Abstract
Purpose
To report the functional and cosmetic results of cases with Sprengel deformity (SD) who underwent a novel technical modification in Woodward procedure for severe SD comprising of lowering of the scapula followed by stabilization of scapula with application of anchoring sutures.
Materials and methods
Twenty-three children were operated at a mean age of 6.6 ± 2.5 years (range 3.4–11 years) and followed up for a mean period of 5.51 ± 1.98 years.
Results
The mean Cavendish score of the patients before procedure of 3.2 ± 0.45 was reduced to 1.7 ± 0.56 (p < 0.001) after the surgery. All patients improved in global shoulder abduction. The mean preoperative abduction of 97° ± 15.2° increased to a mean of 149° ± 14.1° post-operatively (p < 0.001). Significant improvement was also observed in mean Rigault grades after surgical intervention using anchoring suture modification of Woodward procedure. The mean Rigault grade of 2.7 ± 0.47 prior to surgery was reduced to 1.3 ± 0.47 (p < 0.001) at final evaluation. Age of the patients negatively correlated with the functional outcome (r = – 0.48; p < 0.02). The presence of an omovertebral bar or associated congenital anomalies did not affect the functional outcome.
Conclusion
The modification of Woodward procedure for correction of SD by application of anchoring sutures was a reliable method for correction of SD particularly at younger age. The procedure was associated with significant improvement in shoulder abduction, cosmetic appearance and radiological grades over 5 year follow-up.
Keywords: Sprengel’s deformity, Cavendish score, Rigault grade, Woodward procedure, Congenital high scapula, Omovertebral bone
Introduction
Congenital high scapula, commonly known as Sprengel deformity (SD) was initially described by Eulenberg [1] in 1863 and later by Sprengel [2] in 1891. This rare congenital deformity of unknown etiology occurs due to interruption in caudal migration of the scapula in early fetal development resulting in an elevated and hypoplastic scapula. In 25–50% of cases, a bony structure referred to as the ‘omovertebral bone’ or a fibrous and/or cartilaginous structure is interposed between the scapula and the cervical spine [3, 4]. Surgical management of SD involves resection of omovertebral bone or its fibrous or cartilaginous equivalent for improvement in shoulder function and cosmetic appearance.
A number of surgical options are available for the management of severe SD. The management strategies include partial scapulectomy or scapulotomy [2, 5, 6] and scapular translocation involving replacement of insertions of shoulder girdle muscles on scapula [7–9] or by translocation of origins of shoulder girdle muscles on vertebrae [5, 10–13]. These procedures are performed as a single procedure or in combination with an osteotomy of the clavicle to avoid brachial plexus injury.
Over the years, several modifications in Woodward technique have been introduced [10, 11]. Prior to this modification, Woodward procedure [12] was performed for correction of SD. Persistence of cosmetic deformity, limitation of abduction and brachial plexus palsy prompted the need for introduction of modification in Woodward procedure. We present a retrospective evaluation of a novel technical modification in Woodward technique for severe SD comprising of lowering of the scapula followed by stabilization of scapula with application of anchoring sutures. Following surgical intervention data prior to and after surgery were compared for functional, radiological and cosmetic outcomes.
Patient and Methods
This was a retrospective analysis of a novel modification of Woodward procedure performed for correction of SD among 23 patients between 2008 and 2017 at King Khalid University Hospital Riyadh. This group of patients comprised of 14 female and 9 male patients with the mean age of 6.6 ± 2.5 years. Data for demographic and clinical details were extracted from the patients’ medical records. Each patient underwent a thorough physical examination along with radiological assessment of the chest, cervical and thoracic spines before and after surgical intervention. Clinical assessment was performed and Cavendish [13] classification was used to grade the cosmetic appearance of the deformity in each patient both prior to and after surgery. Based on Cavendish grading, 6 scapulae were assigned grade 1 V (severe) and 17 were assigned grade III (moderate) at the time of diagnosis. Radiographic classification of the deformities was performed using Rigault and Pouliquen’s [14] scoring system into three grades based on the location of supero-medial angle of the hypoplastic scapula. The deformity was classified as grade 1 when the supero-medial angle of the scapula was located between the transverse apophysis of the second and the fourth thoracic vertebra. Location of supero-medial angle of scapula between the transverse apophysis of the second thoracic and the fifth cervical vertebra was classified as grade II, whereas the deformity with location of supero-medial angle of the scapula above the transverse apophysis of the fifth cervical vertebra was classified as grade III. Of the 23 scapulae, 16 were classified as grade III and 7 had grade II deformity prior to surgical intervention.
