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Review Article Shoulder Problems in Children With Brachial Plexus Birth Palsy: Evaluation and Management  

Review Article

Shoulder Problems in Children With Brachial Plexus Birth Palsy:

Evaluation and Management

 

Abstract

Michael L. Pearl, MD

Traction injury to the brachial plexus sustained during the birth process that results in impaired neuromuscular function of the upper extremity continues to occur despite advances in modern obstetric care. The most common pattern of injury usually results in motor weakness of shoulder external rotation, leading to internal rotation contractures and subsequent deformity of the skeletally immature glenohumeral joint. Understanding of these deformities and effective surgical intervention have advanced greatly over the past decade. Restoration of balance between internal and external rotation forces around the shoulder has great potential for remodeling of the glenohumeral joint in the young child. Arthroscopic-directed release of the contracture, with select use of latissimus dorsi transfer to provide external rotation power, has proved to be effective for many children with these contractures.

Dr. Pearl is Shoulder and Elbow Surgeon, Kaiser Permanente, Los Angeles Medical Group, Los Angeles, CA, and Assistant Clinical Professor, Department of Orthopaedic Surgery, University of Southern California, Los Angeles.

Supported in part by research grants from Kaiser Permanente Southern California and American Shoulder and Elbow Surgeons. Dr. Pearl or a member of his immediate family has received royalties from Zimmer.

Reprint requests: Dr. Pearl, Kaiser Permanente, Los Angeles Medical Center, 4760 Sunset Boulevard, Los Angeles, CA 90027.

J Am Acad Orthop Surg 2009;17:

242-254

Copyright 2009 by the American Academy of Orthopaedic Surgeons.

E pidemiologic studies show that

brachial plexus palsy occurs in 1

per 1,000 1 to 4.6 per 1,000 births. 2 Conventional thinking that 80% to 90% of children recover must be tempered by the understanding that there are differences in what is meant by “recovery.” A completely normal arm free of any sequelae is probably less common than often realized. In one study, persistent restriction in passive range of shoulder motion was observed in 54% of children who did not demonstrate complete neurologic recovery by 3 weeks, de- spite many of them going on to a “good functional recovery.” 3 The number of such children who will have contractures requiring surgical treatment also varies from center to center, but in one cohort 20 of 74 children referred in early infancy from an established registry were in this category. 4 Likely twice this many

have some degree of abnormal gleno- humeral anatomy on magnetic reso- nance imaging (MRI). 5 Despite advances in obstetric care, the incidence of brachial plexus birth palsy is increasing, speculated to be the result of increasing birth weights. 1,2 The neurologic injury in newborns may involve the entire plexus but most often involves the upper trunk, with varying degrees of severity. Injuries may be transient, with nearly complete neurologic re- covery (ie, antigravity biceps and del- toid function usually observed by age 2 months), or they may result in a permanently flail arm (usually in as- sociation with a complete plexus le- sion and avulsion of the cervical spi- nal nerve roots). For these two extremes, there is little controversy that the early-recovery group does not need surgical intervention and that the latter group will fare poorly

242

Journal of the American Academy of Orthopaedic Surgeons

Michael L. Pearl, MD

Figure 1

Michael L. Pearl, MD Figure 1 Internal rotation contracture (–20° of external rotation) in an 11-month-old

Internal rotation contracture (–20° of external rotation) in an 11-month-old child. A, Clinical photograph demonstrating the degree of contracture with the arm at the side. B, Restriction of passive external rotation was confirmed under anesthesia at the time of surgery. (Reproduced with permission from 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;86:564-574.)

without it. When the nerve roots are avulsed, microsurgical options are limited to nerve transfers from unin- volved areas, such as the intercostal and spinal accessory nerves. The in- dications for neurosurgery on the plexus for injuries that are intermedi- ate to these two extremes remain controversial. Depending on the medical center, recommendations typically involve plexus exploration and grafting from 3 to 9 months fol- lowing birth. With or without nerve repair or transfer, internal rotation contrac- ture of the shoulder is the most com- mon problem requiring treatment in children with incomplete recovery 3-10 (Figure 1). This contracture results from an imbalance between the strength of the relatively unaffected internal rotators and the paralytic external rotators (primarily the in- fraspinatus). Untreated, it usually leads to progressive glenohumeral deformity characterized by posterior displacement of the humeral head on an increasingly dysplastic and de- formed glenoid. Treatment protocols vary widely, making it challenging to

compare the literature and establish definitive indications for both early microsurgery and late secondary or- thopaedic procedures.

