Audit Section

Management of congenital talipes equino varus in Scotland: a nationwide audit

D.J. Chesney¹, S.L. Barker², M.F. Macnicol³ R.W. Porter⁴ and N. Maffulli^5
1Freeman Hospital, Newcastle-upon-Tyne,UK, ²Aberdeen Royal Infirmary, Aberdeen, UK, ³Royal Hospital for Sick Children, Edinburgh, UK, ⁴University of Aberdeen, UK, ⁵University of Keele, UK

Correspondence to: D.J. Chesney, The Old Schoolhouse, High Westwood, Newcastle-upon-Tyne, NE17 7RD. Email: djchesney@rcsed.ac.uk

Introduction Results Management

Discussion References

Keywords: Talipes, clubfoot, management, audit
Surg J R Coll Surg Edinb Irel., 2 February 2004, 47-51

The Scottish Talipes Register is a record of all newly diagnosed cases of idiopathic congenital talipes equinovarus (CTEV) in Scotland over a four-year period. Two hundred and sixteen children were entered into the register, which recorded initial management and all subsequent interventions, both non-operative and operative. The data, therefore, represent a record of the different management regimens used within Scotland highlighting the variations in duration of conservative management and variations in surgical intervention following different methods of conservative management

INTRODUCTION
The Scottish Talipes Register, set up in August 1993, is a record of the birth of children with congenital talipes equinovarus (CTEV) in Scotland. The method of management of CTEV, both at the outset, and subsequently, is also recorded.¹

Every hospital in Scotland involved in the management of CTEV was invited to participate, and 100% participation was achieved. In each of the 18 centres, all new cases of CTEV over a four-year period (1993-1997) were entered. Compliance was ensured by regular visits to each centre by a dedicated research nurse. The original entries in the CTEV register included the following information:-

• Name/postcode of affected child

• Sex

• Date of birth

• Sibling order

• Twin status

• Family history of CTEV (entered as yes or no for siblings, parents, aunts, uncles and cousins)

• Birth weight

• Gestation

• Method of delivery

• Affected side/sides

• Presence of any other congenital abnormalities

• Name of the managing surgeon

• Management, with pages for the entry of all interventions, including any surgery, at varying times after the initial presentation.

Additional sheets were supplied for each child entered into the register, allowing subsequent management interventions to be documented. Repeat visits to each centre by a dedicated research nurse ensured completeness of the data. All the information was stored centrally in Aberdeen. No assessment of the degree of deformity or the degree of correctability was performed prior to enrolment into the register. However, assuming that there is no variation in the severity of CTEV across Scotland, each surgeon will see a spectrum of disease, and variation in outcome may reflect management rather than variation in disease.

The Scottish Talipes Register represents a complete record of the management of clubfoot across Scotland, in a group of children aged three to seven years. 

RESULTS
Over the period 1993-1997, 216 patients were entered into the register (Table 1 and Figure 1).

Of the 18 centres originally included in the register, two entered no children. Stirling Royal Infirmary had no new cases of CTEV over the four-year period, and the Borders General Hospital ceased to treat patients with CTEV in mid-1993 because of anaesthetic limitations.Variations in the number of cases entered into the register by different Health Boards reflect differences in population and birth rate. Table 1 shows number of births in 1995 by health board, and approximate incidence of CTEV by Health Board is also presented.

EPIDEMIOLOGY 
The incidence of CTEV in Scotland for the period covered by the Register was 0.9 affected children per 1000 live births. The male to female ratio was 2.5. Ninety nine (45.7%) children had bilateral CTEV, 52 (24.3%) were left-sided, and 65 (30%) were right-sided. Bilateral and left-sided cases maintained a sex ration of 2.5 to 1. Right-sided cases showed a sex ratio of almost one to one (35 male to 29 female).

Figure 1: Map of Scotland by Health Board 

Key:
1 Argyll & Clyde
2 Ayrshire & Arran
3 Dumfries & Galloway
4 Fife
5 Highland
6 Greater Glasgow
7 Grampian
8 Lanarkshire
9 Lothian
10 Tayside

The first-born child was affected more frequently than children born lower in birth order, since 46% of 204 children in whom birth order was documented were first-born; 31% were second-born.

Of 198 infants for whom the method of delivery was recorded, 123 (62.2%) had spontaneous vertex deliveries, 38 (19.2%) were born by Caesarean section, 17 (8.6%) were delivered by forceps, 7 (3.5%) were delivered by the Ventouse method, and one (0.5%) was a breech delivery.

