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ORIGINAL ARTICLE

A Single-center Prospective Evaluation of the


Ponseti Method in Nonidiopathic Congenital
Talipes Equinovarus
Paul J. Moroney, FRCSI (Tr. & Orth.), Jacques Noe¨l, FRCSI (Tr. & Orth.),
Esmond E. Fogarty, FRCSI, and Paula M. Kelly, FRCSI (Tr. & Orth.)

pathologic condition exists. We have found there to be


Background: The Ponseti method has revolutionized the man- 2 broad groups of nonidiopathic CTEV—those charac-
agement of idiopathic congenital talipes equinovarus (CTEV). terized by soft tissue laxity (eg, connective tissue disorders,
However, nonidiopathic CTEV is still often primarily treated by trisomy 21) and those in whom the soft tissues are ex-
extensive surgical soft tissue release. We believe that non- ceptionally tight and resistant to corrective treatment.
operative treatment of these patients using the Ponseti method Patients in this latter group often have a neurological
may give very satisfactory results. etiology such as arthrogryposis.
Methods: We examined the demographics of nonidiopathic In the past, aggressive surgical correction was advo-
CTEV and the success of the Ponseti method in this population cated for all patients.3–6 Long-term results of such treat-
over a 5-year period. We treated 29 patients with 43 nonidiopathic ment, however, have not been favorable. Dobbs et al7 have
and 97 patients with 138 idiopathic CTEV feet. Patients with radiologically documented unacceptable levels of ankle
nonidiopathic CTEV made up 23% of all cases. The commonest and foot arthrosis with correspondingly poor levels of
etiologies were arthrogryposis (5 cases), trisomy 21 (4 cases), and patient satisfaction. The management of idiopathic CTEV
spina bifida (3 cases). Average follow-up was 39 (nonidiopathic has been revolutionized by the work of Ignacio Ponseti.8,9
group) and 35 months (idiopathic group). His method of gentle, systematic manipulation and casting,
Results: The Ponseti method was initially successful in 91% of followed by Achilles tenotomy and long-term bracing has
nonidiopathic and 98% of idiopathic feet. Recurrence of de- yielded outcomes that are superior to those after extensive
formity occurred in 44% of nonidiopathic and 8% of idiopathic surgical releases.7,10 Many short-term and long-term studies
feet. Thirty-seven percent of nonidiopathic feet required extensive have demonstrated that the Ponseti method produces pain-
surgical release compared with 2% in the idiopathic group. less, flexible feet.8,9,11 His method has been adopted world-
Conclusions: Although the success rate of the Ponseti method in wide and now constitutes the gold standard treatment.
nonidiopathic CTEV is inferior to that in idiopathic CTEV, Despite the success of the Ponseti method in idio-
63% of our nonidiopathic patients did not require extensive pathic CTEV, the published literature has been slow to
surgery. We believe that the Ponseti method should be used in recommend it for the management of nonidiopathic
all cases of nonidiopathic CTEV. CTEV. The historical opinion was that nonidiopathic
Level of Evidence: Level III—prospective cohort study. CTEV feet were too rigid to respond favorably to manip-
Key Words: congenital talipes equinovarus, nonidiopathic, ulation and casting; and several authors continued to ad-
Ponseti vise destructive surgical procedures such as talectomy.12–15
In recent years, however, some groups have reported sat-
(J Pediatr Orthop 2012;32:636–640) isfactory results in their cohorts of patients with non-
idiopathic CTEV treated by the Ponseti method.16–18
The purposes of this study were to document the

