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Surgical outcome with single stage procedure for neglected congenital talipes equinovarus

Congenital talipes equinovarus (CTEV) is one of the commonest congenital anomalies. Incidence is globally 0.6 to 1.5 per 1000 live births. Most of the times, it is an isolated problem but it may be associated with some other anomalies in 6-8% of the cases, of which developmental dysplasia of the hip (DDH) and arthrogryposis multiplex congenital (AMC) are more common. In developing countries like India still there are many myths, culture & awareness issues regarding this deformity. So we find many neglected cases that represent in very late ages for the hope of treatment.


INTRODUCTION
Congenital talipes equinovarus (CTEV) is one of the commonest congenital anomalies. Incidence is globally 0.6 to 1.5 per 1000 live births. Most of the times, it is an isolated problem but it may be associated with some other anomalies in 6-8% of the cases, of which developmental dysplasia of the hip (DDH) and arthrogryposis multiplex congenital (AMC) are more common.
In developing countries like India still there are many myths, culture & awareness issues regarding this deformity. So we find many neglected cases that represent in very late ages for the hope of treatment.
The neglected clubfoot, by definition is a foot which has experienced no or minimal surgical or non-surgical management. 1 (neglected, resistant, or relapsed clubfoot) include the likely related situations of no treatment; inadequate, insufficient, or discontinued treatment; resistant cases (most often syndromic or teratologic); or relapsed and refractory to traditional treatments, whether conservative or surgical. 2 NC is a common problem in developing or underdeveloped countries. Approximately 80% of children with congenital clubfoot (CCF) are born in developing countries, many of those with limited and/or late access to the healthcare system and trained specialists for treatment. 3 Neglected CTEV is common problem in rural area due to lack of health education, awareness and socioeconomic factors related to it. Many patients get initial treatment with casting but tend loose follow ups in this prolonged treatment. Often times many patients don't follow the bracing protocol which again leads to relapse. In our demography patients come in delayed stages of neglect with severe equino cavo varus deformities. The presentation age may vary from 1 year to 25 years of age.
Management of these patients is challenging in terms of compliance to the treatment, follow ups & spillage protocols. Usually they are not willing for prolonged treatment like JESS or Ilizarov correction and demand for single stage procedure.
Conventional soft tissue/bony procedure do not suffice and they require combination of procedures to get maximum results in single stage surgery. Only bony procedures have risk of AVN, early arthritis and stiffness. Only soft tissue procedures lack to give results for remaining bony deformations. To tackle all these problems and give best possible results in this scenario, the technique for single stage procedure which includes bony and soft tissue repair gives excellent results, we have taken care to preserve cosmetic by using small incisions.
The aim of this study is to provide a single stage procedure for the cases of relapse/neglected clubfoot and study its outcome.

METHODS
This is retrospective longitudinal study conducted at Ujjwal paediatric orthopaedic hospital for 3 years from October 2016-September 2019. A total of 13 children of age between 5-25 years (mean 13 years) were studied for 6 months to 3 years (mean 13 months) who underwent soft tissue and bony procedure in single sitting.
Inclusion criteria were relapse clubfeet children with Grade III, IV deformities (Dimeglio et al). 4 The patients with neurological clubfeet and age below 5 years were excluded from the study. Preoperative and post ability of squatting was observed along with length of foot. Postoperative pain duration and time to regain maximum functional abilities recorded. All procedures were done on single foot at a single sitting.

Operative technique
All surgeries are done under regional anaesthesia -spinal/ epidural anaesthesia. The patient is placed supine; tourniquet is applied and inflated with adequate pressure, after proper painting and draping. Percutaneous plantar fascia is released with no.15 blade correcting the cavus deformity. Tendoachilles Z plasty is done with postero medial incision which helps to unlock heel. To get optimal release of ankle equines, posterior capsular release is done.  Through medial transverse incision over TN joint extending to navicular bone TN joint capsule release is done. Tibialis anterior tendon (full thickness) is harvested by releasing its insertion over navicular bone.
Lateral oblique incision is taken over dorsolateral aspect of foot (parallel to crease) to expose cuboid. Dorsal soft tissues with neurovascular structures are lifted and preserved with thin periosteal elevator. Using special mini saw, a dorsal closing wedge (1 cm) osteotomy of cuboid and lateral cuneiform is performed through lateral incision and 1st, 2nd cuneiform osteotomy through medial incision by apex of wedge facing plantar.
The harvested tibialis anterior tendon is routed to lateral aspect of foot and buried in lateral cuneiform with tendon fixed over planter side over a button. Wedge closed and fixed with two k wires whose ends are kept outside the skin. The wounds are closed in layers after looking for adequate foot correction. Thigh foot angle and foot progression angles checked on

Post-operative management
Above knee loose cast given to maintain correction, the cast changed on post op day 3 to check wounds. This plaster removed after 1 month, X-rays done for seeing union. After confirming union, k wires were removed.
Below knee walking cast was given for 3 weeks. This is followed by splints (orthosis) for 6 months.
Clinical examination to check correction, outcomes was done as per preformed protocol.
Post op X-rays for radiological evaluation done for confirming outcomes.

RESULTS
A total 13 children out of whom 9 (69.23%) were male and 4 (30.77%) were female, all having unilateral deformity were studied. The age of the patients was between 5-22 years (mean 13.5±5.84) years.
Among 13 children 06 were of the neglected type and 07 were of the relapsed type.