Clinical Evaluation

Assessing the motor function of the infant and young child is difficult. Electromyography has not been reli- able for most clinical investigations in this patient population. As a re- sult, the physical examination, with all its inherent limitations, is the mainstay of analysis. Fear of the ex- amination, inability to comprehend directions, and lack of coordination from undeveloped motor function in the very young patient all challenge the examiner trying to assess active range of motion (ROM) and strength. The examiner must engage the child in activities and then ob- serve motor function. Young chil- dren and especially infants can often be prompted to reach for objects overhead (eg, lollipop, shiny keys), providing an indication of active ele- vation (Figure 2). The effectiveness

of similar maneuvers in other direc- tions is less predictable. By necessity, therefore, the examination only ap- proximates a complete motor exami- nation, depending on the child’s age and ability to cooperate. Alternatives to the muscle grading systems that are commonly used for adults, such as the British Medical Council 5-point scale, are necessary. The Hospital for Sick Children in To- ronto, Canada, introduced an exami- nation scale, the Active Movement Scale (AMS), to address the limitations in the British Medical Council system, specif- ically that it is incongruous to grade the strength of a weak muscle against grav- ity if it is not clear that the muscle can function with gravity eliminated. 11 Thus, the first four grades of strength in the AMS are devoted to achieving full ROM with gravity eliminated (Table 1). This system is particularly useful for infants under evaluation for potential neurologic surgery, because they are especially weak, rarely have contractures, and have motor grades that must be based on observation alone because they cannot comply with commands.

Shoulder Problems in Children With Brachial Plexus Birth Palsy

Figure 2

in Children With Brachial Plexus Birth Palsy Figure 2 Same child as in Figure 1 at

Same child as in Figure 1 at the same presurgical consultation demonstrating 120° of active elevation in reaching for her mother’s wristwatch. Note the effect of internal rotation contracture on the ability to reach.

Table 1

 
 

The Active Movement Scale 11

 

Muscle

 

Observation

Grade

Gravity Eliminated* No contraction Contraction, no motion Motion ½ range Motion >½ range Full motion Against Gravity Motion ½ range Motion >½ range Full motion

0

1

2

3

4

5

6

7

* Active motion through a full range with gravity eliminated must be demonstrated before advancing to a score that denotes motion through the range in the presence of gravity. Reproduced with permission from Curtis C, Stephens D, Clarke HM, Andrews D:

The active movement scale: An evaluative tool for infants with obstetrical brachial plexus palsy. J Hand Surg [Am] 2002;27:

470-478.

Conversely, this system is a limited tool for measuring strength in the context of marked limitations in pas- sive ROM. When contractures are present, they are not assessed in this system, and grades of strength may not sufficiently represent motor func-

tion. Furthermore, it is unclear that the ability to achieve full ROM with gravity eliminated is requisite for generating appreciable quantities of force in specific joint positions in all circumstances. For example, a mus- cle with a low score (ie, incomplete ROM with gravity eliminated) may be stronger in certain midrange posi- tions than muscles graded higher simply because they can achieve a full range. In an ongoing effort to standardize evaluations, Bae et al 12 performed a reliability study of three major scor- ing systems: Mallet classification, Toronto Test Score, and Hospital for Sick Children AMS. Two trained ex-

aminers twice evaluated 80 children with brachial plexus birth palsy. Re- sults were evaluated for intraob- server and interobserver reliability as well as test-retest reliability. Positive intraobserver and interobserver cor- relations were noted, and test-retest reliability was excellent. The authors concluded that the modified Mallet classification, Toronto Test Score, and AMS are reliable instrument s for assessing upper extremity func- tion in patients with brachial plexus birth palsy. It is not clear, however, how the findings of two examiners from the same institution translate to the findings of multiple examiners from different medical centers over long time periods. More important, the effectiveness and extent to which these scores reflect muscle function and recovery of the limb is not ad- dressed by measures of reliability. No perfect scale or score exists that is applicable to children of all ages un- der consideration for neurosurgical or orthopaedic intervention. Accordingly, investigators and clinicians treating af- fected children are limited to neurologic examination tools in combination with measures of passive and active ROM of all upper extremity joints. With regard to active ROM, the assessment is by ne- cessity an approximation for young children who cannot reliably follow commands.

Imaging Studies

Ultrasonography, 13,14 arthrography, 6 and MRI 5,7,15-19 have all been used to study the morphology of the gleno- humeral joint in children with bra- chial plexus palsy. The exact role and relative advantages of each of these modalities is debatable, al- though most centers now favor MRI. Ultrasonography is real-time and noninvasive, but the level of detail is lower than that of other modalities. Arthrography offers more detail than

Michael L. Pearl, MD

ultrasonography but is invasive; however, it can be done at the time of surgical intervention, under the same anesthesia (Figure 3). MRI of- fers the most detail and potential for standardization but are costly and often require general anesthesia in young children. Regardless of the imaging modality

used, it is difficult to justify any clini- cal study that does not assess the sta- tus of glenohumeral development. It

is now evident that the internal rota-

tion contracture of the diseased shoulder commonly leads to gleno- humeral deformity. Posteriorly di- rected forces displace the humeral head in the same direction. In grow-

ing children, skeletal changes ensue.

