ORIGINAL ARTICLE
Salvage Options in the Cerebral Palsy Hip: A Systematic Review Samuel E. Kolman, MD, Joseph J. Ruzbarsky, MD, David A. Spiegel, MD, and Keith D. Baldwin, MD, MPH, MSPT
Background: No preferred procedure exists for the chronically painful, unreconstructable subluxated or dislocated hip in cerebral palsy. The purpose of this study was to compare pain relief and complication rates of salvage procedures in cerebral palsy for ambulatory and nonambulatory populations. Methods: We searched Medline, Embase, and Cochrane databases using the search terms “cerebral palsy” and “hip dislocation.” Inclusion and exclusion criteria were established to maintain data quality for analysis. A systematic review yielded 28 studies. Relevant information for postoperative pain and complications were extracted from each study and described. Our initial search identified 721 articles. Two hundred twenty duplications were excluded. Five hundred one were screened by title and abstract. One hundred articles underwent further full text and reference evaluation, yielding 25 studies. An additional 3 studies were then identified from the list of 25, yielding a total of 28 studies, which met our inclusion criteria. Results: Among nonambulators, femoral head resection (FHR), valgus osteotomy (VO), and total hip arthroplasty (THA) were found to relieve pain better than arthrodesis [odds ratio (OR) 7.3, 95% confidence interval (CI), 2.2-24.8; OR 5.9, 95% CI, 1.6-22.8; OR 11.7, 95% CI, 1.1-297.5, respectively]. Arthrodesis had a significantly higher complication rate than FHR, VO, THA, and shoulder prosthetic interposition. No significant differences in complication rate were found between FHR and VO. Pain relief rates among nonambulators for FHR, VO, THA, shoulder prosthetic interposition, and arthrodesis were 90.4%, 88.4%, 93.8%, 90.9%, and 56.3%, respectively. Complication rates among nonambulators were 24.0%, 33.3%, 35.3%, 28.6%, and 106.3%, respectively. Comparison of pain relief and complication rates among ambulatory cerebral palsy patients in all procedures except THA was not possible because the populations could not be separated from nonambulators in numbers sufficient to perFrom The Children’s Hospital of Philadelphia, Richard D. Wood Center, Philadelphia, PA. S.E.K. and J.J.R. contributed equally. Sources of support: There were no sources of support for this study. The authors declare no conflicts of interest. Reprints: Keith D. Baldwin, MD, MSPT, MPH, Children’s Hospital of Philadelphia, 34th Street and Civic Center Blvd, Richard D. Wood Center, 2nd Floor, Philadelphia, PA. E-mail: baldwink@email. chop.edu. Supplemental Digital Content is available for this article. Direct URL citations appear in the printed text and are provided in the HTML and PDF versions of this article on the journal’s Website, www. pedorthopaedics.com. Copyright r 2015 Wolters Kluwer Health, Inc. All rights reserved.
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form statistical analysis. Data were available for 32 confirmed cases of THA in ambulators and was associated with a 93.3% pain relief rate and a 38.2% complication rate. Conclusions: Among nonambulators, the available literature suggests that FHR, VO, and THA may be superior at relieving pain than arthrodesis. FHR had the lowest absolute percentage of complications; however, no significant differences in complication rate or pain relief were found in nonambulators undergoing FHR or VO. Most of the complications for VO were implant related, and potentially amenable to hardware removal versus complications in FHR, which were related to the procedure itself such as proximal migration and heterotopic bone formation. THA in nonambulators was associated with complications such as dislocation and revision. Arthrodesis in nonambulators was associated with >100% complication rate and inferior pain relief compared with other procedures. Ambulatory patients had excellent pain relief with THA; however, the complication rate is higher than can be expected with non-neurological populations. Insufficient data exist to support use of other salvage procedures in ambulators. These conclusions should be interpreted with caution as all studies involved level IV evidence. Level of Evidence: IV (systematic review of level IV studies). Key Words: femoral head resection, valgus osteotomy, arthrodesis, total hip arthroplasty, pain relief, ambulators, nonambulators, cerebral palsy, hip dislocation, hip subluxation (J Pediatr Orthop 2015;00:000–000)
H
ip dysplasia and dislocation is common in patients with cerebral palsy (CP), particularly in the nonambulatory population (GMFCS 4-5). It has been estimated that GMFCS V patients have a 90% risk of developing a migration percentage of >33%.1 Progressive neuromuscular hip displacement is often treated by soft tissue or bony reconstructive surgery, with the goal of achieving and maintaining mobile, painless, reduced hips. When this treatment strategy fails, or when reconstructive surgery is precluded by delayed presentation or advanced joint degeneration,2 salvage options include proximal femoral resection-interposition arthroplasty (Castle or Girdlestone procedure), valgus osteotomy (VO) with or without resection of the femoral head and neck, arthrodesis, and total joint replacement or prosthetic interposition.3 The available literature comparing options for salvage is limited and consists mainly of case series of relatively small numbers www.pedorthopaedics.com |
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of patients with short-term to medium-term follow-up without uniformity with regard to management or outcome measures.3 The goal of this systematic review is to compare complication rates and pain relief for these treatment strategies, to provide the clinician with information to provide families when considering these salvage options for their child with CP with the chronically painful dislocated or subluxated unreconstructable hip.
