Tag Archives: clubfoot

Clubfoot in children: An overview

by Mohammad A. Hegazy1, Hossam M. Khairy1, Abdelmonem A. Hegazy2*, Sherif M. El-Aidy1

The Foot and Ankle Online Journal 13 (4): 10

Clubfoot is a case of complex defects affecting mostly newborns and children. Its management represents a great challenge especially in cases of severe resistant cases. Unfortunately, it did not receive the necessary attention in text-books and literature regarding its all aspects under one title. In this article, we aimed to highlight clubfoot focusing pathological anatomy of clubfoot and consequent management options. This might help physicians and surgeons in proper management. Clubfoot shows main anatomical defects including foot cavus, adduction, varus and equinus; the most prominent bony defect has been found in foot talus. Most cases respond to conservative reconstruction including the Ponseti method. However, some cases including those associated with other congenital anomalies are severe and resistant to such conservative management. These cases are suggested to be managed by talectomy that represents a salvage procedure to give a plantigrade foot.

Keywords: clubfoot, anatomy, pathology, talectomy

ISSN 1941-6806
doi: 10.3827/faoj.2020.1304.0010

1 – Orthopedic Surgery Department, Faculty of Medicine, Zagazig University, Zagazig, Egypt.
2 – Anatomy and Embryology Department, Faculty of Medicine, Zagazig University, Egypt.
* – Corresponding author: dr.abdelmonemhegazy@yahoo.com

Clubfoot called talipes equinovarus represents a global health problem affecting 1-2/1000 of live births all over the world. It involves both feet in about half of the reported cases. In unilateral cases, the club foot slightly more affects the right side than the left. It also affects males more than females. The case is mostly managed through conservative measures including the Ponseti method [1]. However, such conservative methods are often unsuccessful for correction of deformity in cases of severe rigid clubfoot [2].

Because of unresponsiveness to many options of management, cases of severe rigid equinovarus deformity represent a challenging problem for orthopedic surgeons. Understanding the pathological anatomy of such cases facilitates their management. There are many surgical options for correction of the deformity including release of soft tissues, Ilizarov correction using an external fixator and triple arthrodesis [3]. However, all these measures also fail to obtain a stable plantigrade foot. Therefore, talectomy has been adopted as a salvage procedure aiming to correct or minimize the deformity. This procedure was used for treatment of deformities of paralytic calcaneovalgus [4].

Although many researchers have tried to clarify the pathogenesis of congenital clubfoot, the exact cause is still obscure. Most of cases (about 80%) occur in normal physical and mental children [5].

In this review, we highlight the clubfoot and its pathological anatomy in a trial for more understanding of its pathogenesis, and hence its proper management. In addition, the options for management were reviewed with a focus on the role of talectomy in severe and resistant cases.


The clubfoot was investigated through the database of PubMed, Google Scholar, Web of Science, Scopus and others. Data were collected, represented and discussed into the following main subtitles: epidemiology, etiology, pathological anatomy, diagnosis, classification, features and management.

Results and Discussion

Epidemiology of Clubfoot

Clubfoot is one of the commonest and oldest recognized orthopedic anomalies. It was recognized as early as the ancient Egyptians. It has been reported that Egyptian Pharaohs Siptah and Tutankhamun were suffering from clubfoot. The first medical document describing the case was produced in 400 BC by Hippocrates [6].

Clubfoot is encountered in about one per thousand of the live births, varying from one race to another. It affects approximately 150,000-200,000 of newborns all over the world each year. About 80% of cases are encountered in developing countries. It affects both limbs in about 50% of cases. The unilateral clubfoot occurs in the right lower limb more than the left one with a ratio of 2-1 respectively [7]. It might be isolated congenital defect or associated with other serious anomalies especially if it is severe and bilateral [8].

Moreover, the incidence is more in males than females with a ratio of 2-1 respectively. The prevalence in Western Europe and the USA accounts 1-1.4/1000 live births. A lower incidence has been detected in Chinese and Japanese but a higher amongst Polynesian as well as South Africa black populations [5].

Etiology of clubfoot

Clubfoot is classified into two main types; congenital and acquired. Acquired form isn’t inborn-error. It might be caused by associated diseases. These include vascular causes such as Volkmann Ischemic Paralysis and neurogenic diseases comprising poliomyelitis, meningitis, sciatic nerve damage. Congenital clubfoot could be subdivided according to their causes into idiopathic or non-idiopathic types. Idiopathic clubfoot is mostly an isolated birth defect. The causes of non-idiopathic clubfoot include teratologic anomalies, generalized syndromes (e.g. diastrophic syndrome) and neurological diseases of known defects (such as spina bifida). The cases of non-idiopathic clubfoot are commonly associated with the presence of other anomalies with poor response to management either conservative or operative treatment [9].

Many theories have been proposed to explain causes of idiopathic type of clubfoot occurring in normal newborns. One of these theories is the mechanical one laid by Hippocrates which assumes that clubfoot might be caused by an increased intrauterine pressure during pregnancy [10]. This theory is disputed because of absence of association of clubfoot with most cases of overcrowded uterus such as cases of twins, large babies or polyhydramnios [9].

Smoking of mothers at pregnancy might be a cause of increased risk of clubfoot [11]. Another cause might be presence of an aberrant muscle, noticed at surgery to be inserted into the deep fascia of foot opposite the medial side of calcaneus [12]. Zimny, et al., found abnormal contracted plantar fascia with fibroblastic contracture similar to that found in Dupuytren’s disease [13].

There are some findings supporting the genetic factor in etiology of clubfoot. These observations include the increased incidence in cases of previous family history. Such history was found in about 25% of isolated cases of clubfoot. Moreover, there is a coincidence of clubfoot of monozygotic twins of about 33% compared with only 3% in dizygotic. It has been suggested that a variety of apoptotic genes are involved in cell death cascade and consequent shaping the defects in clubfoot [14].

Another study attributed the idiopathic clubfoot to be due a disturbance in the germ cells causing arrest of the foot development at the 5-week stage of fetal life. At this period, called physiological clubfoot stage, the foot bones resemble the shape and position of clubfoot [15]. Similarly, Victoria-Diaz and Victoria-Diaz, stated that the development of the human foot passes into three stages in-utero [16]. In the first (the 15-mm embryo length) stage, the foot appears in the same line with the leg. In the second “embryonic” stage (30-mm embryo length), the lateral side of the leg elongates more in relation to the medial aspect causing the foot to assume the clubfoot position. By the third “fetal” stage (50-mm), the medial side of the leg and foot develops to correct the position assuming that seen normally in the newborn.


It is a common cause for rigid talipes equinovarus found since birth. Its disorders include multiple joints so is commonly called arthrogryposis multiplex congenita. It affects two or more joints with no obvious cause or pathogenesis. There is no specific diagnosis, but this depends mainly on clinical findings [17]. The affected joints involve the large joints of limbs such as hip, knee, ankle, shoulder, elbow and wrist as well as the joints of the foot and hand. The joints of the ankle and foot are mostly affected leading to deformity of clubfoot. Such deformity is resistant to manipulation and conservative measures. It could be improved by radical soft tissue surgery; however, the relapses’ rate is high. Therefore, talectomy is recommended as a salvage or even primary procedure in such cases of severe rigid clubfoot [18].

Spina bifida

Spina bifida is a congenital anomaly in which there is failure of fusion of the two halves of the neural arch of one or more vertebrae. It includes many types [Figure 1]. Spina bifida occulta is a non-symptomatic condition and was discovered at investigation. Other types include meningocele with protrusion of sac of meninges through the vertebral defect and meningomyelocele comprising the nervous tissue and meninges’ protrusion [19]. It represents one of the non-idiopathic causes of rigid and resistant clubfoot. This foot deformity occurs in about 30-50% of cases of spina bifida. It is evident at birth; and its incidence differs according to the site of spina bifida lesion. Clubfoot is noticed in about 90% of cases in case thoracic and lumbar spina bifida whilst the sacral region is associated with clubfoot in 50% of patients.

Figure 1 Types of spina bifida.

Figure 2 Talo-calcaneal (TC) and talo-first metatarsal (TF) angles: A) Normal foot; B) Clubfoot.

Since the case is rigid, non-surgical management including stretching, splinting and serial casting commonly fails. It might be an indication for surgical interference [20].

Pathological anatomy of clubfoot

Despite the cause of the clubfoot is still uncertain to treat the cases accordingly, knowledge of the pathological anatomy remains the main guide in repair and management. Many theories were proposed to explain the bony changes occurring in the foot.

Scarpa [21] mentioned that the osseous defects in the talus are the primary causative factor in pathogenesis of the clubfoot. This suggestion has been supported by other studies later on. They added that talus defects are noticed in all clubfoot types [15].

Other authors postulated that the calcaneus is the primary fault in pathogenesis of clubfoot attributing their suggestion to the ossification of the calcaneus that noticed to appear before that of talus [22].

In clubfoot, there is a complex musculoskeletal alteration in the foot to be directed down and medially resembling the club used to hit the golf ball. The anatomical deviations are summarized by the 4 letters of the word “cave” (C=Cavus, A=Adduction, V=Varus and E=Equinus) (Figure 2) [8].

The main features include the followings (Figures 2,3):

1- Cavus: It is the increased convexity (or longitudinal arch) of the foot. This is caused by increased plantar flexion of the first metatarsal bone in relation to the hindfoot. It is caused by contracture of the plantar aponeurosis. Also, there is contracture of the plantar and spring ligaments.

