The Journal of Bone and Joint Surgery 80:659-67 (1998)
© 1998 The Journal of Bone and Joint Surgery, Inc.
Glenoid Deformity Secondary to Brachial Plexus Birth Palsy*
MICHAEL L. PEARL, M.D. and
BRADFORD W. EDGERTON, M.D. , LOS ANGELES, CALIFORNIA
Investigation performed at Kaiser Permanente, Los Angeles Medical Center, Los Angeles
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Abstract
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The association between internal rotation contracture secondary to brachial plexus birth palsy and deformity and posterior dislocation of the glenohumeral joint has been known for a long time. The precise nature of these deformities and their pathogenesis, however, remain unclear. Twenty-five children, ranging in age from 1.5 to 13.5 years, had an operation to release an internal rotation contracture secondary to brachial plexus birth palsy; eleven had a latissimus dorsi transfer to augment external rotation power as well. Arthrograms were made intraoperatively in order to clarify the pathological changes that occur in the glenohumeral joint during growth in patients who have this condition.
Seven children had a concentric glenohumeral joint (the humeral head was well centered in the glenoid fossa). The remaining eighteen children (72 per cent) had a deformity of the posterior aspect of the glenoid. Five of these children had flattening of the posterior aspect of the glenoid, seven had a biconcave glenoid with the humeral head articulating with the posterior of the two concavities, and six had a so-called pseudoglenoid (the most severe deformity, in which the humeral head articulated with a distinct, retroverted, posterior articular surface).
Internal rotation contracture secondary to brachial plexus birth palsy may lead to glenoid deformity that is severely advanced by the time that the child is two years old. In patients who have such a contracture, we recommend early imaging of the shoulder with arthrography or some other modality to allow visualization of the skeletally immature glenohumeral joint.
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Introduction
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Deformities of the glenohumeral joint have long been reported in association with brachial plexus birth palsy. Zancolli and Zancolli documented deformities and posterior subluxation or dislocation of the glenoid in sixty-two (72 per cent) of eighty-six patients who had operative treatment for an internal rotation contracture secondary to brachial plexus palsy33. Other authors also have noted these deformities4,5,11,17,20,26,30; however, to our knowledge, their morphology has not been accurately characterized and their prevalence has not been established.
The need for a greater understanding of deformities of the glenoid associated with brachial plexus birth palsy is evident in view of the fact that, in most clinical studies, operative release of tight muscles and transfer of others has been recommended only if deformity or incongruity of the joint was not present7-9,18,24,25,29,33. Confounding this understanding has been the difficulty in imaging the skeletally immature shoulder. The earlier investigators relied primarily on plain radiographs, which do not show early changes of the cartilaginous, unossified glenoid. To overcome this limitation, we performed intraoperative arthrography on all children at our institution who had an operation for the treatment of residual paralysis of the shoulder secondary to brachial plexus birth palsy.
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Materials and Methods
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Twenty-five consecutive children who had residual paresis and contracture secondary to brachial plexus birth palsy had operative intervention between July 1994 and May 1997 (Table I). The children ranged in age from 1.5 to 13.5 years. The goal of the operation was to release the internal rotation contracture and, in older children, also to restore external rotation strength. Plain radiographs of the shoulder that had been made preoperatively did not adequately demonstrate the largely unossified glenohumeral joint. Arthrograms therefore were made intraoperatively in order to determine whether or not the joint was dislocated.
The protocol for this study was approved by the Institutional Review Board of the Southern California Permanente Medical Group.
In fourteen younger children, the contracture was released without a muscle transfer. The release was accomplished by detaching the subscapularis from its origin and reflecting it distally along the anterior surface of the scapula7-9. The operative approach was along the posterior axillary line, anterior to the ventral border of the latissimus dorsi muscle.
In eleven older children, we transferred the latissimus dorsi to change its line of pull to that of an external rotator in addition to releasing the internal rotation contracture. Five of these children needed an anterior operative approach through the deltopectoral interval in order to release the contracted anterior aspect of the joint capsule and rotator interval tissue as well as the subscapularis muscle.
At the time of the operation, an arthrogram was made with the patient under general anesthesia. With use of image intensification, a 20-gauge needle was inserted into the glenohumeral joint, and four to six milliliters of 50 per cent Renografin 60 (diatrizoate sodium and diatrizoate meglumine) was injected. A total of three arthrograms (one anteroposterior and two axillary) were made. One axillary arthrogram was made with the shoulder in 90 degrees of abduction and the other, with it in slight flexion (roughly 60 degrees of elevation in the 45-degree thoracic plane).