METHODS A literature review was conducted using the Medline, Embase, and Cochrane Database of Systematic Reviews using the terms “cerebral palsy” and “hip dislocation.” Articles were initially screened by title and abstract (J.R., K.D.B.), and those that met our inclusion criteria were evaluated in full text. Our criteria for inclusion in the study included (1) English language, (2) focus on salvage or palliative procedures, (3) reporting or grading of hip pain preoperatively and at follow-up, (4) reporting of complications, and (5) average follow-up >1 year. In cases of disagreement between reviewers, the third reviewer (D.A.S.) was used to break the tie. References from the relevant articles were then scrutinized for inclusion. The literature search yielded in 280 citations in Medline, 16 in the Cochrane Database of Systematic Reviews, and 425 in Embase. Consolidation yielded a total
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of 501 articles. Screening based upon title and abstract yielded 100 articles. Full text review narrowed the field to 25 total articles. Analysis of the references of these remaining 25 articles yielded 12 more relevant studies, 9 of which were excluded due to being written in a non-English language. The final review included 28 studies4–30 (Fig. 1). Overall rates of pain relief were mostly based on proxy assessments from parents or caregivers; however, of the 7 studies that contained some ambulatory patients, at least 4 reported that the patients themselves were able to indicate whether they were in pain.8,18,23,26 The instrument used in pain assessment was not clearly indicated in all studies, but when indicated included either a questionnaire with a pain scale rating from 1 to 10 or a scaled assessment based on degree of grimacing, posturing, or agitation. When applicable, parents and caregivers were used to evaluate and scale the pain. For the purpose of data aggregation and analysis, only study patients who could be identified as ambulatory (functionally ambulatory at a household level or better) versus nonambulatory (nonfunctional ambulators or total care wheelchair-bound patients) were aggregated and analyzed. The patients who could not be separated within a reasonable doubt were included only for descriptive purposes, not for statistical analysis. Complications and pain relief rates of each procedure were aggregated and compared against each other using simple unadjusted odds ratios. The percentage of
FIGURE 1. Flow chart for inclusion and exclusion of studies. This figure outlines the systematic review process used to select studies for inclusion.
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complications was calculated as a function of the number of procedures, whereas pain relief rates were calculated on the basis of those that reported preoperative pain. Significance in comparison was evaluated using either the Pearson w2 or the Fisher exact test. A list of the studies included for each procedure and relevant extracted information can be found in Appendix A (Supplemental Digital Content, http://links.lww.com/BPO/A34). No assessment for publication bias or study quality was possible due to the noncontrolled and noncomparative nature of the member studies, and the level of evidence of the member studies was low (Appendix A, Supplemental Digital Content, http://links.lww.com/BPO/A34).
Salvage Options in the Cerebral Palsy Hip
TABLE 1. Types and Quantities of Complications Sorted by Procedure in Nonambulators
Procedure
Total Complications/ No. Procedures
FHR
75/313
VO
42/126
RESULTS Nonambulatory Patients Studies that distinguished pain relief4–17,20–22,24–26,28,30 and complication types and rates4–17,19–22,24–26,28,30 among confirmed nonambulators were included in the analysis. Pain relief rates for femoral head resection (FHR), VO, total hip arthroplasty (THA), shoulder prosthetic interposition (SPI), and arthrodesis were 90.4%, 88.4%, 93.8%, 90.9%, and 56.3%, respectively. FHR, VO, and THA all had better rates of pain relief compared with arthrodesis [odds ratio (OR) 7.3, 95% confidence interval (CI), 2.2-24.8; OR 5.9, 95% CI, 1.6-22.8; OR 11.7, 95% CI, 1.1-297.5, respectively]. Table 1 lists the types of complications based on procedure. Complication rates for FHR, VO, THA, SPI, and arthrodesis were 24.0%, 33.3%, 35.3%, 28.6%, and 106.3%, respectively. Seventy-five complications occurred in 313 FHR procedures. The most common was proximal femoral migration (7.7%), which required revision 12.5% of the time, followed by heterotopic ossification (3.2%), and pressure ulcer formation (2.6%). Forty-two complications occurred in 126 VO procedures. The most common was symptomatic implants (13.5%), which required removal in 5.9% of cases, followed by infection (5.6%). Six complications occurred in 17 THA procedures that were most commonly revisions (17.6%). Four complications occurred in 14 SPI procedures. The most common was femur fracture (14.3%). Seventeen complications occurred in 16 arthrodeses, most commonly revision for nonunion (25.0%). Patients undergoing arthrodesis had significantly higher complication rates than patients undergoing FHR (P < 0.001), VO (P < 0.001), and THA (P < 0.001). There was no statistically significant difference between the complication rate of FHR and VO, and no other differences in complication rates between procedures were found to be significant.
