ORIGINAL ARTICLE
Hip Reconstruction in Children With Unilateral Cerebral Palsy and Hip Dysplasia Oussama Abousamra, MD, Mehmet S. Er, MD, Kenneth J. Rogers, PhD, ATC, Tristan Nishnianidze, MD, Kirk W. Dabney, MD, and Freeman Miller, MD
Background: Highly functioning children with unilateral cerebral palsy (CP) who have hip involvement (type IV hemiplegia) may present with hip dysplasia during their adolescence. The aim of this report is to assess the outcomes of combined femoral and acetabular reconstruction in this population. Methods: This study is a retrospective review of all patients with unilateral CP, Gross Motor Function Classification System types I and II, who had hip reconstruction for unilateral dysplasia between 1989 and 2013. Clinical variables (pain and hip passive range of motion) were reviewed. Hip morphology was assessed radiographically according to Melbourne Cerebral Palsy Hip Classification System. Three-dimensional gait analyses were also reviewed to evaluate the effect of surgery on these patients’ gaits. Results: Twelve patients were included with a mean age at surgery of 14 years (range, 7 to 19 y) and follow-up mean of 4 years (range, 1 to 8 y). Nine hips were improved according to Melbourne Cerebral Palsy Hip Classification System. Migration percentage decreased significantly (P < 0.001) from 45% (30% to 86%) to 15% (0% to 28%). Neck shaft angle decreased (P < 0.001) from 144 degrees (range, 129 to 156 degrees) to 125 degrees (range, 114 to 139 degrees). Tonnis angle and Sharp angle also decreased significantly. All patients were pain free at the last visit. Overall level of gait function as measured by Gait Deviation Index and Gait Profile Score [78 (61 to 89) and 12 (8 to 16), respectively] for all patients was maintained without significant changes. Conclusions: In hemiplegic type IV CP, with high functional level (Gross Motor Function Classification System I and II), hip dysplasia is a rare occurrence during adolescent years. Combined hip reconstruction improves hip morphology, relieves pain, and maintains a high level of function. Level of Evidence: Level IV—therapeutic study. Key Words: cerebral palsy, hemiplegic, hip dysplasia (J Pediatr Orthop 2015;00:000–000)
From the Department of Orthopedics, Nemours/Alfred I. duPont Hospital for Children, Wilmington, DE. The authors declare no conflicts of interest. Reprints: Freeman Miller, MD, Department of Orthopedics, Nemours/ Alfred I. duPont Hospital for Children, 1600 Rockland Road, Wilmington, DE 19803. E-mail:
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hildren with cerebral palsy (CP) frequently develop hip displacement with variation in the approach and management.1 Hip dysplasia has variable severity and progression of the deformity in children and adolescents with CP.2 During fetal life, the hip develops normally,2 and displacement is believed to be caused by abnormal joint reaction forces, which lead to further subluxation and possible dislocation.1,3 The incidence of hip displacement is most directly related to the gross motor function and age of the child as defined by the Gross Motor Function Classification System (GMFCS).2,4–6 Population-based studies show that one third of children with CP have some level of hip displacement; however, it is rare to find significant displacement in highly functioning GMFCS I and II level children.4,6,7 Many papers discuss hip displacement in GMFCS IV and V, nonambulatory children with CP1,4,6; however, there is only 1 report treating severe hip dysplasia in GMFCS I and II functional level children.5 In population-based studies, the prevalence of hip dysplasia among children with spastic unilateral hip involvement was found to be 10%; however, these studies reported mild subluxation identified on screening.5,8 Most of the reported surgery for children with unilateral CP focuses on gait improvement after unilateral multilevel reconstructions.5,9 The goal of this study is to report a series of children with unilateral CP and highlevel physical function (GMFCS I and II) with severe hip dysplasia. The outcome of the hip reconstruction is recorded including the effect on pain relief, gait, and radiographic changes.