All patients with Cavendish grade III and IV deformity underwent CT scan evaluation and 3D reconstruction of the shoulder and the scapula was performed to exclude associated omovertebral connections. The primary objective of these investigations was to detect associated anomalies such as scoliosis, anomalies of ribs or vertebral segments and omovertebral bone connections. In addition, all patients with congenital scoliosis were offered magnetic resonance imaging (MRI) for assessment of associated spinal cord anomalies. All patients with SD were included in the study as a consecutive case series and this novel anchoring suture modification of Woodward procedure was performed by a single surgeon.
Anchoring Suture Modification of Woodward Procedure for Surgical Treatment of Sprengel Deformity
Surgical Technique
The novel modification of Woodward procedure with application of anchoring sutures involved detachment of the origins of the trapezius and rhomboid muscles from the spinous process and caudal displacement of the elevated scapula after excision of omovertebral bone or fibrous connection with the scapula. The muscles from the superior and medial borders of the scapula were reflected extraperiosteally and scapular release along with mobility to full extent was achieved. Guided by the preoperative 3D-reconstruction based on CT or MRI findings scapuloplasty was performed with excision of the superiomedial border of the scapula for reshaping the deformed scapular contour. Scapula was then displaced distally down to the level of the normal side using the superomedial angle of the scapula as a landmark instead of the hypoplastic tip of the scapula. No attempt was made to bring the inferior angle of scapula down to the level of the normal side to avoid traction on the brachial plexus because of the smaller size of the scapula. Once the scapula was in its corrected position and full scapular rotation was achieved by abduction of the arm. The scapula was retained in the caudal position by application of stout absorbable No 2 Vicryl suture anchoring scapula with spinous process of the relevant vertebra, either T11 or T12. The scapular end of the suture was placed close to the center of rotation of the scapula 1 cm lateral to the vertebral edge at the junction of the upper and lower halves of the scapula (Fig. 1). A similar study reported application of scapular ends of sutures at the superomedial scapular border at the junction of upper and middle third of the scapula [15]. The suture was then brought distally at about a 30–40 degrees angle and was tied to the spinous process by making a hole with a towel clip. To achieve adequate orientation of the glenoid and increased range of abduction, the suture was tightened. Before tying the suture, the arm was carried through full range of abduction to ensure that the suture was tight enough and allowed adequate scapular rotation. The angle of suture application, therefore, varied between 30 and 40° as opposed to fixed suture angulation of 45° in a study reported by Ahmed in 2010 [15] (Table 1).
Fig. 1.
a Intraoperative photograph showing rotation and relocation of the hypoplastic left scapula. b Schematic intraoperative procedure depicting: (1) Downward tilting of the glenoid. (2) Resection of superomedial boarder of scapula and omovertebral bar. (3) Application of stout sutures
Table 1.
Comparison of novel modifications in Woodward Procedure with previously published anchoring suture technique (Ahmad AA 2010) [15]
S. no | Modification | Current study from Riyadh City | Previous study by Ahmad AA 2010 from Palestine [15] |
---|---|---|---|
1 | Placement of sutures | Close to the center of rotation of the scapula 1 cm lateral to the vertebral edge at the junction of the upper and lower halves of the scapula | Superomedial scapula about 1 cm from the edge at the junction of upper and middle third |
2 | Angle of sutures | Angle varied between 30°–40° depending upon the achievement of maximum shoulder abduction | Fixed angle of 45° |
3 | Post-operative immobilization | For two weeks | No immobilization |
If the rotation was noticed to be limited by the angle of the suture it was adjusted accordingly to ensure the full range of movement. For additional, strength, a second stout suture was applied in a similar fashion. The scapula and detached muscles were relocated with heavy sutures. No clavicle osteotomy or external fixation was performed in any patient.
Post-operatively, a Velpeau bandage was applied for 2 weeks and active-assisted range of movement exercises were commenced 2 weeks after the surgery. All patients were examined at one, three, six, nine and 12 monthly intervals and thereafter annually for a mean period of 5.51 ± 1.98 years. The clinical appearance at the last available follow-up including assessment of shoulder abduction, Cavendish and Rigualt grades was recorded for each patient.
The appearance of the scar was considered normal if it matched the adjoining tissue, hypertrophic if it was thicker than the adjoining tissue and keloid if it was abnormal, thicker and different in color and texture. Outcome assessment was done by measuring the range of abduction at the shoulder joint at final follow-up and compared to similar preoperative measurements.
Similarly, Cavendish grades and Rigault grades at final review were compared to those before surgery. Any associated malformation and the presence or absence of omovertebral bar was sought and its impact on the outcome was assessed. The effect of age at the time of surgery, preoperative disease severity and final cosmetic and functional outcomes were also evaluated. A subjective inquiry at the last follow-up examination was directed to both the parents and the patients to rate the overall satisfaction in terms of not satisfied, satisfied and highly satisfied.
