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Is the modified Ponseti method effective in treating atypical and complex clubfoot? A systematic review

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PurposeIn 2006, Ponseti modified the standard technique to treat cases of “atypical” and “complex” clubfoot. To determine the outcomes of Ponseti’s modified method to treat complex idiopathic clubfoot patients, we asked the following: (1) What is the deformity correction success rate? (2) What is the relapse rate after the correction? (3) What is the incidence of complications?Materials and methodsWe performed a systematic review by searching the EMBASE, MEDLINE, Cochrane Library, and Web of Science databases from inception to March 1, 2021. All studies on idiopathic, complex, and atypical clubfoot that assessed Ponseti’s modified technique were included. Of 699 identified articles, ten met the inclusion criteria. The mean index for non-randomized studies score for the included studies was 11.8 ± 1.7.ResultsEarly detection of the deformity and modifying the standard protocol, as described by Ponseti, resulted in a high rate of success. Initial correction occurred in all children, with a mean ankle dorsiflexion of 15°. Relapse occurred often ranging between 10.5 and 55%. The incidence of complications associated with the modified Ponseti method ranged from 6 to 30%.Conclusions Studies using the modified Ponseti technique have shown high initial correction rates and a smaller number of relapses. However, studies with prospective designs and long-term follow-up are required to conclude whether these observations are due to properly performing the modified method or if higher rates of relapse increase with further follow-up.
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International Orthopaedics
https://doi.org/10.1007/s00264-021-05092-4
REVIEW ARTICLE
Is themodified Ponseti method effective intreating atypical
andcomplex clubfoot? Asystematic review
OmarA.Al‑Mohrej1,2 · FawazN.Alshaalan3· ThamerS.Alhussainan3
Received: 3 May 2021 / Accepted: 24 May 2021
© SICOT aisbl 2021
Abstract
Purpose In 2006, Ponseti modified the standard technique to treat cases of “atypical” and “complex” clubfoot. To deter-
mine the outcomes of Ponseti’s modified method to treat complex idiopathic clubfoot patients, we asked the following: (1)
What is the deformity correction success rate? (2) What is the relapse rate after the correction? (3) What is the incidence
of complications?
Materials and methods We performed a systematic review by searching the EMBASE, MEDLINE, Cochrane Library, and
Web of Science databases from inception to March 1, 2021. All studies on idiopathic, complex, and atypical clubfoot that
assessed Ponseti’s modified technique were included. Of 699 identified articles, ten met the inclusion criteria. The mean
index for non-randomized studies score for the included studies was 11.8 ± 1.7.
Results Early detection of the deformity and modifying the standard protocol, as described by Ponseti, resulted in a high rate
of success. Initial correction occurred in all children, with a mean ankle dorsiflexion of 15°. Relapse occurred often ranging
between 10.5 and 55%. The incidence of complications associated with the modified Ponseti method ranged from 6 to 30%.
Conclusions Studies using the modified Ponseti technique have shown high initial correction rates and a smaller number of
relapses. However, studies with prospective designs and long-term follow-up are required to conclude whether these obser-
vations are due to properly performing the modified method or if higher rates of relapse increase with further follow-up.
Keywords Clubfoot· Idiopathic· Atypical· Complex· Modified Ponseti· Relapse· Systematic review
Introduction
Since the emergence of the Ponseti method, the idiopathic
clubfoot surgical release rate has decreased significantly [1,
2]. Idiopathic clubfoot is a group of deformities that involve
the hindfoot varus, cavus, metatarsus adductus, and equi-
nus. The estimated incidence of idiopathic clubfoot ranges
from 0.5 to 2 per 1000 live births [3]. Within the idiopathic
clubfoot designation, “atypical” and “complex” clubfeet
have been identified [4]. Atypical idiopathic clubfeet are
distinguished by significant features that are not present in
“simple” idiopathic clubfeet. These include severe plantar
flexion of all metatarsals, a rigid equinus with a deep crease
above the heel, and a deep plantar crease across the full
width of the sole of the foot with high cavus and a short
hyperextended big toe [5, 6]. The exact incidence of atypical
clubfoot remains unclear, ranging from 6.5 to 21% accord-
ing to various studies [7, 8]. As for complex clubfoot, it
has been postulated that improper casting of predisposed
feet can convert a typical clubfoot into a one that resembles
the features of atypical idiopathic clubfoot termed “complex
iatrogenic clubfoot” [10].
The aetiology of atypical clubfeet remains inadequately
understood. Nevertheless, shortening and tightness of the
gastrocsoleus muscles and fibrosis of the deep plantar
intrinsic muscles of foot are attributable for this deformity
[9]. Moreover, risk factors for complex deformity include
Level of evidence: Level III
* Omar A. Al-Mohrej
mohrejo@gmail.com
1 Division ofOrthopaedics, Department ofSurgery, McMaster
University, Hamilton, ON, Canada
2 Section ofOrthopedic Surgery, Department ofSurgery,
King Abdullah Bin Abdulaziz University Hospital,
Princess Nourah Bint Abdul Rahman University, Riyadh,
SaudiArabia
3 Department ofOrthopedic Surgery, King Faisal Specialist
Hospital andResearch Centre, Riyadh, SaudiArabia
International Orthopaedics
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a severe clubfoot based on the Pirani classification system,
which includes a short, stubby clubfoot. Furthermore, many
of the complex clubfeet cases appear to be iatrogenic, occur-
ring after the typical idiopathic clubfoot slips in its stretch-
ing casts [10].
Many clinicians, including Ponseti, have attempted vari-
ous methods to treat complex clubfoot. In 2006, Ponseti
modified his standard technique [5]. In comparison to the
standard Ponseti method, plantar flexion of the metatarsals
is corrected by holding the ankle with both hands while the
thumbs are placed under the metatarsals, which are pushed
dorsally to correct the forefoot plantar flexion. Furthermore,
to avoid slippage of the cast, the knee is flexed at least 110
degrees in comparison to the usual 90 degrees [5].
The number of studies regarding complex and atypical
idiopathic clubfoot has increased rapidly since Ponseti modi-
fied his technique to treat these subgroups, and most have
shown consistent success in initial correction rates [5, 916].