A “false” articulation often forms on

the posterior aspect of the glenoid that becomes progressively retro- verted, leading to a potential array of deformities that have been described

as flat, biconcave, and convex (ie, pseudoglenoid). Various classifica- tion systems have been pro- posed. 3,7,14,20,21 They have in common the fact that with increasing defor- mity, there is increasing posterior displacement of the glenohumeral joint from its normally centered and concentric position, and the normal concave shape of the glenoid be- comes increasingly convex. In ad- vanced deformities, the humeral head articulates with the posterior aspect of the convex glenoid and be- comes increasingly misshapen and retroverted itself 18 (Figure 4).

Surgical Management

A detailed review of the indications

and techniques for microscopic sur- gical intervention for these injuries is beyond the scope of this discussion, but it is important to recognize that microsurgical options are applicable only to the most severe injuries; ap- ply only to patients in the first year

Figure 3

ap- ply only to patients in the first year Figure 3 A, Axillary arthrogram of a

A, Axillary arthrogram of a concentric glenoid fossa in a 10-month-old child with a relatively normal glenohumeral joint. B, Line tracing of panel A outlining the glenoid (GL). Nearly the entire glenoid is unossified cartilage. C, T2-weighted gradient-recalled echo MRI scan of the concentric glenoid in the same patient. Hyaline cartilage appears white (broken arrow) and labral tissue black (solid arrow) on this sequence. The humeral head is round. D, Line tracing of panel C showing the glenoid, scapular center-line, and approximate humeral head center of rotation (white dot). HH = humeral head. (Reproduced with permission from Pearl ML, Edgerton BW, Kon DS, et al:

Comparison of arthroscopic findings with magnetic resonance imaging and arthrography in children with glenohumeral deformities secondary to brachial plexus birth palsy. J Bone Joint Surg Am 2003;85:890-898.)

of life, after which these options are thought not to be effective; and never result in complete neurologic improvement, always leaving resid- ual impairment. Thus, secondary or- thopaedic procedures are a potential consideration for all children with incomplete recovery, regardless whether they receive microsurgical intervention. For avulsion injuries, the prognosis for natural recovery is so poor that early (<3 months) mi-

crosurgical intervention is recom- mended. The options are limited to various forms of nerve transfer be- cause grafting is not applicable in the absence of a healthy proximal nerve root. 22,23 For intraplexus ruptures, return of antigravity biceps strength remains the primary factor in determining the need for brachial plexus exploration and nerve reconstruction. Contro- versy persists regarding the need for

Shoulder Problems in Children With Brachial Plexus Birth Palsy

Figure 4

in Children With Brachial Plexus Birth Palsy Figure 4 T2-weighted gradient-recalled echo MRI scan of the

T2-weighted gradient-recalled echo MRI scan of the child in Figures 1 and 3, demonstrating pseudoglenoid configuration. The glenoid contour and scapular center line are enhanced with line tracing. (Reproduced with permission from 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;86:564-574.)

and timing of microsurgical interven- tion, with recommendations ranging from age 3 to 9 months. 24,25 There is no doubt that with increasing delay in return of biceps function less spontaneous recovery will occur. Wa- ters 26 has shown that children who do not develop this ability by age 5 months do not do as well under con- servative management as those who receive microsurgical nerve interven- tion (grafting or neurotization). Ef- forts continue to refine surgical indications, with some centers rec- ommending a more comprehensive evaluation that includes muscles other than the biceps to predict re- covery. 27 Discerning which neurologic le- sions will improve equally well with late secondary orthopaedic interven- tion and no early microsurgery, as opposed to early microsurgery fol- lowed later by secondary procedures, is difficult. No study to date can de-

finitively claim that microsurgery in combination with secondary ortho- paedic procedures results in better outcomes than secondary ortho- paedic procedures alone, although much of current practice is predi- cated on this belief. Smith et al 24 pre- sented long-term follow-up of 22 children who had no brachial plexus surgery and for whom biceps recov- ery was delayed until age 3 to 6 months. These children demon- strated comparable function to that reported for children who had mi- crosurgical repair or grafting to the brachial plexus. Adding to the confu- sion is the wide array of secondary procedures that have been associated in published accounts with clinical success.