Statistical Analysis
Statistical analysis was performed using SPSS v.19 software (SSPS Inc., Chicago, IL, USA). Wilcoxon signed-rank test was used for the paired analysis of preoperative and final values of Cavendish grades and range of abduction. Impact of omovertebral bar and associated malformations on the end result was evaluated by the Mann–Whitney test. Pearson’s correlation coefficient was used to evaluate relationship between age, functional improvement and radiographic improvement. A p-value < 0.05 was considered statistically significant.
Results
Table 2 describes characteristic features of 23 patients with SD included in the study. This group of patients included 14 (60.8%) male and 9 (39.2%) female patients with the mean age of 6.6 ± 2.5 years (range 3.4–11 years). The deformity involved left shoulder among 14 patients, whereas the right shoulder was affected among 9 patients. Associated anomalies included omovertebral bar in 10 (43.4%) patients, Klippel-Feil syndrome in 9 (39.1%), spina bifida in 3 (13%), camptodactyly in 3 (13%), scoliosis in 3 (13%) and cleft palate in 2 (8.6%) children.
Table 2.
Characteristics features of patients with severe Sprengel deformity
S. no | Sex | Age at surgery (years) | Side | Omovertebral bar | Associated congenital Anomalies |
---|---|---|---|---|---|
1 | M | 7 | R | Omovertebral bar | K.F.S, S. Bifida, Scoliosis, FRs |
2 | M | 6 | L | None | |
3 | M | 3.8 | R | Omovertebral bar | |
4 | M | 8 | L | None | S. Bifida |
5 | F | 7 | L | Omovertebral bar | K.F.S |
6 | F | 9 | L | None | K.F.S |
7 | M | 11 | R | Omovertebral bar | K.F.S Scoliosis, FRs |
8 | M | 6 | L | None | |
9 | M | 5 | R | None | |
10 | F | 5 | R | None | K.F.S |
11 | M | 4 | L | None | |
12 | M | 6 | R | Omovertebral bar | K.F.S |
13 | M | 11 | R | None | |
14 | M | 13 | L | Cartilaginous Bar | K.F.S, S. Bifida |
15 | M | 4.2 | R | Omovertebral bar | Scoliosis, FRs |
16 | F | 7 | L | Cartilaginous bar | |
17 | F | 6.2 | L | Fibrous bar | |
18 | F | 5.5 | L | None | |
19 | F | 6 | L | None | |
20 | F | 5 | L | None | K.F.S |
21 | F | 3.8 | L | Fibrous bar | |
22 | M | 3.4 | L | None | |
23 | M | 9 | R | None |
Table 3 compares pre-operative and post-operative data for cosmetic and functional outcomes among patients treated with the novel anchoring sutures modification of Woodward’s procedure. A significant cosmetic improvement of at least 2 Cavendish grades was observed in all the patients following surgical intervention. The mean Cavendish score of the patients before anchoring suture modification of Woodward’s procedure of 3.2 ± 0.45 was reduced to 1.7 ± 0.56 (p < 0.001) after the surgery. Assessment at the follow-up revealed that out of 23 patients 8 had Cavendish grade 1 of cosmetic improvement 14 had grade II and only one patient had grade III Cavendish score. All patients experienced an improvement in global shoulder abduction. The mean preoperative abduction of 97° ± 15.2° increased to a mean of 149° ± 14.1° post-operatively (p < 0.001). Figure 2 shows representative images of preoperative Cavendish grade 4 deformity (Fig. 2a) reduced to Cavendish grade 1 postoperatively (Fig. 2b). Significant improvement was also observed in mean Rigault grades after surgical intervention using anchoring suture modification of Woodward procedure. Figure 3 shows preoperative radiograph of a patient with Rigault III deformity (Fig. 3a) improved to Rigault I following surgery (Fig. 3b). The mean Rigault grade prior to surgery was 2.7 ± 0.47 and at the final evaluation it was reduced to 1.3 ± 0.47 (p < 0.001).
Table 3.