METHODS This retrospective study reviewed the records of 12 patients with CP who were classified as type I or II according to the GMFCS,10 indicating that they are able to walk independently indoors and outdoors without assistive devices (full community ambulators), and who had a unilateral combined femoral and acetabular reconstruction at our hospital between 1989 and 2013. Data were collected after obtaining approval from our institutional review board. Surgical indications in these patients were radiographic evidence of acetabular dysplasia and hip subluxation with or without hip pain. The procedures were
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TABLE 1. Patients’ Demographics and Soft-Tissue Procedures That Were Done Simultaneously With Hip Reconstruction Patient No.
Sex
Age at Surgery (y)
Side
Preoperative Pain
GMFCS
Simultaneous Soft-Tissue Procedures
1 2
M M
15.4 12.9
R L
Yes Yes
1 2
3 4 5
M M M
17.5 11 13.1
R L L
Yes No Yes
1 2 2
6 7 8 9 10
F M M M F
19 6.9 17.9 15.8 14.9
L R R R R
Yes No Yes Yes Yes
2 2 2 1 1
11 12
M M
11.6 16.9
L R
No Yes
2 2
ham, add, gastroc ham, tibialis posterior transfer, peroneus brevis advancement ham, add add ham, gastroc, rectus transfer, subtalar fusion, Akin procedure, tibial osteotomy, talonaviculocuneiform fusion, flexor hallucis longus lengthening add, gastroc, calacaneal lengthening None add, ham, rectus transfer, gastroc, knee capsulotomy gastroc, calacaneal lengthening, Akin procedure ham, calcaneal lengthening, Achilles lengthening, Akin procedure, tibialis posterior advancement None rectus transfer, ham, gastroc, subtalar fusion, tibialis anterior transfer, Akin procedure, talonavicular fusion, tibialis posterior advancement
add indicates adductors lengthening; gastroc, gastrocnemius lengthening; GMFCS, Gross Motor Function Classification System; ham, hamstrings lengthening; L, left; R, right.
performed by the 2 senior authors. Soft-tissue contractures were assessed and dealt with specifically for each case. Combined femoral and acetabular bony reconstruction was done in the same manner for all patients. A femoral varus osteotomy was performed using the Arbeitsgemeinschaft fur osteosynthesefragen blade plate for femoral osteotomy fixation as described in previous studies11,12 aiming for a 120-degree neck shaft angle (NSA). The acetabular osteotomy was peri-ilial as previously described.12,13 This osteotomy cuts posteromedially toward the center of the triradiate cartilage eventually cutting both iliac tables down to the triradiate. The osteotomy site can be easily hinged open with the osteotome in the most posterior aspect of it. For the skeletally mature patients, the peri-ilial osteotomy is directed down to the center of the radiographic teardrop. To make the hinging easy, the osteotomy starts with a bicortical cut just superior to the anterior inferior iliac spine and continues as a unicortical cut. When levered open, the osteotomy often produces a greenstick fracture in the inferior third of the acetabulum below the weightbearing surface and it might end up extending into the medial wall of the acetabulum with an intra-articular component. Postoperatively, sitting and physical therapy for hip and knee range of motion were started on the
second day after surgery. Patients were encouraged to stand and bear weight as tolerated with a walker before hospital discharge by postoperative day 3 or 4. No immobilization of the hip was used. We reviewed the clinical charts, radiographs, and gait analyses before the surgery and at the most recent follow-up visit. Physical examination findings were recorded to evaluate the changes in hip joint passive range of motion and to note any complications the patients had during follow-up. To assess radiographic outcomes and effects of reconstruction on hip morphology, anteroposterior pelvic radiographs were reviewed. We classified the hips according to the Melbourne Cerebral Palsy Hip Classification System,14,15 which describes hip pathology around skeletal maturity. It is a valid and reliable 6-grade system based on migration percentage of Reimers (MP),16 Shenton’s line disruption, femoral head deformity, acetabular deformity, and pelvic obliquity.15 We also assessed acetabular roof orientation and acetabular inclination using Tonnis angle and the acetabular angle of Sharp.17–19 As the surgical procedure included a varus osteotomy of the proximal femur, changes in the NSA were also calculated on the anteroposterior pelvic radiographs.17
