565049

research-articleXXXX

FASXXX10.1177/1938640014565049Foot & Ankle SpecialistFoot & Ankle Specialist

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〈 Case Report 〉 Clubfoot Associated With Congenital Constriction Band

Anil Agarwal, MS (Orthopaedics), Abbas Shaharyar, MS (Orthopaedics), and Anubrat Kumar, MS (Orthopaedics)

The Ponseti Method Perspective Abstract: Background. The clubfoot and congenital constriction band is a known association in which the clubfoot is considered as being rigid, responding poorly to casting, and requiring surgical interventions. Objective and Methods. The series describes 3 cases of clubfoot with deep ipsilateral congenital constriction bands managed with Ponseti method and immediate outcomes achieved. Results and conclusions. The feet responded favorably to casting although equinus correction required more extensive surgical interventions. The preexisting edema in distal limb often precluded a snuggly fitting cast increasing the cast numbers. Careful monitoring of vascularity is essential. Simultaneous procedures of release of constriction bands, correction of hand anomalies and equinus can be combined to the advantage of the patient. Levels of Evidence: Case report, Level IV Keywords: club foot; congenital constriction bands; Ponseti method

C

ongenital constriction band (also commonly known as Streeter’s dysplasia or amniotic band

syndrome) is rare birth disorder causing technique and the management issues related with it. circumferential constrictions of the limbs.1-4 The depression in soft tissues is of varying degrees ranging from simple Case 1 constriction to the extent of complete 3,4 A 6-month-old male child presented intrauterine amputations. Congenital with bilateral congenital constriction constriction band can be frequently bands in distal third leg with left accompanied with other anomalies, clubfoot. There was absence of bilateral including clubfeet, acrosyndactyly, and great toes with a bulbous left second toe craniofacial malformations.2 and dorsal edema (Figure 1). Both the The prevalence of clubfoot with lower limb bands were deep to fascia congenital constricting band ranges from and Pirani score at beginning of 12% to 56%.1-4 In fact, clubfeet and hand anomalies are considered a typical triad when associated with congenital Congenital constriction band can be constricting bands.3 The frequently accompanied with other clubfoot associated with constriction rings are anomalies, including clubfeet, considered to be rigid, responding poorly to acrosyndactyly, and craniofacial casting and the surgical rate in them may be malformations.” significantly high.2,3,5 The described series in literature reported these treatment was 5/6. Following application outcomes when Ponseti method was not of second cast, there was diminished commonly used. Ponseti casting method vascularity in toes. The cast was has revolutionized the management of immediately removed and foot was both idiopathic and syndromic clubfoot.6 observed several hours before We describe a series of 3 clubfeet reapplication of cast. A total of 13 casts associated with ipsilateral congenital were required before the equinus constriction bands, use of Ponseti casting



DOI: 10.1177/1938640014565049. From the Department of Pediatric Orthopaedics, Chacha Nehru Bal Chikitsalaya, Delhi, India. Address correspondence to: Anil Agarwal, MS (Orthopaedics), Department of Pediatric Orthopaedics, Chacha Nehru Bal Chikitsalaya, Geeta Colony, Delhi 110031, India; e-mail: [email protected]. For reprints and permissions queries, please visit SAGE’s Web site at http://www.sagepub.com/journalsPermissions.nav. Copyright © 2014 The Author(s)

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Figure 1. Case 1. (a) Bilateral congenital constriction bands in distal third leg with left clubfoot. Note dorsal edema in left foot and absence of bilateral great toes. (B) Correction following open tendoachilles lengthening and posterior release. (C) The constriction band was also released in same sitting.

acrosyndactyly hand on left side was treated with Ponseti technique (Figure 3). Dorsal foot edema was associated. There was 1 episode of slippage of cast after 24 hours of its application for which reapplication of cast was done, this time more snuggly fitting and more flexion at knee. The final surgical correction involved an open tendoachilles lengthening and posterior soft tissue release along with constriction band release which extended to fascia. The acrosyndactyly was also tackled in same sitting. No undesirable event took place after surgery.

Discussion correction was undertaken. In anticipation that a percutaneous tendoachilles tenotomy might not correct the equinus,3 the patient was planned for an open tendoachilles lengthening. The constriction band release in both lower limbs was also planned under same anesthesia. Intraoperatively, the open tendoachilles lengthening corrected equinus till neutral only and a further posterior release was necessary to achieve dorsiflexion till 20°. The postoperative course was uneventful.

Figure 2. Case 2. (A) The congenital constriction band was released 1 year prior elsewhere. The clubfoot was managed with Ponseti method. (B) Associated hand anomalies. (C, D) A total of 12 casts were required before a surgical intervention of posterior release corrected equinus.

Case 2 A 2.6-year-old male child with congenital constriction band distal third (intervened prior) and clubfoot left leg with absent second, third, fourth, and fifth toes and hypoplastic second and third toes right foot was treated with Ponseti method. The congenital constriction ring has been operated previously elsewhere 1 year ago without interventions for clubfoot (Figure 2). This child also had absent second digit left hand with rudimentary second and third digits right hand. The Pirani score at beginning of treatment was 4/6. Total cast required were 12. Even an open tendoachilles lengthening was not able to correct the deformity and posterior soft tissue release was required. A dorsiflexion of 20° was finally achievable. Adductus however reoccurred rapidly.

