Clinical, Radiological, Pedobarographic, and Quality of Life Outcomes of the Ponseti Treatment for Clubfoot: A Prospective Study from Iran

Background: Talipes equinovarus, also known as clubfoot, is a congenital anomaly that affects one newborn per 1000 live births. Its standard treatment strategy is the Ponseti casting management. This study aims to report the long-term outcomes of the Ponseti treatment in Iran. Methods: A prospective cohort study was enrolled to evaluate clinical outcomes, radiological results, pedobarographic measurements, and quality of life after the Ponseti treatment in patients with clubfoot who were followed for at least ve years. Results: In this study, 25 clubfeet of 18 patients were included. Signicant reductions in Pirani, Dimeglio, and CAP scores, improved ankle dorsiexion, and acceptable pedobarographic indices were observed in this study. From the radiological evaluation indices, the calcaneal pitch and lateral talus-rst metatarsal angles were signicantly reduced. The quality of life of patients after ve years of treatment was favorable, which was better in females. This study showed that the results of the Ponseti treatment remained acceptable after ve years. Conclusions: The Ponseti management for clubfoot in the long term appears to maintain signicant improvements. However, the rate of recurrence – albeit without disruption to daily activities - cannot be ignored.


Background
Talipes equinovarus, also known as clubfoot, is a congenital anomaly in the lower extremities that may be part of a syndrome [1]. The disorder, which is one of the most common problems in pediatric orthopedics, affects one newborn per 1000 live births with varying degrees of severity [2,3]. The prevalence of clubfoot in male infants is almost twice that of female ones [2]. It has a wide range of problems, depending on its severity, the treatment, and the success of that treatment [4,5].
Achieving plantigrade locomotion with normal and painless function is the goal of clubfoot treatment [6].
Varieties of treatments are suggested for patients [7]. The standard and benchmark treatment strategy is the Ponseti method of management, which involves manipulation and weekly casting, percutaneous Achilles tenotomy in most patients, and maintenance of correction with braces for several years [4,8]. The rationale for using braces is that recurrence of clubfoot is common even after successful correction [9].
There are various methods for patients' follow-up, including scoring severity of deformities, scoring clinical examination with the proposed protocols, radiological evaluations and measuring radiographic angles, measuring the pressure exerts by the sole on the ground using a pedobarograph, and assessing the patient's quality of life [5,10].
Although about 80% of patients with clubfoot are from developing countries, a signi cant number of patients are missed in these countries, and genetics and race play a role in this disorder, to the best of our knowledge, there was no study to evaluate the long-term results of Ponseti treatment in Iran and the Eastern Mediterranean region [5,11,12]. One of the goals of this study is to evaluate the durability of the results because a number of previous studies have reported remarkable results in the short term using this treatment [11,13]. This prospective cohort study aims to report the long-term clinical outcomes, radiological results, pedobarographic measurements, and quality of life of patients with clubfoot who were treated with the Ponseti method.

Study design and setting
A prospective cohort study was designed to evaluate the long-term results and outcomes of the Ponseti method in patients with clubfoot from August 2013 until September 2015. After each visit, the patient's information was transferred from the medical records to our hospital information system, and nally, the information required for the study was collected from the hospital information system. This study is in accordance with the Declaration of Helsinki and its subsequent revisions [14]. Our institutional review board approved this study. Written informed consent was obtained from each patient at the beginning of the study.

Participants
Through the records sampling, all patients with clubfoot who were treated with the Ponseti method to correct the deformity and were followed for at least ve years were included in the study. All patients were visited and treated by two pediatric orthopedists (BP and MHN). Assessments a. Demographic information was collected, including age at the time of data analysis and gender.
b. Pirani and Dimeglio scoring systems were used to assess the deformity severity in the rst and last follow-up visits. The Pirani score has six parameters and evaluates midfoot and hindfoot. This scoring system is given a score of 0 to 6 for each patient, and the higher the score, the more severe the deformity [15]. The Dimeglio scoring system has eight parameters, and the closer the nal score is to 20, the more severe the anomaly. The parameters of this scoring system for severity evaluation include equinus, varus, supination, and adductus (each from 0 to 4) and posterior crease, medial crease, cavus, and deviant muscle function (each from 0 to 1) [16].
c. At each visit, patients underwent physical examinations to assess functional and structural status, and a standard instrument was used for more precise assessments. The clubfoot assessment protocol (CAP) consists of twenty parameters in four subsets, including passive mobility, muscle function, morphology, and motion quality, and have su cient reliability [17]. Evaluation of the foot structure and function during clubfoot treatment was also performed by the method introduced by Bensahel et al. and the International Clubfoot Study Group (ICSG). In this system, scores of 0 and 60 indicate the best and worst results, respectively [18]. d. Radiological examinations were performed by radiographs in anteroposterior (AP) and lateral views of the foot in stress dorsi exion position and measuring calcaneal pitch angle, lateral talocalcaneal angle, kite angle, lateral talus-rst metatarsal angle, and AP talus-rst metatarsal angle [19]. e. Pedobarography was another method of evaluating the treatment results, which is measuring the pressure on the sole. The device pedobarograph has a pressure-sensitive plastic plate that converts the foot image into visible pressure patterns [20].
f. The PedsQL that is a standard quality of life questionnaire was used, too. This questionnaire has 23 questions in four areas including health and physical function (8 questions), emotional function (5 questions), social function (5 questions), and academic function (5 questions). Parents scored each question from 1 to 5. Finally, the scores of the areas were added, and the lower the nal score, the better the quality of life [21].
g. Recurrence was de ned as the reappearance of one to all previous deformities after successful initial treatment [4].
Statistical analysis IBM SPSS statistics 25 (IBM corporation, Armonk, NY, USA) was used to perform all statistical analyzes. Descriptive statistics such as mean, standard deviation, and percentage were used to describe the data, Independent samples t-test was used to compare means, and Chi-square test was used to examine the relationship between qualitative variables. The One-sample Wilcoxon signed-rank test was used for variables that did not have a normal distribution. The Mann -Whitney U test was used to compare the quality of life of males and females. A p-value of less than 0.05 was considered signi cant.