Comparison of pre and post-operative data for changes in abduction, Cavendish and Rigault grades among 23 patients treated with modified Woodward’s procedure
S. no | Cavendish classification Pre-op | Cavendish classification Post-op | Abduction in degrees Pre-op | Abduction in degrees Post-op | Rigualt Grade Pre-op | Rigualt Grade Post-op |
---|---|---|---|---|---|---|
1 | Grade IV | Grade II | 90 | 135 | 3 | 2 |
2 | Grade III | Grade I | 100 | 140 | 3 | 2 |
3 | Grade III | Grade I | 110 | 160 | 3 | 1 |
4 | Grade IV | Grade I | 115 | 160 | 2 | 1 |
5 | Grade III | Grade II | 80 | 110 | 3 | 2 |
6 | Grade III | Grade II | 80 | 140 | 3 | 2 |
7 | Grade III | Grade II | 125 | 145 | 3 | 2 |
8 | Grade III | Grade I | 90 | 150 | 3 | 1 |
9 | Grade III | Grade II | 110 | 140 | 2 | 1 |
10 | Grade III | Grade II | 90 | 150 | 3 | 1 |
11 | Grade III | Grade I | 140 | 160 | 2 | 1 |
12 | Grade III | Grade II | 90 | 150 | 2 | 1 |
13 | Grade III | Grade II | 90 | 140 | 3 | 2 |
14 | Grade III | Grade II | 110 | 130 | 3 | 2 |
15 | Grade III | Grade I | 80 | 140 | 3 | 1 |
16 | Grade III | Grade I | 100 | 170 | 2 | 1 |
17 | Grade III | Grade I | 100 | 160 | 3 | 1 |
18 | Grade IV | Grade II | 90 | 150 | 3 | 1 |
19 | Grade III | Grade II | 90 | 160 | 2 | 1 |
20 | Grade III | Grade II | 100 | 150 | 2 | 1 |
21 | Grade IV | Grade III | 90 | 170 | 3 | 1 |
22 | Grade IV | Grade II | 90 | 170 | 3 | 1 |
23 | Grade IV | Grade II | 80 | 150 | 3 | 1 |
Mean | 3.2 ± 0.45 | 1.7 ± 0.56 | 97.3 ± 15.2 | 149 ± 14.1 | 2.7 ± 0.47 | 1.3 ± 0.47 |
p value | p < 0.001 | p < 0.001 | p < 0.001 |
Fig. 2.
a Preoperative clinical photograph showing Cavendish grade 4 deformity. b Follow-up clinical photograph after 2.5 years with Cavendish grade 4 deformity reduced to grade 1 and shoulder abduction
Fig. 5.
Parental response to surgical outcome of anchoring suture modification of Woodward procedure for correction of SD at the final follow-up visit at 5.5 ± 1.98 years after surgery (n = 23)
Anchoring suture modification of Woodward’s procedure failed to achieve adequate improvement in abduction at the shoulder among older children satisfactory cosmetic outcome, however, was achieved. Age of the patients negatively correlated with the functional outcome (r = – 0.48; p < 0.02) Fig. 4. None of the children suffered from postoperative brachial plexus palsy.
Fig. 3.
a Preoperative anteroposterior radiograph showing elevated scapula on the left with spine of the scapula at the level of the sixth cervical vertebra (Rigault III), also seen is the omovertebral bar (arrow). b Follow-up radiograph after 4.5 years of the same patient with spine of the scapula at the level of the second thoracic vertebra (Rigault I). Note the de-rotation of the scapula
Table 4 describes data comparing the functional, cosmetic and radiological outcomes among 23 patients with SD who underwent anchoring suture modification of Woodward procedure in the presence or absence of omovertebral bar. Comparison of the surgical outcomes between 11 patients without omovertebral bar with a group of 12 children with omovertebral bar revealed that the presence of omoveretebral bar did not influence the surgical outcomes of shoulder abduction, Cavendish and Rigault grades. Similar observations were also made for the presence of associated anomalies other than omovertebral bar. Shoulder abduction, Cavendish and Rigault grades were no different between the groups (Table 5).
Table 4.
Comparison of data for children with Sprengel deformity treated with modified Woodward procedure either in the presence or absence omovertebral bar
Change | OVB absent (n = 11) | OVB present (n = 12) | P value |
---|---|---|---|
Abduction (mean ± sd) | 48.1 ± 21.3 | 55 ± 14.9 | 0.38 |
Cavendish score (mean ± sd) | − 1.36 ± 0.5 | − 1.75 ± 0.4 | 0.06 |
Rigualt score (mean ± sd) | − 1.27 ± 0.4 | − 1.5 ± 0.5 | 0.28 |
n 23, OVB Omovertebral bar, sd standard deviation
Table 5.
Comparison of data for children with Sprengel deformity treated with modified Woodward procedure either in the presence or absence of associated anomalies
Change | Associated anomalies absent | Associated anomalies present | P value |
---|---|---|---|
Abduction (mean ± sd) | 55.77 ± 6.81 | 46.5 ± 19.44 | 0.23 |
Cavendish score (mean ± sd) | − 1.46 ± 0.52 | − 1.7 ± 0.48 | 0.27 |
Rigualt score (mean ± sd) | − 1.46 ± 0.52 | − 1.3 ± 0.48 | 0.45 |
n 23, Number of patients with no associated anomalies 13, number of patients with associated anomalies 10, sd standard deviation
Patient Satisfaction
Figure 5 shows data for parental response at the last follow-up visit (mean 5.5 ± 1.98 years) to the surgical outcome of anchoring suture modification of Woodward procedure. Majority of the parents (13; 56.5%) were highly satisfied followed by 9 (39.3%) parents who were satisfied, whereas 1 (4.2%) parent was not satisfied with the surgical outcome with regards to their children’s daily activities and participation in sports.