Secondary Interventions

Internal Rotation Contracture Release

Internal rotation contractures and the resultant glenohumeral deformity have been documented in children as young as age 5 months. 5 Treatment

of the contracture can be undertaken

at any time because it will not re- cover spontaneously, although a pe-

riod of formal physical therapy may

with or without latissimus trans- fer. 10,33,34 These approaches variably combine some form of contracture release with or without a muscle transfer to augment external rotation power. For children with extensive glenohumeral deformity, the prevail- ing recommendation is an external rotational osteotomy of the humerus, rotating the arm into a more func- tional position of external rotation. 35 How can these seemingly disparate approaches be reconciled? One way is to recognize that the internal rota- tion contracture results from the loss of the normal balance between exter- nal rotation and internal rotation, principally because of infraspinatus weakness. Different types of proce- dures that restore muscle balance and stability by reducing internal ro- tation strength and/or augmenting external rotation strength may dem- onstrate effectiveness. Similarly, a bony procedure that increases exter- nal rotation will improve function. It is also likely that commonly used clinical scoring systems, most nota- bly the Mallet, are too crude to dis- tinguish the outcomes among these approaches.

Formal Anterior Approach

be

required for the parents to accept

The first surgical releases described

this fact. Four soft-tissue procedures and one bony procedure constitute the majority of surgical experience over the past two decades: (1) ante- rior capsular release, Z-plasty lengthening of the subscapularis ten- don with or without transfer of mus- cles for external rotation; 28 (2) pecto-

in the early 20th century by Fair- bank 36 and later modified by Sever 37 used a traditional anterior deltopec- toral approach. To reduce the recur- rence rate of the internal rotation contracture and add external rota- tion power, most centers added some version of the L’Episcopo transfer of

ralis major release with transfer of the latissimus and teres major as ad- vocated by Hoffer and colleagues; 29,30

the latissimus dorsi and teres major tendons. Modified versions of these approaches are still the preferred

(3) subscapularis slide with and without a latissimus transfer as origi- nally described by Carlioz and Brahi-

method of treatment. Kirkos et al 28 reported a 30-year mean follow-up on 10 children who underwent a re-

mi

31 and recommended by Gilbert et

lease of the upper half of the pecto-

al;

32 and (4) arthroscopic release of

ralis major and the entire subscapu-

the internal rotation contracture

laris and anterior capsule, along with

Michael L. Pearl, MD

Figure 5
Figure 5

Schematic illustration of anterior release, pectoralis transfer, and latissimus dorsi transfer as described by Zancolli and Zancolli. 39 A, Incision. B, Detachment of the subscapularis, pectoralis major, and latissimus dorsi. C, Rerouting of the latissimus tendon and reinsertion of the pectoralis into the subscapularis tendon. (Reproduced with permission from Zancolli EA, Zancolli ER III: Reconstructive surgery in brachial plexus sequelae, in Gupta A, Kay SPJ, Scheker LR [eds]: The Growing Hand. London, England: Mosby, 2000, pp 805-823.)

rerouting (transfer) of the latissimus dorsi and teres major tendons to the pectoralis major stump. Gains in ex- ternal rotation deteriorated over time, and five patients had signifi- cant degenerative changes of the gle- nohumeral joint. Among the concerns regarding the anterior approach were that it re- sulted in poor cosmesis and poten- tially led to anterior dislocation of the glenohumeral joint and/or func- tionally significant external rotation contractures. Several authors have modified this approach with an aim to address some of these issues. Zan- colli and Zancolli 38,39 have long ad- vocated an anterior approach. Their clinical reports extensively describe indications and surgical technique but provide limited data analysis. To minimize cosmetic concerns, these surgeons use an incision in the skin lines from the coracoid to the axilla (Figure 5, A). The latissimus transfer is done by performing a step cut of the tendon insertion, rerouting the released tendon posteriorly, and se- curing it to the remaining latissimus

tendon anteriorly (Figure 5, B and C). The subscapularis is released, and sometimes the pectoralis and teres major are released as well. These authors warn against releasing the anterior capsule. Satisfactory re- sults have been reported with this technique, with an increase of 50° average abduction and 45° average external rotation. 39 In the presence of an incongruent joint, which typically is seen in children older than age 3 or 4 years, Zancolli and Zancolli 39 recommend a rotational osteotomy of the humerus and warn against any “attempt to reduce a posterior sub- luxation surgically when the joint’s surfaces are already deformed.” Recent reports by other authors who use an anterior approach and who warn against releasing the ante- rior capsule have acknowledged that despite their preoperative intentions, at surgery it was not possible to re- store external rotation and reduce the glenohumeral joint without re- leasing the capsule. 40,41 In the series of van der Sluijs et al, 40 15 of 19 pa- tients with internal rotation contrac-

tures required release of the anterior capsule to achieve external rotation and reduce the glenohumeral joint.