Participants
In this study, 25 clubfeet of 18 patients with mean age ± standard deviation (SD) of 6.88 ± 1.68 years at the time of data analysis were included. 13 and 5 of the patients were male and female, respectively, and their ages did not differ [p-value = 0.104 using the independent t-test].
Outcomes Table 1 has detailed the measurements of the scoring systems, clinical examinations, and radiological evaluations variables. The mean ± SD reductions in the Pirani and Demeglio scoring systems from the rst visit to the last follow-up visit was 3.05 ± 1.95 and 6.55 ± 3.22, respectively, which were signi cant using the one-sample t-test and the Wilcoxon test, respectively. Also, the mean CAP score of patients during the follow-up period had a signi cant decrease of 10.38 ± 10.34 using the one-sample t-test. In addition, the Wilcoxon test showed that the dorsi exion angle of the patients' feet was signi cantly improved. The mean ± SD of ICSG at the last follow-up visit was 9.38 ± 10.86 -this variable was not calculated at the rst visits. All radiologically evaluated angles were signi cantly reduced after ve years of treatment using the one-sample t-test, except for the AP talus-rst metatarsal angle, which decreased by an average of 0.30 ± 0.73 -it was not signi cant using the Wilcoxon test.  Table 2 reports the results of the pedobarographic evaluations of patients. These assessments were performed only at the time of the last follow-up visit. The mean PedsQL of all patients, males, and females were 8.69 ± 8.70, 10.73 ± 9.40, and 3.14 ± 1.21, respectively. The observed difference between males and females was statistically signi cant, indicating that in long-term follow-up, the quality of life in females was signi cantly higher than in males [p-value = 0.003 using the Mann-Whitney U test]. Clubfoot recurrence was observed in 3 feet of 3 patients.

Discussion
In the current study, with a ve-year follow-up of patients with clubfoot who were treated with Ponseti method, Pirani scores decreased, Dimeglio decreased, scores CAP scores decreased, ankle dorsi exion improved, several radiological indicators improved, patients' foot condition was acceptable determined by the pedobarographic device, and patients' quality of life was favorable. The only adverse outcomes were lateral talocalcaneal angle and kite angle. This study showed that the results of the Ponseti treatment remained acceptable after ve years. The results of this study are weaker than the short-term studies that have reported remarkable outcomes, and this difference may indicate a recurrence -albeit without functional problems -in patients [11,13].
In this study, the mean Pirani score of patients remained signi cantly reduced and acceptable after ve years of follow-up. In Azimi and Narouie's study of 32 clubfeet, the mean Pirani score dropped from 5.53 to 0.09 after 4 to 14 months of treatment, which was to be expected, given the scoring shortly after treatment [13]. Kumar et al. have also reported a decrease in mean Pirani score from 5.3 to 0.8 following six to twenty months of Ponseti management [22].
Also, the patients' Demeglio score decreased signi cantly and remained acceptable after ve years in this study, which indicates continuation of good function in the long run. Hallaj-Moghaddam et al. by designing a prospective study in patients with severe clubfoot reported a signi cant reduction in the mean Demeglio score from 16 to 1.6 [23]. In another prospective study on 110 idiopathic clubfeet, Bouchoucha et al. reported a reduction in a mean of this score from 12.9 to 1.3 [24].
Improvement of ankle dorsi exion following treatment and remaining this outcome for ve years was one of the ndings of this study. Spiegel et al. and Sanghvi and Mittal reported an average dorsi exion angle of 12.5 and a maximum dorsi exion angle of 12, respectively [25,26].
In this study, children's quality of life was assessed ve years after the start of treatment. The mean score of the standard questionnaire of PedsQL indicates the high quality of life in children -especially in female ones -following clubfoot treatment with the Ponseti method. A study by Smith et al. found that children who underwent surgery to treat clubfoot reported more pain than children who underwent Ponseti treatment; however, both groups were functional and had a high quality of life [27]. Causes of decreased quality of life in these patients include prolonged use of braces and consequent inability to play and anxiety and discomfort caused by thinking of others about how they walk [5,28].
Recurrence was observed in 12% of feet in this study. Porecha [32]. A study by Qudsi et al., which examined clinical outcomes and risk factors for the Ponseti method in 168 children with clubfoot, found that female gender and a higher Pirani score increased the risk of recurrence [33].
Although this study has signi cant advantages such as long-term follow-up, evaluation by various assessments, and prospective design, it also has disadvantages that should be noted. First, the number of patients in this study is small. Second, the assessments performed at follow-up visits did not include evaluations through the assessments mentioned in the materials and methods section, so they were not reported. Third, the ICSG assessment was not performed at the rst visit.

Conclusion
Given the decline in Pirani, Dimeglio, and CAP scores, low ICSG scores, improved ankle dorsi exion function, improved several radiological indices, and acceptable quality of life, the Ponseti's approach to treating clubfoot in the medium term appears to maintain signi cant improvements. However, the rate of recurrence using this method and the worsening of clubfoot improvement indices compared to short-term results can not be ignored. Therefore, preventive tools such as using braces should be used for preventing recurrence of deformity. Availability of data and materials: The datasets used and/or analyzed during the current study are available from the corresponding author on reasonable request.

Declarations
Competing interests: All authors declare that there were no competing interests for this study.
Funding: No funding was received for the current study.