Ambulatory Patients A description of the pain relief and complications in those studies8,11,18,24,26,27,29,31 that included ambulatory patients can be found in Table 2. With the exception of 4 studies,8,11,24,26 pain relief and complication rates among nonambulators and ambulators could not be distinguished for various procedures, and therefore could Copyright
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THA
6/17
SPI
4/14
Arthrodesis
17/16*
Complications Proximal femoral migrations (24), HO (10), pressure ulcers (8), infections (7), UTIs (6), revisions (4), traction pin loosenings (4), reexcisions (3), pneumonias (2), deaths (2), DVTs (1), reoperation for appositional growth (1), hematoma (1), neuroma (1), femur fracture (1) Hardware removals (17), infections (7), pressure ulcers (3), pneumonias (3), hardware failures (2), femur fractures (2), seromas requiring drainage (2), revisions (1), abduction contracture (1), UTIs (1), pain requiring femoral head resection (1), dislocations (1), fibrous ankylosis of hip (1) Revisions (3), trochanteric bursitis (1), dislocation (1), pressure ulcer (1) Femur fracture (2), broken reamer tip in canal (1), bilateral abduction contractures (1) Revision for nonunion (4), implant loosening (2), femur fracture (2), loss of osteotomy alignment (2), decubitus ulcer (2), subluxation (1), intraop lesion of femoral vein (1), delayed union (1), adventitial bursitis (1), pseudoarthrosis (1)
*There was an average of >1 complication per procedure. DVT indicates deep vein thrombosis; FHR, femoral head resection; HO, heterotopic ossification; SPI, shoulder prosthetic interposition; THA, total hip arthroplasty; UTI, urinary tract infection; VO, valgus osteotomy.
not be compared. Three studies8,24,26 distinguished ambulators in THA. The aggregated data reported a pain relief rate of 93.3%, as well as 13 complications in 34 THA procedures, wherein the most common complication was need for revision (14.7%). One study11 distinguished ambulators in arthrodesis and all 4 patients had postoperative pain relief with no complications.
DISCUSSION Pain in the hip region may be identified in 22% to 56% of patients with CP, especially in nonambulatory patients with greater degrees of neurological impairment.32–35 Identifying the source of pain may be challenging as many patients are nonverbal, and as a result clinicians must rely on the perception of the parents or caregivers.36 The relationship between hip pain and subluxation or dislocation remains elusive, and chronically subluxated or dislocated hips often exhibit radiographic signs of arthritis, including deformation and/or notching of the femoral head, even if they are asymptomatic. In contrast, some patients with reduced hips have pain in the hip region. Hip pain may result from local compression of skin www.pedorthopaedics.com |
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TABLE 2. Description of Pain Relief Rates and Complications in Ambulatory Patients Reference FHR Leet et al18
No. No. Pain Pain Relief Total Patients Hips Preoperative Post Complications 16
23
Wright et al31 VO Leet et al18
12
19
11
13
Wright et al31 THA Buly et al.8 Raphael et al23
16
21
21z
7 56
7 59
Root et al24
9
Schroeder et al26 Skoff and Keggi27 Weber and Cabanela29
Arthrodesis de Moraes Barros Fucs et al11
Unavailable Unavailable
Ambulators/ Nonambulators
11*
15/1w
5*
Unavailable
11*
10/1w
14
5*
Unavailable
7 56
7 56
0 27
7/0 32/27
9
8
7
8
9/0
16
18
15
14
5
16/0
9
12
9
9
3
Unavailable
16
16
16
14
9
12/3y
4
4
4
4
0
4/0
19z
12
Unavailable Unavailable
Complications (Number) Wound dehiscenses (4), pressure ulcers (3), hardware failures (2), infected hardware (1), death unrelated to surgery (1) Revisions (5) Wound dehiscenses (4), pressure ulcers (3), hardware failures (2), infected hardware (1), death unrelated to surgery (1) Revisions (5) NA Revisions (9), dislocations (8), trochanteric bursitis (5), infections (2), trochanteric nonunion (1), pulmonary embolism (1), periprosthetic fracture (1) Trochanteric bursitis (2), femoral component bending (1), migration of greater trochanter (1), revision for subluxation (1), UTI (1), femoral component loosening (1), dislocation (1) Acetabular revisions (3), revision for infection (1), dislocation (1) Osteolysis requiring revision (1), perineal nerve injury (1), trochanteric bursitis (1) Revision for aseptic loosening (1), ho requiring reoperation (1), femur fracture (1), acetabular fracture (1), pressure ulcer (1), ileus (1), UTI (1), reoperation for alvulsed trochanter (1), reoperation for adducter spasticity (1) NA
Pain relief rates and complications were reported for all patients in those studies that did not distinguish between ambulators and nonambulators. *Wright et al31 and Leet et al18 did not distinguish between complications associated with femoral head resection (FHR) and valgus osteotomy (VO). wOne nonambulator was identified but it was not reported whether this patient underwent FHR or VO. zPreoperative pain was assessed on the basis of the number of hips because some patients underwent revisions. yWeber and Cabanela29 reported 1 patient whose ambulatory status was unknown.