However, most of the available literature reports wide varia-
tions in relapse and complication rates that are not defined in
a consistent fashion, which may limit appropriate treatment
decisions [11]. Moreover, in most of the available litera-
ture, including the original paper by Ponseti, there seems
to be a lack of clear differentiation between the two sub-
groups which might be the main reason for such discrepan-
cies in recurrence/complications. Therefore, we performed
a systematic review to determine the outcomes of Ponseti’s
modified method to treat complex iatrogenic and atypical
idiopathic clubfoot patients and answer the following ques-
tions: (1) What is the success rate for correcting the deform-
ity? (2) What is the rate of relapse after the correction? (3)
What is the incidence of complications?
Materials andmethods
The study was conducted according to the Cochrane Hand-
book for Systematic Reviews of Interventions Version 6.0
[17] and reported as per the Preferred Reporting Items for
Systematic Reviews and Meta-Analyses (PRISMA) guide-
lines [18]. This review was not pre-registered. Our institu-
tional review board waived the requirement for approval for
the reporting of this study. All investigations were conducted
in conformity with ethical principles of research.
Search strategy
On March 1, 2021, the authors searched the electronic data-
bases EMBASE, MEDLINE, Cochrane Library, and Web
of Science from database inception. The search included
the following keywords without any limits: “clubfoot” OR
“congenital talipes equinovarus” OR “CTEV” AND “Pon-
seti” OR “modified Ponseti.” Through consultations with
clinical epidemiologists and librarians, such terms were
generated. The search strategy, adapted to each database,
included terms representing the modified Ponseti concept
along with terms addressing idiopathic clubfoot.
Study selection was performed manually by the two
authors (OAM, TSH). The selection of studies was per-
formed in a stepwise manner, first by title, then abstract,
and then full-text review. A resolve-by-consensus strategy
was utilized for all discrepancies. If consensus could not be
reached, a third senior, independent, blinded reviewer was
consulted. This reviewer was not part of the study.
Using the search strategy described above, 699 studies
were identified from the electronic databases. Following
removal of duplicates, irrelevant articles, and articles with
incomplete data, ten articles were included for quantitative
synthesis. Figure1 contains the PRISMA flow diagram.
Inter-rater agreement at each stage of the screening pro-
cess was calculated using Cohen’s Kappa (κ). Based on
previous literature, interpretation of κ values was set and
ranged from κ ≤ 0.20 = no agreement to 0.90 < κ ≤ = almost
perfect agreement [19]. There was strong agreement for the
title/abstract (k = 0.7, 95% confidence interval: 0.6–0.9) and
full-text screening stages (k = 0.9, 95% confidence interval:
0.7–1.0).
Eligibility criteria
All studies on idiopathic, complex, and atypical clubfoot
that assessed Ponseti’s modified technique as an intervention
were included. All patients, regardless of age and ethnic-
ity, were eligible. Studies that reported at least one outcome
related to Ponseti’s modified technique for complex clubfoot
were eligible for inclusion. To maximize potentially eligible
data, there were no restrictions based on publication date,
study design, language, or follow-up. All bibliographies of
the included articles were assessed for further relevant stud-
ies. Also, we performed manual search of pertinent journals
in the field as well as conference proceedings. Studies that
included clubfoot with any known etiology including but
not limited to myelomeningocele and arthrogryposis were
excluded.
Data collection
Data were extracted by each reviewer independently from
eligible papers into a collaborative web-based spreadsheet
(Google Sheets, 2021. CA, USA: Google LLC.). To ensure
accuracy, the results were audited by both reviewers.
Extracted data included information on basic study char-
acteristics including first author, study design, level of evi-
dence, sex, mean age upon presentation, number of feet,
previous treatment of any kind, number of casts required for
correction, bracing, period of follow-up, follow-up scores,
International Orthopaedics
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final outcomes, number of relapsed feet after initial treat-
ment, and complications. The authors of the studies were
contacted if there were any uncertainties regarding the data.
Risk ofbias andquality assessment
The Methodological Index for Non-Randomized Studies
(MINORS) was used to assess risk of bias in the included
studies [20]. The MINORS scale assigns a score of 0, 1, or
2 for a list of eight questions in non-comparative studies.
Owing to their observational nature, the identified studies
were considered to be of very low-grade to low-grade quality
according to the GRADE criteria [21].
The mean MINORS score for the included studies was
11.8 ± 1.7 out of 16 (median of 12.0 with a range of 8–14).
The majority of the included studies were rated down due
to retrospective data collection and lack of sample size
calculations, and. Table1 contains the detailed MINORS
assessment.
Results
Most of the included studies used a case series study design
to investigate the effects of the modified Ponseti technique
on the correction of complex clubfoot. A total of 266
patients and 360 feet were included in the ten studies; the
collective male to female ratio in all studies was 2.6:1. More-
over, the geographical location varied among the studies and
spanned five continents. Table2 shows the characteristics of
the included studies.
A high rate of initial correction (76.4–100%), which was
achieved in all patients with a mean of five casts (range 5
to 7 casts), was noted [5, 9, 10, 1216]. The initial correc-
tion became evident with a mean ankle dorsiflexion of 15°
(range, 10–25°) in seven studies [5, 9, 1215]. Dragoni etal.
[10] and Duman etal. [16] achieved initial correction with
excellent to good results on the ICFSGS scale. Three studies
reported decreases in the mean Pirani scores at the treatment
start time, first application of braces, and final visit [2, 6,
16], with statistically significant differences in Pirani scores
at the follow-up periods. The most commonly used bracing
tool was the Mitchell–Ponseti brace, which was used in three
studies, and the Dennis-Browne abduction type, which was
used in two studies.
Among the studies, the follow-up duration was
7.5–86.4months, with an average follow-up duration of
39.7months. Although all of the studies have not consist-
ently included the term, relapse occurred often and its inci-
dence ranged at 10.5–55% [5, 9, 10, 1216]. A relapse was
reported in every other patient (53% and 55%) in two studies
that had longer follow-up periods [9, 10].