2- Adduction: The forefoot is adducted. The cuneiforms and metatarsals are deviated towards the midline but they appear of normal shape.

3- Varus: It is the inversion and adduction of the hindfoot. In other words, the heel forms varus angulation. The calcaneus is adducted, plantar flexed and rotated inwards below the talus to lie nearly in the same line.

4- Equinus: The entire foot shows an increased plantar flexion at the ankle joint.

The soft structures on the medial and posterior aspects of the foot are shortened and thickened keeping the position of the foot in adduction and varus with equinus respectively. These structures include deltoid, talonavicular and spring ligaments as well as tibialis posterior tendon medially. Also, there is tightness of the posterior gastrocsoleus complex [8].

Joints of the foot are distorted due to malposition of its bones. The equinus deformity occurs mainly at the ankle joint, but others also particularly subtalar joints contribute to the deformity of the foot. The talo-calcaneo-navicular joint is dislocated with contracture of the soft tissue surrounding it with the ankle. Such contracture includes the joint capsule, ligaments, tendons and their sheaths and muscles. The contractures are noticed in talofibular, calcaneofibular, spring, deltoid and plantar bifurcated Y ligaments as well as the tendo Achilles. Moreover, there are plantar contractures affecting the intrinsic flexors of the toes, abductor hallucis and plantar aponeurosis [9].

Figure 3 Normal foot compared with varus, cavus and equinus.

Figure 4 Diagrams showing superior view of talus in normal and clubfoot.


Rehman and Faruqui found that all clubfeet show nearly the same defects of foot skeleton. The most prominent features are the small sized foot and distorted talus [23]). The talus is distorted in size, shape and orientation in case of clubfoot. Its head and neck are smaller than normal and directed down and medially. Talus is directed in a plantar flexion position. The trochlear articular surface of the body of the talus is less convex. Despite any change in the shape of the talus body, it is reduced in size [22].

The most apparent change in the talus is found in its anterior part (Figure 4). The neck of the talus is always short, directed medially and plantarward on the body. It is sometimes not apparent. The angle between the long axis of the neck and that of the body is much reduced; in clubfoot it is about 115-135° versus normal foot of 150-155°. The articular surface for navicular is no longer directed forwards like that of normal foot, but deviated medially and plantarward [23]. In case of clubfoot, the degree of medial deviation of the head and neck of the talus is more significant than that of the plantar one. Moreover, the volume of talus in congenital clubfoot is much reduced than that of the normal foot [24].

The navicular bone is dislocated medially lying opposite the tibial malleolus. This causes the front of the head of the talus to be uncovered and pointed towards the lateral instead of medial side [5].

Cuboid is also displaced medially along with the anterior end of the calcaneus displaying foot lateral convexity [9].


The calcaneus in clubfoot is generally of normal shape but slightly smaller than that of normal foot. It is shifted into equinus, varus and medial rotation along the distorted neck of talus [23]. Epeldegui also noticed a small-sized calcaneus; but added the presence of another deformity in the calcaneus represented by twisting along its long axis with consequent rotational deformities of foot longitudinal axis [25].

Diagnosis of Clubfoot

Prenatal diagnosis

Although most cases of clubfoot are diagnosed at birth, they could nowadays be recognized prenatally. Ultrasonography (US) advent into health care enables the physicians to recognize the case during the intrauterine life. Prenatal US examination has a positive predictive value over 80% with no false results [8]. However, using US at pregnancy doesn’t differentiate between its grades. Its importance lies in preparing the parents to be ready to the degree of postnatal management as early as possible. Also, discovery of the case in-utero motivates the doctors to search about the other congenital anomalies that might be associated with clubfoot in up to 50% of affected fetuses [26]. Diagnosis of the case prenatally could be recognized around the twenty-two weeks of gestation. Once noticed, assurance of the parents is essential, focusing that the case is treatable and not an indication for termination of pregnancy. The parents should know that the clubfoot is not a disabling condition but requires patience, compliance and frequent visits and follow-up to achieve excellent results. Prenatal counseling also is important to offer for the parents the options of treatment and results expected from each line management [27].

Postnatal clinical examination

Immediately after birth, the typical clubfoot is diagnosed by the orthopedic physician through taking full history from the parents and inspection of the shape of the foot. Then, the doctor does palpation of foot bones and surrounding connective tissues for abnormal position and contractures respectively. The affected cases should be thoroughly investigated from the head to the foot toes to exclude the other associated congenital defects. All body joints are examined for presence of contracture characterizing arthrogryposis [7]. The case also should be differentiated from paralytic clubfoot such as multiple congenital malformations. The main aim of assessment of cases is to differentiate the postural talipes from the true clubfoot and to define its severity. Postural type is usually easily correctable to the normal anatomical state at birth or in the infantile period after manipulative strapping [28]. Assessment of movements of foot such as inversion/eversion, adduction/abduction of the forefoot, supination/pronation is important. Evaluations of the gait in neglected cases, range of movements and static weight bearing alignment as well as noticing the differences in-between the two limbs are also valuable [29].

Radiological assessment

Though many radiological modalities have been introduced in health investigations, the clinical assessment remains the more informative one in cases of clubfoot. Up till now, there is no consensus regarding the great value of x-ray in routine evaluation and management of such cases. The standard radiograph does not give an accurate single method for evaluation and further management of cases of clubfoot [30]. This is because most tarsal bones are not ossified at birth except the talus and calcaneus of which ossified centers appear in the plain radiographs as rounded ossicles. However, the ossification centers of metatarsal bones are evident at birth; and become sufficiently ossified by the age of 3 to 4 months [31].

The information achieved from radiological examination is usually taken from standing anteroposterior and lateral views. Specific measurements are taken for assessment of cases of clubfoot. These include the angle between talus and calcaneus in lateral and anteroposterior planes as well as the relation of equinus of calcaneus to the longitudinal axis of tibia. The angle between the long axis of first metatarsal and that of talus (called Meary’s angle) is an indication for cavus for forefoot [29].

Magnetic resonance imaging (MRI) and ultrasound also have been used in evaluation of management. MRI is introduced particularly to detect the gradual response for conservative treatment. However, it is important to remember that the initial decision of treatment depends mainly on the clinical grounds [8].

It is important to note that even in well-corrected cases, some radiological residual defects might persist for a long time in the well-treated cases. These include a small volume of tarsal bones and flattening of talar dome [8].

Classification and Features of Clubfoot

Although there is no agreement about the methods of scoring or classification of clubfoot till now, it’s essential to adopt one of them to predict the appropriate line of management and to assess the progress of the lesion and its prognosis [31].

There are many methods of classification. One of them was mentioned by Nordin, et al., and Diméglio, et al. [28,32]. They classified the lesion into four degrees as follows:

1. Degree 1, benign “postural or positional” clubfoot: The position of the foot is easily corrected through casting and physiotherapy.

2. Degree 2, moderate “soft more than stiff” clubfoot: This degree accounts about 33% of cases. It responds to casting in more than 50% of cases. The others not responding to this line of treatment within 7-8 months may need surgical interference.

3. Degree 3, severe “stiff more than soft” clubfoot: It occurs in 61% of cases. More than 50% of them don’t respond to conservative treatment and are mostly released surgically.

4. Degree 4, very severe “stiff” clubfoot: It is irreducible; and congenital anomaly. It often occurs in both sides; and necessitates extensive surgical repair (Table 1).

Grade Score Type Reducibility
I < 5 Benign > 90% soft-soft, resolving
II 5 to < 10 Moderate > 50% soft-stiff, reducible, partly resistant
III 10 to < 15 Severe < 50% stiff-soft, resistant, partly reducible
IV 15 to < 20 Very severe < 10% stiff- stiff, resistant

Table 1 Diméglio, et al., classification of congenital talipes equinovarus [32].

Cummings and Lovell mentioned another three degrees of severity [33]:

1- The first “mild” one is called postural clubfoot and needs no great effort in correction.

2- The second degree, called moderate clubfoot mostly does not need surgical interference. The foot is easily flexible with absence of the transverse crease. Cases of this degree usually respond to realignment followed by keeping the foot for a while in plaster cast.

3- The last “severe” degree is called defiant clubfoot. It is characterized by a small foot and tight skin with a transverse crease in the sole. The heel isn’t easily identified because of the fatty tissue covering the calcaneus. Fortunately, this type is less common than the previous type; but is resistant to conservative treatment and surgical interference is nearly inevitable.

Another common scoring or classification has been proposed by Pirani, et al. [34]. They divided the cases into three categories according to the six clinical findings; and gave three scores from 0-1 to each, as follows: 0 for absence, half for mild and one for presence of fixed contracture. The investigated signs are related to the midfoot and hindfoot. Those related to the midfoot are: 1- Curve of the lateral border of foot, 2- Presence of medial foot crease, & 3- Palpation of the head of talus laterally. The hindfoot signs are: 1- Posterior foot crease, 2- Palpation of heel, & 3- Equinus rigidity (Table 2). The clinical investigation is done through looking, feeling and moving the foot.


Clinical examination

Midfoot Hindfoot
Signs Score Signs Score
Looking Lateral border No deviation from straight line 0 Posterior crease No heel crease 0
Medial deviation distally 0.5 Mild heel crease 0.5
Severe deviation proximally 1 Deep heel crease 1
Feeling Head of talus Reduced talonavicular joint 0 Empty heel sign Hard heel (calcaneus is in normal position) 0
Subluxed but reducible talonavicular joint 0.5 Mild softness 0.5
Irreducible talonavicular joint 1 Very soft heel (calcaneus is not palpable) 1
Moving Medial crease No medial crease 0 Rigidity of equinus Normal dorsiflexion 0
Mild medial crease 0.5 Foot is plantigrade with knee extended 0.5
Deep crease altering contour of foot 1 Fixed equinus 1

Table 2 Pirani scoring of clubfo

The high Pirani score, the more severe clubfoot, i.e. score of six means severe case, while zero is of a normal foot [8].