C ongenital talipes equinovarus (CTEV) is the com-


monest congenital deformity of the foot, with an in-
cidence of 1 to 2 per 1000 live births.1,2 The majority of
incidence of nonidiopathic CTEV in our patient pop-
ulation, to identify the underlying etiology, and to assess
the effectiveness of the Ponseti method in their treatment.
cases are isolated deformities of unknown etiology. In a
minority of cases, however, an identifiable underlying
PATIENTS AND METHODS
This study was conducted in the largest pediatric
From the Our Lady’s Children’s Hospital, Dublin, Ireland. hospital in our country. We house the national centers for
None of the authors received financial support for this study. cardiac surgery and for medical genetics, and we provide
The authors declare no conflict of interest. a tertiary referral service for orthopaedics, rheumatology,
Reprints: Paul J. Moroney, FRCSI (Tr. & Orth.), Suite 19, Sports
Surgery Clinic, Santry Demesne, Dublin 9, Ireland. E-mail: paul nephrology, and neurology. In April 2005, a Ponseti clinic
moroney@rcsi.ie. was established to provide formal multidisciplinary treat-
Copyright r 2012 by Lippincott Williams & Wilkins ment for children with CTEV. All patients with suspected

636 | www.pedorthopaedics.com J Pediatr Orthop  Volume 32, Number 6, September 2012


J Pediatr Orthop  Volume 32, Number 6, September 2012 Evaluation of the Ponseti Method in Non-idiopathic CTEV