Records identified from:
EMBASE (n = 146)
MEDLINE (n = 346)
Cochrane Library (n = 153)
Web of Science (n = 54)
Total (n = 699)
Records removed before
screening:
Duplicate records removed
(n = 124)
Records screened
(n = 575)
Records excluded after
title/abstract screening
(n = 541)
Reports sought for retrieval
(n = 34)
Reports assessed for
eligibility
(n = 8)
Reports excluded:
Using methods other than
Ponseti modified technique
(n = 28)
Data were mixed with
other subgroups (n = 1)
Diagnosis other than
idiopathic complex club
foot (n = 5)
Records identified from:
Citation searching (n = 22)
Reports excluded:
Using methods other than
Ponseti modified technique
(n = 6)
Reports assessed for
eligibility
(n = 2)
Studies included in review
(n = 10)
Identification of studies via databasesIdentification of studies via other methods
Identification
Screening
Included
Reports sought for retrieval
(n = 8)
Fig. 1 PRISMA flow diagram
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The rate of these complications varied among the stud-
ies [5, 12, 14, 15, 22], at 6–30%, although five studies did
not report complications. Of the remaining studies, only
two defined their complications, which included erythema,
swelling, midfoot deformities, or cast slippage [5, 14].
Table3 provides the details of the modified Ponseti tech-
nique that was performed in each study.
Discussion
Satisfactory clinical outcomes have been reported for the
Ponseti technique in treating clubfoot, which entails a series
of manipulations and serial sets of casting [2325]. Studies
have demonstrated that outcomes of the modified Ponseti
method for treating complex clubfoot and atypical club-
foot report widely different percentages of success, relapse,
and complications. Therefore, we performed a systematic
review of the outcomes of Ponseti’s modified method to treat
patients with complex idiopathic clubfoot to clarify the fol-
lowing: the deformity correction success rate, relapse rate
after the correction, and incidence of complications.
In 1994, Turco referred to this condition as “atypical
idiopathic clubfoot”; since then, the terms, atypical and
complex, have been used interchangeably [7]. Most of the
included studies did not distinguish between these terms
[5, 9, 1215], which creates doubt regarding whether the
included cases were atypical at birth or if they were typi-
cal at birth but became complex after improper treatment
[10]. Although the modified Ponseti method is successful
irrespective of aetiology, specific definitions are required
to distinguish complex from atypical clubfoot. This raises
an important question regarding whether these subgroups
have different outcomes. Most of the studies clearly agree
that atypical clubfoot shares the following features: severe
plantar flexion of all metatarsals, a rigid equinus with a deep
crease above the heel, and a deep plantar crease across the
full width of the sole of the foot with high cavus and a short
hyperextended big toe. As for complex clubfoot, Dragoni
etal. [10] found that indeed complex clubfoot started as a
typical clubfoot but iatrogenically converted into complex
clubfoot with improper casting and slippage. This slippage
forces the foot into severe equinas and cavus. However, he
also noticed that most of the complex clubfoot cases were
Table 1 Quality assessment results of the included studies according to the Methodological Index for Non-Randomized Studies scale (MINORS)
Study Clearly
stated
aim
Inclusion of
consecutive
patients
Prospective
collection of
data
Endpoint
appropriate
to the study
design
Unbiased
evaluation of
endpoints
Follow-
up period
appropriate
to the major
endpoint
Loss to
follow-up not
exceeding
5%
Prospective
calculation
of sample
size
Total score
Ponseti etal.,
2006 [5]
2 2 0 2 2 2 2 0 12
Yoshioka
etal., 2010
[12]
2 2 0 2 2 1 2 0 11
Gupta etal.,
2015 [13]
2 2 0 1 0 1 2 0 8
Matar etal.,
2017 [9]
2 2 0 2 2 2 2 0 12
Elseddik
etal., 2018
[14]
2 2 2 1 2 1 2 0 12
Mandlecha
etal., 2019
[22]
2 2 2 1 2 1 2 0 12
Dragoni
etal., 2018
[10]
2 2 2 2 2 2 2 0 14
Allende
etal., 2020
[15]
2 2 0 2 2 2 2 0 12
Duman etal.,
2020 [16]
2 2 0 2 2 2 2 0 12
Bozkurt
etal., 2021
[26]
2 2 2 2 2 1 2 0 13
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initially short, stubby feet which might contribute to the risk
of cast slippage.
The outcomes of modified Ponseti protocol were mainly
defined based on ankle dorsiflexion [5, 9, 1215]. Two of the
included studies used International Clubfoot Study Group
Classification System (ICFSGS) scores [10, 16], while three
used the Pirani score [15, 22, 26]. Early detection of the
deformity and modifying the standard protocol, described
by Ponseti etal. [5], resulted in a high rate of success at
final follow-up despite of the outcome tools used [5, 9, 10,
1216]. Although the modified Ponseti method has a suc-
cess reaching to 100%, several studies have proposed dif-
ferent modifications and management plans for complex
clubfoot [4, 11, 27, 28]. To our knowledge, no study has
compared the different methods; therefore, conclusions
cannot be drawn regarding the modified Ponseti protocol
in comparison with others. However, the key to treating
atypical and complex clubfoot is first to recognize it, then
apply the modified Ponseti technique without variation [5].
The included studies did not provide comprehensive details
regarding casting, manipulation, relapse management, and
bracing protocols. Zhao etal. found strict adherence to the
principles and technical details was highly related to the
treatment outcome using the Ponseti method [29]. Detailed
descriptions of the modified Ponseti technique should be
provided to aid comparison among the studies and to deter-
mine whether the authors strictly adhered to the technique’s
technical details [30].