Neglected clubfoot

Although clubfoot is a major concern for the parents and the whole family as it is a major obvious crippling defect affecting the walking of the child, many cases especially in developing countries are neglected. This neglect might be due to difficulty to gain medical care, shortage in skilled surgeons or ignorance of the parents. In these cases, the children are forced to walk on the dorsolateral side of the foot. The affected cases are unable to wear the shoes in their feet aggravating the problem. Neglecting cases are associated with sequelae and complications [35]. The affected neglected foot is usually reduced in size. This is caused by tethering of the abnormal tight ligaments and tendons that tightly catch the foot impeding its further growth. The clubfoot is often associated with a limitation or even absence of the movements in the subtalar and midtarsal joints leading to their stiffness [36]. The complications include pain, thickened pigmented skin and ulceration. Such ulceration could result in osteomyelitis that might lead to amputation of the foot at the end stages [37].

Management of Clubfoot

The goals of management of clubfoot are to obtain an obvious plantigrade, stable and straight foot with no need to modify shoes and a good range of foot motion without pain [5].

From the beginning of the 20th century till now, the management of clubfoot has fluctuated between conservative and surgical procedures. In the early 20th century, treatment was begun for older children; therefore, it was difficult and resistant to correct [38]. Treatment of clubfoot should start soon after birth; initiated within the first week of life. This is because the joints and bones of the infant’s foot are more flexible to allow successful repair. Treatment depends on the clubfoot degree. Mild flexible clubfoot is usually treated by non-operative or conservative treatment. Surgical treatment is reserved for cases not responded to the first line of treatment and for severe rigid clubfoot [39].

Conservative treatment

History of conservative treatment starts since Hippocrates in 400 BC. Conservative measures could be divided into two phases pre- and post-Ponseti eras. The importance of the Ponseti technique does not mean exclusion of other lines of treatment including surgery depending on the condition of each case [9].

There are many methods for conservative treatment; however, the International Clubfoot Study Group, established in 2003, has approved only three of them as the standardized conservative lines for the treatment of clubfoot; Kite, Ponseti and Bensahel techniques [9].

Kite’s technique

This method was introduced in the USA by Dr. Kite in the 1930s. It assumes gradual correction of each of the main deformities separately instead of simultaneous repair. The method depends on a series of manipulations and castings. To start in the next step, the previous one should be completely repaired. The technique starts with correction of med-tarsal adduction, internal rotation and calcaneal varus; and lastly the equinus. The session of manipulation is about five minutes, followed by lower limb immobilization [40]. The immobilization is performed through doing a slipper cast extending to a level below the knee that is changed every week. At the end of the procedure the foot is put in a Denis Browne Bar. The success of this method in various studies ranged from a low as19% to high up to 90% [9]. This method has not been performed longer because of long term treatment, with casting over two years as well the unsatisfactory outcomes in more than 50% of cases [41].

Ponseti method

The method was developed based on extensive anatomical study of the foot. It involves serial manipulations and casting, followed by a further three weeks in a cast. This method is widely adopted as the method of choice in many centers all over the world; however, most of the treated cases have a residual equinus and necessitate tenotomy of the calcaneal tendon. Tenotomy is usually indicated when the hindfoot can’t achieve dorsiflexion for 15 degrees after correction [42].

Relapses can be frequent after treatment by the Ponseti method. Such relapses need the child to wear an abduction brace for three months. Thereafter, the abduction brace is advised to be worn by the child only at night till reaching the age of four years to avoid the relapse. The deformity could be corrected by applying counter pressure on the head of the talus during application of the casts with keeping the foot in abduction and lateral rotation [6]. Failure rate of the Ponseti method accounts for 3-5% of cases; and this needs surgical interference [43].

The parents are recommended to be aware that the treatment by this method extends up to at least four years; and this needs their cooperation and serious commitment throughout the process of management.

Bensahel ‘French’ technique

This method was suggested in France in the 1970s; and entered English literature in the 1980s. It depends on daily sessions of physiotherapy manipulations of the infant’s clubfoot for thirty minutes for two months. This is followed by stimulation of muscles at the foot, especially the peronei, to keep the passive reduction achieved by physiotherapy manipulations; and then catching and holding the foot in the new position by adhesive strips. This daily management is reduced to three sessions per week till the age of six months. Then, taping of the foot continues until the infant begins walking. Thereafter, the foot is supported with a splint at night for another two to three years. About half of cases are totally improved; and the other cases only require simple surgical interference through a posterior release. Disadvantages of this method include the long-term management for many years that necessitates close contact of the child and his parents with the hospital [9]

Richards, et al.. compared the outcome results of non-operative techniques of the Ponseti and French methods [44]. They concluded that non-significant better results were achieved by the Ponseti method; and added that the poor results were the same in both methods accounting for about 16% of cases in each one. However, He, et al., analyzed the clinical outcome of the different conservative measures of clubfoot treatment in a total 1435 cases [45]. They recommended the Ponseti method to be the first method of choice for management of such cases. They added that this method gives better results than the other conservative techniques; and minimizes numbers of the cases requiring surgical intervention.

Complications of conservative treatment

The main complications are summarized in two points; the first is the recurrence or failure of treatment while the second is the false correction leading to a condition called ‘rocker-bottom foot’. In such a situation, there is overstretching of the foot without actual correction of the equinus of the hindfoot, but the foot tends to take up position towards the neutral position. If this is detected during treatment, manipulations should be stopped in particular to equinus correction; and the foot is rested in a supporting splint in equinus to allow healing of breach in the midfoot. Otherwise, more disability and pain will occur with more difficulty treating the deformed foot [14].

Surgical treatment

The best time for surgical interference for treating the clubfoot is still controversial. Many authors suggest that clubfoot is best to be operated at 9-10 months. At this age, the infant begins to pull himself to attain standing and hence beginning to put body weight on the feet. This might benefit from the gravity in the process of repair [14]. Despite other authors agreeing to perform surgery as early as possible, no evidence of better results to those operated early in life has been found [46]. The authors added that in very young ages, there may be difficulty to recognize the small bones and other cartilaginous structures in addition to presence of abundance of fatty tissues. Surgery in such a condition requires a meticulous surgeon to avoid residual scarring and stiffness resulting from dealing with immature structures. Therefore, surgeries must be performed by expert surgeons at specialized clubfoot centers established for such purposes especially in large hospitals receiving thousands of births per year [14].

The list of types of operations performed to correct clubfoot that begin at 1891 till now is endless; and no single one gives long-lasting repair [9].

The surgery in the current use can be divided into three categories; the first involving soft tissue, the second with bones and the last including both soft tissue and bones. Operations including bones are usually done for children at an older age; and are considered salvage procedures [6].

I. Soft tissue surgery

This procedure includes release, lengthening or transfer of tight structures such as ligaments and tendons and/or deforming soft tissue structures, e.g. joint capsules. The deformity should be corrected before further surgery [28].

Posterior release is the simplest soft tissue release surgery. It includes lengthening of the tendoAchilles and capsulotomy of the talocrural and subtalar joints as well as cutting the posterior talofibular and calcaneofibular ligaments. Such ligaments act as a tether for the talus and calcaneus so their contracture might prevent normal foot dorsiflexion [46].

Other comprehensive soft tissue release could involve posteromedial release of the soft contractures of the posterior, medial and subtalar soft tissues. This might allow correct alignment of bones. Also, circumferential release and posterior or tibialis tendon transfer might be performed to permit dynamic balance between the invertor and evertor muscles [28,33].

II. Combined skeletal and soft-tissue procedures

Evan proposed a procedure depending on shortening the lateral column of the foot in order to realign the midtarsal joint [47]. The author performed it through a closing-wedge resection of the calcaneocuboid joint associated with a modified soft tissue release of medial aspect of the foot.

Lunderg mentioned performing an opening-wedge osteotomy through the calcaneus in addition to the insertion of a bone wedge [48]. This is concomitant with posteromedial soft tissue release in order to obtain full repair.

Ilizarov found that gradual distraction of structures e.g. soft tissues and bones especially in young ages could lead to cellular proliferation of such structures [49]. The author stated that using the Ilizarov external fixator for management of clubfoot affords correction at many planes. Other authors investigated such methods with soft tissue release and bone procedures; and concluded that this method might give good results in management of neglected and relapsed clubfoot [50].

III. Skeletal surgery

There is a general consensus that surgery on the bones of the foot is reserved for older children or cases resistant to other measures of treatment [5].

Management of neglected and severe resistant rigid clubfoot represents a great challenge to orthopedic surgeons. This is because the excessive and extensive open surgical manipulations could lead to postoperative scarring and many complications. Therefore, talectomy has been suggested to be a salvage surgical method for correction of such cases of clubfoot [3].


Currently, there is an increasing attention all over the world to the role of talectomy in correction of cases of rigid clubfoot not responding to other measures [3].

Talectomy has been suggested as a salvage procedure to manage severe resistant cases of clubfoot. The operation gives satisfactory results through removing the talus “the most distorted bone”. The patient could wear shoes with a plantigrade foot following talectomy. It is a safe and one-step surgery without major complications [30].


