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ORIGINAL ARTICLE

Operative Versus Nonoperative Treatments for


Legg-Calvé-Perthes Disease: A Meta-Analysis
Nhu-An T. Nguyen, BS,* Guy Klein, DO,w Godwin Dogbey, PhD,* Jessica B. McCourt, PA-C,z
and Charles T. Mehlman, DO, MPHz

had no significant influence on radiographic outcome (OR =


Background: Legg-Calvé-Perthes disease (LCPD) is an idiopathic 1.248; P = 0.486; 95% CI, 0.670-2.325).
avascular necrosis of the femoral head primarily affecting children Conclusions: This meta-analysis suggests that operative treat-
of ages 4 to 12 years. There is no clear consensus on nonoperative ment is more likely to yield a spherical congruent femoral head
or operative treatment protocols for pediatric patients presenting than nonoperative methods among patients 6 years or older.
with LCPD. This study uses meta-analysis and a binary logistic Among patients younger than 6 years, operative and non-
regression model to analyze the radiographic outcomes of these operative methods have the same likelihood to yield a good
treatment modalities in pediatric patients. outcome. Patients who were 6 years or older were treated op-
Methods: Clinical studies describing patients undergoing either eratively, and had the same likelihood of a good radiographic
nonoperative or operative treatment of LCPD published from outcome regardless of treatment with femoral or pelvic proce-
1960 through 2010 were searched electronically and manually. dures. Among patients younger than 6 years, pelvic procedures
Eligible studies consisted of (1) a minimum of 10 patients; (2) were more likely to result in a good radiographic outcome than
listed age at the time of diagnosis or treatment; (3) performed an femoral procedures.
initial severity assessment using the Herring or Catterall classi- Level of Evidence: Level IV meta-analysis.
fication; (4) detailed the type of intervention; and (5) reassess-
ment of radiographic outcome after a minimum of 1 year after Key Words: Perthes, operative, nonoperative
treatment using the Mose or Stulberg classification. (J Pediatr Orthop 2012;32:697–705)
Results: Twenty-three studies, 1232 patients, and 1266 hips met
the inclusion criteria. Among patients younger than 6 years, op-
erative and nonoperative treatments are equally as likely to
results in a successful radiographic outcome [odds ratio (OR) =
1.071; P = 0.828; 95% confidence interval (CI), 7.377-32.937]. In
L egg-Calvé-Perthes disease (LCPD) is an idiopathic
pediatric hip disorder associated with avascular ne-
crosis of the femoral head. The resulting hip deformity
patients older than 6 years, operative treatment is nearly twice as may lead to problems ranging from mild limp to severe
likely to result in a successful radiographic outcome (OR = 1.754; debilitating arthritis. LCPD occurs most commonly
P < 0.0001; 95% CI, 1.299-2.370). For age at treatment less than among children of 4 to 12 years of age with 4 to 5 times
6 years, a patient treated with a pelvic rather than femoral more males than females being affected.1 Although there
procedure was approximately 5 times as likely to have a good are fewer female cases, some studies have found a worse
radiographic outcome (w2 = 4.488; P = 0.034; unadjusted prognosis in females presumably because their bones have
OR = 5.20; 95% CI, 1.021-26.471). Among patients ages 6 or less time to grow and remodel as a result of early puberty
older, pelvic procedures were equally as likely as femoral proce- compared with their male peers.2–5 The potential influ-
dures to yield a successful radiographic outcome (w2 = 1.845; ence of sex on outcome is one of many unanswered
P = 0.174; unadjusted OR = 1.329; 95% CI, 0.881-2.004). Sex questions related to LCPD.
LCPD is the most common form of pediatric hip
disease. The annual incidence of LCPD cases range from
2 to 294 per 1,000,000 children.6 There is no single ex-
From the *Ohio University College of Osteopathic Medicine, Athens;
planation for the wide range of incidence of LCPD
wOrthopaedic Surgery Residency Program, University Hospitals, cases; however, there is similarity in clinical presentation
Richmond Heights; and zDepartment of Pediatric Orthopaedic among children afflicted with this disease. Some of the
Surgery, Cincinnati Children’s Hospital Medical Center, Cincinnati, possible explanations given for the wide incidence include
OH. genetic predisposition, environmental exposures, and so-
Supported in part by the University of Cincinnati Orthopaedic Research
and Education Fund and Division of Pediatric Orthopaedic Surgery cioeconomic factors.6 The most noticeable symptom in a
at Cincinnati Children’s Hospital Medical Center, Cincinnati, OH. LCPD patient is a mildly discomforting limp, which can
The authors declare no conflict of interest. become progressively more painful with increasing phys-
Reprints: Charles T. Mehlman, DO, MPH, Department of Pediatric ical activity. The hip joint mobility is restricted in ab-
Orthopaedic Surgery, 45229 Cincinnati Children’s Hospital Medical
Center, 3333 Burnet Avenue MLC 2017, 45229 Cincinnati, OH.
duction and internal rotation.7 A diagnosis of LCPD is
E-mail: charles.mehlman@cchmc.org. made with a lateral and anteroposterior x-ray view of the
Copyright r 2012 by Lippincott Williams & Wilkins hip and femoral head. Initial severity of the disease is