Also, the studies lacked any definition of relapse; how-
ever, all studies reported a wide range of relapse occurrence
rate. The variations in reported outcomes and relapse rates
might be attributed to the lack of a definition. Relapse has
been defined as the reappearance of any of the components
of the deformity [31]. The reported causes of relapse were
numerous and entailed decreased function of the evertor
muscles, initial deformity severity, and concurrent foot
deformities [5, 7, 10]. Modifications to the Ponseti protocol
were based on a detailed knowledge of the pathoanatomical
Table 2 General characteristics of the included studies
IQR interquartile range
Study Study type Level of
evidence
County Num-
ber of
patients
Sex Mean age upon pres-
entation
Number of
feet
Previous treat-
ments
Ponseti etal.,
2006 [5]
Retrospective
study
Level III Multi-center
study based
in US
50 31M, 19F 3months
(1week–9months)
75 Yes (plaster
casting 31
patients)
Yoshioka
etal., 2010
[12]
Case series
study
Level IV Single-center
study based
in US
5 3M, 2F 17.7months
(2–47months)
5 Yes (plaster
casting ± ten-
otomies)
Gupta etal.,
2015 [13]
Case series
study
Level IV Single-center
study based
in India
16 10M, 6F 3.2months
(1–5months)
16 Yes (plaster
casting)
Matar etal.,
2017 [9]
Case series
study
Level IV Single-center
study based
in UK
11 9M, 2F 3months
(1week–9months)
17 Yes (Ponseti in
3 patients)
Elseddik
etal., 2018
[14]
Case series
study
Level IV Single-center
study based
in Egypt
19 14M, 5F 2.5months
(2weeks–4months)
28 Not reported
Mandlecha
etal., 2019
[22]
Case series
study
Level IV Single-center
study based
in India
16 13M, 3F 4.7months
(1–12months)
27 Not reported
Dragoni etal.,
2018 [10]
Case series
study
Level IV Single-center
study based
in Italy
38 26M, 12F 2.5months
(6weeks–5months)
9 Yes (plaster
casting ± ten-
otomies)
Allende etal.,
2020 [15]
Retrospective
study
Level III Multi-center
study based
in South
America
79 44M, 35F 7months (15, 1–53)—
median (IQR, min–
max)
124 Yes, (plaster
casting ± ten-
otomies)
Duman etal.,
2020 [16]
Retrospective
study
Level III Multi-center
study based
in Turkey
21 15M, 6F 4months
(4–14months)
32 Yes, Achilles
tenotomy in
23 feet
Bozkurt etal.,
2021 [26]
Prospective
study
Level III Single-center
study based
in Turkey
11 10M, 1F 2months (range,
0–6months)
16 Yes (plaster
casting ± ten-
otomies)
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Table 3 Details of the modified Ponseti technique undertaken in each study
Study Number of casts
required
Bracing Period of follow-up Follow-up method/
outcomes
Satis-
factory
outcomes
Number of relapsed
feet/recurrence after
initial treatment
Complications
Ponseti etal., 2006 [5] 5 (1–10) Soft premolded foot–
ankle abduction brace
23months (6 to
46months)
Initial correction was
achieved with mean
ankle dorsiflexion of
15° (range, 10–25°)
75 Seven patients (14%)
had a relapse after
initial successful
treatment, and two
patients had a second
relapse. The average
time from correction
until diagnosis of
the first relapse was
1.5months (range,
2weeks–3months)
Eleven patients (22%)
had complications
including erythema,
slight swelling of the
forefoot and toes, mild
rocker-bottom deform-
ity, midfoot hyperab-
duction, or repeated
downward cast slippage
Yoshioka etal., 2010
[12]
5 (1–8) Foot abduction
brace ± foot–ankle
orthosis
15.8months (6 to
26months)
Initial correction was
achieved with mean
ankle dorsiflexion of
15° (range, 5–20°)
5 One patient had a
relapse after initial
successful treatment
All patients had consider-
able atrophy of the
calf muscles due to the
peroneal dysfunction.
One patient recovered a
little from the peroneal
nerve dysfunction, but
the others did not. No
other complications
were reported
Gupta etal., 2015 [13] 7 (4–9) Dennis-Browne abduc-
tion brace
2years (1 to 3years) Initial correction was
achieved with mean
ankle dorsiflexion of
15° (range, 10–25°)
16 Not reported Not reported
Matar etal., 2017 [9] 7 (5–10) Mitchell–Ponseti brace 7years (3 to 11years) Initial correction was
achieved with mean
ankle dorsiflexion of
15° (range, 10–25°)
13 The overall relapse rate
was 53%
Not reported
Elseddik etal., 2018
[14]
6 (5–9) Locally manufactured
version of Markell
splint
Mean period of
7.5months post brace
removal
Initial correction was
achieved with mean
ankle dorsiflexion of
15° (range, 10–25°)
28 The incidence of
relapse was 10.5%.
Average time from
correction until
diagnosis of the
first relapse was
2.75months
Four patients (21%) had
complications includ-
ing erythema, slight
swelling of the forefoot
and toes, mild rocker-
bottom deformity, mid-
foot hyperabduction, or
repeated downward cast
slippage
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Table 3 (continued)
Study Number of casts
required
Bracing Period of follow-up Follow-up method/
outcomes
Satis-
factory
outcomes
Number of relapsed
feet/recurrence after
initial treatment
Complications
Mandlecha etal., 2019
[22]
7 (6–10) Steenbeek foot abduc-
tion brace
1.2year (6months to
1.8year)
Correction was
achieved with a mean
Pirani score at latest
follow-up being
0.0556
27 The incidence of
relapse was 11.1%
Overall some or the other
complication occurred
in 8 out of 27 feet
(29.63%)
Dragoni etal., 2018
[10]
6 (4–8) Mitchell–Ponseti brace 7.2years (range:
6–9.6years)
Of the nine complex
clubfeet, two showed
an excellent result and
five showed a good
result (77%) accord-
ing to the ICFSGS
scale
9 The incidence of
relapse was 55% at
an average age of
3.6years (range:
3–5years)
Not reported
Allende etal., 2020
[15]
5 (5, 3–13)—median
(IQR, min–max)
Mitchell–Ponseti brace
or standard abduction
brace
49months (12–
132months)
Median ankle dorsiflex-
ion at the last follow-
up was 20 degrees
(IQR, 10 degrees;
min–max, − 5 to − 30
degrees)
122 Thirty-seven feet
(29.8%) had a relapse
after initially success-
ful treatment
Seven feet (6%) presented
minor complications
Duman etal., 2020 [16] 5 (3–6) Dennis-Browne abduc-
tion brace
56months (24–
144months)
Initial correction with
25 (78.1%) showed an
excellent result, and
seven (21.9%) showed
good result according
to the ICFSGS scale
32 Relapse of pathology
occurred in four
feet (12.5%) of two
patients
Not reported
Bozkurt etal., 2021
[26]
7 (5–8) Foot abduction brace 13.3months (10–
16months)
Initial correction was
achieved with mean
a Pirani score of
0.4 ± 0.4 and mean
dorsiflexion of
20.2° ± 4.5
13 Three patients (18.8%)
experienced a relapse,
which was treated
with two to three
casts. However, plan-
tar creases persisted
on two feet (12.5%)
Not reported
ICFSGS the International Clubfoot Study Group Score, IQR interquartile range
International Orthopaedics
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features of complex clubfoot; therefore, meticulous atten-
tion must be paid to the details of this method. Accordingly,
such “a world of difference” in relapse rates could be related
to imprecise adherence to Ponseti’s recommendations [32,
33]. Our review found that relapse rates in complex clubfoot
patients increased with time. Studies with a long follow-up
duration, including those by Matar etal. [17] and Dragoni
etal. [10], had much higher occurrences of relapse. Thus,
studies with longer follow-up time periods are required
[34]. Nevertheless, relapse occurred in many cases due to
treatment noncompliance as shown by Göksan etal. who
examined a subgroup of patients with complex clubfoot [35].