In addition to the presence of con-

tracted anterior soft tissues, these au-

thors postulate that in many cases excessive retroversion of the hu- merus obligates an external rotation contracture once the glenohumeral joint is reduced. In such cases as

these, Birch recommends an internal rotational osteotomy as part of the same surgery (70 of the 183 cases in

his series 41 ).

Hoffer Modification of the L’Episcopo Procedure

A common surgical approach em-

ployed by many medical centers, originally devised by Hoffer, uses a

cosmetic incision in the axillary crease to release the pectoralis major and transfer the combined tendons

of the latissimus dorsi and teres ma-

jor muscles to the posterior rotator cuff 29,30 (Figure 6). Hoffer et al 29 first reported on this technique in 11 pa- tients, who achieved an average gain

Shoulder Problems in Children With Brachial Plexus Birth Palsy

Figure 6
Figure 6
Illustrations of partial pectoralis major release (A) and transfer of combined latissimus dorsi and teres
Illustrations of partial pectoralis major release (A) and transfer of combined latissimus dorsi and teres major tendons to
the posterior rotator cuff (B) as described by Hoffer. (Reproduced with permission from Hoffer MM, Wickenden R,
Roper B: Brachial plexus birth palsies: Results of tendon transfers to the rotator cuff. J Bone Joint Surg Am
1978;60:691-695.)
Figure 7
weakness in the absence of a severe,
firmly fixed internal rotation con-
tracture and glenohumeral defor-
mity.
Release of the
Subscapularis Origin

Illustrations demonstrating subscapularis release (A) and transfer of the isolated latissimus dorsi tendon (B) as described by Gilbert et al. 32 (Reproduced with permission from Gilbert A: Obstetric brachial plexus palsy, in Tubiana R [ed]: The Hand. Philadelphia, PA: WB Saunders, 1993, vol 4, pp 592, 594.

of 64° of abduction and 45° of exter- nal rotation at a minimum 2-year follow-up. A subsequent report 20 years later presented similar success in another eight children. 30 A conten- tion of these authors and others ad- vocating this procedure is that one should avoid releasing the anterior capsule and even the subscapularis for fear of anterior dislocation or

creation of an external rotation con- tracture. However, recent reports from different centers that used this surgical approach have shown that despite improvements in shoulder function, this procedure did not im- prove the glenohumeral deformity present in many of the patients. 9,42 It is likely that this surgical approach is most effective for external rotation

Gilbert et al 32 have long advocated an approach originated by Carlioz and Brahimi 31 in which the subscap- ularis origin is released and the mus- cle reflected distally (Figure 7, A). In a report on 65 patients followed more than 5 years after this pro- cedure, Gilbert et al 32 noted an aver- age gain in external rotation of 70° when children were operated on at younger than age 2 years provided that the joint was congruent and the humeral head was round. Children operated on after age 4 years did not show similar improvement. For these children, and those that failed an ear- lier release, these authors recom- mended transfer of the latissimus dorsi tendon to the posterolateral ro- tator cuff as well (Figure 7, B). Among the stated principles of this surgical approach is that it avoids re- leasing the anterior capsule and only

Michael L. Pearl, MD

partially weakens the subscapularis, preserving some of its function. In so doing, this technique clearly tips the balance between internal and exter- nal rotation strength in favor of the weak external rotators. In one series, however, there was inadequate re- lease of the capsular contracture in 5 of 25 children, requiring an anterior approach at the same surgical set- ting. 6 A recent long-term follow-up analysis noted that many of the func- tional gains observed in the early postoperative period deteriorated with time, resulting in significant functional limitations in adulthood, 8 a seemingly consistent observation of all reported methods in the few very long-term studies available. 28

Arthroscopic Subscapularis Tenotomy and Capsular Release

In 2003, Pearl 33 first reported on ar- throscopic release with promising early results (Figure 8). A subsequent report on 33 children confirmed the utility of this procedure. 10 The surgi- cal protocol used in these studies fol- lowed in part the recommendations of Gilbert et al 32 in that young children (3 years) receive arthroscopic release only and older children receive a simul- taneous latissimus dorsi tendon trans- fer. Nineteen children in this series re- ceived a release only; 14 had a release combined with a latissimus dorsi trans- fer. At a minimum 2-year follow-up, the mean passive external rotation in- creased by 67° (P < 0.005) in the 15 children with a successful arthroscopic release (Figure 9) and by 81° (P < 0.005) in those treated with a primary latissimus dorsi transfer. Four of the 19 younger children failed to improve suf- ficiently and were successfully treated with subsequent latissimus dorsi trans- fer performed for either failure to main- tain sufficient external rotation strength postrelease or for recurrence of the con- tracture. All 14 older children (age >3