Eight children in the register were one of a set of twins (4%). In all twin cases, only one sibling was affected.

Gestational age at the time of delivery was recorded for all the children: 14 male and 8 female infants were premature (sex ratio 1.75 to 1).

MANAGEMENT
All the children were initially managed conservatively, although 114 (53.3%) required surgery (81 (52.9%) males and 33 (54.1%) females).

The method of conservative management was recorded for 143 of the 153 boys entered: 51 (36%) were managed initially with adhesive strapping, 48 (34%) were managed with a combination of strapping for the first few weeks of life, and then plaster of Paris casts, and 42 (29%) were managed with strapping followed by Denis Browne splints and boots.

Of the patients managed with strapping, 69% required surgery, whereas 42% of those managed with plaster casts required surgery, and only 14% of those splinted with Denis Browne boots were referred for surgery.

In girls, information about the method of conservative management was recorded in 56 of the 61 patients: 21 patients were managed with strapping alone, 16 were managed with strapping then plaster casts, and 15 were managed with Denis Browne boots. To date, those children managed with strapping alone have required surgery in 76%, those managed with strapping and then plaster casts in 63%, and those managed with Denis Browne boots in 33% of cases.

Duration of conservative management in children not undergoing surgery ranged from one week to “continuing to present day”, and duration of conservative management prior to surgery was also recorded. For those children managed with strapping, the mean duration of conservative management was 17 weeks (range 3 to 75 weeks: SD+/- 14.4). For children managed with plaster of Paris casts, the mean duration of casting was 24.1 weeks (range 6 to 90 weeks: SD+/ - 20.4), and for children managed with Denis Browne splints and boots, mean duration of treatment was 57.1 weeks (range 13 to 140 weeks: SD+/- 44.1).

In the vast majority of patients requiring surgery, a posterior or posteromedial release was performed. However, one left side affected male and one right side affected female have undergone Achilles tendon lengthening alone. Two children (both bilaterally affected males) have undergone application of an Ilizarov frame, as the primary surgical intervention, following one year of conservative management

Only three boys are recorded as having undergone a second surgical procedure. One boy with right-sided CTEV, was initially managed with strapping and plaster casts, and then a posterior release. He required a second posterior release for residual equinus. A second, bilaterally affected boy underwent a further posterior release bilaterally. The other child was bilaterally affected and had undergone a posterior release. He required a Turco release at one year of age.^2,3

Five girls required a second surgical procedure. Two were bilaterally affected, requiring a repeat posteromedial release bilaterally; one was left-sided, requiring a repeat posteromedial release, and two were right-sided, initially managed with posterior releases, but later requiring posteromedial releases.

One bilaterally affected male has required three surgical procedures. He has undergone two medial releases and a lateral release on each foot. None of the children have undergone bony procedures.

DISCUSSION
The incidence of CTEV in Scotland was 0.9 pre 1000 live births. This is similar to published data.4 The number of cases entered into the register by different Health Boards reflects differences in population size and birth rates but the incidence of CTEV across Scotland does not vary significantly.

The sex ratio of 1 female:2.5 males is similar to other published studies, although interestingly this is not reflected in right-sided cases.⁴ The reasons for this are not clear but may represent a statistical anomaly because of the numbers involved.

Idelberger (1935) reported a concordance between dizygotic twins of 2.9%, and 32.5% for monozygotic twins.⁵ This finding was not seen in the Scottish cohort.

On first inspection of the management data, there appears to be wide variations in the need for surgery. This reflects the world literature, and the predilection of the different surgeons. 

Kite (1970) reported that 92% of 922 patients managed with casts and wedging alone were cured, although the outcomes of 70 years ago may not be acceptable today.⁶ Ponseti and Smoley (1963) found a 56% recurrence occurring 10 months to 5 years after conservative management.⁷ Fripp and Shaw (1967) observed that 71% of 96 patients managed with stretching and strapping, 19% of 105 patients managed in Denis Browne splints, and none of 20 patients managed with serial casts were corrected.⁸ Harrold and Walker (1963) managed 103 patients with serial casts, succeeding in 53.4% of cases.⁹ Similar results were obtained with serial casts in 323 Polish children.¹⁰ Yamamoto and Furuya (1990) were successful in 66% of 91 patients managed with Denis Browne splints.