TABLE 2. Hip Passive Range of Motion Preoperatively and at the Last Visit Preoperative Abduction Internal rotation External rotation Flexion Extension
Last Visit
Mean
Range
Mean
Range
Change (%)
P
25 64 25 105 7
10 to 45 38 to 90 5 to 62 90 to 120 35 to 5
30 47 31 106 3
10 to 50 30 to 67 5 to 60 70 to 120 15 to 10
+19 27 +23 +1 66
0.218 0.004 0.346 0.780 0.238
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Hip Subluxation in Hemiplegic CP
TABLE 3. Temporal Spatial Gait Parameters Preoperatively and at the Last Visit Preoperative Step length (cm) Stride length (cm) Forward velocity (cm/s) Cadence (steps/min) Total support time (%)
Last Visit
Mean
Range
Mean
Range
Change (%)
P
44 84 82 115 58
27-57 56-117 39-115 84-134 53-64
44 86 79 111 58
31-57 61-115 52-112 91-128 53-65
0 +2 3 3 0
0.991 0.756 0.636 0.408 0.839
Instrumented 3-dimensional gait analyses for all patients were done in our hospital. Data that we collected for our study were temporal spatial parameters, kinematic sagittal, coronal, and transverse parameters at the level of pelvis and hip, in addition to foot progression angle. Gait Deviation Index (GDI) and Gait Profile Score (GPS) are closely related measures of overall gait pathology.20,21 Changes in these measures that were related to index surgery were recorded. We compared preoperative and postoperative range of motion and radiographic and gait parameters using paired t test. We used SPSS software (version 22; SPSS Inc., Chicago, IL).
RESULTS Twelve patients with spastic unilateral CP were included, 10 boys and 2 girls with a mean age of 14.3 years (range, 7 to 19 y). Mean follow-up after surgery was 4 years (range, 1 to 8 y). Eight patients were classified as level II on GMFCS and 4 were level I. All of them were full community ambulators with no assistive devices. Seven patients had involvement on their right sides and 5 patients on the left. There were simultaneous softtissue procedures done on the same limb in 10 cases (Table 1). A previous multilevel surgery on the same limb had been performed in 7 cases. This previous surgery included femoral reconstruction (varus derotational osteotomy) without acetabular intervention. During followup, 3 patients needed an additional adductor release. Otherwise, and apart from hardware removal, none of our patients had further surgery on the extremity between the reconstructions and last follow-up. Nine patients presented with hip pain. At the last visit, all 12 patients denied any pain in their involved hips. After surgery, hip passive range of motion in all move-
ment directions became closer to normal. However, these movement changes were not statistically significant except for internal rotation decrease (Table 2). We compared gait analyses results preoperatively and postoperatively. There were no statistically significant changes in temporal spatial parameters and kinematic parameters at the hip and pelvis nor were there significant changes in GDI and GPS (Tables 3 to 5). Radiographically, all hips were grade IV according to Melbourne Cerebral Palsy Hip Classification System. Most hips were in grade III at the last visit. Shenton’s line was disrupted in all hips preoperatively, and it was intact in all of them at the last follow-up visit. Radiographically, femoral head deformity improved in 4 hips and remained the same in 8. However, acetabular deformity improved in 11 hips and remained the same in only 1 hip (Table 6). The MP decreased significantly (P < 0.001) from preoperative mean of 45% (range, 30% to 86%) to last