nonidiopathic CTEV are reviewed by a pediatric neurol- Demographic characteristics and outcome measures
ogist, a neonatologist, and a geneticist. Once referred, all were compared with the entire cohort of idiopathic CTEV
patients are seen at the next available Ponseti clinic patients who attended our clinic during the same time
(usually within 1 to 2 wk of birth). interval.
Patients are reviewed on a weekly basis. At each Statistical analysis was performed using Instat
appointment, the casts are removed, the children are (Graphpad Software Inc., La Jolla, CA). Continuous
bathed, and their feet are scored according to the scoring variables were assessed using unpaired t tests, whereas the
system of Pirani.19,20 Clinical photographs are taken. All Fisher exact test was used to assess categorical variables.
information is recorded prospectively on a computerized All statistical tests were 2-tailed, with a level of sig-
database (MD Analyze; Medtech Global Ltd., Australia). nificance set at Pr0.05.
Manipulation is performed according to the technique
described by Ponseti8,21 and moulded, above-knee casts are RESULTS
then applied. In our early experience, all patients received 6
casts before percutaneous Achilles tenotomy. Now the Demographics
number of casts applied pretenotomy is individualized for All patients who attended our clinic with non-
each patient. Most patients with idiopathic CTEV now idiopathic CTEV during the 5-year period from April
only require 4 or 5 casts to achieve adequate correction. 2005 to April 2010 and who had a minimum of 1-year
Conversely, in some very stiff nonidiopathic feet, tenotomy follow-up were included in the study. Twenty-nine such
is performed early in their treatment. Achilles tenotomy is patients were identified—17 males (59%) and 12 females.
performed in the clinic under local anesthesia whenever During the same period, we treated 97 patients with idi-
<10 degrees of passive ankle dorsiflexion is present. opathic CTEV. Of this group, 80 were male (82%) and 17
After Achilles tenotomy, an above-knee cast is ap- were female. Patients with nonidiopathic CTEV com-
plied for 3 weeks. Thereafter, a foot abduction orthosis, prised 23% of total new attendances.
comprising open-toed, high-top, straight-last shoes (Markell Fifteen of our 29 nonidiopathic patients had bi-
Shoe Company, Yonkers, NY) attached to a Denis lateral CTEV (52%), giving a total of 44 feet. Of the
Browne bar is applied. The feet are held in approximately remaining 14 feet, 8 were left and 6 were right. Bilaterality
70-degree abduction. In cases of nonidiopathic CTEV, we was present in 41 (42%) idiopathic cases, giving a total of
often use Mitchell shoes (MD Orthopaedics Inc., Iowa 138 feet. There was no significant difference in rates of
City, IA) and hold the feet in 60-degree abduction. Pa- bilaterality between idiopathic and nonidiopathic groups
rents are instructed to leave this orthosis in situ for 23 h/d (P = 0.4). In 1 case of proximal femoral focal deficiency
for the first 3 months and nighttime thereafter until the (PFFD), it was decided not to correct the foot on the
age of 4 years. Advice is provided regarding home affected limb, so as to preserve maximum length for later
stretching exercises. fitting of a prosthesis. Therefore, 43 nonidiopathic feet
We believe that it is essential to strive for a planti- were treated according to the Ponseti method.
grade foot in all cases. Even when patients are non- In our cohort, there was a large variety of diverse
ambulators, a plantigrade foot facilitates seating, allows diagnoses (Table 1). The commonest etiology was ar-
the wearing of more comfortable footwear, and reduces throgryposis (5 cases), followed by trisomy 21 (4 cases)
the risk of decubitus ulceration. Therefore, all patients and neural tube defects (3 cases). In 4 clearly non-
who fail conservative management with manipulation idiopathic, neuromuscular cases, the underlying etiology
and casting using the Ponseti method progress to surgical has not yet been established. One child has cerebral palsy
correction. In general, such correction comprises poste- secondary to a postnatal intraventricular cerebral hem-
rior and medial release or lengthening of the contracted orrhage. He was born at 26 weeks’ gestation and had a
soft tissues—including the ankle, subtalar, and talona- structural CTEV at birth. It is possible that his original
vicular joint capsules; the Achilles, tibialis posterior, deformity was “idiopathic” CTEV; however, it is likely
flexor digitorum longus, and flexor hallucis longus ten- that his postnatal foot deformity was driven by his ac-
dons; the talofibular, tibiofibular, and calcaneofibular quired neurological defect. Therefore, we believe that it is
ligaments; the plantar fascia and the abductor hallucis appropriate to include him in this cohort. Arthrogryposis
muscle. The extent of surgical release is decided on an was a clinical diagnosis, arrived at in conjunction with the
individual basis—depending upon the difficulty encoun- neonatology teams. Muscle biopsies were not performed.
tered in achieving correction. Procedures such as tibialis Many children had very significant comorbidities includ-
anterior transfer for correction of residual dynamic ing 14 (48%) orthopaedic, 10 (34%) cardiorespiratory,
forefoot supination are not uncommonly used to fine- 7 (24%) neurological, and 5 (17%) renal.
tune a CTEV foot when the child is older. Such proce- The median age at application of first cast was 12
dures are not considered in our study. days (mean, 25 ± 37; range, 28 to 160) in the non-
The success of the Ponseti method in each patient was idiopathic group. Seventy-three percent of patients were
recorded using the Pirani scoring system.19,20 We docu- seen within 2 weeks of birth. The average follow-up was
mented the rate of successful correction and that of re- 38.7 ± 15.5 months (range, 12 to 64 mo). In the idiopathic
currence. The number of casts required to achieve correction group, the first cast was applied at a median age of 8 days
and the rate of surgical correction were also assessed. (mean, 20 ± 34; range, 0 to 180) (P = 0.55) and the mean

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Moroney et al J Pediatr Orthop  Volume 32, Number 6, September 2012