Figure 8

con- tracture. All 14 older children (age >3 Figure 8 Arthroscopic view of a right shoulder

Arthroscopic view of a right shoulder from a posterior portal demonstrating the level of subscapularis tenotomy with an electrocautery device (arrow). BT = biceps tendon, HH = humeral head. (Reproduced with permission from Pearl ML: Arthroscopic release of shoulder contracture secondary to birth palsy: An early report on findings and surgical technique. Arthroscopy

2003;19:577-582.)

years) treated with simultaneous release and latissimus transfer maintained gains in external rotation. At follow-up, the four younger children who required de- layed latissimus transfer had a mean passive external rotation of 78° and showed no ill effects of delayed trans- fer. This differential approach to man- aging children younger than age 3 years with an isolated arthroscopic release re- mains the author’s preferred method of managing these contractures. In the report by Pearl, 33 arthro- scopic release not only improved ex- ternal rotation but also demon- strated remarkable remodeling of glenohumeral deformity when pres- ent before surgery. Follow-up MRI was available for 15 of 18 children with advanced pseudoglenoid defor- mities at the time of release or trans- fer. All but the three most severe deformities (12 of 15) showed nor- malization of the glenohumeral joint on follow-up MRI scan, as evidenced by increased sphericity of the hu- meral head, restoration of the gle- noid concavity, and centralization of

Figure 9

of the gle- noid concavity, and centralization of Figure 9 Active external rotation 2 years after

Active external rotation 2 years after arthroscopic release in the same child as in Figures 1, 3, and 4. (Reproduced with permission from 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;88:564-574.)

the humeral head on the glenoid fossa (Figure 10). Normalization of glenohumeral anatomy may result from a range of surgical methods of contracture re- lease as long as external rotation is restored and preserved at follow-up. Pedowitz et al 34 showed some im- provement of glenohumeral align- ment on MRI immediately after arthroscopic reduction with the shoulder held in external rotation by

a spica cast. At our center, we have

observed remodeling of deformity by

a variety of surgical methods, open

anterior release, subscapularis slide Figure 11), and now with our cur- rent protocol of arthroscopic release (Figure 10). Hui and Torode 20 also reported improved glenoid retrover- sion for 23 children at an average follow-up of 43 months after open anterior release. Most recently, Wa- ters and Bae 43 reported on 23 chil- dren with 83% showing remodeling of deformity after open soft-tissue procedures that included open gleno- humeral joint reduction (ie, capsulor- rhaphy) . This was in contrast to their

Shoulder Problems in Children With Brachial Plexus Birth Palsy

Figure 10

in Children With Brachial Plexus Birth Palsy Figure 10 A, Preoperative T2-weighted gradient-recalled echo MRI scan

A, Preoperative T2-weighted gradient-recalled echo MRI scan of a 4.7-year- old patient revealing mild pseudoglenoid. B, T2-weighted gradient-recalled echo MRI scan of the same patient 2 years after arthroscopic release and latissimus dorsi transfer, demonstrating remodeling to a concentric joint with a round humeral head well centered on the glenohumeral joint. (Reproduced with permission from 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;86:564-574.)

earlier report on patients treated sim- ilarly who did not receive a capsular release and showed no improvement

in glenohumeral deformity. 9

Procedures that tip the balance of the shoulder rotators toward exter- nal rotation, either by sacrificing in- ternal rotator strength or augment- ing external rotator power, will inevitably weaken internal rotation or diminish internal rotation range.

A loss of internal rotation was ob-

served with the arthroscopic ap- proach, as well. 10 As reported,

The ability to reach up the back was not measured preop- eratively, but it was clearly re- stricted at the time of follow- up, with the children only able to reach between the sacrum and L5 on the average. 10

Earlier reports in the literature de- scribing results from other tech- niques have not addressed the result- ant loss of internal rotation

sufficiently to allow for meaningful comparison. The recent report by

Waters and Bae, 43 describing internal rotation using the Mallet scale hand

to spine score, described a mean im-

provement from a pre-operative 1 to

a postoperative 2 (the ability to

reach S1 postoperatively). From these two reports, it appears that both surgical approaches (arthro- scopic and open capsulorraphy) re- sult in the same amount of internal rotation. 10,43 It is not clear why this was observed as a decrease in func- tion in the arthroscopic study and an increase following capsulorraphy, but the difficulties in examining mo- tor function in young children may play a role. One may conclude from the literature on the various tech- niques of contracture release and tendon transfer, as just discussed, that procedures that do not release the anterior capsule will less consis- tently restore external rotation and improve glenohumeral deformity

