MOHAMED M. HEGAZY, M.D.*; SHERIF M. ISMAIL, M.Sc.**; AHMAD S. HASSAN, M.Sc.** and IBRAHEM S. HANTERA, M.Sc.**

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1 Med. J. Cairo Univ., Vol. 83, No. 2, September: , New Technique for the Single Stage Management of Congenital Vertical Talus by Percutaneous Tenotomies, Open Talonavicular Reduction and Percutaneous K-Wire Fixation MOHAMED M. HEGAZY, M.D.*; SHERIF M. ISMAIL, M.Sc.**; AHMAD S. HASSAN, M.Sc.** and IBRAHEM S. HANTERA, M.Sc.** The Department of Orthopedic Surgery, Faculty of Medicine, Cairo University Teaching Hospitals* and Orthopedic Department, El-Helal Hospital**, Cairo, Egypt Abstract Background: Management of Congenital Vertical Talus (CVT) may depend upon the age of initial presentation. Early management may involve a trial of single stage correction, a two-stage correction, or the addition of subtalar arthrodesis. These procedures are associated with a high complication rates. Methods: A prospective cohort of 2 feet (11 patient) with CVT all under 3 months of age of both idiopathic and teratologic variety is presented. The condition was treated by a novel technique utilizing percutaneous tenotomies of the tibialis anterior, extensor digitorum, extensor hallucis longus, peroneal longus and brevis tendons, along with the Achillis tendon. This was combined with a minimal midfoot incision to allow for reduction and K-wire fixation of the talonavicular and calcaneocuboid joints, followed by plication of the spring ligament. Patients then were casted in an above knee cast for 8 weeks. Following which, a below knee orthotic with medial arch support was applied for one year. Results: Minimum follow-up period was for twenty months. Results were evaluated using the 1-point system developed by Adelaar et al., initial correction was achieved in all cases with significant improvement of radiological parameters (p<.5). There were two cases of relapse (in a bilateral case diagnosed as Escobar syndrome). Conclusion: The described technique appears to give initial promising results. The recurrence rate is statistically related to later age of management and to teratologic varieties. However, this rate still appears to be lower than reported complication rate associated with the more extensive open surgical techniques. Level of Evidence: Level IV case series study. Key Words: Congenital vertical talus Single-stage management Tenotomies percutaneous fixation. Introduction VERTICAL talus is a condition that produces a rocker-bottom deformity of the foot. It has an Correspondence to: Dr. Mohamed M. Hegazy, Mohamedhegazy1971@gmail.com incidence of 1 in 1.. The term vertical talus should be reserved for feet with fixed equinus of the calcaneus and dorsal dislocation of the navicular on the talus. It has also been called congenital convex pes valgus [1-7]. The most appropriate name for the condition is teratologic dorsolateral dislocation of the talocalcaneonavicular joint. The first description was by Henken in 1914, and the characteristic features were well defined by Lamy and Weissman [5,8]. The classic appearance is a rocker-bottom foot, which is a foot with a convex plantar surface and the apex of the convexity at the talar head. The calcaneus is fixed in equinus and the Achilles tendon is contracted. The peronei and anterior tibialis tendons are taut, and the foot is everted into a valgus, externally rotated position. The navicular is palpable as it lies on the talar neck, where it abuts the anterior tibial surface at the front of the ankle joint. There may be some flexibility of the foot, but passive correction of the deformity is not possible [5,6,8]. Vertical talus may be present alone but more commonly is accompanied by other neuromuscular conditions. It is most often present in association with myelomeningocele and arthrogryposis, it also has been found in spinal muscular atrophy, neurofibromatosis [5,8]. Coleman and colleagues distinguished two types of vertical talus, the first with talonavicular dislocation and the second with concomitant dislocation of the cuboid on the calcaneus [9]. Treatment of congenital vertical talus is either non-operative using serial manipulation and casting or operative treatment that may be done as one- 265