Clubfoot is a challenging orthopedic problem especially in severe resistant cases. Such cases are mostly encountered associated with other congenital anomalies. Thorough investigation of cases to exclude the association of other anomalies is essential to determine the line of treatment. Therefore, it is suggested to perform talectomy as a salvage procedure in cases of failure of other conservative measures; and it is also suggested as the first line in cases of severe resistant clubfoot associated with other congenital anomalies. Future studies are also recommended particularly to reveal genetic involvement in its etiology. This might be of benefit to alleviate or minimize occurrence of clubfoot.

Conflict of Interest: None

Funding: None


We would like to thank ALL members of the Orthopedic Surgery Department, Zagazig University. All diagrams are made by the corresponding author “A A Hegazy”.


  1. Smythe T, Mudariki D, Kuper H, Lavy C, Foster A: Assessment of success of the Ponseti method of clubfoot management in sub-Saharan Africa: a systematic review. BMC Musculoskelet Disord. 2017;18:453. https://doi.org/10.1186/s12891-017-1814-8
  2. Meena S, Sharma P, Gangary SK, Lohia LK: Congenital clubfoot. J Orthop Allied Sci. 2014;2:34-39. 10.4103/2319-2585.145593
  3. El-Sherbini MH, Omeran AA: Midterm follow-up of talectomy for severe rigid equinovarus feet. J Foot Ankle Surg. 2015;54:1093-1098. https://doi.org/10.1053/j.jfas.2015.07.004
  4. Joseph TN, Myerson MS: Use of talectomy in modern foot and ankle surgery. Foot Ankle Clin. 2004;9:775-785. 10.1016/j.fcl.2004.06.007
  5. Diepstraten AF: Congenital clubfoot. Acta Orthop Scand. 1996;67:12. 10.3109/17453679608994698
  6. Matuszewski L, Gil L, Karski J: Early results of treatment for congenital clubfoot using the Ponseti method. Eur J Orthop Surg Traumatol. 2012;22:403-406. 10.1007/s00590-011-0860-4
  7. Balasankar G, Luximon A, Al-Jumaily A: Current conservative management and classification of clubfoot: A review. J Pediatr Rehabil Med. 2016;9:257-264. 10.3233/PRM-160394
  8. Cooke SJ, Balain B, Kerin CC, Kiely NT: Clubfoot. Curr orthop. 2008;22:139-49. https://doi.org/10.1016/j.cuor.2008.04.002
  9. Anand A, Sala DA: Clubfoot: Etiology and treatment. Indian J Orthop. 2008, 42:22-8. 10.4103/0019-5413.38576
  10. Alberman ED: The causes of congenital clubfoot. Arch Dis Childh. 1965, 40:548-54. 10.1136/adc.40.213.548
  11. Honein MA, Paulozzi LJ, Moore CA: Family history, maternal smoking, and clubfoot: an indication of a gene-environment interaction. Am J Epidemiol. 2000; 152:658-65. 10.1093/aje/152.7.658
  12. Abo El-Fadl SM: An Unusual aberrant muscle in congenital clubfoot: An intraoperative finding. J foot ankle surg. 2013;52:380-382. 10.1053/j.jfas.2012.12.012
  13. Zimny ML, Willig SJ, Roberts JM, D’Ambrosia RD: An electron microscopic study of the fascia from the medial and lateral sides of clubfoot. J Pediatr Orthop. 1985; 5:577-581. 10.1097/01241398-198509000-00014
  14. Sadler B, Gurnett CA, Dobbs MB. : The genetics of isolated and syndromic clubfoot. J Child Orthop. 2019; 13:238-244. 10.1302/1863-2548.13.190063
  15. Irani RN, Sherman MS: The Pathological Anatomy of Club Foot. JBJS. 1963:45-52.
  16. Victoria-Diaz A, Victoria-Diaz J: Pathogenesis of idiopathic clubfoot. Clin Orthop Relat Res. 1984;5:14-24.
  17. Bamshad M, Heest AEV, Pleasure D: Arthrogryposis: A Review and Update.. J Bone Joint Surg Am. 2009;91:40-46. 10.2106/JBJS.I.00281
  18. Chotigavanichaya C, Ariyawatkul T, Eamsobhana P, Kaewpornsawan K: Results of primary talectomy for clubfoot in infants and toddlers with arthrogryposis multiplex congenita. J Med Assoc Thai. 2015;98:38-41.
  19. Hegazy A: Clinical Embryology for medical students and postgraduate doctors. LAP Lambert. Academic Publishing, Berlin; 2014.
  20. Swaroop VT, Dias L: Orthopaedic management of spina bifida-part II: foot and ankle deformities. J Child Orthop. 2011;5:403-414. 10.1007/s11832-011-0368-9
  21. Scarpa A (1818): A Memoir on the Congenital Club Feet of Children, and on the Mode of Correcting that Deformity. Clinical Orthopaedics and Related Research. 1994, 4-7.
  22. Windisch G, Anderhuber F, Haldi-Brändle V, Exner GU: Anatomical study for an update comprehension of clubfoot. Part I: bones and joints. J Child Orthop. 2007;1:69-77. https://doi.org/10.1007/s11832-006
  23. Rehman F, Faaruqi NA: Morphometric changes in talus of clubfoot – A gross observation in human fetuses. Int J Morphol. 2012;30:217-221. 10.4067/S0717-95022012000100039
  24. Itohara T, Sugamoto K, Shimizu N, et al.: Assessment of talus deformity by three-dimensional MRI in congenital clubfoot. Eur J Radiol. 2005;53:78-83. 10.1016/j.ejrad.2004.03.003
  25. Epeldegui T: Deformity of talus and calcaneus in congenital clubfoot: an anatomical study. J Pediatr Orthop B. 2012;21:10-15. 10.1097/BPB.0b013e32834de59b
  26. Bass A: Update on club foot. Pediatrics and child health. 2011;22:239-242. https://doi.org/10.1016/j.paed.2011.11.007
  27. Sarbu I, Socolov D, Carp S, Ciongradi CI: The prenatal counseling importance in congenital talipes equinovarus treatment. Procedia Social Behavioral Sci. 2015; 205:688-692. https://doi.org/10.1016/j.sbspro.2015.09.108
  28. Nordin S, Aidura M, Razak S, Faisham WI: Controversies in congenital clubfoot: Literature review. Malays J Med Sci. 2002;9:34-40.
  29. Graf A, Wu K, Smith PA, Kuo KN, Krzak J, Harris G: Comprehensive review of the functional outcome evaluation of clubfoot treatment: a preferred methodology. J Pediatr Orthop B. 2011;21:20-27. 10.1097/BPB.0b013e32834dd239
  30. Hegazy MA, Khairy HM, El-Aidy SM: Role of Talectomy in Severe Resistant Clubfoot in Children. J Foot Ankle Surg (Asia Pacific) 2019;6:29-38. 10.5005/jp-journals-10040-1105
  31. Miyagi N, Lisaka H, Yasuda K, Kaneda K: Onset of ossification of the tarsal bones in congenital clubfoot. J Pediatr Orthop. 1997;17:36-40.
  32. Diméglio A, Bensahel H, Souchet P, Mazeau P, Bonnet F: Classification of clubfoot. J Pediatr Orthop B. 1995;4:129-136. 10.1097/01202412-199504020-00002
  33. Cummings RJ, Lovell AW: Current concept review: Operative treatment of congenital idiopathic club foot. J Bone Joint Surg. 1988;70:1108-1112.
  34. Pirani S, Hodges D, Sekeramyi F. : A reliable and valid method of assessing the amount of deformity in the congenital clubfoot deformity (The Canadian Orthopaedic Research Society and the Canadian Orthopaedic Association conference proceeding) . 2008;90-B(Suppl I):53.
  35. Penny JN: The neglected clubfoot. Techniques Orthop. 2005;20:153-166. 10.1097/01.bto.0000162987.08300.5e
  36. Sobel E, Giorgini R, Velez Z: Surgical correction of adult neglected clubfoot: Three case histories. J Foot Ankle Surg. 1996;35:27-38. https://doi.org/10.1016/S1067-2516(96)80009-3
  37. Mirzayan R, Early SD, Matthys GA, Thordarson DB: Single-stage talectomy and tibiocalcaneal arthrodesis as a salvage of severe, rigid equinovarus deformity. Foot Ankle Int. 2001;22:209-213. 10.1177/107110070102200307.
  38. Ippolito E, Farsetti P, Valentini MB: Management of Clubfoot. In: Bentley G. (eds) European Surgical Orthopaedics and Traumatology. Springer, Berlin, Heidelberg; 2014. https://doi.org/10.1007/978-3-642-34746-7_157
  39. Parvizi J: High yield othopaedics. Philadelphia, Pennsylvania, Saunders; 2010.
  40. Derzsi Z, Nagy O, Gozar H, Gurzu S, Pop TS: Kite versus Ponseti method in the treatment of 235 feet with idiopathic clubfoot. Medicine. 2015;94:1-4. 10.1097/MD.0000000000001379
  41. Lovell WW, Farley D: Treatment of congenital clubfoot. Ona J. 1979;6:453-456.
  42. Maranho DA, Volpon JB: Congenital Clubfoot. Acta Ortop Bras. 2011;19:163-169.
  43. Parsa A, Moghadam MH, Jamshidi MHT: Relapsing and residual clubfoot deformities after the application of the Ponseti method: A contemporary review. Arch Bone Joint Surg. 2014:7-10.
  44. Richards BS, Faulks S, Rathjen KE, Karol LA, Johnston CE, Jones SA: A comparison of two nonoperative methods of idiopathic clubfoot correction: the Ponseti method and the French functional (physiotherapy) method. J Bone Joint Surg Am. 2008;90:2313-2321. 10.2106/JBJS.G.01621
  45. He JP, Shao JF, Hao Y: Comparison of different conservative treatments for idiopathic clubfoot: Ponseti’s versus non-Ponseti’s methods. J Int Med Res. 2017;45:1190-1199. 10.1177/0300060517706801
  46. Cummings RJ, Lovell AW: Operative treatment of congenital idiopathic club foot. J Bone Joint Surg Am. 1988;70:1108-1112.
  47. Evan D. : Relapsed Club Foot. J. Bone and Joint Surg. 1961;43:722-733. https://doi.org/10.1302/0301-620X.43B4.722
  48. Lunderg BJ: Early Dwyer Operation in Talipes Equinovarus. Clin Orthop. 1981 Jan-Feb, 223:7.
  49. Ilizarov GA: The tension-stress effect on the genesis and growth of tissues. Part I. The influence of stability of fixation and soft-tissue preservation. Clin Orthop Relat Res. 1989 Jan, 249:81.
  50. Fernandes RMP, Mendes MDS, Amorimb R, Preti MA, Sternick MB, Gaiarsae GP: Surgical treatment of neglected clubfoot using external fixator. Rev Bras Ortop. 2016;51:501-508. 10.1016/j.rboe.2016.08.002