Fig. 4.
Correlation between age and shoulder abduction in degrees
Discussion
The Green [16] and Woodward [12] procedures are commonly performed techniques for correction of SD. The anchoring suture modification of Woodward procedure [12] performed in the present study comprised of lowering of the scapula with a correction of the Varus of the glenoid cavity and use of anchoring sutures to achieve stability of shoulder. Several operative procedures have been described for correction SD but the functional and cosmetic improvement achieved in the present study has not been reported in any previously described modifications of Woodward procedure. Earlier studies (Table 6) describing the surgical outcomes of Woodward procedure for correction of SD reported a mean of 32° improvement in abduction of shoulder. The anchoring suture modification of Woodward procedure performed in the present study increased the shoulder abduction by 45° in the latest follow-up of patients with SD. Moreover, unacceptable rates of incomplete correction, recurrent deformity following simple excision of superomedial part of the scapula and omovertebral bone or distal transplantation of scapula have been reported in a number of studies [13, 19–21]. None of the patients with SD in the present study developed recurrence of deformity during long-term follow-up after having undergone anchoring suture modification of Woodward procedure performed in the present study. The modified Woodward procedure for correction of SD in the present study was associated with a favorable outcome with negligible rate of complications and recurrence of deformity.
Table 6.
Comparison of the observations in the present study with the previously published reports for surgical correction of Sprengel deformity
Author | Cases | Mean age at the time of operation (years) | Function before operation | Function after operation | Cavendish grade before operation | Cavendish grade after operation | Operative technique |
---|---|---|---|---|---|---|---|
Woodward [12] | 9 | 8.1 | 140 | 172 | Woodward | ||
Borges [10] | 15 | 8.3 | 115 | 150 | 3 | 1.53 | Modified Woodward |
Cavendish [13] | 34 | 7.2 | 140 | 154 | 3.1 | 1.7 | All methods in common use |
Wilkinson [17] | 12 | 8.4 | 118 | 164 | 3 | 1.33 | Scapular Osteotomy |
McMurty [6] | 2 | 8.1 | 88 | 132 | 3 | 1.5 | Scapular Osteotomy |
Crha [18] | 18 | 7 | 145 | 151.6 | 2.55 | 1.88 | Woodward |
Grogan [19] | 20 | 6.5 | 110 | 147 | Woodward | ||
Present Study | 23 | 6.6 | 97 | 142 | 3.2 | 1.6 | Modified Woodward |
Brachial plexus palsy remains one of the most dreaded complications of the procedure. Postoperative neurological involvement, either transient or permanent, after the Woodward procedure has be reported in a number of studies [22]. Caudal displacement of the scapula increases the risk of damage to brachial plexus in surgical correction of SD [16, 22]. Clavicular osteotomy is performed to minimize the occurrence of neurovascular compression, particularly in severe cases among older patients [23]. In this study, clavicular osteotomy was not performed and none of the patients developed brachial plexus palsy or winging of scapula post-operatively. The role of clavicular osteotomy in avoiding brachial plexus palsy, however, remains elusive [19]. There is no direct evidence to suggest that clavicular osteotomy prevents neurological complications. Clavicular osteotomy, however, helps in mobilization of scapula in older children, where the joints are not supple enough to allow a good surgical descent [24]. A number of studies have recommended clavicular osteotomy as a routine part of the procedure to diminish the risk of neurovascular compression between the clavicle and deformed chest wall [24, 26].
The observations of the present study are in agreement with Grogan et al. [19] recommending that as long as the correction is not overly vigorous, clavicular osteotomy is usually not necessary to achieve favorable outcome. We propose that excision of the omovertebral connection followed by a scapuloplasty and proper but not forceful caudal relocation of the scapula may circumvent the possibility of neurological complications. Based on the observations of the present series, it appears that the anchoring suture modification of the Woodward procedure involving excision of prominent superomedial border of the scapula accomplishes these objectives and offer patients the opportunity for cosmetic and functional improvement.