The findings showed reduced patient compliance, with high
relapse rates based on 11 cases of complex clubfoot [35].
Substantial deviations were noted in the details reported by
the studies regarding compliance with braces, the types of
braces, application time, period of application, and weaning
off. These differences should be analyzed to determine the
effects of different bracing protocols on the final outcomes.
A stepwise pattern of treatment for relapse is recommended
and involves repeated casting, tenotomies, tendon transfer,
and posterior release, if necessary. Ponseti etal. [5] noted
that after successful intervention, seven patients relapsed,
three of whom underwent a second Achilles tenotomy.
Dragoni etal. [10] treated a case of relapse with Achilles
tenotomy; however, three patient underwent Achilles tendon
lengthening. Bozkurt etal. [26] and Mandlecha etal. [22]
required application of a further two to four casts. Matar
etal. treated approximately half of those with relapses with
soft-tissue releases [9]. Due to a lack of sufficient details,
more studies on relapse management are required as well.
We suggest conducting studies with a prospective design that
examine the causes of relapse after performing the modified
Ponseti method because they may offer a reliable, valuable,
and effective understanding and management of such cases.
In our review, the complications associated with the
modified Ponseti method included casting complications,
slippage, and pressure sores. The later suggests that poor-
fitting casts can cause a complex deformity [7]. Five studies
did not report complications [9, 10, 13, 16, 26], which might
have introduced errors in the reported incidence of compli-
cations. Studies that reported complications most commonly
reported those related to casting. Slipping of casts was the
most common complication; consequently, it is recom-
mended to bend the knee more than 110 degrees. None of
the studies reported complications related to the tenotomy.
Complex and atypical clubfoot, even though still uncom-
mon, represent a challenge for paediatric orthopaedic surgeons.
Studies regarding modified Ponseti technique have shown high
initial correction rates and a smaller number of relapses.
Our review has several limitations. The lack of randomized
controlled trials (RCTs) and prospective and comparative
studies on this topic impeded statistical data pooling and
meta-analysis. The level of evidence of the included stud-
ies was low due to inadequate prospective studies and RCTs.
However, it is difficult to conduct RCTs for a rare disease
entity. The included studies were associated with several
important types of bias. These biases may lead to overesti-
mation of treatment benefits, non-reporting may minimize the
complication rate, and a very short follow-up may increase
survival estimates. Also, one of the most important limita-
tions of the included studies was their lack of clarity regard-
ing whether the patients had atypical or iatrogenic complex
clubfoot due to previous standard Ponseti treatment failure
[10]. Studies with prospective designs and long-term follow-
up are required to determine whether our observation of a
wide range of relapse rates is due to performing the modified
method properly or whether this deformity has higher relapse
rates with further follow-up. However, satisfactory outcomes
might only be achieved if the method is followed without vari-
ation, with proper compliance, and brace protocol adherence.
Acknowledgements We would like to thank prof. Nassser Al-Sanea
general director at King Abdullah Bin Abdulaziz University, Riyadh,
Saudi Arabia, for allowing us to perform the investigations at his center.
We sincerely acknowledge Prof. Al-Sanea for his support and the con-
structive feedback that he always provides. His willingness to give his
time so generously has been very much appreciated.
Author contribution OAM: Study design, data analysis, and process-
ing, writing, and approval of final draft.
FNA: Data collection and processing and writing—original draft.
TSA: Data curation, methodology, validation, visualization, project
administration, and approval of final manuscript.
Data availability The datasets used and/or analyzed during the current
study are available from the corresponding author on request.
Code availability Not aplicable.
Declarations
Ethics approval Each author certifies that his institution waived
approval for the reporting of this investigation and that all investiga-
tions were conducted in conformity with ethical principles of research.
Investigation was performed at Section of Orthopedic Surgery, Depart-
ment of Surgery, King Abdullah Bin Abdulaziz University Hospital,
Princess Nourah Bint Abdul Rahman University, Riyadh, Saudi Arabia.
Consent to participate Not applicable.
Consent for publication Not applicable.
Competing interests The authors declare no competing interests.
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... However, slippage of casts remains a limiting factor in successful treatment leading to pressure sore formation, an increased number of visits to the clinic, and an economic burden on the family. Although accepted as a common problem, the incidence of cast slippage in complex clubfeet appears to be underreported in the literature [9]. ...
... The true incidence of cast slippage has been poorly reported in the literature. Flexing the knee beyond 110° was recommended in a systematic review by Al-Mohrej et al. [9] for management of complex clubfoot. ...
Article
Full-text available
Background The clinical entity of complex clubfoot poses a significant challenge to correction by slippage of casts which further complicates the deformity and prolongs the treatment. A static and dynamic component associated with this deformity causing cast slippage was recognized. The purpose of this study was to evaluate the clinical outcomes at the end of the casting period while addressing these issues.MethodsA retrospective study of 17 patients with 25 complex clubfeet over a period of 2 years was conducted. Tug test was used to ascertain the snugness of the cast. To address the dynamic component, distal extent of the cast was limited to metatarsal heads.ResultsThe mean age of patients at diagnosis was 4.41 months (2–7 months). The mean pre-casting Pirani score was 4.8 (4–6) and post casting Pirani score was 0.4 (0–1). A total of 128 casts were applied to correct 25 complex clubfeet. The average number of casts required to achieve correction by the modified Ponseti technique was 5.12 (4–7). Overall, four incidences of cast slippage occurred.Conclusion The modified Ponseti technique is effective in the correction of complex clubfoot. Tug test can detect casts which are prone to slippage. Limiting the distal extent of the cast to the metatarsal heads can reduce cast slippage by reducing the repeated downward pressure by the toes on the cast.Level of evidence Level 4.