Ponseti (1992) observed that the most popular method of conservative management was plaster of Paris casting, although any method (casts, Denis Browne splints, stretching and strapping) could be effective if applied correctly.¹² Success of conservative management varied with the age of the patient, the severity of the initial deformity, and the skill of the surgeon. In his hands, a combination of serial casts combined with limited operative intervention yielded positive results in 89% of children. Poor results of manipulation reflected poor technique rather than a failure of conservative management, although the severity of the deformity obviously influences outcome. More recently, the underlying neurological defect has been shown to be associated with severity of clubfoot deformity, and its response to treatment.^6,13

There is obviously a wide variation in outcome after conservative management. This may be attributed to variation in severity of the deformity, differences in length of follow-up and different methods of outcome assessment. Indeed, it is well recognised that CTEV is a spectrum of disease, ranging from a postural deformity that is easily correctable, to a severe stiff foot, resistant to most forms of management.^14-16

While most authors agree that initial management should be conservative, there is little evidence to demonstrate the best method of conservative management, or the optimal duration of conservative management. If one method of conservative management can be shown to be more effective than another, this would represent an important breakthrough in the management of CTEV. However, it is clear that a prolonged period of splintage is required if the surgeon decides for a conservative approach.

While there are many studies evaluating the management of CTEV, all are limited by lack of a standardised method of assessment, and few studies compare outcome for different methods of management.^14,15,17-20

As no classification of the CTEV affected children was performed prior to enrolment into the Scottish Talipes Register, comparison of different methods of conservative management is difficult. However, while a variety of conservative methods are used by different surgeons, each surgeon uses the same method for all the children in his care. Conservative management, therefore, may depend upon geography and local practice, rather than severity of deformity. Each surgeon will see a spectrum of disease and in each centre all children are initially managed in the same way. If one assumes that, within Scotland, there is no significant variation in the spectrum of severity of the condition, then, when taken as a whole by centre, differences in response to conservative management may reflect the management itself rather than the initial condition. The available information from the Scottish Talipes Register, therefore, suggests that a prolonged period of conservative management with Denis Browne splints and boots limits the need for surgery, but at the cost of prolonged splintage and possible stiffness or skeletal deformity. Because of the lack of a standardised classification of severity of CTEV at the time of presentation, however, the best method of conservative management remains unproven.

Only by means of a multicentre prospective long-term study of CTEV, including a universally accepted and applied classification of the condition on presentation, will it be possible scientifically to determine the optimum method of management in CTEV.

REFERENCES
1. Turco V.J. Surgical correction of the resistant clubfoot: a one stage posteromedial release with internal fixation: a preliminary report. Journal of Bone & Joint Surgery 1971; 53A: 497.
2. Turco V.J. Resistant congenital clubfoot. One stage posteromedial release with internal fixation. Journal of Bone & Joint Surgery 1979; 61A: 805-814.
3. Wynne-Davies R. Family studies and the cause of congenital clubfoot. Journal of Bone & Joint Surgery 1964; 46B: 445-463.
4. Idelberger, K. Die Ergebnisseder Zwillingsforschung beim angeborenen Klumpfuß. Verhandlungen der Deutschen Orthop per thousand dischen Gesellschaft 1939; 33: 272-276.
5. Kite, J.H. Non-operative treatment of congenital clubfeet: a review of one hundred cases. Southern Medical Journal 1930; 23: 337-345.
6. Ponseti, I.V. and Smoley E.N. Congenital clubfoot: the results of treatment. Journal of Bone & Joint Surgery 1963; 45-A: 261-273.
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16. Porter RW. Clubfoot: congenital talipes equinovarus. J R Coll Surg Edinb 1995; 40: 66-71.
17. Haasbeek, J.F. and Wright, J.G. A Comparison of the Long-Term Results of Posterior and Comprehensive Release in the Treatment of Clubfoot. Journal of Paediatric Orthopaedics 1997; 17: 29-35.
18. Laaveg, S.J. and Ponseti, I.V. Long-term results of the Treatment of Congenital Club Foot. Journal of Bone & Joint Surgery 1980; 62: 23-31.
19. Lau, J.H.K., Meyer, L.C. and Lau, H.C. Results of surgical treatment of talipes equinovarus congenital. Clinical Orthopaedics & Related Research 1989; 248: 219-226.
20. Magone, J.B., Torch, M.A., Clark, R.N. and Kean, J.R. Comparative review of surgical treatment of the idiopathic clubfoot by 3 different procedures at Columbus Children’s Hospital. Journal of Paediatric Orthopaedics 1989; 9: 49-58.

Copyright: 9 November 2003