Compared with the previously reported cases in other series the mean patient age at the time of surgery in our study was 6.6 years, which was less than the mean age reported in literature (Table 6). Currently consensus regarding the optimal age for correction for SD is lacking. Surgical correction before 3 years of age is technically more challenging compared to older children [21]. Advancing age tends to adversely affect the outcome [10–13, 19, 21], where modified Woodward procedure performed in older patients was shown to improve the cosmetic appearance but not the function. Similar observations were made in the present study, where a negative correlation between advancing age the functional outcome was observed. Moreover, it has been recommended that the best outcome of surgical intervention is achieved between the ages of 3 and 8 years [19, 22] On the contrary modified Green’s operation for correction of SD has been recommended soon after attainment of 2 years of age on the pretext that better outcome is expected due to flexibility of soft tissues allowing continued growth and development [27]. A number of studies tend to oppose early surgical intervention in SD for being more challenging due to difficulty in identification of anatomical structures [19, 22, 28]. Moreover, surgical intervention for SD has also been recommended before the ages of 4 [29] and 6 years [30]. The issue relating to determination of optimal age for surgical intervention in SD is further complicated by the fact that it has also been reported that the patient’s age and the presence of omovertebral bone do not influence the surgical outcome in SD [11]
Scapular attachments limiting the movement are either due to the presence of omovertebral bar or simple fibrous bands. Presence of omovertebral bar has been shown not to be involved in limiting the shoulder movement in SD; however, fibrous bands play a critical role in limiting scapulothoracic movements [17]. The fibrous bands run within the rhomboid muscles and when attached to the inferior angle of the scapula, they cause gross mal-rotation which can easily be corrected by division of the fibrous bands [17].On the contrary there is evidence that excision of omovertebral bar improves shoulder abduction significantly [10]. The presence or absence of omovertebral bar in the present study did not show any association with limitation of movements at the shoulder joint. Moreover, no differences were observed in improvement of Cavendish or Regault grades in the presence of omovertebral bar in the present study.
The prevalence of skeletal deformities in this study was 43% and the most common anomalies were congenital scoliosis, Klippel-Feil deformity and the absent or fused ribs among patients with SD. Higher prevalence rates of skeletal deformities among patients with SD ranging between 67 and 100% have previously been reported [10, 31, 32]. It is generally believed that co-existing skeletal anomaly is a pejorative predictive factor. Presence of Klippel-Feil syndrome among patients with SD deformity has been shown to be associated with poor outcome after surgical intervention [33]. Similar observations have also been reported among SD patients with spinal deformities by Khairouni et al. [11]. The findings of the present study, however, revealed that the presence or absence of associated anomalies did no influence the surgical outcome.
Several modifications of the surgical techniques have been proposed for achieving improvements in the functional and cosmetic outcome in SD. These modifications involved application of percutaneous wire or spring system maintained by a plaster jacket [34] and fixation of scapula to the contralateral iliac crest using wires. Whereas the plaster casts proved cumbersome for patients wire breakages also required second procedure in several patients. Ahmad [15] proposed a modification to the Woodward’s procedure by external rotation of scapula with lateral displacement of inferior angle, thereby achieving correction of glenoid vara. This was followed by application of absorbable sutures at the superomedial portion of the scapula at the junction of the upper and middle third by anchoring of sutures to either T11 or T12 vertebrae at an angle of 45°. Functional and cosmetic improvement was reported among all cases in agreement with observations of the present study. In the present study the absorbable sutures were applied close to the center of rotation of the scapula and anchored to the T11 and T12 vertebrae at 30–45°. This double correction associated with immediate postoperative mobilization of scapulothoracic joint may have contributed to improved functional outcome of the shoulder after realignment.
Conclusion
In conclusion the anchoring suture modification of Woodward procedure performed in this study for correction of SD was associated with significant improvement in shoulder abduction, cosmetic appearance and radiological grades over 5 year follow-up. The procedure was devoid of any major complication and the level of parental satisfaction was high.
Acknowledgements
The authors are grateful to the Deanship of Scientific Research, King Saud University, Saudi Arabia, for funding this work through the Voice Deanship Research Chair. I would like to express my deep gratitude to Professor Zahid Shakoor department of immunology for his enthusiastic encouragement and useful critiques of this research work.
Compliance with Ethical Standards
Conflict of interest
The authors declare that they have no conflict of interest.
Ethical standard statement
This article does not contain any studies with human or animal subjects performed by the any of the authors.
Informed consent
For this type of study informed consent is not required.
Footnotes
Publisher's Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Contributor Information
Abdulmonem M. Alsiddiky, Email: alsiddiky@hotmail.com
Zulqurnain Rafiq, Email: drzulqurnain@gmail.com.
Khalid A. Bakarman, Email: Kbakarman@yahoo.com
Fahad S. Alhuzaimi, Email: alhuzaimi@gmail.com
Muhammad Asif, Email: doctorbajwa@gmail.com.