TABLE 1. Diagnosis and Comorbidities in Patients With TABLE 2. Results of Ponseti Treatment in Nonidiopathic and
Nonidiopathic Congenital Talipes Equinovarus Idiopathic Congenital Talipes Equinovarus Feet
Case Nonidiopathic Idiopathic
Number Bilateral Diagnosis Associated Features (N = 43) (N = 138) P
1 Yes CHILD Scoliosis, oligodactyly, absent Starting Pirani 5.1 ± 1 (1.5-6) 4.9 ± 1 (1.5-6) 0.311
syndrome kidney, meningocele score*
2 Yes Arthrogryposis DDH, camptodactyly, VSD, Final Pirani score 1.49 ± 1.46 (0-4) 0.12 ± 0.21 (0-0.5) < 0.0001
epilepsy, developmental delay No. casts 6.42 ± 2.25 (3-12) 5.14 ± 1.54 (3-11) < 0.0001
3 Yes Trisomy 21 ASD, PDA, hypothyroidism Initial correctionw 39 (91%) 135 (98%) 0.056
4 No Trisomy 21 Hearing impairment Recurrence 17/39 (44%) 11/135 (8%) < 0.0001
5 No Trisomy 21 PDA, UDT, thrombocytopenia, Ponseti success 27 (63%) 135 (98%) < 0.0001
hypoglycaemia Extensive soft tissue 16 (37%) 3 (2%) < 0.0001
6 No Trisomy 21 ASD release
7 Yes Arthrogryposis Microcephaly
8 Yes Arthrogryposis — *Continuous data are presented as the mean ± SD with the range in paren-
theses.
9 Yes Neuromuscular* Coxa valga wThe data for categorical variables are given as the number of patients with the
10 No Cerebral palsy Vocal cord palsy, tracheostomy frequency in parentheses.
11 Yes Nail patella Radial head dislocation
syndrome
12 Yes Larsen Scoliosis, windswept knees, cleft
syndrome palate, cardiac anomalies On average, 6.4 ± 2.2 pretenotomy casts were required to
13 No Arthrogryposis Contralateral calcaneovalgus achieve initial correction in the nonidiopathic group
foot, camptodactyly, clinodactyly (range, 3 to 12) compared with 5.1 ± 1.5 (range, 3 to 11)
14 Yes PFFD Short right femur in the idiopathic group (P < 0.0001). The Ponseti method
15 Yes Marden-Walker ASD, UDT,
syndrome tracheobronchomalacia, was initially successful in correcting 39 of 43 non-
cardiomegaly idiopathic feet (91%). In 17 feet (44%) (12 patients), re-
16 No Ito syndrome Syndactyly, UDT, hearing currence of deformity occurred. A further series of casting
impairment corrected 3 feet, and repeat Achilles tenotomy was nec-
17 Yes Spina bifida DDH essary in 1 bilateral case. Twelve relapses eventually re-
18 No Spina bifida Knee contractures
19 No Spina bifida — quired extensive surgical release. All recurrences were
20 Yes Moebius Cardiorespiratory, associated with problems in wearing of the abduction
syndrome developmental delay orthosis. In the idiopathic cohort, the Ponseti method was
21 No Caudal — initially effective in 135 of the 138 feet (98%) (P = 0.056).
regression
22 No Ehlers-Danlos — Recurrence occurred in 8.1% of cases (P < 0.0001).
syndrome After a course of weekly manipulations and casts,
23 No Tibial hemimelia — the final Pirani score in our cohort of nonidiopathic
24 No Tibial hemimelia — CTEV feet was 1.49 ± 1.46 (range, 0 to 4.5). For those
25 Yes DiGeorge Cystinosis, pulmonary atresia, who did not progress to extensive corrective surgery, the
syndrome ASD, VSD, PDA
26 No Neuromuscular* — average Pirani score was 0.35. The mean Pirani score in
27 Yes Neuromuscular* Blind, DDH those who did require extensive surgical releases was 3.7
28 No Arthrogryposis Torticollis, cleft palate at the time of surgery. The final Pirani score in the idio-
29 Yes Neuromuscular* ASD, scoliosis pathic group was 0.12 ± 0.21 (range, 0 to 0.5). A total of
*Neuromuscular conditions where an exact diagnosis has not yet been 16 out of 43 (37%) nonidiopathic CTEV feet underwent
identified. extensive surgical release. At the end of the study period,
ASD indicates atrial septal defect; CHILD, congenital hemidysplasia with
icthyosiform erythroderma and limb defects; DDH, developmental dysplasia of no other patients had been scheduled for future surgery.
the hip; PDA, patent ductus arteriosus; PFFD, proximal femoral focal deficiency; This number represents 10 patients—8 of whom had bi-
UDT, undescended testes; VSD, ventricular septal defect. lateral CTEV and 2 with unilateral pathology. Six of the 8
bilateral cases underwent surgery to both feet. The un-
follow-up was 34.9 ± 16.4 months (range, 12 to 60 mo) derlying diagnoses in the surgical cases were: CHILD
(P = 0.19). (congenital hemidysplasia with ichthyosiform erythroderma
and limb defects) syndrome, Ito syndrome, Larsen syn-
Treatment drome, Marden-Walker syndrome, Moebius syndrome,
For the purpose of this discussion, we considered caudal regression syndrome, PFFD, cerebral palsy, spina
individually the 43 feet managed with the Ponseti method. bifida, and tibial hemimelia.
Results are displayed in Table 2. In the idiopathic group, 3 feet (2%) (3 patients)
At presentation, the mean Pirani score in our non- required extensive surgical release (P < 0.0001).
idiopathic group was 5.1 ± 1.0 (1.5 to 6) compared with
4.9 ± 1.0 (1.5 to 6) in the idiopathic group. Only 3 feet DISCUSSION
had initial scores <4. One patient with trisomy 21 had a The modern management of idiopathic CTEV, us-
score of 3, whereas the patient with DiGeorge syndrome ing the Ponseti method, delivers excellent results. Long-
had scores of 1.5 and 2 after initial treatment elsewhere. term follow-up studies by Ponseti himself and others have