Figure 11

rotation and improve glenohumeral deformity Figure 11 T2-weighted gradient-recalled echo MRI scan taken 3.5 years

T2-weighted gradient-recalled echo MRI scan taken 3.5 years postoperatively of a child who had a pseudoglenoid prior to extra- articular subscapularis slide at age 2 years. The scan shows a round humeral head well centered on a concave glenoid.

than those that target the joint capsule. Accordingly, historic con - cerns regarding release of the sub- scapularis and the anterior capsule must be tempered by the evidence that not doing so will not release all contractures and will leave behind many posteriorly displaced, de- formed glenohumeral joints without the possibility of remodeling. The ar- throscopic release allows for select attention to the subscapularis and anterior capsule in contrast to the earlier open procedures that required release of multiple superficial struc- tures as well.

Rotational Osteotomy

For older children or those with ad- vanced glenohumeral deformity, the prevailing recommendation has been to avoid soft-tissue procedures at the joint in favor of rotational osteot- omy of the humerus. The utility of this procedure was confirmed in a re- cent report by Waters and Bae. 35 These authors reported on 27 pa-

Michael L. Pearl, MD

tients who underwent rotational os- teotomy with plate fixation and achieved an average improvement of 64° in external rotation and im- proved 5 points on a modified Mal- let scale. No clear guidelines exist, however, as to what age or degree of deformity should dictate this form of surgical treatment. As it is now evi- dent that many children younger than age 6 years are capable of sub- stantial remodeling, the decision to perform an osteotomy is all the more complex. Humeral osteotomy does improve the functional position of the hand, but it leaves the shoulder in a posteriorly dislocated position and eliminates all hope of remodel- ing. At present, the surgeon must make a judgment call regarding the growth potential of the child, the se- verity of the deformity, and her or his own surgical abilities in deciding to perform a soft-tissue procedure that recenters the humeral head on the glenoid fossa (Figure 12).

Other Problems

Poor Active Elevation

Weakness of elevation is functionally limiting when active elevation is <90°. Affected children have much better passive than active ROM. Some reports of anterior capsular re- lease and muscle transfer to provide or increase external rotation claim significant improvement in abduc- tion with these procedures. 29,30,39,44 However, we have observed only modest improvements in active ele- vation with any of the aforemen- tioned procedures and believe that much of the improvement noted in other studies reflects the difficulties in measuring motion in young chil- dren as well as the apparent in- creased reach resulting from proce- dures that improve external rotation. Among the specific procedures desig- nated to improve active elevation are

Figure 12

desig- nated to improve active elevation are Figure 12 A, Preoperative T2-weighted gradient-recalled echo MRI scan

A, Preoperative T2-weighted gradient-recalled echo MRI scan of an 11-year- old boy with a functionally disabling internal rotation contracture. His age and extensive glenohumeral deformity made significant skeletal remodeling and improvement with soft-tissue procedures unlikely. B, Postoperative anteroposterior radiograph demonstrating that rotating the humerus 60° into external rotation can dramatically improve appearance and function.

upper trapezius transfer, in which that muscle is detached from the acromion and inserted distally on the humerus, and bipolar transfer of the latissimus dorsi and pectoralis major to replace the deltoid. No recent clin- ical series have demonstrated success with any of these procedures. Currently, shoulder elevation in brachial plexus birth palsy is largely an unsolved problem, and aggressive surgical intervention is likely to be disappointing. However, for a child with limited elevation and a signifi- cant internal rotation contracture, improving the range of external rota- tion can markedly improve the child’s ability to reach, by allowing the hand to be placed in a higher po- sition (Figure 13).

Scapular Dyskinesia

Asymmetric and apparently abnor- mal movement of the scapula is a frequent cause of concern to parents. It is most often associated with an internal rotation contracture but will

accompany stiffness or limited excur- sion of the glenohumeral joint in any direction. Zancolli and colleagues 38,39 refer to the “scapular elevation sign” and have noted that it accompanies internal, external, and abduction contractures. The sign, historically called the “Putti sign,” is seen when the superior angle of the scapula bulges into the trapezius on forced external rotation of the arm in the patient with an internal rotation con- tracture. Conversely, the so-called re- verse Putti sign demonstrates aber- rant scapular motion with forced internal rotation in the presence of an external rotation contracture. Surgical release of the contractures improves scapular motion, but in- completely. For external rotation and abduction contractures involving the supraspinatus and the posterior rota- tor cuff, surgical release must include these important motors of the shoul- der. Zancolli and colleagues 38,39 have reported such a surgical technique but did not report on clinical results.