... 14 Complex clubfeet have the same feature as the atypical ones, as well as edema, redness, and hyperesthesia. 15 Other authors making the same distinction are Al-Mohrej et al., 16 Allende et al., 17 and Duman et al. 18 Duman et al. and Dragoni M. et al. named the latter group "iatrogenic complex clubfeet." Dragoni et al. 15 found that all feet with slipping casts were not converted from typical to complex clubfeet, but some were, probably because of a combination of some intrinsic predisposing factors (high Pirani score, short and stubby clubfoot, partly fibrotic muscles in the plantar compartment, and a short triceps surae) and slipping casts. ...
Article
Purpose To follow children with a clubfoot by ultrasonography during the entire treatment period up to 4 years and compare with controls. Method Thirty clubfeet in 20 children treated using the Ponseti method and 29 controls were followed by repeated ultrasonography investigations from neonates to the age of 4 years. The previously established coronal medial and lateral, sagittal dorsal and posterior projections were used. Changes over time, correlations to the Diméglio score, and the course of treatment were studied. Results The medial malleolus–navicular distance was shorter, while the talar tangent–navicular distance and the talo-navicular angle were larger in clubfeet than in controls even after the initial correction. The healthy feet in unilateral cases did not differ significantly from the controls. The range of motion in the talo-navicular joint was approximately 20° less in clubfeet than in controls during the first four years of life. The medial malleolus–navicular distance ( r = –0.58) and the talo-navicular angle ( r = 0.66) at the first ultrasonography showed the highest correlation to the number of casts needed to correct the deformities. Conclusion Ultrasonography can be used to evaluate the initial degree of deformities in clubfeet and to follow the progress of the treatment and growth. Ultrasonography showed a clear difference between clubfeet and controls during the first four years of life. Although it was not possible to define specific limit values as benchmarks in the treatment, dynamic ultrasonography can provide valuable support in the decision-making process when complementary treatment may be needed. Level of evidence III
... Earlier on, most of the children needed surgical interventions; however, from the last twenty years, the Ponseti treatment is perceived as a standard treatment of clubfoot across the world. [1][2][3] Traditionally, clubfoot is treated clinically but physiotherapy has also been proven effective even in non-idiopathic and complicated cases. 4 The survey data suggests the drop in conduction of extensive surgeries for treating clubfoot from 70% in 1996 to only about 10% in 2006 in the USA. 5 However, it is surprising to find out that only a few studies have been conducted in Pakistan that compared Ponseti treatment with earlier treatment methods. ...
Article
Full-text available
Objectives: To compare the outcomes of Ponseti treatment with the traditional treatment method for clubfoot. Methods: A cross-sectional comparative study was conducted at the orthopedic department of Nishtar Medical Hospital & University Multan for one year. The study included 40 children (29 clubfeet) treated with conventional treatment (pre-Ponseti group) who were compared with 55 Ponseti-treated children (72 clubfeet) (Ponseti group). All children were aged under five years. The traditional treatment involved casting and surgery (if required). All the participants were evaluated by a single orthopedic surgeon. The questionnaire was administered to the parents to collect relevant data. X-ray studies were conducted of all feet and patients' records were checked for surgical history. Results: Children in the pre-Ponseti group had a significantly higher number of surgeries (54) than those in Ponseti group eight. According to the reports of the parents, children in Ponseti group had significantly better motion in the ankle, lesser pain, and higher satisfaction (p<0.05.whereas, the pre-Ponseti group had a higher incidence of moderate or severe talar flattening rate (p=0.01). Conclusion: Ponseti treatment is better than earlier treatment in terms of lesser need of surgeries, higher flexibility of ankle or foot, and lower presence of X-ray guided talar flattening.
... Al-Mohrej et al. have conducted a systematic review on atypical or complex clubfeet treated using modified Ponseti casting protocol; however, while they have described the available studies in detail, a statistical analysis in the form of meta-analysis lacks in their report. 23 This meta-analysis has some limitations. We could not find studies of a higher level of evidence (better than case series) comparing complex clubfoot with idiopathic clubfoot. ...
Article
Background/purpose Atypical clubfeet are distinct from idiopathic clubfeet. It is resistant to correction by conventional casting methods and often requires a modification of Ponseti's casting technique. Although the initial correction rates are reasonable, relapse and complications are frequent. There is limited literature on the results of modified Ponseti casting of these feet. We conducted this meta-analysis to study a few important aspects of atypical/complex clubfeet treatment by the modified Ponseti technique. Research question What are the results of atypical or complex clubfeet after treatment by the modified Ponseti technique? Methodology Five electronic databases (PubMed, Embase, Scopus, Ovid, and Cochrane Library) were searched for articles reporting on the results of atypical/complex clubfeet treated by the modified Ponseti technique. Details of the number of casts required for correction, rate of percutaneous Achilles tenotomy (PAT), other soft tissue procedures required, complications, and relapse rates were extracted into spreadsheets, and meta-analysis was carried out using OpenMeta Analyst software. Results Ten studies were included for analysis with a total of 240 patients with 354 clubfeet. The initial correction was achieved in all feet. A pooled analysis of the data showed that a mean of six casts was required for the initial correction. The rate of PAT was 98.3%. The overall complication rate was 16.8%. 7.2% required an additional soft tissue procedure apart from the PAT, and relapse of the deformity was observed in a mean of 19.8% cases. Conclusion Modified Ponseti technique is effective in the initial management of atypical/complex clubfeet. Although the PAT rate is slightly higher in the Modified Ponseti technique, the remaining result parameters are comparable with the results of idiopathic clubfoot managed with the Ponseti method of casting. However, these children should be kept under follow-up for a longer duration to find the exact relapse rates.
... The frequency of post-traumatic ankle arthrodesis has increased in recent years due to sports practice; however, sports activity level [10] following an ankle fusion is decreased, and many patients will not return to their pre-injury level of activity. The clubfoot pathology rarely recurs in adulthood when corrected in childhood with the classical Ponseti technique [11,12] or with a new technology [13] with sensors integrated in the brace. However, a neglected clubfoot [14] or persistent deformities with arthritic pain may require further arthrodesis during adulthood. ...