References
- 1.Eulenberg, M. (1863). Casuistsche Mittheilungen aus dem Gebiete der Orthopädie. Langenbecks Arch KlinChir 4, 301–311.
- 2.Sprengel, R.D. (1891). Die angeborenen Vershiebung des Schulterblattes nach oben. Langenbeks Arch KlinChir 42,:5456.
- 3.Dilli A, Ayaz UY, Damar C, Ersan O, Hekimoglu B. Sprengel deformity: magnetic resonance imaging findings in two pediatric cases. Journal Clinical Imaging Science. 2011;1:17–20. doi: 10.4103/2156-7514.76691. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 4.Samilson RL. Congenital and developmental anomalies of the shoulder girdle. Orthopedic Clinics of North America. 1980;11:219–231. doi: 10.1016/S0030-5898(20)31474-7. [DOI] [PubMed] [Google Scholar]
- 5.Mears DC. Partial resection of the scapula and a release of the longhead of triceps for the management of Sprengel’s deformity. Journal of Pediatric Orthopedics. 2001;21:242–245. [PubMed] [Google Scholar]
- 6.McMurtry I, Bennet GC, Bradish C. Osteotomy for congenital elevation of the scapula (Sprenel’s deformity) Journal of Bone and Joint Surgery. British Volume. 2005;87(7):986–989. doi: 10.1302/0301-620X.87B7.16148. [DOI] [PubMed] [Google Scholar]
- 7.Andrault G, Salmeron F, Laville JM. Green’s surgical procedure in Sprengel’s deformity:cosmetic and functional results. Orthopaedics and Traumatology Surgical and Research. 2009;95:330–335. doi: 10.1016/j.otsr.2009.04.015. [DOI] [PubMed] [Google Scholar]
- 8.Aydinli U, Ozturk C, Akesen B, Ozer O. Surgical treatment of Sprengel’s deformity: a modified Green procedure. Acta Orthopaedica Belgica. 2005;71:264–268. [PubMed] [Google Scholar]
- 9.Schrock, R. D. (2008). Congenital elevation of the scapula: Robert D. Schrock MD (1884-1960). The 9th president of the AAOS 1940. Clinical Orthopaedics and Related Research, 466, 74–80. [DOI] [PMC free article] [PubMed]
- 10.Borges JL, Shah A, Torres BC, Bowen JR. Modified Woodward procedure for Sprengel deformity of the shoulder: long-term results. Journal of Pediatric Orthopaedics. 1996;16:508–513. doi: 10.1097/01241398-199607000-00018. [DOI] [PubMed] [Google Scholar]
- 11.Khairouni A, Bensahel H, Csukonyi Z, Desgrippes Y, Pennecot GF. Congenital high scapula. Journal of Pediatric Orthopedics Part B. 2002;11:85–88. doi: 10.1097/00009957-200201000-00014. [DOI] [PubMed] [Google Scholar]
- 12.Woodward JW. Congenital elevation of the scapula: Correction by release and transplantation of muscle origins. Journal of Bone and Joint Surgery American Volume. 1961;43:219–228. doi: 10.2106/00004623-196143020-00010. [DOI] [Google Scholar]
- 13.Cavendish ME. Congenital elevation of the scapula. Journal of Bone and Joint Surgery British Volume. 1972;54:395–408. doi: 10.1302/0301-620X.54B3.395. [DOI] [PubMed] [Google Scholar]
- 14.Rigault P, Puoliquen JC, Guyonvarch G, Zujovic J. Congenital elevation of the scapula in childhood. Revue de chirurgie orthopedique et reparatrice de l'appareil moteur. 1976;62:5–26. [PubMed] [Google Scholar]
- 15.Ahmad A. Surgical correction of severe Sprengel deformity to allow greater postoperative range of shoulder abduction. Journal of Pediatric Orthopedics. 2010;30(6):575–581. doi: 10.1097/BPO.0b013e3181e4f5a6. [DOI] [PubMed] [Google Scholar]
- 16.Green WT. The surgical correction of congenital elevation of the scapula (Sprengel’sdeformity) Journal of Bone and Joint Surgery American Volume. 1957;39:149–151. [Google Scholar]
- 17.Wilkinson JA, Campbell D. Scapula osteotomy for Sprengel’s shoulder. Journal of Bone and Joint Surgery British Volume. 1980;62:486–490. doi: 10.1302/0301-620X.62B4.7430229. [DOI] [PubMed] [Google Scholar]
- 18.Crha B. Surgical treatment of Sprengel’s deformity of the scapula. SCRIPTA MEDICA (BRNO) 2001;74:245–254. [Google Scholar]