visit mean of 15% (range, 0% to 28%). The NSA decreased significantly (P < 0.001) from 143 degrees preoperatively (range, 129 to 156 degrees) to 124 degrees (range, 114 to 139 degrees) at last visit. There were also significant changes in the acetabular angles of Tonnis and Sharp. The Tonnis angle mean decreased from 23.42 degrees (range, 15 to 35 degrees) to 12.25 degrees (range, 3 to 19 degrees), and the Sharp angle mean decreased from 53.41 degrees (range, 42 to 61 degrees) to 43.58 degrees (range, 35 to 52 degrees). In contrast, pelvic obliquity measurements in these patients did not change significantly (Table 7). In our population, there were no postoperative infections, hardware failures, or nonunions. Seven patients had some degree of limb-length discrepancy after surgery, ranging from 1 to 3 cm with a mean of 1.7 cm. In 1 case, in which the discrepancy was 3 cm, the patient needed an
TABLE 4. Kinematic Parameters, Sagittal, Coronal, and Transverse Parameters at the Level of Pelvis and Hip and Foot Orientation Angle Preoperative Pelvic tilt (maximum stance) Pelvic rotation (maximum stance) Hip flexion angle (maximum stance) Hip rotation angle (average stance) Hip adduction angle (average stance) Foot orientation (average foot contact)
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Last Visit
Mean
Range
Mean
Range
Change (%)
P
19 1 38 19 5 0
11 to 29 18 to 8 29 to 47 11 to 38 5 to 14 29 to 38
20 2 42 15 2 6
5 to 33 13 to 12 31 to 55 15 to 74 13 to 10 29 to 3
+5 +300 +11 21 60 600
0.743 0.277 0.103 0.478 0.171 0.449
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TABLE 5. GDI and GPS Preoperatively and at the Last Visit Normal Values GDI GPS
100 ± 10 5±2
Preoperative
Last Visit
Mean Range Mean Range 78 12
61-89 8-16
75 11
Change (%)
53-107 9-15
4 8
GDI indicates Gait Deviation Index21; GPS, Gait Profile Score.20
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TABLE 7. Radiographic Measurements Preoperatively and at the Last Visit Preoperative
P 0.294 0.092
Last Visit
Mean Range Mean Range Migration (%) Pelvic obliquity Neck shaft angle Tonnis angle Sharp angle
45 5 144 23 53
30-86 2-13 129156 15-35 42-61
15 4 125 12 44
0-28 1-9 114139 3-19 35-52
Change (%)
P
67 28 13
< 0.001 0.271 < 0.001
48 19
< 0.001 < 0.001
epiphysiodesis on the contralateral extremity to reduce the limb-length inequality.
DISCUSSION It is widely accepted that hip displacement is rare in highly functioning patients with hemiplegic CP.4,5,7,22 Therefore, hip surveillance programs have not recommended ongoing radiologic surveillance for this group of patients.4,22,23 However, it is not clear from previous studies how common hip dysplasia is among children who are classified as having hemiplegia type IV according to Winters classification for spastic hemiplegia24 indicating that they have motor dysfunction of the entire lower limb including a restricted motion of the hip. It has been reported as high as 10%5,8; however, this is mild dysplasia in young children. The current reported severe dysplasia is usually not recognized until late when symptoms occur in adolescence, which is also consistent with our series.5 It is hard to calculate the incidence from our series as most of these patients were referred when the hip was already symptomatic. On the basis of our number of patients we followed, our gross estimate is that one in 25 to 50 children with GMFCS I or II and type IV hemiplegia develop severe hip subluxation. For all children with hip subluxation due to CP who require hip reconstruction, this
TABLE 6. Radiographic Hip Deformities According to MCPHCS Preoperatively and at the Last Visit No. Patients MCPHCS
Preoperatively
Last Visit
0 0 0 12
1 1 7 3
0 4 8
12 0 0
0 8 4
4 5 3
0 5 7
9 2 1
Grade 1 Grade 2 Grade 3 Grade 4 Shenton’s line Intact Broken 5 mm Femoral head deformity Round head Mildly flattened Variable deformity Acetabular deformity Normal acetabulum Mildly dysplastic Variable deformity
MCPHCS indicates Melbourne Cerebral Palsy Hip Classification System.