Article
Full-text available
Introduction The treatment of complex atypical clubfoot poses many challenges. In this paper, we report on the course of complex clubfoot, primary correction using the modified Ponseti method and midterm outcomes. Special consideration is given to clinical and radiological changes in cases of relapse. Materials and methods Twenty-seven cases of complex, atypical, non-syndromic clubfoot were treated in 16 children between 2004 and 2012. Patient data, treatment data, functional outcomes and, in the relapse cohort, radiological findings were documented during the course of treatment. The radiological findings were correlated with the functional outcomes. Results All atypical complex clubfeet could be corrected using a modified form of the Ponseti method. Over an average study period of 11.6 years, 66.6% (n = 18) of clubfeet relapsed. Correction after relapse showed an average dorsiflexion of 11.3° during a 5-years’ follow-up period. Radiological results showed residual clubfoot pathologies such as a medialized navicular bone in four clubfeet. There were no instances of subluxation or dislocation of the talonavicular joint. Extensive release surgery was not necessary. Nevertheless, after 2.5 preoperative casts (1–5 casts), bone correction was performed in n = 3 feet in addition to Achilles tendon lengthening and tibialis anterior tendon transfer. Conclusion Good primary correction of complex clubfoot using the modified Ponseti technique results in a high recurrence rate in the medium term. Relapse treatment without peritalar arthrolysis procedures produces good functional results even though minor residual radiological pathologies did persist in a minor number of cases.
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Objectives: This study aims to present the results of complex clubfoot patients treated with modified Ponseti method and put forth the warning clinical signs of complex deformities. Patients and methods: A total of 11 patients (10 males, 1 female; mean age 60.1±49.7; range, 2 to 180 days) with 16 complex clubfeet treated with modified Ponseti method were included in this study conducted between January 2016 and June 2019. All the data of the patients were collected prospectively and reviewed retrospectively. Demographic features, clubfoot severity, number of casts, position of each foot before cast removal, ankle dorsiflexion (DF), complications, and additional procedures were noted at all clinical visits. Results: Eleven (11.2%) of 98 patients had complex clubfoot deformity. Six (7.7%) of 78 newly diagnosed patients and five (25%) of 20 referred patients had complex clubfeet. We treated 16 complex clubfeet of 11 patients. The mean follow-up period was 13.3 (range, 10 to 16) months. All deformities were initially corrected using a mean of seven (range, 5 to 8) casts and Achilles tenotomy. Relapses occurred in three (18.75%) patients, but all recovered after recasting. The creases above the heels disappeared in all of the patients, whereas plantar creases persisted on two (12.5%) feet. Pirani scores and DF improved statistically significantly after treatment, and DF improved significantly between tenotomy and the final visit. Conclusion: The modified Ponseti method is an effective treatment for complex clubfoot. Classical clinical appearance, treatment-resistant deformities and referred patients should be warning signs for complex clubfoot.
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Background: The Ponseti method is the preferred technique to manage idiopathic clubfoot deformity; however, there is no consensus on the expected relapse rate or the percentage of patients who will ultimately require a corrective surgical procedure. The objective of the present systematic review was to determine how reported rates of relapsed deformity and rates of a secondary surgical procedure are influenced by each study's length of follow-up. Methods: A comprehensive literature search using the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines was performed to identify relevant articles. The definition of relapse, the percentage of patients who relapsed, the percentage of feet that required a surgical procedure, and the mean duration of follow-up of each study were extracted. Pearson correlations were performed to determine associations among the following variables: mean follow-up duration, percentage of patients who relapsed, percentage of feet that required a joint-sparing surgical procedure, and percentage of feet that required a joint-invasive surgical procedure. Logarithmic curve fit regressions were used to model the relapse rate, the rate of joint-sparing surgical procedures, and the rate of joint-invasive surgical procedures as a function of follow-up time. Results: Forty-six studies met the inclusion criteria. Four distinct definitions of relapse were identified. The reported relapse rates varied from 3.7% to 67.3% of patients. The mean duration of follow-up was strongly correlated with the relapse rate (Pearson correlation coefficient = 0.44; p < 0.01) and the percentage of feet that required a joint-sparing surgical procedure (Pearson correlation coefficient = 0.59; p < 0.01). Studies with longer follow-up showed significantly larger percentages of relapse and joint-sparing surgical procedures than studies with shorter follow-up (p < 0.05). Conclusions: Relapses have been reported to occur at as late as 10 years of age; however, very few studies follow patients for at least 8 years. Notwithstanding that, the results indicated that the rate of relapse and percentage of feet requiring a joint-sparing surgical procedure increased as the duration of follow-up increased. Longer-term follow-up studies are required to accurately predict the ultimate risk of relapsed deformity. Patients and their parents should be aware of the possibility of relapse during middle and late childhood, and, thus, follow-up of these patients until skeletal maturity may be warranted. Level of evidence: Therapeutic Level IV. See Instructions for Authors for a complete description of levels of evidence.
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Introduction: Serial Ponseti casting achieves deformity correction in early presenting idiopathic clubfoot cases normally in around 7 casts. However, there are resistant patients where correction requires more casts than usual. In such patients a modification in standard technique might be required right from the beginning. Such patients were collectively called as difficult clubfoot. The aim of this study was to assess the outcome of our modification to Ponseti technique in difficult clubfoot. Methods: All idiopathic clubfoot cases who were 75th percentile or more in WHO age for weight chart (chubby infants) or untreated clubfoot patients presenting for first time to our clinic at more than 5 months age (late presenters and neglected cases) were included in the study. Patients who had been previously surgically intervened elsewhere, patients over 7 years of age, patients with syndromic clubfoot or clubfoot associated with neurological conditions were excluded from the study. The patients were treated by early tenotomy of tendoachillis and a plantar fascia release before starting serial casting by Ponseti technique. Post correction, strict bracing protocol was followed with regular follow up. Pirani scoring was done at each stage. Measurement of Talocalcaneal angle on AP radiograph, maximum degree of abduction and dorsiflexion was noted once every year. Results: There were total 28 patients in our study. In all, 47 feet were subjected to modified Ponseti protocol. There were 21 male patients. Median age at presentation was 4 months. Mean centile of weight for age as per WHO growth chart was 64. Mean Pirani score at presentation was 5.86 (S.D. ± 0.34). Mean number of casts required for correction was 3.75 ± 1.10. Maximum followup period was 25 months. Conclusion: This modification of Ponseti casting for difficult clubfoot patients achieves correction in shorter duration with less number of casts.