Distal lower extremity manifestations in spina bifida patients of the Yucatan Peninsula: A 24-year retrospective case series

by Alexandra Heidtmann, BS1; Lahari Madulapally, BS, MA1; Luis Rodriguez Anaya, DPM2*; Daniel Cawley, DC, MS2

The Foot and Ankle Online Journal 13 (4): 8

Spina Bifida, a rare congenital disorder with an incidence of 7.85 per 10,000 births in Mexico. It results from the failed closure of the neural tube leading to the incomplete development of the neural arches. This case series is part of the Yucatan Crippled Children’s Project that began in 1996 by Charles Southerland, Doctor of Podiatric Medicine and former professor of Barry University’s School of Podiatric Medicine. All patients in this study were assessed and treated at the Red Cross hospital in the city of Merida, Yucatan, Mexico. Attendings, residents and medical students travel to the Yucatan Peninsula four times a year for a period of one week. Given that this study was built from reports of medical mission trips that occur four times a year with limited resources and time, the lack of documentation of treatment plans and follow-ups made it difficult to identify surgical procedures and assess the success of surgeries. Additionally, we did not have access to the patients birth records or their mothers medical records to accurately determine the etiology of their deformities. Based on our data, we conclude that intervention should be considered as early as possible in any flexible deformity to prevent them from becoming rigid.

Keywords: Spina Bifida, talipes equinovarus, clubfoot, adductovarus, calcaneo valgus, Ponseti, osteomyelitis

ISSN 1941-6806
doi: 10.3827/faoj.2020.1304.0008

1 – Fourth Year Medical Student at Barry University School of Podiatric Medicine, Miami, Florida.
2 – Assistant Professor Barry University School of Podiatric Medicine
* – Corresponding author: LARodriguez@barry.edu

Spina Bifida, a rare congenital disorder with an incidence of 7.85 per 10,000 births in Mexico. It results from the failed closure of the neural tube leading to the incomplete development of the neural arches [1]. Spina bifida is the result of genetic and non-genetic factors that interfere with the folding and closure of the neural tube. In its most severe form, meningomyelocele, the neurons of the spinal cord are exposed to amniotic fluid resulting in neuronal death. In addition, the spinal cord and meninges protrude through the midline bony defect of the back [2].

The clinical manifestation of a meningomyelocele is dependent on the spinal level of involvement and the presence of cerebral involvement and hydrocephalus [3]. Sensory and motor impairments are commonly present below the level of the lesion causing alterations in the bowel and bladder function, muscle paresis and paralysis, and sensory loss. Impairment is classified by the level of neurosegmental involvement determined by the strength of specific muscle groups [3]. Nearly all patients with spina bifida will experience manifestations in their feet, especially those cases involving the thoracic and lumbar spinal regions [4,5]. Previous studies have reported that the most common manifestation of spina bifida in the feet are talipes equinovarus, equinus, vertical talus, calcaneal deformities, and cavovarus [4,6,7]. The aim of this study is to analyze the incidence of various distal lower extremity manifestations and their long-term effects on spina bifida patients of the Yucatan Peninsula.


This case series is part of the Yucatan Crippled Children’s Project that began in 1996 by Charles Southerland, Doctor of Podiatric Medicine and former professor of Barry University’s School of Podiatric Medicine [8]. All patients in this study were assessed and treated at the Red Cross hospital in the city of Merida, Mexico. Attendings, residents and medical students travel to the Yucatan Peninsula four times a year for a period of one week.

From 1999 to 2020, we retrospectively analyzed 1,489 patients that were seen by physicians from the Yucatan Crippled Children’s Project. Among the total, we identified 25 patients, 17 male and 8 female, with history of Spina Bifida and concomitant lower extremity deformities. From the 25 patients, 15 patients had bilateral lower extremity deformities and 10 patients had unilateral deformities, leading to a total of 40 limbs. The ages ranged from 3 months old to 43 years old, with a total average age of 11.33 years. The mean age for rigid deformities was 15.4 years, while the mean age for flexible deformities was 8.8 years.

We analyzed 3 cases of patients with a history of spina bifida and lower extremity deformities according to the clinical notes collected from the Yucatan Crippled Children’s Project.

Case Presentation 1

Case 1 is a 2-year-old male who presented to the Yucatan Crippled Children’s project clinic in March of 2007 with a chief complaint of difficulty ambulating. Patient’s family reports past medical history of birth at 38 weeks and significant time spent in the NICU due to hydrocephalus and spina bifida. Upon initial assessment, the patient was alert and oriented and showed no additional symptoms. Patient was diagnosed with bilateral flexible clubfoot deformity, as seen in Figures 1 and 2. Considering the age and the flexibility of the deformity, the conservative Ponseti serial casting technique was performed on the patient.

Figures 1 and 2 Plantar and dorsal views of bilateral clubfoot prior to Ponseti method.

Figures 3 and 4 Patient at 12 years old, 10 years after Ponseti method. AP Radiograph of bilateral feet. Pre-operative clinical picture of bilateral feet.

Figures 5, 6, 7 Intraoperative picture of left foot before and after external fixator application. AP radiograph of left foot.

The patient and his family were instructed to follow – up with the local doctor. The patient returned to the clinic in November of 2017, at 12 years old, with chief complaint of continued difficulty ambulating due to the progression of the windswept deformity (Figures 3 and 4). After assessment, the left foot was diagnosed with adductovarus and talipes equinovarus deformity. The right foot was diagnosed with forefoot adduction, midfoot abduction, and calcaneovalgus which are the three components of complex skew foot. Given the Ponseti technique applied ten years ago had failed, the deformity has worsened and progressed from flexible to rigid. The procedure consisted of application of external fixation with medial motor for gradual correction of adductovarus deformity on the left foot (Figures 5-7).

Figure 8 Bilateral flexible cavovarus deformity with ulcer on dorsolateral aspect of right foot.

Figures 9 and 10 Nine year follow-up shows rigid bilateral cavovarus deformity. The patient is confined to a wheelchair.

The patient presents to the clinic in February of 2018 for a 3 month postoperative visit after application of external fixation. It was noted that the toes were not fixated during the external fixator surgery and they developed flexion contractures within the reduction frame. The patient developed clinodactyly of all five digits of the left foot. At this date, the frame was removed and the patient began physical therapy in an attempt to reduce flexion contractures.

Case Presentation 2

Case 2 is an 8-year-old female with a past medical history of spina bifida and sensory neuropathy bilaterally. The patient presented to the clinic in July of 2011 with a chief complaint of a wound on the right foot. Upon physical exam, an open ulcer was noted on the dorsolateral aspect of the right foot along the cuboid-5th metatarsal joint (Figure 8). The wound has a beefy red base with friable granulation tissue and a circumferential macerated periwound with suspected areas of hyperkeratotic tissue. Mild hyperpigmentation and erythema is noted proximally towards the dorsum of the ankle. The patient was diagnosed with a pressure ulcer on the right foot and bilateral flexible cavovarus deformity. Ulcer was managed during the patient’s first visit prior to any surgical intervention. Upon healing of the ulcer, surgery was performed; however, the surgical technique was not recorded.

The patient was virtually contacted during Covid-19 2020 Pandemic, and sent Figures 9 and 10. The patient stated she still has insensate feet and is unable to ambulate. Deformity has progressed to rigid and she is waiting until the next Yucatan Medical Mission Trip to possibly undergo another surgery that would allow her to ambulate.

Case Presentation 3

Case 3 is a 24-year-old male with a past medical history of spina bifida, insensate feet, and chronic lymphangitis. The patient presented to the Yucatan Project Clinic in April of 2005 with a chief complaint of wounds on the right foot. Upon physical exam, ulcer on the lateral aspect of the head of the 5th metatarsal of the right foot was noted to have a 50/50 granular fibrotic base with slough in the center. The periwound consisted of hyperkeratotic tissue on the plantar aspect of the 5th metatarsophalangeal joint. Hyperpigmentation is present extending proximally on the dorsum of the foot. The second wound, located on the lateral aspect of the 5th metatarsal tuberosity of the right foot, appeared to have 75/25 fibrotic granular base with regular borders. The patient was diagnosed with active infected ulcers and bilateral cavovarus deformity (Figures 11 and 12). Initial treatment consisted of ulcer debridement and offloading of the right foot with a 3D Walker. The patient was seen in November of 2005, 7 months after initial treatment. The right foot still remained dysfunctional with chronic non-healing wounds. Radiographs show radiolucency from mid-shaft of 4th and 5th metatarsal distally to the 4th and 5th distal phalanges and thickened periosteum of the proximal end of the mid shafts of 4th and 5th metatarsals on Figure 13, suggesting osteomyelitis.