J Pediatr Orthop ! Volume 32, Number 7, October/November 2012 www.pedorthopaedics.com | 697


Nguyen et al J Pediatr Orthop ! Volume 32, Number 7, October/November 2012

assessed by radiographic methods using the Catterall or text articles were reviewed to identify additional studies
the Herring system involving necrosis in the femoral head, that potentially met the inclusion criteria.
whereas final assessment is graded with the Mose or
Stulberg classification. Study Selection
Although discovered over 100 years ago, the precise Inclusion Criteria
etiology of LCPD remains unclear. In some cases, the Studies that met the inclusion criteria for the meta-
disease has the capacity to resolve on its own, but for analysis had the following characteristics: (1) a minimum
many other patients the long-term prognosis may be of 10 patients; (2) initial radiographs documenting se-
substantially worse because of residual hip deformities. verity at the time of diagnosis using the Catterall or
Many patients develop osteoarthritis as early as their Herring classification; (3) a minimum of 1-year radio-
mid-40s.1,8–10 As a result, multiple nonoperative and op- graphic follow-up using the Mose or Stulberg criteria; (4)
erative treatment methods have been performed with the age of patients listed at the time of diagnosis or at the
goal of altering the natural history of the disease. time of treatment; and (5) the type of intervention used.
Multiple studies have evaluated nonoperative and
operative treatments with variable conclusions. This lack Exclusion Criteria
of consensus stems from the small sample size and vari- Excluded studies from our analysis had the follow-
ability between studies in the age of LCPD study pop- ing characteristics: (1) the outcomes of predicted meas-
ulation and severity of femoral head involvement upon ures were not fully discernable and data were impossible
presentation. In addition, the lack of intraobserver or to pool and analyze; (2) the combination of Z2 operative
interobserver reliability in classifying radiographic stages techniques not conventionally used but only reserved as
has weakened the ability to draw consensus opinions salvage technique; and/or (3) the patient population
from the LCPD literature.11–13 The primary aim of this consisted of other hip diseases not limited to LCPD.
meta-analysis was to compare outcomes of nonoperative
and operative treatments for LCPD. An additional goal Data Extraction
was to determine if long-term prognosis was predicted by Two reviewers (N.T.N. and J.M.) independently
age, sex, or disease severity at initial presentation. assessed the level of evidence of the included studies using
the guidelines of the Oxford Centre for Evidence Based
Medicine to determine the level of evidence of the study.
METHODS Data that were independently extracted from the selected
studies included: number of patients, number of affected
Search Strategy hips, sex, average and range of age at diagnosis, follow-up
A literature search was conducted using PubMed age, side of affected hip, type of treatment, and final
MEDLINE (1960 to August 2010), Science Citation radiographic outcome.
Index (1965 to August 2010), and The Cochrane Library Discrepancies of the extracted data and eligibility of
databases (1960 to August 2010) aimed at identifying studies for inclusion were resolved by discussion with a
studies examining LCPD treatment results. The following third reviewer (C.T.M.). Nine authors were contacted to
terms were used: “Perthes” OR “Coxa Plana” and furnish additional information regarding their published
“treatment” OR “non-operative” OR “conservative” OR results. Three additional studies were included for anal-
“surgery.” Only full-text articles published in the English ysis through this method.
language were considered. We searched for any current or
past clinical trials on LCPD on http://www.clinicaltrials.gov, Disease Severity Measures
current controlled trials (http://www.controlled-trials.com), Patients were grouped as having mild or severe
http://www.asiaclinicaltrials.org, and the World Health disease presentation. Under the Herring classification,
Organization portal of clinical trials (http://www.who.int/ those individuals with class A or B radiographs repre-
trialsearch). In addition, abstracts from the following sented mild progression of the disease, whereas those with
annual conferences were searched: European Paediatric B/C or C were grouped in the more severe stage.14 For
Orthopaedic Society (2006 to 2010), Pediatric Orthopae- studies using the Catterall classification, type 1 or 2 rep-
dic Society of North America (2007 to 2010), and resented a mild presentation, whereas type 3 or 4 depicted
American Academy of Orthopaedic Surgeons (2008 to a more severe presentation.1
2010). Bibliographies of 3 major pediatric orthopaedic
textbooks were screened for relevant studies identified Outcome Measures
through chapters related to LCPD (Pediatric Orthopedic The primary data gathered from the studies con-
Deformities by Frederic Shapiro, Lovell and Winter’s sisted of radiographic outcome defined by the Mose or
Pediatric Orthopaedics by Morrissy and Weinstein, and Stulberg classification. To pool the data for statistical
Tachdjian’s Pediatric Orthopaedics by Herring). Titles and analyses, the radiographic outcomes were dichotomized
abstracts that were deemed relevant to this review, were as good or poor. Under the Mose criteria, a good out-
retrieved as full-text articles to be considered in the come consists of a spherical femoral head that has 0 mm
analysis. Authors were contacted in instances when ad- in deviation of concentric circles between the frog-leg
ditional data were needed. References from selected full- lateral and anteroposterior views, whereas a fair outcome