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Background: Also known as clubfoot, idiopathic congenital talipes equinovarus (ICTEV) is the most common pediatric deformity and occurs in 1 in every 1000 live births. Even though it has been widely researched, the etiology of ICTEV remains poorly understood and is often described as being based on a multifactorial genesis. Genetic and environmental factors seem to have a major role in the development of this disease. Thus, the aim of this review is to analyze the available literature to document the current evidence on ICTEV etiology. Methods: The literature on ICTEV etiology was systematically reviewed using the following inclusion criteria: studies of any level of evidence, reporting clinical or preclinical results, published in the last 20 years (1998-2018), and dealing with the etiology of ICTEV. Results: A total of 48 articles were included. ICTEV etiology is still controversial. Several hypotheses have been researched, but none of them are decisive. Emerging evidence suggests a role of several pathways and gene families associated with limb development (HOX family; PITX1-TBX4), the apoptotic pathway (caspases), and muscle contractile protein (troponin and tropomyosin), but a major candidate gene has still not been identified. Strong recent evidence emerging from twin studies confirmed major roles of genetics and the environment in the disease pathogenesis. Conclusions: The available literature on the etiology of ICTEV presents major limitations in terms of great heterogeneity and a lack of high-profile studies. Although many studies focus on the genetic background of the disease, there is lack of consensus on one or multiple targets. Genetics and smoking seem to be strongly associated with ICTEV etiology, but more studies are needed to understand the complex and multifactorial genesis of this common congenital lower-limb disease.
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Background: Complex clubfoot does not respond to ponseti method. In 2006 Ponseti et al published the results of treatment of such complex club foot by modified ponseti technique, since then it has become standard method of treatment for complex clubfoot. There has been only few published result of this method and hence, here we are evaluating our experience with 16 patients (27 clubfeet) with complex clubfeet treated at our center by modified ponseti method. Method: Parents of patient fulfilling the criteria for complex clubfoot were consented and registered under the study. Pirani score at presentation, at prescription of foot abduction brace and at final follow up was noted. total number of casts required for desired correction, number of cast before and after tenotomy, need of tendoachilles tenotomy, relapse and complications were documented. Result: Average follow up duration was 14.762 months (Range 6 month to 22 months). Of the total 16 patients 11 had bilateral complex clubfoot and 5 had unilateral complex clubfoot, the mean pirani score at the time of presentation was 5.5741 (range 4.5-6), Mean pirani score at latest follow up was 0.0556. Average no. of casts required for the complete correction with modified ponseti method was 7.44 (ranging from 6 to 10 casts). All 27 feet (100%) required tendo achillies tenotomy. Percutaneous tenotomy was done in 19 feet while 8 feet required Mini-Open tenotomy (due to thick pad of fat tendon was not palpable). Relapse rate was 11.11% (3 feet) [all had relapse of equinus, fore foot adduction treated by remanipulation by modified ponseti technique, retenotomy and casting]. An excellent result was achieved with at final follow-up in all 27 feet. Conclusion: In our experience modified ponseti technique for treatment of complex clubfoot is a successful method of treatment if aided with tendoachilles tenotomy, also it has reduced the requirement of surgical intervention in such patients.Level of Evidence - Level IV.
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Background: Complex clubfoot is a term used to describe those feet that present after treatment with a short first metatarsal, severe plantar flexion of all metatarsals, rigid equinus, and deep folds through the sole of the foot and above the heel. Ponseti has described a modification of his original technique for the treatment of the deformity. Few series have reported the treatment outcomes of this group of patients. The purpose of this study is to analyze mid-term results and complications of a large multicenter cohort. Methods: Patients with complex clubfoot treated at 6 tertiary-care institutions with a minimum of 1-year follow-up were retrospectively analyzed. Demographic data, previous treatment, number of casts, Achilles tenotomy, recurrences, complications, and additional procedures were documented. The patients were clinically evaluated at the time of presentation, after treatment, and at the last follow-up according to the Pirani score. All variables had a nonparametric distribution and are thus described as median (interquartile range (IQR), minimum-maximum). A comparison between the variables was performed using a Mann-Whitney U test, the change within each group was performed with a Wilcoxon-designated range test. A P-value <0.05 was used to indicate statistical significance. Results: One hundred twenty-four feet (79 patients) were evaluated. The median age at initial treatment was 7 months (IQR, 15; min-max, 1 to 53 mo). The mean follow-up was 49 months (IQR, 42; min-max, 12 to 132 mo). A median of 5 casts (IQR, 5; min-max, 3 to 13) was required for correction. Percutaneous tenotomy of the Achilles tendon was performed in 96% of the feet. One hundred twenty-two feet (98%) were initially corrected; 2 feet could not be corrected and required a posteromedial release. The Pirani score improved significantly from a pretreatment mean of 6 points (IQR, 1; min-max, 4.5 to 6) to 0.5 (IQR, 0.5; min-max, 0 to 2.5) at the last follow-up (P <0.001). Seven feet (6%) presented minor complications related to casting. Relapses occurred in 29.8% (37/124). In this subgroup, the number of casts required at initial treatment was higher (6; IQR, 5; min-max, 1 to 12 vs. 4 IQR, 4; min-max, 1 to 13; P<0.001), and follow-up was significantly longer (62 mo; IQR, 58; min-max, 28 to 132 vs. 37 mo; IQR, 48, min-max, 7 to 115; P<0.001). Conclusions: Ponseti method is safe and effective for the correction of complex clubfeet. Early diagnosis and strict adherence to the Ponseti principles are key to achieve deformity correction. Patients with complex clubfoot require frequent follow-up because of a higher recurrence rate. Level of evidence: Level III-therapeutic study.
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The revised edition of the Handbook offers the only guide on how to conduct, report and maintain a Cochrane Review ? The second edition of The Cochrane Handbook for Systematic Reviews of Interventions contains essential guidance for preparing and maintaining Cochrane Reviews of the effects of health interventions. Designed to be an accessible resource, the Handbook will also be of interest to anyone undertaking systematic reviews of interventions outside Cochrane, and many of the principles and methods presented are appropriate for systematic reviews addressing research questions other than effects of interventions. This fully updated edition contains extensive new material on systematic review methods addressing a wide-range of topics including network meta-analysis, equity, complex interventions, narrative synthesis, and automation. Also new to this edition, integrated throughout the Handbook, is the set of standards Cochrane expects its reviews to meet. Written for review authors, editors, trainers and others with an interest in Cochrane Reviews, the second edition of The Cochrane Handbook for Systematic Reviews of Interventions continues to offer an invaluable resource for understanding the role of systematic reviews, critically appraising health research studies and conducting reviews.