2 266 New Technique for the Single Stage Management of CVT by Percutaneous Tenotomies stage procedure or two-stage procedure. Most authors prefer one-stage procedures [6]. Several authors beginning with Osmond-Clark in 1956, Herndon and Heyman in 1963, and Coleman in 197, described a staged, 2-incision reconstructive surgery [1,9,1]. After noting a high rate of complications with the two staged procedure, Ogata and Shoenecker [11] recommended a single stage procedure with a medial approach. In 1987, Seimon [12] described a single stage dorsal approach with the talonavicular joint reduced and held with k-wire and the Achillis tendon lengthened percutaneously. Stricker and Rosen (1997), have published their experience with this technique, as have Mazzocca and Thomson [13], and both groups have noted excellent results with minimal complications. Dunacn and Fixsen transferred tibialis anterior to the neck of talus and claimed good results [14]. Review of the published literature on the extensive open release performed for the treatment of CVT has shown a high rate of complications [15,16]. Consequently, some authors reported recently using the reversed Ponseti technique of casting followed by percutaneous reduction and fixation [17,18], however, a high recurrence rate was reported, that lead some authors to suggest inclusion of limited capsulotomy during the management of such conditions [19]. It was hypothesized that performing percutaneous tenotomies for the involved tendons, combined with a minimal medial incision to reduce the talonavicular joint plus percutaneous fixing both talonavicular and calcaneocuboid joint and augmenting the reduction by spring ligament plication; might reduce adequately equal results as well as reducing the stiffness and other complications associated with the relatively more extensive open incisions. Material and Methods A prospective study of 2 feet in 11 patients was carried out after obtaining the institutional review board committee approval. The study was undertaken between the years 21 and 213. The study constituted of 7 females (all with bilateral presentations), and 4 males (2 bilateral cases). There were 1 right feet and 1 left feet. The age at initial procedure ranged from 9-36 months with average age of months ± Seven feet were 1 year old or less, four feet ranged between 1-2 years, 9 feet were more than 2 years. Eleven feet were idiopathic, while 7 feet (4 patients) were associated with arthrogryposis multiplex congenital. There was also one patient with bilateral presentation diagnosed as Escobar syndrome. All the arthrogrypotic patients were presents bilaterally, however one of these patients presented to us after having the other foot treated via an open technique in another hospital outside of our institution. Four of these patients were associated with flexion contracture of the knee, two of which were successfully treated before presenting initially for foot correction. Also four of these arthrogrypotic patients were also associated with bilateral thumb in palm deformity. Eight feet had previously undergone serial manipulation and casting. Sex feet underwent a total of five casts, and two feet underwent a total of four casts (Tables 1,2) shows the various demographic data and characteristics of the involved cases. Table (1): Demographic data and characteristics of the involved cases. No. Sex: Female 14 7 Male 6 3 Age: Range 9-36 Mean ± SD 21.25± 1.21 Table (2): Side distribution of cases. Side No. % Left Right Total 2 1 Clinical examination was performed for all the included subjects to assess presence of other congenital anomalies, and to evaluate the general condition of the feet to detect skin breakage, callosities on pressure areas. In addition, the tightness of the tendons around the ankle joint were also symmetrically assessed and evaluated as to whether the tightness was mainly anterior, lateral or both. The range of motion of the ankle, subtalar, and mid-tarsal joints were recorded using a hand-held goniometer. All patients were subjected to a series of. AP and lateral of both feet were performed in addition to forced planterflexion and forced dorsiflexion lateral views. Radiographs were routinely assessed for the following %