Figures 11 and 12 Dorsolateral view of right foot showing ulcer along 5th metatarsal and medial view of the right foot showing cavovarus deformity.

Figure 13 AP Radiograph of bilateral feet.

Figures 14 and 15 Preoperative view of the right foot. Intraoperative picture of the right foot after Lisfranc amputation.

Figure 16 Dorsolateral view of right foot after amputation.

Due to the lack of access to other diagnostic tools, combined with the request from the patient for a permanent solution, a LisFranc amputation was performed on the right foot and osteoset beads with vancomycin were inserted to treat the infection (Figures 14 and 15).

The patient was seen in February of 2006, 3 months after LisFranc amputation of the right foot. The surgical site healed well with good results (Figure 16). However, the cavovarus deformity remained on the left foot (Figure 17).

The patient was seen again in November of 2006 for the last time, 12 months after LisFranc amputation of the right foot. The patient redeveloped an equinus deformity of the right foot and ulcers under styloid processes bilaterally (Figure 18). The patient was treated with well-padded plastazote ankle foot orthosis (AFO).

Figure 17 Dorsolateral view of left foot.

Figure 18 Ulcer under styloid process of the right foot after amputation.


The most common lower extremity manifestation was ulcerations. In 17 ulcerated limbs, 8 were insensate and 5 developed osteomyelitis. Out of the 40 limbs, 5 ulcerated limbs had no reported gross deformity, and therefore were not included in the graphs. The remaining 35 limbs were biomechanically classified as rigid and flexible. In the 13 rigid limbs, there were 4 equinus, 3 talipes calcaneus, 7 cavus, and 2 planus feet. These were further subclassified into 6 more categories: equinovarus, pes cavocalcaneus, cavovalgus, cavovarus, pes planovalgus and no additional deformity. Rigid deformities subgroups can be seen in Graph 1. In the 22 flexible limbs, there were 9 equinus, 5 talipes calcaneus, 6 cavus, and 2 planus feet.

Graph 1 Rigid deformity of the foot.

Graph 2 Flexible deformity of the foot.

They were then subclassified into 5 more categories: calcaneovalgus, calcaneovarus, equinovarus, cavovarus, and no additional deformity. Flexible deformities are illustrated in Graph 2.


Lower extremity manifestations due to spina bifida are difficult to be classified and the rate of misdiagnosis and mistreatment is high [9]. Similarly to previous findings, the most frequent foot deformity in our study was flexible equinus [4,5,10]. However, we found that rigid pes cavus was the second most predominant foot deformity in the Yucatan Peninsula, contrary to previous reports. Based on the limited medical access in the area and the higher average age of patients presenting with rigid deformities (15.4 years) when compared to flexible deformities (8.8 years), we suggest that this is possibly due to years of leaving the deformity untreated.

The prevalence of spina bifida was 7.85 per 10,000 births or 0.0785% in the country of Mexico [1]. However, in the Yucatan Peninsula the prevalence was found to be significantly higher. In this study, out of the 1,489 total cases analyzed from years 1999 to 2020, 25 patients with spina bifida were identified. This shows a prevalence rate of 167.90 per 10,000 births or 1.68%. Folic acid is a nutrient that is essential to the development of the fetus. Spina bifida and other birth defects form within the initial 28 days after conception. These congenital deformities can be prevented by ensuring sufficient blood folate levels in the mother during fertile years and early fetal development [11]. In the literature, North America has been shown to have the lowest incidence of spina bifida while Asia has the highest incidence. This could be due to Canada and the United States being the first countries to mandate folic acid fortification. In addition, even though mandatory fortification with folate has been implemented in many countries, it might not be enough folic acid to reach the daily recommended dosage of 400 micrograms. Therefore, it is important for mothers before conception and in the early fetal developmental months to supplement their folic acid intake [12].

Case 1 illustrated a patient with talipes equinovarus, also known as clubfoot deformity on the left foot. This triplanar deformity includes 3 components: ankle equinus, hindfoot varus, and forefoot adduction. Traditionally, there is a higher prevalence of clubfoot in males with a ratio of 2:1 to female and approximately 50% of the cases are bilateral. A few etiologies have been described in the literature, mainly divided into idiopathic and non-idiopathic. Idiopathic consists of limited intrauterine position due to a larger size of the fetus or smaller frame of mothers, while non-idiopathic includes a history of congenital deformities such as spina bifida, cerebral palsy, and meningitis. Clinical presentation of patient 1 at birth predisposed him to a higher risk of developing clubfoot given he is a male with a history of spina bifida. We do not have additional history of patient 1 such as birth weight and height, however, these factors could have also played a role in the patient developing clubfoot [13]. The Ponseti technique, a conservative treatment, was attempted when the patient was 2 years old; however, this technique is only proven to be successful in patients with flexible clubfoot up to 120 days of age [14]. On the right foot, the patient has had a long-standing complex skewfoot with forefoot adduction, midfoot abduction, and calcaneovalgus. The unsuccessful result of the Ponseti method on the left foot, combined with years of the patient not returning for medical assistance, led the bilateral deformity to become rigid on both feet.

Case 2 presented a 2-year-old female with history of spina bifida, insensate feet, active ulcer on the right foot, and flexible bilateral cavovarus deformity. Cavovarus involves a high longitudinal plantar arch, hindfoot varus, forefoot equinus, and pronated first ray in the stance phase of gait. If this deformity is present bilaterally, the most likely etiology is a neurological condition; however, if it is present unilaterally the etiology can be related to trauma such as pilon fractures or talar neck fractures [15]. In a flexible cavovarus foot, surgical correction could be achieved through extensive plantar release and metatarsal osteotomies. However, at the time of the patient’s first visit, physicians from the Yucatan Project prioritized the management of the ulcer prior to correcting any gross deformity. Due to the long period of treatment for ulcer management and limited medical access in the Yucatan Peninsula, the patient did not seek medical help for many years. Recent literature has described that if left untreated, cavovarus deformity can progress into fibrosis of the plantar fascia, shortening and tightening of the achilles tendon leading to excess pressure under metatarsal heads, overloading of the lateral aspect of the foot leading to stress fractures of the 5th metatarsal and more rarely, the cuboid. In addition, it can cause inadequacy of the lateral ligaments and tendons leading to instability of anterolateral ankle and lateral talus [15]. During the 2020 Covid-19 pandemic, we reached out through social media and discovered the patient was no longer ambulating. The patient described a rigid deformity with insensate feet and showed interest in undergoing another surgery, so she could possibly walk again. In a mature foot, surgical intervention might require aggressive techniques including midtarsal osteotomies, calcaneal osteotomies and triple arthrodesis [16]. Final decision for a surgical procedure will only be done in person once full updated history and radiographs are taken.

Case 3 showed the most severe result that could come from insensate lower extremity in spina bifida patients if left untreated for long periods of time: amputation. This patient was first seen at 24 years old, when his rigid cavovarus deformity was present since birth. This deformity caused chronic non-healing wounds that developed into osteomyelitis. Osteomyelitis can be defined as an infectious agent which causes inflammation of the bone. The hallmark of chronic osteomyelitis is the progression of inflammation to tissue necrosis and destruction of bone trabeculae and bone matrix caused by an infectious agent. This is usually accompanied by fragments of bone lacking blood supply which can become separated to form sequestra and continues to host and spread bacteria despite antibiotic treatment. The fifth metatarsal, first metatarsal, calcaneus, and first digit distal phalanx are the four structures with the highest incidence of developing osteomyelitis in the foot [17]. This case emphasizes the need of spina bifida patients with concomitant lower extremity deformities to seek medical help at a young age to avoid the progression of the deformity and consequently loss of a limb.

Given that this study was built from reports of medical mission trips that occur four times a year with limited resources and time, the lack of documentation of treatment plans and follow-ups made it difficult to identify surgical procedures and assess the success of surgeries. Additionally, we did not have access to the patients birth records or their mothers medical records to accurately determine the etiology of their deformities.


The types of foot and ankle deformities seen in spina bifida are diverse in etiology, age and gender of the patients. We discovered the most common lower extremity manifestations of spina bifida in the Yucatan Peninsula are flexible equinus and rigid pes cavus. The mean age of patients with rigid deformities was almost twice as the mean age of the patients with flexible deformities. Zang, et al., concluded that equinovarus requires immediate treatment while valgus deformities can have delayed intervention [15]. Based on our data, we conclude that intervention should be considered as early as possible in any flexible deformity to prevent them from becoming rigid.


We would like to thank all attendings, residents and students involved in the Yucatan Crippled Children Project along with the International Foot & Ankle Foundation for Education and Research. Additionally, we would like to thank the local Red Cross Hospital in the city of Merida.