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group represents between 1% and 2% of the total. Although it is rare, it has a large effect on a child who has close to normal gross motor function. On the basis of this, we believe that the use of screening radiographs every 2 years from age 6 through adolescence has merit for early identification. Three of our patients (patients no. 4, 7, and 11 in Table 1) had the reconstruction before adolescent age. This might be due to the awareness that has been raised as the first few cases of hemiplegic late hip dysplasia were encountered in our practice. As a result, children with unilateral CP and hip motion involvement (Winters hemiplegia type IV) started having their hips radiographically surveilled. Therefore, early diagnosis of hip dysplasia became possible before the patients present with hip pain (as listed in Table 1, these 3 children did not develop hip pain preoperatively). At the time of reconstruction, these 3 children did not have muscle contractures severe enough to indicate a multilevel surgery, so they had fewer simultaneous surgeries than the other children. However, one of them (patient no. 4) had previous soft-tissue releases and many of the other patients had previous soft-tissue releases at earlier ages. Unfortunately, no clear explanation can describe this finding based on the 2 cases mentioned above. Although only 2 female patients were encountered in our population, no definitive conclusion could be drawn due to the small sample size. Moreover, in the previous study that reported a similar population,5 there were 5 girls of 11 patients. This indicates that no definitive statement can be currently based on this finding. In our sample, 7 patients had previous femoral osteotomy at a mean age of 7 years (Table 8), and it did not prevent the development of hip dysplasia. Their acetabuli, however, were not felt to have significant dysplasia at the time of primary surgery to require reconstruction. This finding supports what was reported previously5 and confirms that femoral varus derotation did not prevent the development of dysplasia and subluxation during adolescent growth. Mean age at reconstruction for these 7 patients who had a previous femoral osteotomy (15.2 y) was not significantly different than the mean age at reconstruction for the other 5 patients who did not have a previous femoral osteotomy (13.3 y) (P = 0.4). To our knowledge, acetabular reconstruction results have not been previously reported in this specific Copyright
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M M M M F M F
All bony and soft-tissue procedures before the combined hip reconstruction (index surgery) were recorded. Patients were assigned the same numbers as in Table 1. Surgeries before hip reconstruction were performed either with the previous femoral osteotomy or at a different time. The number within parentheses indicates the age at which listed surgery was performed. Achilles indicates Achilles tendon lengthening; gastroc, gastrocnemius lengthening; ham, hamstrings lengthening; rectus, rectus transfer.
ham, gastroc (6) None None None None
Surgeries Before Hip Reconstruction Sex
M M M M M 5 7 8 11 12
Patient No. Surgeries Before Hip Reconstruction Sex Patient No.
1 2 3 4 6 9 10
Age at Previous Osteotomy (y)
gastroc (5) ham, gastroc, rectus (6) ham, gastroc (8) ham, gastroc (4); rectus, tibial osteotomy (7) ham, gastroc, rectus (10); add (16) Achilles, ham (6) ham, gastroc (7)
5 Patients
8 6 8 7 7 6 7
Children Without a Previous Femoral Osteotomy
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Children With a Previous Femoral Osteotomy
TABLE 8. Two Groups of Children, With and Without a Previous Femoral Osteotomy
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population. However, many studies reported significant pain reduction after acetabular reconstruction in children with CP and more severe involvement.12,25,26 Our findings are consistent with these previous reports; however, on the basis of the high functional level of our patients, these hips are not likely to wear normally, and we warn the patients that they should expect eventual arthritis and will likely need total hip replacement. From a functional standpoint, overall gait measures using GDI and GPS20,21 did not change from preoperative level. As the goal of surgery was not to improve gait, we believe that keeping the same high level of function postoperatively is a good result itself and confirms that this reconstruction did not have negative effects on these patients’ gaits. In previous studies,5,9 significant improvement in gait was reported; we had similar postoperative results with different preoperative starting points. We had significant improvement in hip internal rotation only, which was similarly reported in other studies.5,9 Improvements in hip morphology were frequently reported after combined femoral and acetabular reconstruction.12,13,25 In addition to changes in radiographic measurements, and similar to what was reported before,12 we had changes in femoral head deformity toward a more normal shape in 4 cases. In our study, these 4 patients were the youngest in the group with a mean age of 11 years. On the basis of this, we can address a potential for deformity remodeling after acetabuloplasty at young ages, supporting a similar finding in a recent report5 and supporting the authors’ recommendations for a systematic radiographic follow-up and aggressive hip dysplasia management in this specific population as early as clear dysplasia is recognized. In the same paper,5 pelvic obliquity was reported to be always high on the affected side despite limb shortening. We had the same findings in all our patients. There was no significant change in obliquity measurements at the last follow-up as it remained high on the affected side. It was suggested that spasticity plays a role in pelvic obliquity, which itself may contribute to the etiology of the hip dysplasia.5,27 The limitation of this study is the small sample size that spans over 24 years of a large CP practice. We cannot determine the potential long-term outcomes such as development of degenerative arthritis and recurrence of dysplasia, as this will require follow-up to mid-life. In summary, this study presents the results of unilateral combined hip reconstruction in highly functioning patients with hemiplegic CP. After surgery, these patients kept their high level of function with improved hip morphology and stayed pain free throughout the mid-term follow-up. In conclusion, severe hip dysplasia in highly functioning children with type IV hemiplegic CP is rare. Normal early screening and early femoral reconstruction do not prevent the risk of hip pathology in adolescence. Hip reconstruction can maintain ambulatory function, reduce the hip dysplasia, and relieve the pain (Fig. 1).