698 | www.pedorthopaedics.com r 2012 Lippincott Williams & Wilkins


J Pediatr Orthop ! Volume 32, Number 7, October/November 2012 Operative Versus Nonoperative Treatments

has <2 mm in deviation. In contrast, a poor outcome intervals (CIs) were obtained. A sensitivity analysis was
comprises a nonspherical femoral head with variability performed on all eligible studies that used hip spica as the
>2 mm between the 2 projected radiographic views.15 sole nonoperative technique or Chiari pelvic osteotomy
A good radiographic outcome under a Stulberg I or II (CPO) as the operative treatment modality. This was
classification is defined by a spherical femoral head that carried out to assess whether or not hip spica casting
has a normal hip joint or has a larger femoral head, influenced the overall analysis and change any of the
shorter femoral neck, or steep acetabulum (Table 1).8 possible predictor variables in measuring outcome varia-
Poor outcomes correlate with radiographic grades of bles as the method does not allow molding of the femoral
Stulberg III, IV, or V that demonstrate a nonspherical head within the acetabulum because of lack of weight
femoral head in conjunction with abnormal features in bearing compared with other nonoperative treatment
the femoral neck and acetabulum. Radiographs demon- methods. Patients who are selected for CPO tended to
strating the different stages of Stulberg classification are have a severe disease profile (Catterall 3 and 4), thus a
shown in Figure 1. Consistent with previous studies that sensitivity analysis was performed to detect whether or
utilized the Stulberg or Mose classifications to assess ra- not exclusion of the treatment modality would alter any
diographic outcome, a Stulberg I or II grade is equivalent of the possible predictor variables to be nonsignificant in
to a good or fair Mose rating and was categorized as a the overall analysis.
good outcome in our analysis, whereas Stulberg III-V was A 6-year age threshold was chosen as a cut-off for
equated to a poor Mose rating and categorized as a poor the pediatric population, based on previous studies that
outcome (Table 1).8,14,16–23 have demonstrated that children who present with LCPD
before the age of 6 fared better in radiographic outcomes
Statistical Methods than those who acquire the disease at a later age.24–26
Meta-analysis was conducted using Comprehensive Operative treatments were categorized as: “femoral,”
Meta-Analysis v2.0 (Biostat, Englewood, NJ). Confidence “pelvic,” or a “combined procedure.” A “femoral” pro-
limits were set at 95% and data were pooled in a random cedure consisted of femoral varus osteotomy (FVO), a
effect model. In addition, statistical analysis using a bi- “pelvic” procedure of an innominate osteotomy (IO),
nary logistic regression model was performed with shelf acetabuloplasty/arthroplasty, or CPO, whereas a
PASW/SPSS v.18 (Predictive Analytic Software/Stat- “combined” procedure used both femoral and pelvic
istical Package for the Social Sciences) (Chicago, IL) to procedures on the same affected hip.
analyze the pooled patient-level data. The binary logistic To measure heterogeneity between the studies, Com-
regression model was used with radiologic outcome prehensive Meta-Analysis was used to calculate the I2 sta-
(good/poor), derived from the Stulberg and Mose classi- tistic. An I2 value of 0%, 25%, 50%, and 75% represents 0,
fications, as the dichotomous outcome variable. Four low, moderate, and high heterogeneity, respectively.27
predictor variables were used: sex, age at treatment (di-
chotomized into less than 6 y or 6 y and older), initial RESULTS
radiographic status defined by Herring and Caterall cri-
teria (dichotomized into mild or severe), and treatment Literature Search
(dichotomized into operative or nonoperative). The use of Our electronic search strategy resulted in 2069 ar-
binary logistic regression afforded us the opportunity to ticles, of which 88 studies were deemed relevant for full-
pool and use patient-level data from the studies in de- text evaluation. Six additional studies were found from
termining predictors of the dichotomized radiographic textbook references. Out of the 94 studies, 23 met the
outcomes. Odds ratios (OR) along with 95% confidence inclusion criteria (Fig. 2). One included study28 reported

TABLE 1. Description of Stulberg and Mose Classification


Stulberg Class Features Femoral Head Mose Prognosis
I Normal hip joint Spherical congruency Good Good
II Spherical femoral head with Z1 abnormalities: Spherical congruency Fair Good
coxa magna
shorten femoral neck
steep acetabulum
III Nonspherical but not flat femoral head with Ovoid, mushroom shaped Poor Poor—slower onset of
Z1 abnormalities: osteoarthritis
coxa magna
shorten femoral neck
steep acetabulum
IV Flat femoral head and abnormal femoral head, Flat Poor Poor—moderate onset of
neck, and acetabulum osteoarthritis
V Flat femoral head with normal femoral neck and Flat Poor Poor—early onset of
acetabulum osteoarthritis
Adapted from Stulberg et al.8

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Nguyen et al J Pediatr Orthop ! Volume 32, Number 7, October/November 2012

FIGURE 1. Frog-leg radiographs of Stulberg I to V. A and B represent Stulberg I and II (good results). C, D, and E represent
Stulberg III, IV, and V (poor results).

some of the same patients included in another long-term comes with other hip conditions, 3 studies had <10 pa-
paper10; data from these 2 studies were extracted to yield tients reported, and 3 did not use the Catterall or Herring
the latest results without including the same patients in classification as an initial assessment.
the analysis twice.
Of the excluded studies, 28 did not report radio- Population Characteristics
graphic outcome using Mose or Stulberg criteria, 24 did Twelve of the 23 eligible studies evaluated radio-
not report age at diagnosis or at treatment, 8 studies in- graphic outcomes at skeletal maturity.3,4,10,16,19–21,29–33
volved >1 treatment procedures where outcomes were Radiographic assessment using Mose or Stulberg criteria of
not differentiated by treatment, 5 studies described out- LCPD patients ranged from 1 to 64 years after treatment.