Statistical Methods
The distribution of continuous variables was tested for normality with use of the Shapiro and Wilk test28. Comparisons of continuous variables (age of the patient and preoperative passive external rotation) among the four different appearances of the glenoid (ranging from normal to a distinct pseudoglenoid) were based on the exact probability of the Kruskal-Wallis test, conducted with use of StatXact 3 software (CYTEL Software, Cambridge, Massachusetts). The Fisher exact test, conducted with use of SAS software (version 6.12; SAS Institute, Cary, North Carolina) was used to compare the binary variable of whether an anterior operative approach was needed among the four groups of patients. Comparison of the ages of the patients who needed a more extensive operative release with those who did not was performed with the exact probability of the Wilcoxon-Mann-Whitney test.
The results were recorded as the median (and range) for the continuous variables and as the cell size for the categorical variables unless otherwise stated. All analyses were two-tailed. Type-I error was set at the 0.05 level.
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Results
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The axillary arthrogram best displayed the shape of the glenoid and was the most useful for this study. In children who had severe deformity, the anteroposterior arthrogram showed the overlapping shadows of the glenoid and the humeral head that are typically seen with a posterior dislocation but it was not otherwise useful (Fig. 1).
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Fig. 1: Case 23, a 6.7-year-old girl. Anteroposterior arthrogram (shown with and without a line-tracing) demonstrating posterior dislocation of the joint secondary to a pseudoglenoid deformity. The overlapping shadows of the humeral head and the glenoid are characteristic of a posterior dislocation. Ac = acromion, CL = clavicle, HH = humeral head, and GL = glenoid.
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Seven children had a concentric glenohumeral joint (a round humeral head centered in a glenoid with a matching curve) (Figs. 2-A and 2-B). The other eighteen children (72 per cent) had a deformity of the posterior aspect of the glenoid. There were three consistent patterns. Five children had flattening of the posterior aspect of the glenoid (Figs. 3-A and 3-B). Seven children had a biconcave glenoid with the humeral head articulating with the posterior of the two concavities (Figs. 4-A and 4-B). In some children, the glenoid appeared flat on one axillary view but biconcave on the other. The glenoid was considered flat only if it appeared so on both axillary projections. Six children had a pseudoglenoid, the most severe deformity, with the posterior concavity distinct and separate from the remaining, original articular surface (Figs. 5-A, 5-B, and 5-C). This deformity was distinguished from the biconcave glenoid in that the posterior articular cavity was clearly retroverted in relationship to the original glenoid. Intraoperatively, the pseudoglenoid was seen as adjacent articular surfaces, which gave the glenoid a bifid appearance. Both articular surfaces were covered with articular cartilage but were separated by a sharp cleft at the line of intersection. The humeral head rotated smoothly and congruently on the posterior articular surface until it relocated to the original anterior articular surface after release of the contracture. After sufficient soft-tissue release, the humeral head either rotated against one of the articular surfaces or popped freely from one to the other, depending on the position and motion of the arm. Positioning the arm in external rotation caused the humeral head to relocate to the original anterior articular surface. This position was maintained with use of a spica cast.
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Figs. 2-A and 2-B: Case 25, a 1.5-year-old girl (the youngest child in the series) who had a concentric glenohumeral joint.
Fig. 2-A: Plain axillary radiograph showing the relative alignment of the humerus and the scapula, which suggests a centered, located glenohumeral joint. In children of this age, the humeral head and the glenoid consist mainly of unossified cartilage.
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Fig. 2-B: Axillary arthrogram (shown with and without a line-tracing), made with the arm in external rotation, demonstrating the concentric glenohumeral joint. With the arm in full external rotation, the tight anterior aspect of the capsule pushes the humeral head onto the posterior aspect of the glenoid. Ac = acromion, HH = humeral head, and GL = glenoid.
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Figs. 3-A and 3-B: Arthrograms (shown with and without a line-tracing) demonstrating a flat glenoid. The relative alignment of the humerus and the scapula suggests posterior positioning of the humeral head on the flattened glenoid. Ac = acromion, HH = humeral head, GL = glenoid, and Co = coracoid process.
Fig. 3-A: Case 19, a 2.0-year-old boy.
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Figs. 4-A and 4-B: Arthrograms (shown with and without a line-tracing) demonstrating a biconcave glenoid. The relative alignment of the humerus and the scapula suggests that the humeral head articulates with the posterior of the two concavities. Ac = acromion, HH = humeral head, GL = glenoid, and Co = coracoid process.
Fig. 4-A: Case 15, a 2.1-year-old girl.
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Figs. 5-A, 5-B, and 5-C: Images (shown with and without a line-tracing) demonstrating a pseudoglenoid (PsGL). HH = humeral head, GL = glenoid, Ac = acromion, Co = coracoid process, and CL = clavicle.