3 Mohamed M. Hegazy, et al. 267 angles: On the AP radiograph: The talo-calcaneal angle (normally 2-4), and the talar axis-first metatarsal axis angle (normally -3), while in the lateral : The talo-calcaneal angle (normally 25-55), the tibio-calcaneal angle (normally 6-9), and the talar axis first metatarsal axis angle (normally -2). A single investigator performed the radiographic measurements, in order to minimize the possible measurement errors [2]. Patients were clinically and radiologically assessed and evaluated according to the 1-point scoring system described by Adelaar in 198 [21]. Adelaar described five clinical (with a maximum of sex attainable points) and five radiological points (maximum four attainable points). These points were assessed both pre-operative and post-operative. The maximum obtainable score was 1 with one point subtracted for each abnormality noted either clinically or radiologically. A score of 1 points was considered excellent, a score of 7, 8, or 9 points good, a score of 4, 5, or 6 points, fair; and a score of, 1, 2, or 3 points, poor. Management technique: Upon initial presentation in our institution, the initial line of management was percutaneous tenotomies combined with small medial incision for reduction of talonavicular joint plus percutaneous fixation with k-wire. Under general anesthesia, on a radiolucent operating table the patient is positioned supine. A Tourniquet is applied and patient is draped from the knee to the toes. For correction of congenital vertical talus we will use a new technique of percutaneous tenotomy of the following tendons: 1- The tibialis anterior at the level of ankle joint. 2- The extensor halluces longus just distal to the ankle joint. 3- The extensor digitorum longus just distal to the ankle joint. 4- The peroneal longus and brevis at lateral aspect of foot just distal to the lateral malleolus. 5- The achilis tendon at posterior aspect of ankle joint just above its insertion for correction of equines deformity. The approach (the medial incision): The patient is supine and the medial incision is made from a point inferior to the medial malle- olus and the heel, centered over the prominence of head talus and navicular Fig. (1). The tibialis posterior tendon is traced behind and posterior to the medial malleolus and distally to its insertion into the navicular bone and cut wit a part of the periosteum over the navicular Fig. (2). The talonavicular joint is opened medially and dorsally (capsulotomy). The redundant spring ligament is preserved plantar to the talar head and isolated from plantar attachment. Now the hindfoot can be reduced on the forefoot. This will be followed by fixation of the reduced joint by two k-wires: The first for the calcaneocuboid joint and is introduced postero-anteriorly under fluoroscopy with foot is held in maximum planterflexion and the second wire to fix the talonavicular joint and is introduced antero-posteriorly, in a reteograde fashion by insertion first in the navicular then the joint is reduced followed by advancing the k-wire into the talus under fluoroscopy Fig. (3). The redundant spring ligament is divided and tightened. Then the tibialis posterior will be sutured to the undersurface of the navicular. The wounds are closed in layers with absorbable sutures, and the k.wires are cut off and bent outside the skin, to be removed at the out-patient clinic later Fig. (4). A below knee cast is applied and molded to maintain the medial plantar arch. The cast is changed in the outpatient clinic after 1 days to check for wound closure and subsidence of edema then another will fitting cast is applied. After 8 weeks, the cast and wires is removed and a below knee brace with medial arch is applied for one year (23 hours in the first 4 months, then at bed time in the next 9 months). The patient will be followed in the out-patient clinic every month for one year. In each visit the patient is examined both standing and supine. The range of motion of the ankle and subtalar joint, and AP and lateral are obtained.