Citation
Identified
electronically
(n=2,069)

Article
considered for Citation
Full Text Identifed
Review manually
(n=94) (n=6)

Studies Excluded:
Did not use Mose/Stulberg (n=28)
Did not use Catterall/Herring (n=3)
Less than 10 patients (n=3)
Missing age of diagnosis/treatment (n=24)
Other concurrent hip disorders (n=5)
Treatment not specified (n=8)

Total Studies
included in
Meta-Analysis
(n=23)

FIGURE 2. Study selection process identifying eligible studies.

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J Pediatr Orthop ! Volume 32, Number 7, October/November 2012 Operative Versus Nonoperative Treatments

Ten studies evaluated operative procedures,18,20,23,30,31,33–37 1.299-2.370). Patients with mild disease severity were twice
7 studies examined nonoperative treatment,4,10,22,24,28,29,32 as likely as those with severe disease presentation to have a
and 6 studies compared operative and nonoperative treat- good radiographic outcome than a poor outcome among
ments.3,16,19,21,38,39 All studies included in the meta-analysis patients 6 years or older (OR = 2.014; P < 0.0001; 95%
were retrospective in design. CI, 1.427-3.842).
The sample size of eligible studies ranged from 10 to Furthermore, controlling for sex, treatment, and
358 patients, which yielded 1232 patients and 1266 hips age, patients with lesser rather than greater disease se-
for analysis. A total of 783 hips were treated non- verity were twice as likely to have a good rather than poor
operatively and 483 hips were treated operatively. At the outcome (OR = 2.036; P = 0.001; 95% CI, 1.326-3.132).
time of initial evaluation, patients ranged from 1.3 A patient having a good or bad outcome was not de-
through 15.2 years old. Among studies that listed the side pendent on or associated with sex. Males were equally as
of affected hip, the ratio distribution of left to right was likely as females to have good or bad outcomes regardless
165:183. For studies that detailed patients’ sex, there were of age, treatment, and initial disease severity (OR =
599 (87%) male and 91 (13%) female patients. Table 2 1.248; P = 0.486; 95% CI, 0.670-2.325).
describes the population characteristics of the included Additional results from subgroup analysis using the
studies. w2 test of association indicated a statistically significant
relationship between surgical types [femoral (FVO)/pelvic
Statistical Analysis (shelf, IO, or CPO) procedures] and the radiologic out-
Meta-analysis based on study-level data examining comes for patients at age less than 6. For age at treatment
treatment type (operative or nonoperative) was performed less than 6 years, a patient treated with pelvic rather than
on the eligible 23 studies. This subgroup analysis of treat- a femoral procedure was about 5 times as likely to have
ment type compared radiographic outcome among 783 hips good radiographic outcomes (w2 = 4.488; P = 0.034; un-
treated with nonoperative methods to 483 hips that un- adjusted OR = 5.20; 95% CI, 1.021-26.471). For patients
derwent operative treatment. The success rate of treatment aged 6 or older, pelvic procedures were equally as likely as
for operative treatment was 51.7%, whereas nonoperative femoral procedures to yield a successful radiographic
treatment had a success rate of 52.2% in restoring femoral outcome (w2 = 1.845; P = 0.174; unadjusted OR = 1.329;