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FIGURE 1. Patient no. 9 in Table 1. This boy was followed from age 2 until age 6 when his gait pattern had reached a plateau. He was at GMFCS level I of function with a type IV hemiplegic gait pattern, toe walking, increased knee flexion in stance, and internal rotation throughout the whole gait cycle. A and B, At this time, he had a femoral osteotomy to correct his right femoral internal torsion, and he had gastrocnemius and hamstrings lengthening. There was no significant acetabular dysplasia and, therefore, no further hip radiographs were taken. C, Ten years later, he presented with hip pain and radiographic hip dysplasia. Hip reconstruction was indicated. D, Hip radiograph after the reconstruction with femoral varus osteotomy and peri-ilial pelvic osteotomy. E, Hip radiograph 4 years after the reconstruction, and the patient is pain free. His right lower limb was 1 cm shorter than his left with a shoe lift needed to resolve this discrepancy. Preoperatively, GDI was 86.33 and GPS was 9.18. Postoperatively, GDI was 105.80 and GPS was 8.70. He plays tennis at the high school competitive level, during which time he sustained a metatarsal stress fracture without any hip pain. GDI indicates Gait Deviation Index; GMFCS, Gross Motor Function Classification System; GPS, Gait Profile Score.
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16. Reimers J. The stability of the hip in children. A radiological study of the results of muscle surgery in cerebral palsy. Acta Orthop Scand Suppl. 1980;184:1–100. 17. Goldstein RY, Kaye ID, Slover J, et al. Hip dysplasia in the skeletally mature patient. Bull Hosp Jt Dis (2013). 2014;72: 28–42. 18. Laborie LB, Engesaeter IO, Lehmann TG, et al. Radiographic measurements of hip dysplasia at skeletal maturity—new reference intervals based on 2,038 19-year-old Norwegians. Skeletal Radiol. 2013;42:925–935. 19. Tonnis D, Brunken D. Differentiation of normal and pathological acetabular roof angle in the diagnosis of hip dysplasia. Evaluation of 2294 acetabular roof angles of hip joints in children. Arch Orthop Unfallchir. 1968;64:197–228. German. 20. Baker R, McGinley JL, Schwartz MH, et al. The Gait Profile Score and movement analysis profile. Gait Posture. 2009;30:265–269. 21. Schwartz MH, Rozumalski A. The Gait Deviation Index: a new comprehensive index of gait pathology. Gait Posture. 2008;28:351–357. 22. Dobson F, Boyd RN, Parrott J, et al. Hip surveillance in children with cerebral palsy. Impact on the surgical management of spastic hip disease. J Bone Joint Surg Br. 2002;84:720–726. 23. Robb JE, Hagglund G. Hip surveillance and management of the displaced hip in cerebral palsy. J Child Orthop. 2013;7:407–413. 24. Winters TF Jr, Gage JR, Hicks R. Gait patterns in spastic hemiplegia in children and young adults. J Bone Joint Surg Am. 1987;69:437–441. 25. Al-Ghadir M, Masquijo JJ, Guerra LA, et al. Combined femoral and pelvic osteotomies versus femoral osteotomy alone in the treatment of hip dysplasia in children with cerebral palsy. J Pediatr Orthop. 2009;29:779–783. 26. Sankar WN, Spiegel DA, Gregg JR, et al. Long-term follow-up after one-stage reconstruction of dislocated hips in patients with cerebral palsy. J Pediatr Orthop. 2006;26:1–7. 27. Black BE, Hildebrand R, Sponseller PD, et al. Hip dysplasia in spastic cerebral palsy. Contemp Orthop. 1994;29:101–108.
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