or subcutaneous tissues from asymmetric seating, muscle spasticity and/or contracture, joint degeneration, and referred pain from an intra-abdominal or pelvic source.36 The management of progressive hip displacement in patients with CP includes both reconstructive or salvage strategies, and the latter are indicated for a chronically painful subluxated or dislocated hip when reconstructive options are precluded. Salvage options include femoral head and neck resection, VO with or without resection of the femoral head and neck, arthrodesis, and total joint replacement or prosthetic interposition.3 Our systematic review included 722 hips treated by a variety of procedures in both ambulators and nonambulators, including FHR (369, 51.1%), VO (160, 22.2%), THA (139, 19.3%), SPI (32, 4.4%), and arthrodesis (22, 3%). Our analysis was limited by the fact that many studies did not separate ambulators from nonambulators,18,23,27,29,31 and some studies did not separate pain relief by type of procedure performed.18,31 We performed an aggregated analysis on
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the subset of patients for whom separation could be performed. Our aggregated data suggest that no preferred procedure exists for nonambulatory patients with painful chronic subluxated or dislocated hips; however, arthrodesis results in >100% complication rate and a pain relief that is significantly lower than other procedures, and hence cannot be recommended in this group. FHR, VO, and THA are associated with excellent pain relief rates but different complication profiles. FHR is most commonly associated with proximal migration of the femur and heterotopic bone formation. VO is associated with complications mainly related to implants, treated in the respective series by removal of implant. Prosthetic replacement/interposition was most commonly associated with revision, dislocation, and femur fracture (in the case of SPI). Our analysis of ambulatory patients indicated that conclusions could only be drawn in the THA group because it was the only group of ambulators to have a large enough Copyright
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cohort of patients. Our data suggest that THA has an excellent pain relief rate (93.3%), but a higher rate of complications than can be expected in the general population of patients for whom this procedure is performed. Boldingh et al2 recently performed a similar study on palliative hip surgery in severe CP. Their inclusion criteria consisted of all types of palliative surgeries (FHR, total hip replacement, arthrodesis, VO), a diagnosis of CP with GMFCS IV or V, a diagnosis of pelvis/femur relation by radiograph, a stated indication for surgery, articles that explained the surgical procedure and reported on the effect in terms of indication, follow-up of at least 1 year, and articles in English, Dutch, and German. Their results showed that no one procedure was favorable. Eight studies were included in the review, and as such, they were unable to parse out differences between procedures. Our review used broader search criteria, thereby broadening our potential for studies that could be included. We also analyzed ambulatory and nonambulatory patients individually, allowing us to examine each procedure in the context of the patients’ functional capacity. Lastly, we included SPI as a distinct entity from THA in our analysis, which allowed us to analyze associated pain relief and complication rates separately. Limitations of our study include the quantity and quality of studies on which our review is based, all of which provided level IV evidence. In addition, identifying the pain generator and the magnitude of discomfort from “hip pain” is often challenging, as many of the patients do not communicate verbally, and instead require parents and/or caregivers to help provide the history and grade the symptoms. This undermines our primary outcome of hip pain relief. User variability is also a likely contributor to both the pain relief and complication rates of these procedures. Surgeons who are more experienced in performing certain salvage procedures are likely to have better outcomes than those who are less experienced. Therefore, this review may either overestimate or underestimate associated pain relief and complication rates. Prospective, comparative studies would be helpful to assess the true effect of these procedures on pain relief, severity of complication, and complication rate, although these would be difficult to produce owing to the rarity of the procedures even in large centers. In conclusion, identifying the source of hip pain remains a challenge especially in children with greater degrees of neurological impairment, who are often noncommunicative (GMFCS IV and V) and rely on parents and/or caregivers to represent them. Each procedure for salvage surgery for unreconstructable hips in CP represents a good prognosis for pain relief and improved sitting function, and each is associated with unique sets of challenges. On the basis of our findings, we cannot recommend arthrodesis in the nonambulatory population because of poor pain relief and high complication rate with relatively serious complications, although THA in ambulatory patients may be appropriate after a thorough discussion with the patient and patient’s family of risks and benefits. Copyright
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25. Schejbalova A, Havlas V, Trc T. Irreducible dislocation of the hip in cerebral palsy patients treated by Schanz proximal femoral valgus osteotomy. Int Orthop. 2009;33:1713–1717. 26. Schroeder K, Hauck C, Wiedenhofer B, et al. Long-term results of hip arthroplasty in ambulatory patients with cerebral palsy. Int Orthop. 2010;34:335–339. 27. Skoff HD, Keggi K. Total hip replacement in the neuromuscularly impaired. Orthop Rev. 1986;15:154–159. 28. Van Riet A, Moens P. The McHale procedure in the treatment of the painful chronically dislocated hip in adolescents and adults with cerebral palsy. Acta Orthop Belg. 2009;75:181–188. 29. Weber M, Cabanela ME. Total hip arthroplasty in patients with cerebral palsy. Orthopedics. 1999;22:425–427. 30. Widmann RF, Do TT, Doyle SM, et al. Resection arthroplasty of the hip for patients with cerebral palsy: an outcome study. J Pediatr Orthop. 1999;19:805–810.
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31. Wright PB, Ruder J, Birnbaum MA, et al. Outcomes after salvage procedures for the painful dislocated hip in cerebral palsy. J Pediatr Orthop. 2013;33:505–510. 32. Hodgkinson I, Jindrich ML, Duhaut P, et al. Hip pain in 234 non-ambulatory adolescents and young adults with cerebral palsy: a cross-sectional multicentre study. Dev Med Child Neurol. 2001; 43:806–808. 33. Jozwiak M, Harasymczuk P, Koch A, et al. Incidence and risk factors of hip joint pain in children with severe cerebral palsy. Disabil Rehabil. 2011;33:1367–1372. 34. Moreau M, Drummond DS, Rogala E, et al. Natural history of the dislocated hip in spastic cerebral palsy. Dev Med Child Neurol. 1979;21:749–753. 35. Schwartz L, Engel JM, Jensen MP. Pain in persons with cerebral palsy. Arch Phys Med Rehabil. 1999;80:1243–1246. 36. Spiegel DA, Flynn JM. Evaluation and treatment of hip dysplasia in cerebral palsy. Orthop Clin North Am. 2006;37:185–196.