head sphericity (Fig. 3). Without taking into account the 95% CI, 0.881-2.004).
effects of any other variables, such as sex, age, or disease A sensitivity analysis was performed on studies that
severity, the analysis revealed no significant differences be- included hip spica casting or CPO as treatment modality
tween the 2 treatment types (P = 0.678). The I2 values for to assess whether or not the inclusion or exclusion of
operative and nonoperative treatment were 75% and 91%, these methods would affect the other variables in pre-
respectively, thus indicating high heterogeneity across the dicting radiographic outcomes. Using cases that excluded
studies. With such high levels of variability across and treatment with a hip spica cast in the sensitivity analysis;
within the studies, further statistical analysis was performed both Herring (P = 0.008) and Caterall (P = 0.027) clas-
to incorporate the effects of multiple variables through sifications significantly predicted radiographic outcomes
regression models. regardless of treatment. Thus, the inclusion or exclusion
The results from the logistic regression analyses in- of hip spica cases did not alter the result where initial
dicated that after controlling for age, sex, and disease severity predictors (Herring/Catterall) were still sig-
severity, patients who had operative treatment were about nificant as measures of disease severity in predicting
twice as likely as those who had nonoperative treatment outcome. Similarly, excluding CPO cases, initial severity
to have a good outcome (OR = 2.01; P = 0.001; 95% CI, measures [Herring (P = 0.002) and Caterall (P = 0.025)]
1.309-3.09). Similarly, after adjusting for sex, disease se- were significant predictors of outcome. This suggested
verity, and treatment type, patients of age less than 6 that our results were robust with or without CPO as
years were almost 4 times as likely as those 6 years or treatment modality when Herring or Caterrall was used
older to have a good radiographic outcome (OR = 3.882; as initial disease severity measures.
P < 0.0001; 95% CI, 2.026-7.36).
Controlling for treatment type, patients less than 6
years old with mild disease severity were 15 times as likely DISCUSSION
to have a good radiographic outcome (OR = 15.564; Children diagnosed with LCPD have an increased
P < 0.0001; 95% CI, 7.377-32.937) than their counterparts risk in developing debilitating osteoarthritis by early
with severe disease condition. When disease severity was adulthood. To prevent or delay the onset of osteo-
controlled for, patients younger than six years did not have arthritis, the goal of nonoperative or operative treatment
any difference in radiographic outcomes when operative has focused on restoring a spherical femoral head.
treatment was compared with nonoperative treatment However, despite the plethora of literature on non-
(OR = 1.071; P = 0.828; 95% CI, 0.582-1.968). Con- operative and operative methods in treating LCPD, there
versely, adjusting for disease severity among patients age 6 still exists a lack of consensus among studies regarding
or older, operative treatment was almost twice as likely to the risk factors that influence patient outcomes and the
yield a good radiographic outcome in comparison with optimal treatment of the condition. Without taking into
nonoperative treatment (OR = 1.754; P < 0.0001; 95% CI, account any risk factors, our pooled meta-analysis of