Shoulder Problems in Children With Brachial Plexus Birth Palsy

Figure 13

in Children With Brachial Plexus Birth Palsy Figure 13 Postoperative active elevation of 140° (20° gain

Postoperative active elevation of 140° (20° gain from the preoperative state) 2 years after arthroscopic release in the same child as shown in Figures 1, 3, 4, and 9. Note the improved ability to reach, with improved external rotation.

Scapular dyskinesia may exist even when the internal rotation contrac- ture is not severe enough to compel surgical treatment. Although some weakness of the rhomboid and serra- tus anterior muscles may contribute to this scapular dyskinesia, these muscles are usually functional. The asymmetric scapular motion more likely relates to persistent stiffness of the glenohumeral joint and learned patterns of movement. As with a fro- zen shoulder, the ROM in these neu- rologically damaged shoulders is glo- bally restricted. Tightness in the superior structures such as the su- praspinatus will result in an abduc- tion contracture of the humerus rela- tive to the scapula. In severe cases, as

the child lowers the arm to the side, the scapula will elevate. With move- ment, as the child tries to position the hand in space, 38,39 the scapula will be carried along in an asymmet- ric manner. This abnormal scapular movement does not represent a mo- tor problem of the scapular muscles, but is secondary to changes at the glenohumeral joint in the most ex- treme cases. The acronym SHEAR has been used to refer to the deformity of scapular hypoplasia, elevation, and rotation. 45,46 Nath and colleagues 45,46 postulate that this deformity is the cause, not the result, of the ensuing medial rotation contractures and gle- nohumeral deformity. They offer a sur- gical protocol aimed at correcting this root cause. Although this approach does focus interest on a perhaps ne- glected concern of the parent or guard- ian (ie, unusual scapular motion), its theoretical premise conflicts with much of our current understanding of the pathophysiology affecting the shoulder in brachial plexus birth palsy. Brachial plexus lesions have much less effect on the muscles that move the scapula (and none on the trapezius and levator scap- ulae) than on the muscles that exter- nally rotate the shoulder. It is incongru- ous to attribute an internal (medial) rotation contracture to aberrant scap- ular motion when the more proximate cause is readily evident.

External Rotation Contractures

External rotation contractures pre- sent a smaller subset of the problems encountered from birth palsy than internal rotation contractures, with 14% reported by Zancolli and Zan- colli. 38 This contracture does not re- sult in posterior displacement of the glenohumeral joint or in significant deformity, but it can be functionally disabling. External rotation contrac- ture may also occur iatrogenically

following release of internal rotation contractures. Surgical intervention is indicated when functional limita- tions in personal care are severe and the child has great difficulty reaching the midline. Zancolli and Zancolli 39 described surgical release and length- ening of the posterior-superior rota- tor cuff for this contracture. Few others have experience with this ap- proach, and caution is advised before further weakening important motors of external rotation and elevation in affected children. However, internal rotational osteotomy of the humerus has been effective in helping appro- priately selected children both cos- metically and functionally in terms of their ability to reach the midline to perform activities of personal care. 10,41

Summary

Traction injuries to the brachial plexus during the birth process result in residual orthopaedic problems that most commonly affect the shoulder. Although the role of micro- surgical intervention has become clearer and the techniques refined, complete restoration of neuromuscu- lar function remains elusive. For motor weakness that follows the neurologic injury, internal rotation contractures most commonly occur as a result of external rotation weak- ness. This leads to deformity of the glenohumeral joint (ie, glenohumeral dysplasia, posterior humeral head subluxation). As a result, secondary orthopaedic evaluation and correc- tive procedures are an integral part of the treatment of these children. Internal rotation contractures must be managed aggressively to avert and even correct deformity of the gleno- humeral joint. A variety of surgical approaches to restore balance be- tween external rotation and internal rotation power has demonstrated

Michael L. Pearl, MD

clinical success. Currently, clinical studies do not definitively favor one approach, but existing evidence sup- ports early correction of the internal rotation contracture and reduction of the glenohumeral joint by open or arthroscopic means, even if it re- quires release of the anterior capsule. Surgeons who have experience with both open and arthroscopic methods usually find that the arthroscopic- guided release allows for equal if not greater contracture release, with re- duced morbidity. Regardless of the method used for anterior capsule re- lease, most surgeons who treat bra- chial plexus birth palsy favor supple- menting the release with a transfer of the latissimus dorsi tendon, whether in all children or in children older than age 3 years. Consideration of any of the proce- dures to improve shoulder function should include a realistic assessment of the child’s ability to use the hand and of the limitations at the elbow. A nonfunctional hand is unlikely to be more useful even when it can be placed in a functional position. Simi- larly, an elbow that cannot extend because of a persistent flexion con- tracture or an absent triceps may im- pede a child’s reach to a far greater extent than limitation of shoulder motion.

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