Case 3 Another 6-month-old male child with constriction band distal third leg, clubfoot (initial Pirani score 5/6) and

Clubfoot associated with congenital constriction ring is reported by several authors.1-5 Associated anomalies include Patterson’s combinations (constriction ring alone or in combination with deformity of distal part of limb, with or without lymphedema, fusion of distal portions, or intrauterine amputation), cleft lip and palate, scoliosis, and limb length discrepancy.7 Although a wellrecognized association, the clubfoot associated with congenital constriction bands has posed great challenges for the treating clinicians. The results of casting are believed to be poor regardless of whether the constricting band was in the affected leg or proximally elsewhere.3 The clubfeet associated with the constrictions were shown to be rigid, requiring frequent surgical corrections and treatment studded with complications.2-5 The accumulated experience in various series points to a poorer outcome in presence of a rigid equinovarus deformities, neurological deficits, presence of more than 1 band in 3 extremities, deep bands located in ipsilateral distal third leg with associated secondary changes such as cyanosed extremity, superficial skin ulcers, osteomyelitis, edema, or marked calf atrophy.2-4 There seems to an unverified consensus that constriction rings should be treated primarily and clubfoot intervened later to prevent circulatory compromise.2-5

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Figure 3. Case 3. (A, B, C) The triad of congenital constriction bands, club foot, and hand anomalies. (D, E) The procedures of posterior release for equinus correction, Z-plasty for congenital constriction band and release of acrosyndactyly were combined. (F) Abduction brace fitting after equinus correction.

This series is a communication of an extended application of Ponseti method for treatment of syndromic clubfoot associated with ipsilateral congenital constriction bands and immediate favorable outcomes achieved. The Ponseti method has revolutionized the management of idiopathic clubfoot and better results are also reported with use of the method in syndromic and atypical feet as well.6 Only one previous report mentions use of Ponseti method for the clubfoot associated with congenital constriction bands.8 In that report, 7 casts were given to both children aged 6 and 8 months before a tendoachilles

tenotomy was done under general anesthesia. The release of constriction bands was performed 6 months later. No complications were noted and the patients after 1 year of follow up had good results. Our experience with the 3 cases described above shows that there are several issues that should be taken care of while managing clubfoot associated with ipsilateral constriction bands. A detailed examination of the patient is a must as several other anomalies are frequently associated.2 The severity of the band, distal vascularity and neurological deficits should be carefully assessed.2 The preexisting edema in distal limb often precludes a snuggly fitting cast (Figure 3) and cast slippage can be more frequent (case 3). However, a well molded cast and more flexion of knee can take care of this problem. Following casting, the child should be monitored more closely for distal circulation and capillary filling. Absent toes which is a common finding in congenital constriction band poses difficulty in casting as it makes manipulation of foot and watch of vascularity of foot becomes more difficult especially by parents. Parents should be educated that there should not be slightest hesitancy to remove the cast on doubt of decreased vascularity. Because of casting limitations and inert rigidity, the number of casts can increase (up to 13 in the present series). We recommend an open tendoachilles lengthening for these patients in view of need of anticipated extensive surgical procedures for equinus correction. As noticed by other authors, there are chances that a percutaneous tenotomy might not correct the equinus deformity and supplementary procedures are required.4,5 Hennigan and Kuo2 have described use of average 2.1 surgeries per clubfoot in their series of 37 feet with congenital constriction bands. Of these 57 were soft tissue releases, 19 were combined bony procedures and soft tissues releases and 2 were below knee amputations.2 We combined the

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release of constrictions bands (multiple Z-plasties) and acrosyndactyly in the same sitting as equinus correction in the patients. The midline posterior incision can easily be combined with Z-plasty in lower third leg. We noted no complications by combing the 2 procedures.2 An additional operation, anesthesia and ultimately financial burden could hence be avoided by correcting both the pathologies in one sitting. No bony procedures were necessary in our series. It is essential that the parents are educated and prognosticated about the pathogenesis and manifestations of congenital constriction bands. The available management options, difficulties and complications of clubfoot associated with congenital constriction bands should be discussed in details with them. They should be made active partners in management and care of clubfoot while using the Ponseti method. They should be cautioned regarding danger signs and provided the emergency helpline number.

Conclusions Ponseti method can yield favorable results in syndromic clubfoot associated with ipsilateral congenital constriction bands. A snugly fitting cast and more flexion at knee can prevent cast slippage. The distal circulation requires close monitoring after casting. The release of constriction bands can be combined with equinus correction surgery without added risks. An open tenotomy is recommended in view of need of additional surgical procedures to correct equinus.

References 1. Askins G, Ger E. Congenital constriction band syndrome. J Pediatr Orthop. 1988;8:461-466. 2. Hennigan SP, Kuo KN. Resistant talipes equinovarus associated with congenital constriction band syndrome. J Pediatr Orthop. 2000;20:240-245.

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3. Gomez V. Clubfeet in congenital annular constricting bands. Clin Orthop Relat Res. 1996;(323):155-162.

5. Tada K, Yonenobu K, Swanson AB. Congenital constriction band syndrome. J Pediatr Orthop. 1984;4:726-730.

4. Allington N, Kumar S, Guille J. Clubfeet associated with congenital constriction bands of the ipsilateral lower extremity. J Pediatr Orthop. 1995;15:599-603.

6. Ford-Powell VA, Barker S, Khan MS, Evans AM, Deitz FR. The Bangladesh Clubfoot Project: the first 5000 feet. J Pediatr Orthop. 2013;33:40-44.

7. Patterson TJ. Congenital ring constrictions. Br J Plast Surg. 1961;14:1-31. 8. Ozkan K, Unay K, Goksan B, Akan K, Aydemir N, Ozkan NK. Congenital constriction ring syndrome with foot deformity: two case reports. Cases J. 2009;2:6696.

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Clubfoot associated with congenital constriction band: the Ponseti method perspective.

The clubfoot and congenital constriction band is a known association in which the clubfoot is considered as being rigid, responding poorly to casting,...
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