4 268 New Technique for the Single Stage Management of CVT by Percutaneous Tenotomies Fig. (1): Medial approach. Fig. (2): Tracing of tibialis posterior tendon. Fig. (3): Percutaneous fixation of the calcaneocuboid joint. Fig. (4): Immediate post-operative view after skin closure. Statistical analysis: Data were analyzed using Statistical Program for Social Science (SPSS) version 18.. Quantitative data was expressed as mean ± Standard Deviation (SD). Qualitative data were expressed as frequency and percentage. The following tests were done: Chi-square ( χ 2 ) test for significance was used in order to compare proportions between two qualitative parameters. Paired sample t-test of significance were used when comparing between related samples. Pearson correlation coefficient (r) test was used for correlating data. Probability (p-value) was also used, where p-value <.5 was considered significant, and a p-value <.1 was considered as highly significant. Results The follow-up period of the involved cases was for a mean of months (range, 2-24). There was good range of motion in all the involved foot joints except in the patient diagnosed as Escopar syndrome (two feet). Also, one patient had a mild superficial infection of wound that healed appropriately with antibiotic therapy and frequent dressing. Two feet suffered from recurrence of deformity, which was noted radiologically as dorsal sublaxation of navicular on the head of talus that occurred at the average of five months after the initial correction. Both these were in the patient that was diagnosed as Escopar syndrome. The mean radiographic measurements made at time of initial presentation were compared with the same measurements made after one year postoperatively and were also measured both in the standing and non positions (Table 3). According to the grading system proposed by Adelaar et al., [25], was documented 11 feet with good or excellent results, and nine feet with poor and fair results. On further statistical analysis of the data, it was found that the sex of the involved cases had no statistical effect on the results of the procedure. On the other hand, it was found that the primary diagnosis of the patients affect the results, as we found that patients diagnosed to have

5 Mohamed M. Hegazy, et al. 269 idiopathic CVT gave statistically better results than those diagnosed as having arthrogryposis multiplex congenita, while patient diagnosed to have Escopar syndrome (a variant of arthrogryposis multiplex congenita) gave poor results (Table 4). Similarly, by further analysis of the measured radiographic angles, and comparing the preoperative measurements with the standing one year after the procedure; a significant improvement in the lateral talo-calcaneal angle, a highly significant improvement in the lateral tibiocalcaneal angle, and a highly significant improve- ment in the lateral talar axis-base of first metatarsal axis angle was shown (Table 5). Also comparison between standing and non standing post-operative lateral performed at one year follow-up, showed that the measures angles become worse with standing. We attributed this to the hypermobility that presents between forefoot and midfoot (Table 6). The age at which the initial management is performed was also shown to be statistically significant (p-.5), with younger patients gave better results (Table 7). Table (3): Descriptive data of the study group. Min. Max. Mean ±SD : Anteroposterior talocalcaneal angle Anteroposterior talar axis-first metatarsal base angle Lateral talocalcaneal angle Lateral tibiocalcaneal angle Lateral talar axis-first metatarsal base angle -non: Lateral talocalcaneal angle Lateral tibiocalcaneal angle Lateral talar axis-first metatarsal base angle : Anteroposterior talocalcaneal angle Anteroposterior talar axis-first metatarsal base angle Lateral talocalcaneal angle Lateral tibiocalcaneal angle Lateral talar axis-first metatarsal base angle Table (4): Relation between result and primary diagnosis. 12 Primary diagnosis Result Excellent Fair Good Poor χ Total Arthrogryposis: No % Escopar: No % Idiopathic: No % Total: No % p-value % Excellent Fair Good Poor 5 Arthrogryposis Escopar Idiopathic Data are expressed as frequency and percentage data. χ 2 Chisquare test, p-value <.5 significant.

6 27 New Technique for the Single Stage Management of CVT by Percutaneous Tenotomies Table (5): Comparison between pre-operative and after 1 year standing as regard. Mean ±SD Paired differences Paired samples test Mean ±SD t p-value Anteroposterior talocalcaneal angle: Anteroposterior talar axis-first metatarsal base angle: Lateral talocalcaneal angle: (S) Lateral tibiocalcaneal angle: <.1 (HS) Lateral talar axis-first metatarsal base angle: <.1 (HS) Table (6): Comparison between after 1 year non standing and standing as regard. Mean ±SD Paired differences Paired samples test Mean ±SD t p-value Lateral talocalcaneal angle: -non standing <.1 (HS) Lateral tibiocalcaneal angle: -non standing <.1 (HS) Lateral talar axis-first metatarsal base angle: -non standing (S) non standing standing non standing standing non standing standing t-paired sample t-test; p-value <.1 HS; p-value <.5 S; p-value >.5 NS. Data are expressed as mean ± SD for parametric data. t-paired sample t-test; p-value <.1 HS; p-value <.5 S.