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J Pediatr Orthop  Volume 32, Number 6, September 2012 Evaluation of the Ponseti Method in Non-idiopathic CTEV

demonstrated excellent results in patients with idiopathic surgical soft tissue release. There are still relatively few
CTEV treated according to his method.9,11 Nonetheless, reports of use of the Ponseti method in nonidiopathic
the principles of treatment of CTEV in nonidiopathic CTEV. Most of these studies concern arthrogryposis.
cases have remained the same for decades. Various authors have published rates of surgery in ar-
Menelaus12 first advocated the use of talectomy in throgrypotic feet managed by the Ponseti method that
cases of nonidiopathic CTEV in 1971. Drummond and range from 8% to 60%16,26 Gerlach et al17 reported a 14%
Cruess13 reviewed the treatment of arthrogrypotic foot rate of extensive soft tissue release in 28 CTEV feet sec-
deformity in 1978. Manipulation and cast was of benefit ondary to myelomeningocele. In a cohort of similar size
in only one of 32 patients. Talectomy was found to be the and diversity to ours, Janicki et al18 cited a rate of surgical
most successful surgical technique in their series. intervention of 28%.
More recent studies have questioned the long-term Within our cohort of nonidiopathic CTEV, we
outcome after talectomy. Legaspi et al14 reported only found great variations in response to manipulation and
33% good results, with 42% fair and 25% poor at 20-year casting. Typically, patients with conditions characterized
follow-up. Similarly, radical soft tissue release has also by ligamentous laxity corrected very easily. Our 4 patients
been proven to have significantly poorer long-term results with trisomy 21 and 1 with the collagen disorder Ehlers-
in idiopathic CTEV than the Ponseti method.7 Several Danlos syndrome were all successfully treated using the
authors16–18 have since reported encouraging successes in Ponseti method. Deformity did not recur in any case.
treating cases of nonidiopathic CTEV using the Ponseti Conversely, patients with conditions typified by extremely
method. tight soft tissues achieved only incomplete correction
In our series, 23% of all CTEV patients had non- through nonoperative means. Our patients with Larsen
idiopathic CTEV. This figure is similar to those quoted in syndrome, Marden-Walker syndrome, and Moebius
studies by Janicki et al,18 Gurnett et al,22 Paton et al,23 syndrome all had bilateral surgery. Clearly, these num-
and Madzivire et al24 (range, 13.5% to 30.5%). Given the bers are small due to the rarity of many of these
frequency of nonidiopathic CTEV, it is surprising that conditions. Although they reflect our experience, they do
how little individual attention has been given to the study not constitute proof that these trends will apply in all
of its management. We believe that the spectrum of eti- cases.
ologies documented in our series is an accurate reflection It is worth noting, nonetheless, that the Ponseti
of the relative importance of each condition. Our patient method has significant benefits even for those patients
population is not biased toward or against any 1 syn- who ultimately require extensive surgical release. In this
drome or disease. subgroup, the Pirani score fell from a mean score of 5.8
Compliance with brace wearing has been identified initially to 3.7 before surgery. In small deformed feet,
as the most critical factor in relapse of deformity in idi- where access to the posteromedial aspect of the foot
opathic CTEV.25 Despite an initial success rate of 91% in is difficult, such an improvement is a huge advantage