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Nguyen et al J Pediatr Orthop ! Volume 32, Number 7, October/November 2012

TABLE 2. Study Population Characteristics


Range Mean
Onset Age Length Range
Mean Age Range of Mean Age Treated Follow-up Follow-up No. No. Good Female
References Intervention Onset (y) Age (y) Treated (y) (y) (y) (y) Patients Hips Outcome (%)
Alves and Nonoperative 6.8 4-11.0 — — 19 10-33.0 43 49 27 19
Santili35
Arkader et al16 Nonoperative, 10.8 9-14.0 — — 10 5-22.0 43 43 7 9.30
FVO, CPO
Crutcher and FVO 7.6 4.0-10.5 8.33 4.5-10.8 8 5-10.5 14 14 7 14.29
Staheli18
Ghanem et al36 Shelf, FVO 7.5 6.2-11.0 8.6 7.1-13.0 9.5 5.2-12 30 30 19 26.67
Gower and Nonoperative 8.3 2-15.0 8.9 2.9-14 44.6 30-48 36 37 — 13.89
Johnston28w
Ippolito et al29 Nonoperative 7.5 2.4-15.0 — — 25 7-56 61 61 25 11.48
Kamegaya FVO, 9.2 6.25-11.25 — — — 16-20* 36 36 17 0.00
et al19 nonoperative
Kamegaya FVO 8.4 4.1-12.1 8.7 4.4-12.2 18.5* SM 37 38 20 8.11
et al30
Kelly et al4 Nonoperative 6.3 2-12.0 — — 22.4 SM 80 80 73 —
Kim et al22 Nonoperative 7.1 3.5-11.1 — — 7.8 6.3-10.8 20 20 17 15.00
Kruse et al38 Nonoperative 7 4.0-10.0 — — 28 7-45 17 18 11 13.89
Shelf 8 2.0-14.0 11 7-15.0 19 2-47 19 20 14
McAndrew and Nonoperative 8.2 4.7-15.0 — — 47.7 39-64 28 28 14 14.29
Weinstein10w
Noonan et al20 FVO — — 10.6 9.5-13.9 10 4.2-17.8 17 18 6 29.41
Oh et al37 Shelf — — 8.7 6-12.0 5 3-14.0 20 20 8 20.00
Osman et al3 Nonoperative, 9.75 8-14.3 — — 6.4 2.2-20 44 48 9 18.18
FVO, shelf
Pecquery et al34 Shelf 7.2 2.9-12.4 8 4.5-13.0 4.25 1.0-8.75 21 21 14 9.52
Reddy and CPO — — 8.5 6.0-11.0 6.1 — 21 22 15 19.05
Morin31
Robinson et al32 IO 6.25 2.6-12.2 8 4.8-13.2 8.2 5.0-16.3 25 25 23 7.41
Rosenfeld et al24 Nonoperative 4.6 2-5.9 — — — > 1.0 172 188 152 —
Segev et al33 Arthrodiatasis — — 12.3 9.4-15.1 5.7 4.3-7.8 10 10 0 23.08
Sharma et al21 Nonoperative, 6.75 4-8.0 — — — SM 28 28 7 14.29
FVO, shelf,
IO
Sponseller et al23 FVO, IO 7.2 3.5-13.2 — — 8.9 5-13 88 91 62 —
Wiig et al39 Nonoperative, 5.8 1.3-15.2 — — — > 5.0 358 358 176 —
FVO
*Mean age range at follow-up.
wGower and McAndrew studies had overlapping patients.
CPO indicates Chiari pelvic osteotomy; FVO, femoral varus osteotomy; IO, innominate osteotomy; SM, skeletal maturity.