7 Mohamed M. Hegazy, et al. 271 Table (7): Correlation between age and score. Age (month) r Score p-value Age (month) Score p-value <.5 significant, r-pearson correlation coefficient. Discussion The current study seems to provide encouraging short term results for the use of the newly described technique in management of cases with CVT of both the idiopathic and teratologic variety. Generally, and in comparison to congenital talipus equinovarus, CVT is relatively rare deformity [2] and is more commonly associated with a more diverse variety of associated congenital abnormalities [5,6,8]. In addition, the guidelines for standard management of talipus equinovarus have been more adequately laid out. The ultimate goal of the treatment of CVT is the restoration of the normal anatmiacl relationships between the talus, navicular, and calcaneus. To achieve such goal by serial casting alone has been established to be of very limited value, and at best it contributes to the stretching of the soft tissues of the foot [17,18]. Most treatment protocols have involved more complex reconstructive procedures, either one-stage or two-stage procedures [6,7,8,2]. However, these extensive procedures also been linked with a large number and variety of complications as talus avascular necrosis [5,6], stiffness of ankle and subtalar joint, pseudoarthrosis, and under correction of the deformity that may require secondary procedures [17,18]. Our institution is a tertiary referral center for the more complicated and often neglected cases. In addition, family compliance and the ability of follow up the children regularly for serial casting, are not strictly adhered to. This explains the rela- tively higher age at initial presentation in our case series. Inclusion of percutaneous tenotomies in our case series was aimed to reduce the number of capsulotomies that might be required to achieve correction and thus prevent post-operative adhesions and stiffness, and decrease the risk of development of talar necrosis. Also, it allows for amore supple ankle and foot for post-operative casting and thus maintenance of the attained reduction. The inclusion of fixation by k-wires for cases of CVT is not a new concept. Simon utilized it in 1987, and he added percutaneous Achillis tenotomy [8]. Recently, Dobbs et al., in 26 recommended the use of talonavicular k-wire and noted that no recurrence occurred in all of the seven patients in which the talonavicular wire was used, while recurrence occurred in six out of seven cases in which k-wire fixation was not used [22]. Even more recently in 212, the same investigator advocated the use of a combination of percutaneous Achillis tenotomy combined with either k-wire fixation of the talonavicular joint or limited capeulotomies of both the talonavicular and anterior subtalar joints [16]. Excellent short-term results were reported, with recurrence in five feet that were described to have had "initial sublaxation of the calcaneocuboid joint" [16]. In our series, the addition of the second k-wire (the calcaneocuboid wire) offered some advantages. It is inserted through the calcaneus to cuboid in an antigrade fashion, and consequently offers a stronger bony hold than if it is inserted only in the cartilaginous navicular. In addition, this k-wire was purposefully left approximately an inch protruded of the posterior calcaneal heel skin, and this was used during the immediate post-operative casting to provide for an adequate lever to maneuver the calcaneus in a plantar direction, and thus cause the ankle to dorsiflex a little more and maintain the required correction during the sitting of the cast. The newly described technique of percutaneous tenotomies, pin fixation of both the talonavicular joint and calcaneocuboid joint; combined with a limited open capsulotomy and reduction of the talonavicular joint seems to provide satisfactory early results (at a mean of 2 months after the procedure) as regards the clinical appearance of the foot, and radiographic evidence of correction. Longer follow-up is necessary to determine maintenance of the correction, as well as possibly randomized comparative studies with other methods