correcting cases of nonidiopathic deformity, our overall intraoperatively.
success rate (those who did not require extensive surgical In our idiopathic cohort, the initial correction rate
release) was only 63%. However, it should be recognized of 98%, relapse rate of 8%, and rate of extensive cor-
that children and their parents face many challenges in rective surgery (2%) are comparable with those in pa-
maintaining compliance with the abduction orthoses. tients reported by Ponseti10 (98%, 11%, and 2.5%,
Slippage and potential blistering are ever-present con- respectively).
cerns. In our series, 1 case of recurrence occurred in a This is a relatively large, prospective study. Ap-
patient with PFFD who was unable to wear an abduction proximately two thirds of patients with nonidiopathic
orthosis due to her significant leg length discrepancy. CTEV avoided extensive surgical soft tissue release due to
Moreover, increasing difficulty in tolerating an orthosis a successful period of Ponseti manipulation and casting.
may be secondary to (rather than the cause of) pro- At present, the results are only of a short-term nature.
gression of the underlying condition. Given the natural history of many of these conditions, it
Despite the altered sensation present in several of is likely that there will be a number of late recurrences in
our patients, we had no significant skin problems in our future years. A further study at some time in the future
series. We attribute this to a combination of factors. We may confirm or refute this. Equally, we are unable to
now use “soft cast” plasters that may be easily removed comment on the long-term outcomes of the patients who
by parents if they feel that their child is in pain due to a underwent surgical correction.
slipped cast. When patients have progressed to boots, we Nonetheless, we believe that the Ponseti method
often opt to use Mitchell boots that are made of a softer should now be widely adopted for the treatment of non-
material than the Markell boots and may be padded over idiopathic CTEV. The proven inferior results of tale-
bony prominences. Most importantly, we try to educate ctomy and extensive soft tissue release7,14 need not
parents on skin care and provide good support for any be inflicted on all patients with nonidiopathic CTEV.
concerns or queries that they may have, even outside With the support of an educated and well-motivated
normal working hours. multidisciplinary team, the Ponseti method may be most
At an average follow-up of 38.7 months, 37% of our successful in the care of these challenging, but very
treated nonidiopathic CTEV feet had undergone extensive rewarding, children.

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Moroney et al J Pediatr Orthop  Volume 32, Number 6, September 2012

ACKNOWLEDGEMENTS 13. Drummond DS, Cruess RL. The management of the foot and ankle
The management of these challenging children is very in arthrogryposis multiplex congenita. J Bone Joint Surg Br.
1978;60:96–99.
much a multidisciplinary process. It would be impossible 14. Legaspi J, Li YH, Chow W, et al. Talectomy in patients with
without the skill and enthusiasm of our 2 clinical nurse recurrent deformity in club foot. A long-term follow-up study.
specialists—Catherine Howells and Olga Gallagher—and J Bone Joint Surg Br. 2001;83-B:384–387.
the rest of our team. 15. Hennigan SP, Kuo KN. Resistant talipes equinovarus associated
with congenital constriction band syndrome. J Pediatr Orthop.
2000;20:240–245.
16. Boehm S, Limpaphayom N, Alaee F, et al. Early results of the
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