth palsy. J Bone Joint Surg Am. 2009;91:2367-75.

    8. Hogendoorn S, van Overvest KL, Watt I, Duijsens AH, Nelissen RG. Structural changes in muscle and glenohumeral joint deformity in neonatal brachial plexus palsy. J Bone Joint Surg Am. 2010;92:935-42.

    9. van Gelein Vitringa VM, Jaspers R, Mullender M, Ouwerkerk WJ, van der Sluijs JA. Early effects of muscle atrophy on shoulder joint development in infants with unilateral birth brachial plexus injury. Dev Med Child Neurol. 2010 Sep 15 [Epub ahead of print].

    Francisco Soldado
    Posted on October 05, 2010
    Nonoperative Treatment of Shoulder Internal Rotation Contracture and Shoulder Dysplasia after NBPP
    Hospital Universitari Vall d´Hebron, Passeig Vall D´Hebron, Barcelona, Spain

    To the Editor:

    I read with interest the paper by Ezaki et al. entitled, "OnabotulinumtoxinA Injection as an Adjunct in the Treatment of Posterior Shoulder Subluxation in Neonatal Brachial Plexus Palsy" (2010;92:2171-7). It describes nonsurgical treatment of glenohumeral dysplasia following NBPP which combines injection of botulinum toxin A into internal rotator muscles of the shoulder, followed by casting. Shoulder problems to address in these patients are muscle imbalance, shoulder internal rotation contracture (SIRC, muscle and soft tissue contracture) and joint deformity. In this paper, muscle imbalance is addressed with the use of botulinum toxin A, while soft tissue and joint abnormalities are addressed with manipulation and casting. It seems that the authors consider that botulinum toxin A injection and thus, muscle rebalancing, is the key factor in this treatment. In our opinion, casting is the main factor contributing to the improvement in SIRC although shoulder rebalancing might be necessary to avoid recurrence.

    Unpublished data from our research group shows that isolated Botulinium toxin injection into shoulder internal rotator muscles does not prevent SIRC in a rat model. Currently, in line with Dr. Ezaki, the policy of our unit is to nonoperatively treat children with Waters type I to III joint deformities (Figure 1A). Our protocol is similar: shoulder casting is done with 90 degrees of shoulder abduction to allow external rotation. This treatment has been successful in soft tissue stretching with excellent passive shoulder external rotation outcomes (Fig 1B). In Ezaki's paper, children have been imaged with ultrasound although data has not been discussed. MRI imaging of our patients showed no postoperative joint remodeling (Figure 1C) despite the excellent clinical results.


    Fig 1.
    A: MRI showing left glenohumeral dysplasia (waters type III) in a 7-month-old girl.
    B: Shoulder passive external rotation 6 months after nonoperative treatment with casting.
    C: Postoperative MRI showing no joint remodeling despite the excellent shoulder motion.

    Considering Ezaki's paper and our preliminary results it appears that soft tissue contractures are successfully treated nonoperatively while the effect on the glenohumeral abnormalities needs further study. The main advantage of nonoperative treatment is sparing the subescapular muscle and thus avoiding loss of midline function which is the main complication associated with soft tissue release in surgical treatment. It is our opinion the reported non-operative management is the future direction of treatment of shoulder problems after NBPP but comparative studies are necessary.

    The author did not receive any outside funding or grants in support of his research for or preparation of this work. Neither he nor a member of his immediate family received payments or other benefits or a commitment or agreement to provide such benefits from a commercial entity.

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