  1. Gunay H, Sozbilen MC, Gurbuz Y, Altinisik M, Buyukata B. Incidence and type of foot deformities in patients with spina bifida according to level of lesion. Childs Nerv Syst. 2016;32(2):315-319. doi:10.1007/s00381-015-2944-7.
  2. Swaroop VT, Dias L. Orthopaedic management of spina bifida-part II: foot and ankle deformities. J Child Orthop. 2011;5(6):403-414. doi:10.1007/s11832-011-0368-9.
  3. Sharrard WJ, Grosfield I. The management of deformity and paralysis of the foot in myelomeningocele. J Bone Joint Surg Br. 1968;50(3):456-465.
  4. Atta CA, Fiest KM, Frolkis AD, et al. Global Birth Prevalence of Spina Bifida by Folic Acid Fortification Status: A Systematic Review and Meta-Analysis. Am J Public Health. 2016;106(1):e24-e34. doi:10.2105/AJPH.2015.302902.
  5. Frischhut B, Stöckl B, Landauer F, Krismer M, Menardi G. Foot deformities in adolescents and young adults with spina bifida. J Pediatr Orthop B. 2000;9(3):161-169. doi:10.1097/01202412-200006000-00005.
  6. Krähenbühl N, Weinberg MW. Anatomy and Biomechanics of Cavovarus Deformity. Foot Ankle Clin. 2019;24(2):173-181. doi:10.1016/j.fcl.2019.02.001.
  7. Westcott MA, Dynes MC, Remer EM, Donaldson JS, Dias LS. Congenital and acquired orthopedic abnormalities in patients with myelomeningocele. Radiographics. 1992;12(6):1155-1173. doi:10.1148/radiographics.12.6.1439018.
  8. Yucatán Crippled Children’s Project. (2012). Retrieved from (https://www.internationalfootankle.org/philanthropy/crippled-childrens-project/).
  9. Mandell JC, Khurana B, Smith JT, Czuczman GJ, Ghazikhanian V, Smith SE. Osteomyelitis of the lower extremity: pathophysiology, imaging, and classification, with an emphasis on diabetic foot infection. Emerg Radiol. 2018;25(2):175-188. doi:10.1007/s10140-017-1564-9.
  10. Cavalheiro S, da Costa MDS, Moron AF, Leonard J. Comparison of Prenatal and Postnatal Management of Patients with Myelomeningocele. Neurosurg Clin N Am. 2017;28(3):439-448. doi:10.1016/j.nec.2017.02.005.
  11. McCluskey WP, Lovell WW, Cummings RJ. The cavovarus foot deformity. Etiology and management. Clin Orthop Relat Res. 1989;(247):27-37.
  12. Feldkamp, M., Sanchez, E., & Canfield, M. (2014). International Clearinghouse of Birth Defects Surveillance and Research – Annual Report 2014.
  13. Awang M, Sulaiman AR, Munajat I, Fazliq ME. Influence of Age, Weight, and Pirani Score on the Number of Castings in the Early Phase of Clubfoot Treatment using Ponseti Method. Malays J Med Sci. 2014;21(2):40-43.
  14. Copp AJ, Adzick NS, Chitty LS, Fletcher JM, Holmbeck GN, Shaw GM. Spina bifida. Nat Rev Dis Primers. 2015;1:15007. Published 2015 Apr 30. doi:10.1038/nrdp.2015.7.
  15. Zang J., Qin S., Shi L. (2020) Lower Limb Deformity Caused by Spina Bifida Sequelae and Tethered Cord Syndrome. In: Qin S., Zang J., Jiao S., Pan Q. (eds) Lower Limb Deformities. Springer, Singapore.
  16. Kancherla V. Countries with an immediate potential for primary prevention of spina bifida and anencephaly: Mandatory fortification of wheat flour with folic acid. Birth Defects Res. 2018;110(11):956-965. doi:10.1002/bdr2.1222.
  17. Broughton NS, Graham G, Menelaus MB. The high incidence of foot deformity in patients with high-level spina bifida. J Bone Joint Surg Br. 1994;76(4):548-550.


‘Fast Casts’: Evidence Based and Clinical Considerations for Rapid Ponseti Method

by April Sutcliffe1, Kolini Vaea2, John Poulivaati2, Angela Margaretpdflrg Evans3,4

The Foot and Ankle Online Journal 6 (9): 2

The Ponseti method of correction of congenital clubfoot is recognized as the preferred management technique for this pediatric deformity. The original method has been subtly modified over time in response to clinical experience and research findings. Most recently, two randomized controlled trials have shown that less time is needed for each serial cast immobilization. Clinical cases from the Kingdom of Tonga are presented to illustrate the clinical use of more rapid plaster cast changes – the ‘fast casts’ modification incorporating increased manual manipulation time, within the Ponseti method. The Pirani score was used to monitor the clubfoot correction between each plaster cast change for each baby. In all feet the Pirani scores reduced sequentially with shorter periods of casting. Shorter duration of cast immobilization – ‘fast casts’ – can be used with many advantages for the clinical setting. Less time in plaster can at least halve the corrective phase of Ponseti management without compromising results. In addition, there are possible benefits for families from distant locations, for babies being less prone to skin irritations, and less difficult day-to-day baby care related to long leg plaster casts. These factors may benefit compliance and overall treatment outcomes.

Key words: clubfoot, Ponseti, pediatric, foot

Accepted: August, 2013
Published: September, 2013

ISSN 1941-6806
doi: 10.3827/faoj.2013.0609.002

Address correspondence to: Department of Podiatry, Lower Extremity and Gait Studies (LEGS) Research Program, La Trobe University, Bundoora, Melbourne, Australia. Email: angela.evans@latrobe.edu.au

1Sydney Children’s Hospital, High Street, Randwick, Sydney, Australia
2Vaiola Hospital, Tonga
3Department of Podiatry, Lower Extremity and Gait Studies (LEGS) Research Program,
La Trobe University, Bundoora, Melbourne, Australia
4Health and Rehabilitation Research Institute, AUT University, Auckland, New Zealand

The Ponseti method has taken the developed world by storm in the last decade, becoming acknowledged as the optimal treatment for congenital clubfoot deformity.[1,2]

Cited as the most significant and potentially debilitating congenital pediatric orthopaedic deformity, talipes equino varus, has littered the pages of historic tomes, medical journals and textbooks alike.[3] The Egyptian boy king, Tutankhamen; the tragic poet, Lord Byron; and celebrated stage and screen actor, Dudley Moore; eponymously all male, were born and/or lived with clubfoot deformity. [4]

Whilst management with splints, binding, and plaster casts has been evident across the hundreds and thousands of years in which clubfoot deformity is referenced, the 20th century saw such conservative measures subsumed by surgical correction, and notably the posterior medial release (PMR). [5-7] The PMR is a joint invasive procedure, which also severs to lengthen, all the soft tissue structures found contracted on the medial and posterior aspects of the infant clubfoot.[8]

In the 21st century, surgical correction of clubfeet has been firmly denounced.[9] Both retrospective concerns and reviews, and prospectively designed studies have shown the poor outcomes, in terms of pain and function, resulting from the PMR and akin surgical procedures.[7]

Simultaneously, the Ponseti method, developed and named after the orthopedic specialist Ignacio Ponseti[10], has been investigated both retrospectively and in many prospective randomized controlled trials (RCTs), and found not only to give the best clinical outcomes, but to also be a more economical management, when compared to surgery – the rare health care setting finding of a ‘win:win’.[11]

Much investigation of Ponseti’s original method has occurred in the last decade.[7,12,13] Whilst it’s superior outcomes for management of congenital clubfoot has met with universal consensus, this has also resulted in considerable refinement of the technique.[11,14,15]

The original Ponseti method

The duration of each serial plaster cast, a fundamental aspect of the basic weekly casts which made up the original Ponseti method[16] now has good evidence for amendment.

The original method described by Ponseti involves a series of plaster casts changed weekly for a period of five to six weeks, followed by percutaneous elongation of the Achilles tendon and application of a final cast for three weeks. The foot abduction bracing phase, is commenced immediately after the post tenotomy cast is removed.

There is now strong evidence to suggest that accelerated frequency of cast changes has comparable outcomes to those of the original Ponseti method.[17] with the benefit of limiting time spent is casts during the corrective phase of treatment.

The evidence for, and implication of, ‘fast casts’

It was first revealed that casts changed every five days, instead of the originally prescribed seven days, gave the same results – potentially saving ten to 12 days in the initial casting phase.[18] Two more recent RCTs have shown that casts changed twice (even three times) each week attain the same correction as weekly casts. [17, 19]

The halving of the casting phase from an average six weeks to three weeks, without compromising results, has clear advantages. Less time immobilized in plaster casts is intuitively preferable for the baby, and their parents or caregivers. Shorter durations of each corrective cast reduces the likelihood and extent of undetected skin pressure lesions, and at least halves the overall corrective phase, such that babies commence the (virtually) full-time boots and bar phase over three months, at a younger and possibly more amenable age. With the consistently demonstrated and positive correlation between successful use of the maintenance boots and bar, and lessened relapse of clubfoot correction – starting the boots and bar habit earlier within the rapid development that hallmarks infancy – may be more helpful than at first glance considered.[20-22]

How can the notion of ‘fast casts’ be applied clinically, and what are the possible pitfalls as well as benefits?

Illustrative use of the ‘fast casts’ technique

Two cases from Tonga, the country with the world’s highest incidence of congenital clubfoot deformity[23], are included in this review. In Tonga, a pacific island country geographically comprised of numerous islands, clinical use of the ‘fast casts’ method facilitates coordination with the availability of surgical expertise to perform Achilles tenotomies, as well as accelerated progress of babies through the casting stage. Both of the case-study babies were cast and re-cast four times in one week. This is more rapid and intense than might normally occur due to the visit from the off-shore surgeon occurring the following week (the local surgeon has now undertaken training for tenotomy procedures).