Fig. 5-A: Case 20, a 2.0-year-old boy, the youngest child who had a pseudoglenoid in the series. The humeral head is seen to articulate with a retroverted, posterior articular surface (the pseudoglenoid) on this arthrogram.
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Fig. 5-B: Case 13, a 13.5-year-old boy, the oldest child who had a pseudoglenoid in the series. The child is old enough for the anatomy to be visualized on this plain radiograph without contrast material. The axillary radiograph shows the humeral head articulating with a retroverted pseudoglenoid.
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Fig. 5-C: Postoperative axillary radiograph of the same patient as in Fig. 5-B. The humeral head is relocated in a diminutive, underdeveloped glenoid. The anteroposterior dimensions of the scapula, from the coracoid to the acromion, are markedly narrowed. Metallic suture anchors were used to secure the latissimus dorsi transfer to the humerus.
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The presence and type of glenoid deformity were significantly associated with the severity of the internal rotation contracture (p = 0.003). Preoperatively, the median range of passive external rotation was -45 degrees for the patients who had a pseudoglenoid, -10 degrees for those who had a flat or biconcave glenoid, and 0 degrees for those who had a concentric glenoid (Table II). Glenoid deformity was also associated with the patient's age (p = 0.041), although all types of deformity were seen at all ages.
Older children were more likely to need a more extensive release that involved an anterior operative approach than were younger children (p < 0.001). The median age of the children who needed such an approach was 10.9 years (range, 4.8 to 13.5 years) compared with 2.8 years (range, 1.5 to 9.2 years) for those who did not need an anterior approach.
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Discussion
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The previous literature documenting the morphology of the glenoid in children who have an internal rotation contracture secondary to brachial plexus birth palsy is essentially limited to a case report of one patient who had a single arthrogram31 and a study of five patients who were examined with magnetic resonance imaging11,12. The arthrogram, of a four-month-old infant, showed that the glenohumeral joint dislocated posteriorly with internal rotation31. The magnetic resonance imaging study revealed changes to the posterior aspect of the glenoid in all five patients; these changes included damage to the posterior aspect of the labrum, thinning of the posterior articular cartilage, blunting of the posterior corner, and subluxation of the joint11,12.
The findings of these studies are consistent with those presented here. We hypothesize that a persistent, posteriorly directed force on the growing glenoid may either erode the posterior aspect of the glenoid or inhibit its development. In patients who have the most severe deformity, a pseudoglenoid, the humeral head articulates with a retroverted, posterior articular surface. The pseudoglenoid is in many ways analogous to the pseudoacetabulum of congenital hip dysplasia, in which the femoral head subluxates superiorly and may come to lie in a distinct articulation formed at the base of the iliac wing, superior to the original, true acetabulum3,15,21.
Harryman et al., in a cadaver model, showed that tightening the glenohumeral joint capsule imparts a force on the humeral head away from the capsule, a mechanical property defined as capsular constraint13,14. An internal rotation contracture secondary to tight anterior soft tissues therefore would apply a posteriorly directed force to the humeral head as the soft tissues tighten2,6. Wear of the posterior aspect of the glenoid secondary to an internal rotation contracture also has been well described in the adult shoulder1,16,23. Osteoarthrosis of the glenohumeral joint frequently is seen with an internal rotation contracture and eccentric wear of the posterior aspect of the glenoid. The glenoid may appear biconcave on inspection23. In somewhat younger adults, this mechanism has been observed secondary to repairs for instability that resulted in overtightening of the anterior soft tissues, a condition known as capsulorrhaphy arthropathy1,16.
Zancolli and Zancolli, in their system for the classification of children who have an internal rotation contracture secondary to brachial plexus birth palsy, distinguished between those who had a glenoid deformity or posterior subluxation (or dislocation), or both, and those who did not33. We further divided the deformities into three distinct groups: flat glenoids, bi-concave glenoids, and pseudoglenoids (Fig. 6). Zancolli and Zancolli recommended that patients who have a deformity be managed with an external rotation osteotomy of the proximal aspect of the humerus to improve the position of the arm33. This procedure leaves the humeral head in the posteriorly located pseudoglenoid; thus, it is not possible for the joint to develop normally.
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Fig. 6: Schematic drawings showing the spectrum of the glenoid morphology that was seen in this series. The findings ranged from a normal concentric concavity to a deformed, retroverted articular surface (a pseudoglenoid [PsGL]). Ac = acromion, GL = glenoid, and Co = coracoid process.