8 272 New Technique for the Single Stage Management of CVT by Percutaneous Tenotomies of treatment. This described technique may allow children with CVT to avoid traditional extensive surgical releases while maintaining flexibility if the foot. References 1- GRUNDY M., TOSH P.A., McLEISH R. and SMIDT L.: An investigation of the centers of pressure under the foot while walking; J. Bone Joint Surg. Br., 57: 98, MORRIS J.M.: Biomechanics of the foot and ankle; Clin. Orthop. Relat. Res., 1: 1: 22, KITAOKA H.B., LUO Z.P. and AN K.N.: Threedimensional analysis of normal ankle and foot mobility; Am. J. Sports Med., 25: , McCLAY I. and MANAL K.: The influence of foot abduction on differences between two-dimensional and three-dimensional rear foot motion; Foot Ankle Int., 19: 26-31, JOHN ANTHONY HERRING: Chapter 38. Tachdjians Pediatric Orthopedics, 4 th edition, an imprint of Elsevier inc, ALAEA F., DOBBS M.B. and BOEM S.: A new approach for the treatment of congenital vertical talus; J. Child. Orthopaedic, 1: , NAPIONTEK M.: Congenital vertical talus: A retrospective and critical review of 32 feet operated on by peritalar reduction; J. Pediatr. Orthop. B, 4: , OSMOND-CLARKE H.: Congenital vertical talus; J. Bone Joint Surg. Br., 1: , COLEMAN S.S., STELLING 3 rd F.H. and JARRETT J.: Pathomechanics and treatment of congenital vertical talus; Clin. Orthop. Relat. Res., 7: 62-72, WIRTH T., SCHEULER P. and GRISS P.: Early surgical treatment of congenital vertical talus; Arch. Orthopaedic. Trauma Surgery, 113: , OGATA K., SHEOENECKER P.L. andsheridan J.: Congenital vertical talus and its familial occurrence: An analysis of 36 patients; Clin. Orthop., 139: , SEIMON L.P.: Surgical correction of congenital vertical talus under the age of 2 years; J. Paediat. Orthop., 7: 45-11, MAZZOCCA A.D., THOMSON J.D., DELUCA P.A. and ROMNESS M.J.: Comparison of the posterior approach versus the dorsal approach in the treatment of congenital vertical talus; J. Pediatr. Orthop., 21: 212-7, DERNNAN J.C.: Congenital vertical talus; Instr. Course Lect., 45: , MATHEW P.J., SPONER P., KARPAS K. and SHAIKH H.H.: Mid-term results of one-stage surgical correction of congenital vertical talus; Bratisl. Lek. Listy, 9: 39-3, CHALAYON O., DOBBS M.B. and ADAMS A.: Minimally invasive approach for the treatment of non-isolated congenital vertical talus. Journal of Bone and Joint Surgery, 73: 1-7, DAVID M.G.: Simultaneous correction of congenital vertical talus and talipus equinovarus using Ponseti method. The Journal of foot and Ankle Surgery, 5: 494-7, WRIGHT J., COGGINGS D., MAIZEN C. and RAM- ACHANDRAN M.: Reverse Ponsetti-type traetment for children with congenital vertical talus. The Bone and Joint Journal, 96: 274-8, BRAND R.A.: 5 years ago in CORR: Congenital vertical talus Tom Outland M.D. and Henry H. Sherk M.D.; Clin. Orthop. Related Research, 468: , VANDERWILDE R., STAHELI L.T., CHEW D.E. and MALAGON V.: Measurements on of the foot in normal infants and children; J. Bone Joint Surg. Am., 7: 47-15, ADELAAR R., WILLIAMS R.M. and GOULD J.S.: Congenital convex pes valgus: results of an early comprehensive release and a review of congenital vertical talus at Richmond Crippled Children's Hospital in the University of Alabama in Birmingham. Journal of Foot and Ankle, 1: 62-73, DOBBS N.B., NUNLEY R. and SCHOENECKER P.L.: Long term follow-up of patients with congenital vertical talus treated with extensive soft tissue release; J. Bone and Joint Surgery A, 88: , 26.

9 Mohamed M. Hegazy, et al. 273

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