Fig 1 Baby J

Figure 1 Baby J, whose data is presented in table 2.


Table 1 Baby J – left congenital clubfoot. The use of ‘fast casts’ saw this baby’s corrected and ready for the tenotomy procedure after six days (4 casts).

Fig 2 Baby S

Figure 2 Baby S, whose data is presented in table 3.


Table 2 Baby S – bilateral congenital clubfeet. The use of ‘fast casts’ corrected the cavus and adduction of the clubfoot deformity, but made no change to the equinus component, which required the tenotomy for correction (as indicated by the initial Pirani score).

As the Tables 1 and 2 show, both babies showed consistent correction of their foot deformity with manipulation and casting. (Fig. 1 and 2) The Pirani scores reduced consistently within the initial corrective phase, showing the value of using this demonstrably reliable and objective measure. Further, the initial Pirani scores of 5 and 5.5 respectively, heralded the very likely need for tenotomies.[24] Indeed, the hindfoot scores equaled or approximated the total Pirani scores after the casting phase, signaling the residual equinus aspect of the deformity. It must be stated that similarly to the findings of the clinical trial by Xu et al[17], that these Tongan cases also underwent ‘more rather than less’ manipulation prior to casting. Whilst the effect of manipulation time has not been formally studied, histological investigation directs maintained loading of ligaments to promote the lengthening or ‘uncrimping’ of these structures.[25] Might it be that more attention to, and time spent, carefully manipulating clubfoot correction is able to render cast time less relevant?

Considerations, variations, and further questions

There are many factors to consider when contemplating the use of ‘fast casts’ as part of the Ponseti clubfoot correction method.

Firstly, there is now very good evidence to support shortening cast time[17] for the typical, congenital clubfoot deformity.

Secondly, the convenience for parents travelling with infants to distant clinics for treatment which necessitates time away from home, work, and family, a common occurrence in developing countries, may be greatly improved.[26-28] If, as on average, a baby requires six casts, the time away from home/work may be reduced from six weeks to two weeks. This could provide great savings for costs incurred whilst living away from home, and time lost from work. In turn, compliance may also benefit.

Thirdly, less time immobilized in plaster is probably advantageous for the baby in terms of reduced skin sore issues, easier bathing, more normal motor development and possibly lessens the risk of osteopenia.[29]

Notable in the current findings on faster casting is the longer manipulation time, (two minutes) specified by Xu, et al., [17], an additional departure from the original Ponseti protocol, and also the long follow up time of this study, as opposed the otherwise similar Malawi trial.[19]

It is important to appreciate that all accelerated casting studies and trials have addressed the typical congenital clubfoot, and that the effects and use in syndromic[11] or complex clubfoot types[30] are unknown.

The application of best available evidence to any health care setting is important, particularly if there are clear benefits to the recipients of this care. The rescheduling of the weekly clubfoot clinic for casting, to at least twice weekly, is now a possible shift in contemporary evidence based practice.


The Ponseti method continues to be the best approach to correction of the typical congenital clubfoot. There is now high-level evidence to support changing casts after three days or less, which greatly reduces the time infants spend immobilized in plaster.

The pre-casting manipulation is important and indications are that more time spent may be beneficial in correcting the clubfoot deformity.

In developing countries where travelling to clinics necessitates time away from home, work, and family, the adoption of ‘fast casts’ can reduce costs to families, and perhaps help to improve compliance and overall outcomes.


1.           Steinman S, Richards BS, Faulks S, Kaipus K. A comparison of two nonoperative methods of idiopathic clubfoot correction: the Ponseti method and the French functional (physiotherapy) method. Surgical technique. JBJS 2009 91A (Suppl 2): 299-312. [PubMed]
2.           Carroll NC. Clubfoot in the twentieth century: where we were and where we may be going in the twenty-first century. J Pediatr Orthop 2012 21: 1-6. [PubMed]
3.           Dobbs MB, Morcuende JA, Gurnett CA, Ponseti IV. Treatment of idiopathic clubfoot: an historical review. Iowa Orthop J 2000 20: 59-64. [PubMed]
4.           Anand A, Sala D. Clubfoot: Etiology and treatment. Indian J Orthop 2008 42: 22-28.  [PubMed]
5.           Manzone P. Clubfoot surgical treatment: preliminary results of a prospective comparative study of two techniques. J Pediatr Orthop 1999 8: 246-250. [PubMed]
6.           Zionts LE, Zhao G, Hitchcock K, Maewal J, Ebramzadeh E. Has the rate of extensive surgery to treat idiopathic clubfoot declined in the United States? JBJS 2010 A92: 882-889. [PubMed]
7.           Halanski MA, Davison JE, Huang J-C, Walker CG, Walsh SJ, Crawford HA. Ponseti method compared with surgical treatment of clubfoot: a prospective comparison. JBJS 2010 A92: 270–278. [PubMed]
8.           Laaveg S, Ponseti I. Long-term results of treatment of congenital club foot. JBJS 1980 62A:23-31. [PubMed]
9.           Morcuende J, Dolan L, Dietz F, Ponseti I. Radical reduction in the rate of extensive corrective surgery for clubfoot using the Ponseti method. Pediatrics 2004 113: 376-80. [PubMed]
10.         Ignacio Ponseti [Internet]. Wikipedia. [cited 2013 Jan 29]. Available from: http://en.wikipedia.org/wiki/Ignacio_Ponseti 
11.         Dobbs MB, Gurnett CA. Update on clubfoot: etiology and treatment. Clin Orthop and Rel Res  2009 467: 1146-1153. [PubMed]
12.         Niki H, Nakajima H, Hirano T, Okada H, Beppu M. Ultrasonographic observation of the healing process in the gap after a Ponseti-type Achilles tenotomy for idiopathic congenital clubfoot at two-year follow-up. J Orthop Sci 2013 18: 70-75. [PubMed]
13.         Carroll N. Editorial: Clubfoot: What Have We Learned in the Last Quarter Century? J Pediatr Orthop 1997 17: 1-7.  [PubMed]
14.         Rijal R, Shrestha BP, Singh GK, Singh M, Nepal P, Khanal GP, Rai P. Comparison of Ponseti and Kite’s method of treatment for idiopathic clubfoot. Indian J Orthop 2010 44: 202-207. [PubMed]
15.         Andriesse H, Roos EM, Hägglund G, Jarnlo G-B. Validity and responsiveness of the Clubfoot Assessment Protocol (CAP). A methodological study. BMC Musculoskelet Disord 2006 7: 28. [PubMed]
16.         Ponseti I. Clubfoot management. J Pediatr Orthop 2000 20: 699-700.[PubMed]
17.         Xu RJ. A modified Ponseti method for the treatment of idiopathic clubfoot: a preliminary report. J Pediatr Orthop 2011 31: 317-319. [PubMed]
18.         Morcuende J, Abbasi D, Dolan L. Results of an accelerated Ponseti protocol for clubfoot. J Pediatr 2005 25: 623-625. [PubMed]
19.         Harnett P, Freeman R, Harrison WJ, Brown LC, Beckles V. An accelerated Ponseti versus the standard Ponseti method: a prospective randomised controlled trial. JBJS 2011 B93: 404-408. [PubMed]
20.         Garg S, Porter K. Improved bracing compliance in children with clubfeet using a dynamic orthosis. J Children’s Orthopaedics 2009 1: 271-276. [PubMed]
21.         Boehm S, Sinclair M. Foot abduction brace in the Ponseti method for idiopathic clubfoot deformity: torsional deformities and compliance. J Pediatr Orthopaedics 2007 27: 712-716. [PubMed]
22.         Ippolito E, Fraracci L, Farsetti P, Di Mario M, Caterini R. The influence of treatment on the pathology of club foot. CT study at maturity. JBJS 2004 B86: 574-580. [PubMed]
23.         Chapman C, Stott NS, Port RV, Nicol RO. Genetics of club foot in Maori and Pacific people. J Med Genet 2000 37: 680-683. [PubMed]
24.         Shack N, Eastwood D. Early results of a physiotherapist-delivered Ponseti service for the management of idiopathic congenital talipes equinovarus foot deformity. JBJS 2006 88: 1085-1089. [PubMed]
25.         Ponseti I. Treatment of congenital club foot. JBJS 1992 74: 448-454.[PubMed]
26.         Pirani S, Naddumba E, Mathias R, Konde-Lule J, Penny JN, Beyeza T,  Mbonye B, Amone J, Franceschi F.Towards Effective Ponseti Clubfoot Care: The Uganda sustainable clubfoot careproject. Clin Orthop Rel Res 2009 467:1154-1163. [PubMed]
27.         Evans AM, Van Thanh D. A review of the Ponseti method and development of an infant clubfoot program in Vietnam. JAPMA 2009 99: 306-316. [PubMed]
28.         Evans AM. Preliminary evaluation of implementing the Ponseti method for correction of clubfoot in Vietnam. J Children’s Orthop 2010 4: 553-559. [PubMed]
29.         Lourenço AF, Morcuende JA. Correction of neglected idiopathic club foot by the Ponseti method. JBJS 2007 89B: 378-381. [PubMed]
30.         Ponseti IV, Zhivkov M, Davis N, Sinclair M, Dobbs MB, Morcuende JA. Treatment of the complex idiopathic clubfoot. Clin  Orthop Rel Res 2006 451:171-176. [PubMed]