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Recommendations for the management of children who have an internal rotation contracture are widely divergent and have included release of the subscapularis, the anterior aspect of the capsule, the pectoralis major, or more than one of these structures5,7-9,19,27. Other authors have suggested transfer of the latissimus dorsi in addition to release of the tight structures10,18,22,25,29,32. Zancolli and Zancolli33, as well as others7-9,24,29, have cautioned that, in patients who have incongruity of the joint, a full anterior release causes the humeral head to dislocate anteriorly on the deformed glenoid. Sufficient detail regarding the anatomy of the glenoid, however, was lacking in all of these clinical series. Without knowledge of the underlying shape of the glenoid, it is impossible to compare the outcomes of these various reports and operative procedures. Our approach has been to perform adequate soft-tissue releases in order to relocate the humeral head (with the addition of a muscle transfer in older children), in the hope that many of these deformities will be ameliorated.
Knowledge of the degree of the internal rotation contracture may be helpful in assessing the presence and type of glenoid deformity, but it is essential that preoperative or intraoperative magnetic resonance imaging or arthrography be performed. Plain radiographs may give a sense of the relative anteroposterior location of the humeral head and the scapula, but they provide scant detail about the evolving morphology of the glenoid. The inferior ossification center of the glenoid does not ossify until puberty, and it may become markedly deformed before there is radiographic evidence of deformity.
It is interesting to note that not all glenoids deform in the presence of an internal rotation contracture. Strikingly, the prevalence of relatively normal glenohumeral joints both in the current study (seven of twenty-five) and in that of Zancolli and Zancolli (twenty-four of eighty-six)33 was 28 per cent. Factors other than the contracture that were not readily apparent in our study must have contributed to the development of the glenoid in each child. Differences in muscle strength are very difficult to determine through examination of an infant's shoulder. Whether some of our patients had partial innervation of the supraspinatus and infraspinatus and others had none is not ascertainable.
In light of the findings of the current study, we are much more vigilant with regard to progressive internal rotation contractures in children who have brachial plexus birth palsy. A restricted range of passive external rotation may begin to manifest as early as in the first six months of life. At that time, the parents are alerted to the possibility of operative intervention and are instructed about stretching exercises to be performed at home. Intensive occupational or physical therapy also is initiated. If the child does not respond to the intensive exercise program, we recommend operative release of the subscapularis muscle. Although our clinical follow-up data to date are inadequate for critical evaluation of the effectiveness of operative intervention, we believe, on the basis of the anatomical findings presented here, that early release is the rational approach for the treatment of these contractures.
In conclusion, an internal rotation contracture secondary to brachial plexus birth palsy has a high likelihood of being associated with glenoid deformity. For patients who have such a contracture, we recommend early imaging of the shoulder with arthrography or some other modality that will allow visualization of the skeletally immature glenohumeral joint.
NOTE: The authors thank Wansu Chen, B.S., for performing the statistical analysis, and David Apel, M.D., Joel Feigenbaum, M.D., and Paul Kazimiroff, M.D., for reviewing the radiographs with the authors.
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Footnotes
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*No benefits in any form have been received or will be received from a commercial party related directly or indirectly to the subject of this article. Funds were received in total or partial support of the research or clinical study presented in this article. The funding source was the Kaiser Permanente Medical Group.
Department of Orthopaedics, Kaiser Permanente, Los Angeles Medical Center, 4747 Sunset Boulevard, Los Angeles, California 90027. E-mail address: michael.l.pearl@kp.org.
Department of Plastic Surgery, 6041 Cadillac Avenue, Kaiser Permanente, Los Angeles Medical Center, Los Angeles, California 90034. E-mail address: bradford.w.edgerton@kp.org.
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86(10):
2163 - 2170.
[Abstract]
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D. Moukoko, M. Ezaki, D. Wilkes, and P. Carter
Posterior Shoulder Dislocation in Infants with Neonatal Brachial Plexus Palsy
J. Bone Joint Surg. Am.,
April 1, 2004;
86(4):
787 - 793.
[Abstract]
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M. L. Pearl, B. W. Edgerton, D. S. Kon, A. B. Darakjian, A. E. Kosco, P. B. Kazimiroff, and R. J. Burchette
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.,
May 1, 2003;
85(5):
890 - 898.
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A. F. Hoeksma, A. M. ter Steeg, P. Dijkstra, R. G.H.H. Nelissen, A. Beelen, and B. A. de Jong
Shoulder Contracture and Osseous Deformity in Obstetrical Brachial Plexus Injuries
J. Bone Joint Surg. Am.,
January 29, 2003;
85(2):
316 - 322.
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A F Hoeksma, H Wolf, and S L Oei
Obstetrical brachial plexus injuries: incidence, natural course and shoulder contracture
Clinical Rehabilitation,
May 1, 2000;
14(5):
523 - 526.
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Top
Abstract
Introduction