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Salvage Options in the Cerebral Palsy Hip: A Systematic Review Samuel E. Kolman, MD, Joseph J. Ruzbarsky, MD, David A. Spiegel, MD, and Keith D. Baldwin, MD, MPH, MSPT
Background: No preferred procedure exists for the chronically painful, unreconstructable subluxated or dislocated hip in cerebral palsy. The purpose of this study was to compare pain relief and complication rates of salvage procedures in cerebral palsy for ambulatory and nonambulatory populations. Methods: We searched Medline, Embase, and Cochrane databases using the search terms “cerebral palsy” and “hip dislocation.” Inclusion and exclusion criteria were established to maintain data quality for analysis. A systematic review yielded 28 studies. Relevant information for postoperative pain and complications were extracted from each study and described. Our initial search identified 721 articles. Two hundred twenty duplications were excluded. Five hundred one were screened by title and abstract. One hundred articles underwent further full text and reference evaluation, yielding 25 studies. An additional 3 studies were then identified from the list of 25, yielding a total of 28 studies, which met our inclusion criteria. Results: Among nonambulators, femoral head resection (FHR), valgus osteotomy (VO), and total hip arthroplasty (THA) were found to relieve pain better than arthrodesis [odds ratio (OR) 7.3, 95% confidence interval (CI), 2.2-24.8; OR 5.9, 95% CI, 1.6-22.8; OR 11.7, 95% CI, 1.1-297.5, respectively]. Arthrodesis had a significantly higher complication rate than FHR, VO, THA, and shoulder prosthetic interposition. No significant differences in complication rate were found between FHR and VO. Pain relief rates among nonambulators for FHR, VO, THA, shoulder prosthetic interposition, and arthrodesis were 90.4%, 88.4%, 93.8%, 90.9%, and 56.3%, respectively. Complication rates among nonambulators were 24.0%, 33.3%, 35.3%, 28.6%, and 106.3%, respectively. Comparison of pain relief and complication rates among ambulatory cerebral palsy patients in all procedures except THA was not possible because the populations could not be separated from nonambulators in numbers sufficient to perFrom The Children’s Hospital of Philadelphia, Richard D. Wood Center, Philadelphia, PA. S.E.K. and J.J.R. contributed equally. Sources of support: There were no sources of support for this study. The authors declare no conflicts of interest. Reprints: Keith D. Baldwin, MD, MSPT, MPH, Children’s Hospital of Philadelphia, 34th Street and Civic Center Blvd, Richard D. Wood Center, 2nd Floor, Philadelphia, PA. E-mail: baldwink@email. chop.edu. Supplemental Digital Content is available for this article. Direct URL citations appear in the printed text and are provided in the HTML and PDF versions of this article on the journal’s Website, www. pedorthopaedics.com. Copyright r 2015 Wolters Kluwer Health, Inc. All rights reserved.
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form statistical analysis. Data were available for 32 confirmed cases of THA in ambulators and was associated with a 93.3% pain relief rate and a 38.2% complication rate. Conclusions: Among nonambulators, the available literature suggests that FHR, VO, and THA may be superior at relieving pain than arthrodesis. FHR had the lowest absolute percentage of complications; however, no significant differences in complication rate or pain relief were found in nonambulators undergoing FHR or VO. Most of the complications for VO were implant related, and potentially amenable to hardware removal versus complications in FHR, which were related to the procedure itself such as proximal migration and heterotopic bone formation. THA in nonambulators was associated with complications such as dislocation and revision. Arthrodesis in nonambulators was associated with >100% complication rate and inferior pain relief compared with other procedures. Ambulatory patients had excellent pain relief with THA; however, the complication rate is higher than can be expected with non-neurological populations. Insufficient data exist to support use of other salvage procedures in ambulators. These conclusions should be interpreted with caution as all studies involved level IV evidence. Level of Evidence: IV (systematic review of level IV studies). Key Words: femoral head resection, valgus osteotomy, arthrodesis, total hip arthroplasty, pain relief, ambulators, nonambulators, cerebral palsy, hip dislocation, hip subluxation (J Pediatr Orthop 2015;00:000–000)
H
ip dysplasia and dislocation is common in patients with cerebral palsy (CP), particularly in the nonambulatory population (GMFCS 4-5). It has been estimated that GMFCS V patients have a 90% risk of developing a migration percentage of >33%.1 Progressive neuromuscular hip displacement is often treated by soft tissue or bony reconstructive surgery, with the goal of achieving and maintaining mobile, painless, reduced hips. When this treatment strategy fails, or when reconstructive surgery is precluded by delayed presentation or advanced joint degeneration,2 salvage options include proximal femoral resection-interposition arthroplasty (Castle or Girdlestone procedure), valgus osteotomy (VO) with or without resection of the femoral head and neck, arthrodesis, and total joint replacement or prosthetic interposition.3 The available literature comparing options for salvage is limited and consists mainly of case series of relatively small numbers www.pedorthopaedics.com |
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of patients with short-term to medium-term follow-up without uniformity with regard to management or outcome measures.3 The goal of this systematic review is to compare complication rates and pain relief for these treatment strategies, to provide the clinician with information to provide families when considering these salvage options for their child with CP with the chronically painful dislocated or subluxated unreconstructable hip.