1266 hips found that both operative and nonoperative methods. In contrast, patients age 6 or older who were
procedures had the same likelihood of obtaining a good treated nonoperatively fared worse than those treated
result. However, because of the high level of heterogeneity with surgery. This finding is consistent with previous
across the studies and the need to assess the influence of studies that suggested that nonoperative treatment yields
multiple risk factors that may influence the outcome of a favorable outcome among younger patients with mild
patient with LCPD, a binary logistic regression model presentation of the disease, but ineffective especially
was performed to examine which modality of treatment among older patients with known risk factors for poor
yielded a more superior result among nonoperative and prognosis.24,41–44
operative procedures. Nonoperative treatment of LCPD Studies that have examined outcomes after surgical
has a tractable history and long tradition. The pioneer treatments, such as FVO, IO, shelf acetabuloplasty/
physicians—Legg, Calvé, and Perthes, all treated their arthroplasty, and Chiari pelvic osteomy have found better
patients nonoperatively. These treatment options in- results for their patients in comparison with those who
cluded: no treatment except symptom management, bed were treated nonoperatively among both the age
rest, range of motion therapy, weight relief (splints, groups.3,16,21,39,45 Our meta-analysis revealed an associa-
traction, crutches), or containment of the femoral had tion between radiographic outcomes and the type of
using abduction splint or Petrie casting.40 This meta- procedure performed, especially among patients with
analysis found that patients who are younger than 6 years early-onset LCPD. In a subgroup analysis with regard to
had the same likelihood in obtaining a good radio- operative procedures and age, we found that patients
graphic outcome with either nonoperative or operative younger than 6 years old, treatment with a pelvic procedure

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J Pediatr Orthop ! Volume 32, Number 7, October/November 2012 Operative Versus Nonoperative Treatments

Study Subgroup within study Event Event rate and 95% CI


rate
Successful Outcomes /
Total Subjects

Alves Non-Operative 27 / 49 0.55


Arkader Non-Operative 3 / 21 0.14
Ippolito Non-Operative 25 / 61 0.41
Kamegaya Non-Operative 4 / 18 0.22
Kelly Non-Operative 73 / 80 0.91
Kim Non-Operative 17 / 20 0.85
Kruse Non-Operative 13 / 18 0.72
McAndrew Non-Operative 14 / 28 0.50
Osman Non-Operative 4 / 26 0.15
Rosenfeld Non-Operative 152 / 188 0.81
Sharma Non-Operative 3 / 14 0.21
Wiig Non-Operative 134 / 260 0.52
Pooled Estimate of Non-Operative Treatment 469 / 783 0.52
Arkader Operative 4 / 22 0.18
Kamegaya Operative 20 / 38 0.53
Kruse Operative 14 / 20 0.70
Osman Operative 5 / 22 0.23
Sharma Operative 4 / 14 0.29
Wiig Operative 42 / 98 0.43
Crutcher Operative 7 / 14 0.50
Ghanem Operative 19 / 30 0.63
Kamegaya Operative 13 / 18 0.72
Noonan Operative 6 / 18 0.33
Oh Operative 8 / 20 0.40
Pecquery Operative 14 / 21 0.67
Reddy Operative 15 / 22 0.68
Robinson Operative 23 / 25 0.92
Segev Operative 0 / 10 0.05
Sponseller Operative 62 / 91 0.68
Pooled Estimate of Operative Treatment 256 / 483 0.52
0.00 0.50 1.00