Pre operative evaluation
Of 13 feet, 03 were classified as grade III & 10 were grade IV deformity of Dimeglio's clubfoot classification. All were walking on deformed feet and were not able to squat.

Intra operative evaluation
Average time for surgery was 75±15 minutes. No intra op or immediate post op complication happened.

Post operative evaluation
The follow up ranged from 4 months to 48 months (mean 26 months  All except one achieved complete squatting by 4 months of time. One case had difficulty in squatting for 8 months which later could be achieved by compensatory mechanism.

Follow up evaluation
Out of 13 feet, 05 children were younger than 10 yrs and 08 were older than 10 yrs. The results shows that frequency of excellent results was higher in younger children (<10 yrs) than the older (10+ yrs) children.
None of the patients had pain in the foot or operative area in any activity at final follow up, squatting was possible in all the patients.

Tenotomies and myotendinous release
The severity of the deformity and the degree of rigidity appear to be fundamental elements for its indication. However, the limits of severity and rigidity that guide closed arthrodiastasis, the association with tenotomies, and the use of osteotomies are still subjects of discussion. 5 In skeletally immature patients, up to 10 or12 years, there are advantages to this approach, which minimizes damage related to the development and growth of the bones of the tarsus and metatarsus. 5 The soft tissue procedures on feet with severe deformities (Dimeglio III and IV) and in recurrent cases after previous treatments, associated with neurological injuries or neuromuscular diseases and arthrogryposis, appear to have better results, with less overload on the system or even lower incidence of complications, residual deformities, and relapses, in addition to the reduced correction time when compared with the closed treatment. 6 The indications typically include Achilles tenotomy, which can be open or percutaneous (Hoke or White techniques), and plantar fasciotomy. Other tenotomies include tenotomy of the flexor hallucis longus, fingers, and posterior tibial, which can like-wise be performed using percutaneous or minimally invasive, complete, or Z-shaped techniques. The percutaneous tenotomy of the flexors of the fingers and hallux is usually made through the inter phalangeal joint of the fingers, with a No. 15 scalpel blade. 7

Bone procedures and osteotomy
Foot osteotomies are mainly indicated in skeletally mature patients (over 10-12 years) with severe deformities (Dimeglio III and IV), and extremely rigid feet. 8 Osteotomies can be classified as (1) calcaneal (of the posterior tuberosity or below the subtalar joint); (2) talo-calcaneal (anterior talo-calcaneal, V-shaped, Yshaped, or dome-shaped); (3) midfoot (navicular-cuboid or cuboid-cuneiform); and (4) forefoot (metatarsal or phalangeal). The related osteotomies combinations are listed. They can be per-cutaneous (with pre-burring or with a Gigli saw) or open. 9 Cuboidectomy associated with soft tissue procedures presented good results in the series published by Faldini et al. 10 In our study we found excellent results in 77% cases and good in 23% cases which is similar to the other studies mentioned here. The rate of positive results (excellent or good) observed in the comparison between the pre-and post-operative Dimeglio scores is 88%, according to El-Sayed. 11 Makhdoom et al observed good results in 74% of patients, using the scale pro-posed by Renkerand and Carpenter. 12 Refai et al using the AOFAS score as a reference identified 84% good results. 13 Good results were also observed by Ahmed 6,7,9,14,15 Although the numbers of good results were high, the diversity of cases and lack of prospective protocols limits the comparison among the series, as well as prospective evaluation with better statistical value and consequent significance. 15 The scores proposed and used are also quite varied in the literature. Saghieh et al emphasized this limitation and suggested the use of the International Clubfoot Study Group scoring system (ICFSG) as a proposal for prospective evaluation. 16 In 1940, cole popularised a dorsiflexory osteotomy through the lesser tarsus to correct a cavus type condition. 17 Our technique is similar to cole's osteotomy but used for treatment of neglected CTEV correction.
The only disadvantage observed in our study was shortening of the foot in some cases which is comparable to other studies by Graham et al Laaveg et al, Wynne-Davies et al. [18][19][20] We have discussed only neglected cases beyond 5 years of age with Dimeglio Grade III onwards.
Usually all these deformities have contracted TA which is one of the major deforming forces. TA lengthening corrects equinus by unlocking heel. Also it helps to avoid calcaneal osteotomies. Conventional procedures like Dwyer, Evans alone have failed to give complete correction, so combination of different procedures is required.
With our described technique excellent to good results were achieved in all cases with very much cosmetic scar and without undue skin scarring. Since we don't touch any of the articular surfaces in this technique, so no incidence of stiffness or painful joints. No bone grafts are required in this technique. Numbers of follow ups required are less & are placed at minimum 3 weeks duration. Compliance & education of patients like in JESS/ Ilizarov is not required so more patient friendly. It is very much cost effective technique. Only disadvantage is some shortening of foot compared to conservatively treated cases.

CONCLUSION
CTEV is best to be treated at the early age with Ponseti's casting method. In neglected cases many management options are available but most of them require good compliance and extensive patient education for maintenance of correction.
With our technique we can achieve satisfactory outcomes in single sitting surgery with cosmetically optimal foot & minimum patient compliance.
It can be concluded from analyzing this case series that treatment of neglected clubfoot using single stage procedure dorsal close wedge osteotomy with soft tissue release has a high rate of good and excellent results, with low frequency of complications. But still, long term follow up with larger sample size of patient is required for knowing the detailed outcome of this procedure.
Funding: No funding sources Conflict of interest: None declared Ethical approval: Not required