METHODS A literature review was conducted using the Medline, Embase, and Cochrane Database of Systematic Reviews using the terms “cerebral palsy” and “hip dislocation.” Articles were initially screened by title and abstract (J.R., K.D.B.), and those that met our inclusion criteria were evaluated in full text. Our criteria for inclusion in the study included (1) English language, (2) focus on salvage or palliative procedures, (3) reporting or grading of hip pain preoperatively and at follow-up, (4) reporting of complications, and (5) average follow-up >1 year. In cases of disagreement between reviewers, the third reviewer (D.A.S.) was used to break the tie. References from the relevant articles were then scrutinized for inclusion. The literature search yielded in 280 citations in Medline, 16 in the Cochrane Database of Systematic Reviews, and 425 in Embase. Consolidation yielded a total
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of 501 articles. Screening based upon title and abstract yielded 100 articles. Full text review narrowed the field to 25 total articles. Analysis of the references of these remaining 25 articles yielded 12 more relevant studies, 9 of which were excluded due to being written in a non-English language. The final review included 28 studies4–30 (Fig. 1). Overall rates of pain relief were mostly based on proxy assessments from parents or caregivers; however, of the 7 studies that contained some ambulatory patients, at least 4 reported that the patients themselves were able to indicate whether they were in pain.8,18,23,26 The instrument used in pain assessment was not clearly indicated in all studies, but when indicated included either a questionnaire with a pain scale rating from 1 to 10 or a scaled assessment based on degree of grimacing, posturing, or agitation. When applicable, parents and caregivers were used to evaluate and scale the pain. For the purpose of data aggregation and analysis, only study patients who could be identified as ambulatory (functionally ambulatory at a household level or better) versus nonambulatory (nonfunctional ambulators or total care wheelchair-bound patients) were aggregated and analyzed. The patients who could not be separated within a reasonable doubt were included only for descriptive purposes, not for statistical analysis. Complications and pain relief rates of each procedure were aggregated and compared against each other using simple unadjusted odds ratios. The percentage of
FIGURE 1. Flow chart for inclusion and exclusion of studies. This figure outlines the systematic review process used to select studies for inclusion.
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complications was calculated as a function of the number of procedures, whereas pain relief rates were calculated on the basis of those that reported preoperative pain. Significance in comparison was evaluated using either the Pearson w2 or the Fisher exact test. A list of the studies included for each procedure and relevant extracted information can be found in Appendix A (Supplemental Digital Content, http://links.lww.com/BPO/A34). No assessment for publication bias or study quality was possible due to the noncontrolled and noncomparative nature of the member studies, and the level of evidence of the member studies was low (Appendix A, Supplemental Digital Content, http://links.lww.com/BPO/A34).
Salvage Options in the Cerebral Palsy Hip
TABLE 1. Types and Quantities of Complications Sorted by Procedure in Nonambulators
Procedure
Total Complications/ No. Procedures
FHR
75/313
VO
42/126
RESULTS Nonambulatory Patients Studies that distinguished pain relief4–17,20–22,24–26,28,30 and complication types and rates4–17,19–22,24–26,28,30 among confirmed nonambulators were included in the analysis. Pain relief rates for femoral head resection (FHR), VO, total hip arthroplasty (THA), shoulder prosthetic interposition (SPI), and arthrodesis were 90.4%, 88.4%, 93.8%, 90.9%, and 56.3%, respectively. FHR, VO, and THA all had better rates of pain relief compared with arthrodesis [odds ratio (OR) 7.3, 95% confidence interval (CI), 2.2-24.8; OR 5.9, 95% CI, 1.6-22.8; OR 11.7, 95% CI, 1.1-297.5, respectively]. Table 1 lists the types of complications based on procedure. Complication rates for FHR, VO, THA, SPI, and arthrodesis were 24.0%, 33.3%, 35.3%, 28.6%, and 106.3%, respectively. Seventy-five complications occurred in 313 FHR procedures. The most common was proximal femoral migration (7.7%), which required revision 12.5% of the time, followed by heterotopic ossification (3.2%), and pressure ulcer formation (2.6%). Forty-two complications occurred in 126 VO procedures. The most common was symptomatic implants (13.5%), which required removal in 5.9% of cases, followed by infection (5.6%). Six complications occurred in 17 THA procedures that were most commonly revisions (17.6%). Four complications occurred in 14 SPI procedures. The most common was femur fracture (14.3%). Seventeen complications occurred in 16 arthrodeses, most commonly revision for nonunion (25.0%). Patients undergoing arthrodesis had significantly higher complication rates than patients undergoing FHR (P < 0.001), VO (P < 0.001), and THA (P < 0.001). There was no statistically significant difference between the complication rate of FHR and VO, and no other differences in complication rates between procedures were found to be significant.