- 19.Grogan DP, Stanley EA, Bobechko WP. The congenital undescended scapula. Surgical correction by the Woodward procedure. The Journal of Bone and Joint Surgery British Volume. 1983;65:598–605. doi: 10.1302/0301-620X.65B5.6643564. [DOI] [PubMed] [Google Scholar]
- 20.Jeannopoulos, C. L. (1952). Congenital elevation of the scapula. Journal Bone Joint Surgery. America 34, 883–892. [PubMed]
- 21.Jeannopoulos CL. Observations on congenital elevation of the scapula. Clinical Orthopaedics and Related Research. 1961;20:132–137. [PubMed] [Google Scholar]
- 22.Ross DM, Cruess RL. The surgical correction of congenital elevation of the scapula. A review of seventy-seven cases. Clinical Orthopaedics. 1977;125:17–23. [PubMed] [Google Scholar]
- 23.Carson WG, Lowell WW, Whiteside TE. Congenital elevation of the scapula Surgical correction by the Woodward procedure. Journal of Bone Joint Surgery America. 1981;63A:1199–1207. doi: 10.2106/00004623-198163080-00001. [DOI] [PubMed] [Google Scholar]
- 24.Walstra FE, Alta TD, van der Eijken JW, Willems WJ, Ham SJ. Long-term follow-up of Sprengel’s deformity treated with the Woodward procedure. Journal of Shoulder and Elbow Surgery. 2013;22:752–759. doi: 10.1016/j.jse.2012.08.014. [DOI] [PubMed] [Google Scholar]
- 25.Jindal N, Gupta P. Sprengel’s shoulder treated by the Woodward procedure: analysis of factors affecting functional and cosmetic outcome. Journal of Children's Orthopaedics. 2012;6:291–296. doi: 10.1007/s11832-012-0428-9. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 26.Klisic P, Filipovic M, Uzelac O, Milinkovic Z. Relocation of congenitally elevated scapula. Journal of Pediatric Orthopedics. 1981;1:43–45. doi: 10.1097/01241398-198101010-00006. [DOI] [PubMed] [Google Scholar]
- 27.Robinson RA, Braun RM, Mack P, Zadek R. The surgical importance of theclavicular component of Sprengel's deformity. Journal of Bone and Joint Surgery. American Volume. 1967;49:1481. [Google Scholar]
- 28.Wada A, Nakamura T, Fujii T, Takamura K, Yanagida H, Yamaguchi T, et al. Sprengel Deformity: Morphometric Assessment andSurgical Treatment by the Modified Green Procedure. Pediatric Orthopaedics. 2014;34:55–62. doi: 10.1097/BPO.0b013e318288b407. [DOI] [PubMed] [Google Scholar]
- 29.Gonen E, Simsek U, Solak S, Bektaser B, Ates Y, Aydin E. Long-term results of modified Green method in Sprengel’s deformity. Journal of Children's Orthopaedics. 2010;4:309–314. doi: 10.1007/s11832-010-0265-7. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 30.Dendane AM, Amrani A, El Medhi T, El Alami Z, Gourinda H, Miri A. Congenital elevation of the scapula. La Tunisie Medicale. 2004;82(11):1032–1037. [PubMed] [Google Scholar]
- 31.Greitemann B, Rondhuis JJ, Karbowski A. Treatment of congenital elevation of the scapula: 10 (2–18) year follow-up of 37 cases of Sprengel’s deformity. Acta Orthopaedica Scandinavica. 1993;64(3):365–368. doi: 10.3109/17453679308993646. [DOI] [PubMed] [Google Scholar]
- 32.Zoppi AF, Ferreira AAF, Robazzi PSM, Ferreira AAN, Benegas E, Costa AJF. Deformidade de Sprengel-Revisão de 15 casosoperados. Rev Bras Ortop. 1995;30(9):665–668. [Google Scholar]
- 33.Van Heat AE. Congenital disorders of the hand and upper extremity. Pediatric Clinics of North America. 1996;43–5:1119–1121. doi: 10.1016/s0031-3955(05)70453-3. [DOI] [PubMed] [Google Scholar]
- 34.Farsetti P, Weinstein SL, Caterini R, De Maio F, Ippolito E. Sprengel’s deformity: long-term follow-up study of 22 cases. Journal of Pediatric Orthopedics. Part B. 2003;12(3):202–210. doi: 10.1097/01.bpb.0000049568.52224.1e. [DOI] [PubMed] [Google Scholar]
- 35.da Silva Reginaldo, S., de Macedo, R. R., de Andrade Amaral, R., Cardoso, A. L., Araújo, HR4., Daher, S. (2009). Sprengel's deformity: surgical correction by a modified green procedure. Rev Bras Ortop 44(3), 208–213. [DOI] [PMC free article] [PubMed]