Abstract
This study examines the role of talectomy in the management of arthrogrypotic clubfoot in children, based on 26 cases collected over a 12-year period at Rabat Children’s Hospital. The patients, with an average age of 2 years (SD ± 1.2), underwent talectomy as a primary or salvage procedure. The results showed that talectomy achieved a plantigrade, shoeable, and pain-free foot in 81% of cases (95% CI: 65.6%-96.4%), with a 19% recurrence rate (95% CI: 3.6%-34.4%). Multivariate analysis revealed that age at surgery (P = .032) and pre-operative Pirani score (P = .018) were significant predictors of outcome. This research suggests talectomy as a viable option for severe arthrogrypotic clubfoot, while highlighting the need for careful patient selection and close post-operative follow-up to optimize long-term outcomes.
Introduction
Arthrogrypotic clubfoot represents a challenging condition in pediatric orthopedics, characterized by severe, rigid deformities that are often resistant to conservative treatments. Talectomy, a surgical procedure involving the removal of the talus bone, has emerged as a potential solution for these complex cases. 1
The management of arthrogrypotic clubfoot has evolved over the years, with various surgical and non-surgical approaches being explored. However, the severe nature of the deformity in arthrogrypotic cases often necessitates more aggressive interventions. Talectomy, while considered a radical approach, has shown promise in achieving plantigrade feet capable of bearing weight and allowing functional ambulation. 2
This study aims to evaluate the efficacy of talectomy in the treatment of arthrogrypotic clubfoot, analyzing outcomes, complications, and long-term results in a series of 26 cases. By examining these cases, we seek to contribute to the ongoing discussion about the role of talectomy in the management of severe, rigid clubfoot deformities associated with arthrogryposis.
Historical Context
The use of talectomy in the treatment of clubfoot deformities has a long history in orthopedic surgery. Initially described in the late 19th century, it was primarily used to treat severe infections and traumatic injuries of the talus. 3 In the early 20th century, the procedure gained popularity as a treatment for various foot deformities, including those associated with poliomyelitis. 4
The application of talectomy specifically for arthrogrypotic clubfoot began to emerge in the mid-20th century. Menelaus, in his pioneering work in the 1970s, popularized the use of talectomy for rigid clubfoot deformities, particularly in cases of arthrogryposis and myelomeningocele. 5 This marked a significant shift in the management of these complex cases, offering a surgical alternative when conservative methods failed.
Current Perspectives
In recent years, there has been a renewed interest in talectomy as a treatment option for severe, resistant clubfoot deformities in arthrogryposis. While conservative methods like the Ponseti technique have shown success in many cases of idiopathic clubfoot, the unique challenges presented by arthrogrypotic clubfoot often necessitate more invasive approaches.6,7
Contemporary research has focused on refining the indications for talectomy, improving surgical techniques, and evaluating long-term outcomes. The procedure is now typically reserved for cases where conservative methods have failed or for particularly severe deformities that are unlikely to respond to less invasive treatments. 8
This study aims to contribute to this evolving body of knowledge by presenting a comprehensive analysis of talectomy outcomes in a series of arthrogrypotic clubfoot cases, providing insights into its efficacy, complications, and long-term functional results.
Materials and Methods
Study Design and Patient Population
This retrospective cohort study analyzed 26 cases of arthrogrypotic clubfoot treated with talectomy at the pediatric orthopedic department of the Rabat Children’s Hospital over a 12-year period from January 2012 to December 2023.
Inclusion criteria encompassed patients diagnosed with arthrogrypotic clubfoot, who had failed conservative treatment or experienced recurrence after previous interventions. Exclusion criteria included idiopathic clubfoot, clubfoot associated with other syndromes, and incomplete medical records.
Data Collection
Comprehensive patient data were collected from medical records, including demographic information, clinical history, pre-operative deformity severity (using the Pirani scoring system), surgical details, post-operative complications, and functional outcomes.
Surgical Technique
All surgeries were performed by the same team of experienced pediatric orthopedic surgeons (Figure 1). The talectomy procedure was standardized as follows:
Intraoperative image showing the removal of the talus during talectomy procedure.
General anesthesia administration
Supine positioning with a tourniquet applied to the affected limb
Lateral curved incision along the subtalar and talonavicular joints
Careful dissection and protection of neurovascular structures
Complete resection of the talus
Repositioning of the calcaneum to create a neo-articulation with the tibial pilon
Soft tissue release as necessary to achieve adequate correction (typically including Achilles tendon lengthening and posterior capsule release)
Fixation using K-wires
Wound closure and application of a long leg cast
Post-Operative Management
Patients were immobilized in a below-knee cast for 6 weeks, followed by the use of custom-made orthotic devices and intensive physiotherapy. A standardized rehabilitation protocol was implemented, with regular follow-ups scheduled at 2 weeks, 6 weeks, 3 months, 6 months, and then annually.