Ambulatory Patients A description of the pain relief and complications in those studies8,11,18,24,26,27,29,31 that included ambulatory patients can be found in Table 2. With the exception of 4 studies,8,11,24,26 pain relief and complication rates among nonambulators and ambulators could not be distinguished for various procedures, and therefore could Copyright
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THA
6/17
SPI
4/14
Arthrodesis
17/16*
Complications Proximal femoral migrations (24), HO (10), pressure ulcers (8), infections (7), UTIs (6), revisions (4), traction pin loosenings (4), reexcisions (3), pneumonias (2), deaths (2), DVTs (1), reoperation for appositional growth (1), hematoma (1), neuroma (1), femur fracture (1) Hardware removals (17), infections (7), pressure ulcers (3), pneumonias (3), hardware failures (2), femur fractures (2), seromas requiring drainage (2), revisions (1), abduction contracture (1), UTIs (1), pain requiring femoral head resection (1), dislocations (1), fibrous ankylosis of hip (1) Revisions (3), trochanteric bursitis (1), dislocation (1), pressure ulcer (1) Femur fracture (2), broken reamer tip in canal (1), bilateral abduction contractures (1) Revision for nonunion (4), implant loosening (2), femur fracture (2), loss of osteotomy alignment (2), decubitus ulcer (2), subluxation (1), intraop lesion of femoral vein (1), delayed union (1), adventitial bursitis (1), pseudoarthrosis (1)
*There was an average of >1 complication per procedure. DVT indicates deep vein thrombosis; FHR, femoral head resection; HO, heterotopic ossification; SPI, shoulder prosthetic interposition; THA, total hip arthroplasty; UTI, urinary tract infection; VO, valgus osteotomy.
not be compared. Three studies8,24,26 distinguished ambulators in THA. The aggregated data reported a pain relief rate of 93.3%, as well as 13 complications in 34 THA procedures, wherein the most common complication was need for revision (14.7%). One study11 distinguished ambulators in arthrodesis and all 4 patients had postoperative pain relief with no complications.
DISCUSSION Pain in the hip region may be identified in 22% to 56% of patients with CP, especially in nonambulatory patients with greater degrees of neurological impairment.32–35 Identifying the source of pain may be challenging as many patients are nonverbal, and as a result clinicians must rely on the perception of the parents or caregivers.36 The relationship between hip pain and subluxation or dislocation remains elusive, and chronically subluxated or dislocated hips often exhibit radiographic signs of arthritis, including deformation and/or notching of the femoral head, even if they are asymptomatic. In contrast, some patients with reduced hips have pain in the hip region. Hip pain may result from local compression of skin www.pedorthopaedics.com |
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TABLE 2. Description of Pain Relief Rates and Complications in Ambulatory Patients Reference FHR Leet et al18
No. No. Pain Pain Relief Total Patients Hips Preoperative Post Complications 16
23
Wright et al31 VO Leet et al18
12
19
11
13
Wright et al31 THA Buly et al.8 Raphael et al23
16
21
21z
7 56
7 59
Root et al24
9
Schroeder et al26 Skoff and Keggi27 Weber and Cabanela29
Arthrodesis de Moraes Barros Fucs et al11
Unavailable Unavailable
Ambulators/ Nonambulators
11*
15/1w
5*
Unavailable
11*
10/1w
14
5*
Unavailable
7 56
7 56
0 27
7/0 32/27
9
8
7
8
9/0
16
18
15
14
5
16/0
9
12
9
9
3
Unavailable
16
16
16
14
9
12/3y
4
4
4
4
0
4/0
19z
12
Unavailable Unavailable
Complications (Number) Wound dehiscenses (4), pressure ulcers (3), hardware failures (2), infected hardware (1), death unrelated to surgery (1) Revisions (5) Wound dehiscenses (4), pressure ulcers (3), hardware failures (2), infected hardware (1), death unrelated to surgery (1) Revisions (5) NA Revisions (9), dislocations (8), trochanteric bursitis (5), infections (2), trochanteric nonunion (1), pulmonary embolism (1), periprosthetic fracture (1) Trochanteric bursitis (2), femoral component bending (1), migration of greater trochanter (1), revision for subluxation (1), UTI (1), femoral component loosening (1), dislocation (1) Acetabular revisions (3), revision for infection (1), dislocation (1) Osteolysis requiring revision (1), perineal nerve injury (1), trochanteric bursitis (1) Revision for aseptic loosening (1), ho requiring reoperation (1), femur fracture (1), acetabular fracture (1), pressure ulcer (1), ileus (1), UTI (1), reoperation for alvulsed trochanter (1), reoperation for adducter spasticity (1) NA
Pain relief rates and complications were reported for all patients in those studies that did not distinguish between ambulators and nonambulators. *Wright et al31 and Leet et al18 did not distinguish between complications associated with femoral head resection (FHR) and valgus osteotomy (VO). wOne nonambulator was identified but it was not reported whether this patient underwent FHR or VO. zPreoperative pain was assessed on the basis of the number of hips because some patients underwent revisions. yWeber and Cabanela29 reported 1 patient whose ambulatory status was unknown.