FIGURE 3. Effect of operative and nonoperative treatment based on radiographic outcomes. Without taking into consideration
any other modulating variables, the subgroup indicated that operative and nonoperative treatment yielded successful radiograph
outcomes at the rate of 51.7% and 52.2%, respectively. A high level of heterogeneity was found at the study-level data analysis.
CI indicates confidence interval.

was 5 times as likely to result in a spherical femoral head as their spherical shape and remain congruent within the
treatment with a femoral procedure. Pelvic procedures ap- acetabulum.
pear to provide better shelf coverage and containment than Although males are more prone to develop LCPD,
femoral techniques, thus allowing the femoral head to be the influence of sex on outcome has been a topic of dis-
molded within the acetabulum.16 However, among patients agreement in the literature. Some studies suggest a female
6 years or older, pelvic procedures had the same likeli- sex predisposed the patient to a worse prognosis,2–5
hood of yielding a good radiographic outcome as femoral whereas others found that sex did not influence out-
procedures. comes.24,39,47 In this meta-analysis, males outnumbered
This meta-analysis strongly suggests that patients female patients at a rate of 6.5 to 1. However, when the
who present with LCPD at an early age younger than 6, influence of age, disease severity, and treatment methods
tended to have a more favorable outcome than those di- were controlled in our analysis, sex was not a significant
agnosed at a later age. Previous studies have suggested predictor of long-term radiographic outcome.
that this may be because of greater time available for the This meta-analysis focused on radiographic out-
femoral head to remodel before skeletal maturity is comes. The studies that met our inclusion criteria did not
reached.10,21,46 We also found that the initial stage of report clinical outcomes in a standardized manner that
disease presentation is another significant predictor of would enable data pooling. With regard to radiographic
long-term radiographic outcome. Patients with a milder outcome, femoral heads that are more nonspherical and
form of LCPD at the time of diagnosis and treatment incongruent at skeletal maturity are at a higher risk for
have significantly better radiographic outcomes that those developing debilitating osteoarthritis.8,48–50 In a long-
with a more severe involvement. Femoral heads that were term study, Stulberg and colleagues reported a correlation
not as deformed at initial presentation tended to retain between clinical and radiographic outcome among LCPD

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Nguyen et al J Pediatr Orthop ! Volume 32, Number 7, October/November 2012

patients. Stulberg et al8 found that hips categorized as was equivalent for femoral and pelvic procedures. We
Stulberg I and II developed osteoarthritis at a low rate strongly advocate early diagnosis and treatment of pa-
ranging between 0% to 7%, whereas those belonging to tients who present with LCPD as delay in treatment may
Stulberg III, IV, and V have a higher rate of degenerative decrease the likelihood of a favorable outcome.
joint disease with 36%, 67%, and 81%, respectively.
These evaluations at the end of bone remodeling suggest a
good correlation between radiographic outcome and the ACKNOWLEDGMENTS
clinical risk associated with degenerative joint disease. The authors thank Tony Herring, MD, Alex Arkader,
We advocate for a more standardized system to MD, and Makoto Kamegaya, MD for generously providing
grade clinical outcomes and functional mobility in the additional data on published studies. The authors also thank
long-term follow-up of LCPD patients. Until such a Debra Orr, MSS, MLIS and Douglas Mann, PhD for
system has been developed, the evaluation of treatment assistance in preparing the manuscript.
efficacy will continue to focus on the systematic grading
of radiographs.
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