Outcome Measures
Primary outcomes included correction of deformity (assessed clinically and radiographically), functional improvement (ability to wear normal shoes, gait analysis using the Edinburgh Visual Gait Score), and pain reduction (measured using the Wong-Baker FACES Pain Rating Scale).
Secondary outcomes encompassed complications (infection, wound healing issues, recurrence) and the need for additional procedures.
Statistical Analysis
Descriptive statistics were used to summarize patient characteristics and outcomes. Continuous variables were expressed as means with standard deviations or medinals with interquartile ranges, depending on the distribution of data. Categorical variables were presented as frequencies and percentages.
Paired t-tests or Wilcoxon signed-rank tests were used to compare pre- and post-operative measures, depending on data normality. McNemar’s test was used for paired nominal data.
Multivariate logistic regression analysis was performed to identify predictors of successful outcomes, with variables including age at surgery, pre-operative Pirani score, and duration of pre-operative conservative treatment.
All statistical analyses were performed using Jamovi. A P-value < .05 was considered statistically significant.
Results
Patient Demographics
The study included 26 patients (15 males, 11 females) with arthrogrypotic clubfoot who underwent talectomy. The demographic and clinical characteristics are summarized in Table 1.
Patient Demographics and Clinical Characteristics.
Surgical Outcomes
The primary outcomes of the talectomy procedure (Figure 2) are presented in Table 2.
Clinical photographs showing post-operative correction after talectomy.
Primary Surgical Outcomes.
Radiographic Results
Radiographic assessment (Figure 3) showed satisfactory alignment of the calcaneus with the tibial axis in 88% of cases (95% CI: 75.3%-100%). Residual hindfoot varus was noted in 12% of cases (95% CI: 0%-24.7%).
Radiographs demonstrating pre-operative severe equinovarus deformity before talectomy.
Complications
Many complications were faced involving mainly wound healing issues and superficial infections (Table 3).
Post-Operative Complications.
Long-Term Follow-Up
The average follow-up period was 5.3 years (range: 2-10 years).
Multivariate Analysis
Multivariate logistic regression analysis revealed that age at surgery (OR: 0.68, 95% CI: 0.48-0.96, P = .032) and pre-operative Pirani score (OR: 0.42, 95% CI: 0.20-0.87, P = .018) were significant predictors of successful outcomes. Duration of pre-operative conservative treatment was not found to be a significant predictor (P = .24).
Discussion
The management of arthrogrypotic clubfoot remains a significant challenge in pediatric orthopedics. This study demonstrated that talectomy can be an effective surgical option for severe, rigid clubfoot deformities associated with arthrogryposis. The results showed a success rate of 81% in achieving plantigrade, functional feet, which is comparable to other reported series in the literature.8,9
Efficacy of Talectomy
The high rate of plantigrade, shoeable feet achieved in this study (81%, 95% CI: 65.6% - 96.4%) underscores the effectiveness of talectomy in correcting severe deformities. This success rate is particularly significant given the resistant nature of arthrogrypotic clubfoot to conservative treatments. The procedure allows for substantial correction of the equinus and varus deformities, which are often the most challenging aspects of arthrogrypotic clubfoot.
However, it is important to note the 19% recurrence rate (95% CI: 3.6%-34.4%) observed in our series. This highlights the persistent nature of the deformity and the need for long-term follow-up and management. The recurrence rate is consistent with other studies, such as those by Menelaus 5 and Green et al, 10 who reported recurrence rates of 29%.
The Kaplan-Meier survival analysis provides valuable insights into the long-term durability of the correction, with a 76% probability of maintaining correction at 5 years post-surgery. This information is crucial for patient counseling and setting realistic expectations for long-term outcomes.
Functional Outcomes
The improvement in gait, as evidenced by the significant reduction in Edinburgh Visual Gait Scores, and the ability of 92% of patients to wear normal shoes post-operatively are encouraging results. These functional outcomes significantly impact the quality of life for these patients, allowing for improved mobility and independence. The creation of a neo-articulation between the calcaneus and tibia, while not replicating normal ankle mechanics, appears to provide sufficient stability and motion for functional ambulation.