or subcutaneous tissues from asymmetric seating, muscle spasticity and/or contracture, joint degeneration, and referred pain from an intra-abdominal or pelvic source.36 The management of progressive hip displacement in patients with CP includes both reconstructive or salvage strategies, and the latter are indicated for a chronically painful subluxated or dislocated hip when reconstructive options are precluded. Salvage options include femoral head and neck resection, VO with or without resection of the femoral head and neck, arthrodesis, and total joint replacement or prosthetic interposition.3 Our systematic review included 722 hips treated by a variety of procedures in both ambulators and nonambulators, including FHR (369, 51.1%), VO (160, 22.2%), THA (139, 19.3%), SPI (32, 4.4%), and arthrodesis (22, 3%). Our analysis was limited by the fact that many studies did not separate ambulators from nonambulators,18,23,27,29,31 and some studies did not separate pain relief by type of procedure performed.18,31 We performed an aggregated analysis on
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the subset of patients for whom separation could be performed. Our aggregated data suggest that no preferred procedure exists for nonambulatory patients with painful chronic subluxated or dislocated hips; however, arthrodesis results in >100% complication rate and a pain relief that is significantly lower than other procedures, and hence cannot be recommended in this group. FHR, VO, and THA are associated with excellent pain relief rates but different complication profiles. FHR is most commonly associated with proximal migration of the femur and heterotopic bone formation. VO is associated with complications mainly related to implants, treated in the respective series by removal of implant. Prosthetic replacement/interposition was most commonly associated with revision, dislocation, and femur fracture (in the case of SPI). Our analysis of ambulatory patients indicated that conclusions could only be drawn in the THA group because it was the only group of ambulators to have a large enough Copyright
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cohort of patients. Our data suggest that THA has an excellent pain relief rate (93.3%), but a higher rate of complications than can be expected in the general population of patients for whom this procedure is performed. Boldingh et al2 recently performed a similar study on palliative hip surgery in severe CP. Their inclusion criteria consisted of all types of palliative surgeries (FHR, total hip replacement, arthrodesis, VO), a diagnosis of CP with GMFCS IV or V, a diagnosis of pelvis/femur relation by radiograph, a stated indication for surgery, articles that explained the surgical procedure and reported on the effect in terms of indication, follow-up of at least 1 year, and articles in English, Dutch, and German. Their results showed that no one procedure was favorable. Eight studies were included in the review, and as such, they were unable to parse out differences between procedures. Our review used broader search criteria, thereby broadening our potential for studies that could be included. We also analyzed ambulatory and nonambulatory patients individually, allowing us to examine each procedure in the context of the patients’ functional capacity. Lastly, we included SPI as a distinct entity from THA in our analysis, which allowed us to analyze associated pain relief and complication rates separately. Limitations of our study include the quantity and quality of studies on which our review is based, all of which provided level IV evidence. In addition, identifying the pain generator and the magnitude of discomfort from “hip pain” is often challenging, as many of the patients do not communicate verbally, and instead require parents and/or caregivers to help provide the history and grade the symptoms. This undermines our primary outcome of hip pain relief. User variability is also a likely contributor to both the pain relief and complication rates of these procedures. Surgeons who are more experienced in performing certain salvage procedures are likely to have better outcomes than those who are less experienced. Therefore, this review may either overestimate or underestimate associated pain relief and complication rates. Prospective, comparative studies would be helpful to assess the true effect of these procedures on pain relief, severity of complication, and complication rate, although these would be difficult to produce owing to the rarity of the procedures even in large centers. In conclusion, identifying the source of hip pain remains a challenge especially in children with greater degrees of neurological impairment, who are often noncommunicative (GMFCS IV and V) and rely on parents and/or caregivers to represent them. Each procedure for salvage surgery for unreconstructable hips in CP represents a good prognosis for pain relief and improved sitting function, and each is associated with unique sets of challenges. On the basis of our findings, we cannot recommend arthrodesis in the nonambulatory population because of poor pain relief and high complication rate with relatively serious complications, although THA in ambulatory patients may be appropriate after a thorough discussion with the patient and patient’s family of risks and benefits. Copyright
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25. Schejbalova A, Havlas V, Trc T. Irreducible dislocation of the hip in cerebral palsy patients treated by Schanz proximal femoral valgus osteotomy. Int Orthop. 2009;33:1713–1717. 26. Schroeder K, Hauck C, Wiedenhofer B, et al. Long-term results of hip arthroplasty in ambulatory patients with cerebral palsy. Int Orthop. 2010;34:335–339. 27. Skoff HD, Keggi K. Total hip replacement in the neuromuscularly impaired. Orthop Rev. 1986;15:154–159. 28. Van Riet A, Moens P. The McHale procedure in the treatment of the painful chronically dislocated hip in adolescents and adults with cerebral palsy. Acta Orthop Belg. 2009;75:181–188. 29. Weber M, Cabanela ME. Total hip arthroplasty in patients with cerebral palsy. Orthopedics. 1999;22:425–427. 30. Widmann RF, Do TT, Doyle SM, et al. Resection arthroplasty of the hip for patients with cerebral palsy: an outcome study. J Pediatr Orthop. 1999;19:805–810.
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31. Wright PB, Ruder J, Birnbaum MA, et al. Outcomes after salvage procedures for the painful dislocated hip in cerebral palsy. J Pediatr Orthop. 2013;33:505–510. 32. Hodgkinson I, Jindrich ML, Duhaut P, et al. Hip pain in 234 non-ambulatory adolescents and young adults with cerebral palsy: a cross-sectional multicentre study. Dev Med Child Neurol. 2001; 43:806–808. 33. Jozwiak M, Harasymczuk P, Koch A, et al. Incidence and risk factors of hip joint pain in children with severe cerebral palsy. Disabil Rehabil. 2011;33:1367–1372. 34. Moreau M, Drummond DS, Rogala E, et al. Natural history of the dislocated hip in spastic cerebral palsy. Dev Med Child Neurol. 1979;21:749–753. 35. Schwartz L, Engel JM, Jensen MP. Pain in persons with cerebral palsy. Arch Phys Med Rehabil. 1999;80:1243–1246. 36. Spiegel DA, Flynn JM. Evaluation and treatment of hip dysplasia in cerebral palsy. Orthop Clin North Am. 2006;37:185–196.
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