The improvement in PODCI scores from 45.3 to 78.6 further supports the positive impact of talectomy on overall function and quality of life. This improvement is particularly noteworthy given the complex nature of arthrogrypotic clubfoot and the limited treatment options available for these patients.
Complications and Challenges
The complication rate in our series was relatively low, with wound healing issues and superficial infections being the most common problems. The absence of deep infections or neurovascular complications is reassuring and speaks to the safety of the procedure when performed with proper technique and post-operative care.
However, the 7% of patients reporting post-operative pain underscores the need for comprehensive pain management strategies in the post-operative period. Additionally, the residual hindfoot varus observed in 12% of cases highlights the complexity of achieving perfect alignment in these severely deformed feet.
Predictors of Outcome
The multivariate analysis revealing age at surgery and pre-operative Pirani score as significant predictors of outcome provides valuable information for patient selection and surgical planning. Younger age at surgery was associated with better outcomes, which aligns with the general principle in pediatric orthopedics of earlier intervention leading to better results. 11 The association between higher pre-operative Pirani scores and poorer outcomes underscores the importance of careful pre-operative assessment and the potential benefits of pre-operative manipulation or other interventions to improve the initial deformity before proceeding with talectomy.
Comparison With Other Techniques
When comparing talectomy to other surgical approaches for arthrogrypotic clubfoot, it’s important to consider the severity of the deformities typically addressed by this procedure. Soft tissue releases alone are often insufficient for the rigid deformities seen in arthrogryposis. Talectomy offers the advantage of addressing both bony and soft tissue components of the deformity. 12
The study by Cooper and Capello, 13 which reported a 92% success rate, suggests that with refinement of technique and patient selection, even better outcomes might be achievable. Their long-term follow-up (average 20 years) also provides encouraging evidence for the durability of the results.
Limitations and Future Directions
This study is limited by its retrospective nature and the relatively small sample size, which is reflected in the wide confidence intervals for some of our outcomes. Future research should focus on prospective studies with larger cohorts and longer follow-up periods. Additionally, comparative studies between talectomy and other surgical techniques for arthrogrypotic clubfoot would provide valuable insights into the optimal management strategies for these challenging cases.
Further investigation into the factors predicting successful outcomes and those associated with recurrence could help refine patient selection criteria and improve long-term results. The role of post-operative bracing and rehabilitation protocols in maintaining correction and preventing recurrence also warrants further study.
Advanced imaging techniques, such as weight-bearing CT scans and 3D gait analysis, could provide more detailed information about foot biomechanics post-talectomy and guide further refinements in surgical technique. 14 Additionally, the potential role of emerging technologies, such as 3D-printed custom implants or external fixators, in conjunction with talectomy, represents an exciting area for future research. 15
Long-term studies focusing on the impact of talectomy on adjacent joints, particularly the knee and hip, are needed to fully understand the biomechanical consequences of this procedure over time. This information would be invaluable in counseling patients and families about the lifelong implications of talectomy for arthrogrypotic clubfoot.
Conclusion
Talectomy remains a valuable option in the surgical management of severe arthrogrypotic clubfoot. This study demonstrated that the procedure can achieve plantigrade, functional feet in a high percentage of cases, with acceptable complication rates. The 81% success rate in achieving plantigrade, shoeable feet represents a significant improvement in function and quality of life for these patients.
However, the 19% recurrence rate and the presence of residual deformities in some cases highlight the challenging nature of arthrogrypotic clubfoot and the need for careful patient selection, meticulous surgical technique, and long-term follow-up. The procedure should be considered as part of a comprehensive treatment approach, which may include pre-operative manipulation, concurrent soft tissue procedures, and post-operative bracing and rehabilitation.
Our findings suggest that younger age at surgery and lower pre-operative Pirani scores are associated with better outcomes, providing valuable guidance for surgical timing and patient selection. The long-term survival analysis offers important information for patient counseling and expectation management.
While talectomy offers a viable solution for many patients with arthrogrypotic clubfoot, it is not without risks and potential complications. The decision to proceed with talectomy should be made carefully, considering the individual patient’s characteristics, the severity of the deformity, and the family’s expectations and preferences.
As our understanding of arthrogrypotic clubfoot evolves, so too will our ability to provide effective, lasting solutions for this complex condition. Continued research, refinement of surgical techniques, and long-term follow-up studies will be crucial in optimizing outcomes for these challenging cases. The ultimate goal remains to provide these children with functional, pain-free feet that allow for improved mobility and quality of life.
