Abstract
Background
Noncompliance with bracing and its protocol is the most common cause of clubfoot deformity relapse after correction. The literature does not clarify when patient noncompliance with the brace occurs—during which period of the bracing phase, or in which age group. This study aimed to determine when noncompliance is most likely to occur.
Methods
This descriptive observational study included children with idiopathic talipes equinovarus deformity for whom the foot abduction brace was applied to their feet after complete correction via the Ponseti method. Each patient was observed until the end of the brace-wearing program (at the end of the fourth year) and had 18 follow-up appointments. The patient’s age and compliance (yes or no) were recorded at each appointment.
Results
Thirty-four cases (21 males and 13 females) with 58 feet were included in the current study. The mean age at the time of deformity correction and commencement of the brace was 2.3 months (standard deviation [SD], 0.6 months), and that at the final follow-up appointment was 50.6 months (SD, 0.9 months). Noncompliance occurred in 11 of 34 cases (32.4%). It started after the end of the first year of life, when the mean age of the patients was 14.6 months (SD, 0.8 months). A prominent rise in noncompliance was observed after the end of the second year of life, when the mean age was 26.6 months (SD, 0.9; 95% CI, 26.3–26.9). There was a significant positive correlation between instances of noncompliance and both the number of brace phase appointments and the age of the patient (p < 0.001).
Conclusions
Noncompliance with foot abduction brace started at the end of the first year and peaked after the end of the second year.
Keywords: Clubfoot, Talipes equinovarus, Clubfoot brace, Noncompliance
The Ponseti technique became the standard method for treating idiopathic talipes equinovarus clubfoot deformity. If applied correctly, it can eliminate the need for extensive operative correction in over 98% of patients.1,2) However, the success of this method is not complete without the bracing phase, which lasts 4–5 years to maintain correction and prevent relapse of the deformity.3,4)
The Denis Browne brace is the most effective foot abduction brace (2 shoes connected by a bar) used to hold the corrected foot/feet in an abduction, external rotation, and dorsiflexion position according to the Ponseti method recommendations.3,5) The brace-wearing protocol (BWP) recommends wearing the brace for 23 hours per day in the first 3 months, then during naps and at night, with an average of 12–14 hours per day in the remaining period until the age of 4 years.4,6,7)
Noncompliance with bracing and its protocol is the most common cause of relapse.1,8,9) The rate of noncompliance may reach up to 61%.5) The noncompliant patients had greater odds of relapse of their deformity than the compliants.3)
Noncompliance with the brace may be due to skin breakdown (ulceration and blistering), the child’s irritability from the restriction of leg motion, parents fed up or inconvenienced by the brace, especially in unilateral cases, or having 3 or more children.10,11) It has not been proven to be related to family education, culture, or income level.12) The literature does not clarify when noncompliance occurs—specifically, during which period of the bracing phase, or in which age group.
This study aimed to determine when noncompliance is most likely to occur. The objectives were to determine the stage of the bracing phase at which noncompliance is most common, determine the child’s age at which the noncompliance is commonly observed, and check for a possible association between noncompliance and the bracing phase or the patient’s age.
METHODS
The approval to conduct the study was obtained from the Research Ethics Committee/General Directorate of Duhok Health, Duhok/Kurdistan Region, Iraq (No. 1342022-2-4). Informed consent was obtained from the parents/guardians of the children included in the current study. This descriptive observational study was conducted at the Early Detection of Childhood Disability Center in Duhok City, Kurdistan Region, Iraq, from January 2020 to October 2024.
Participants and Sampling
Inclusion criteria were children with idiopathic talipes equinovarus deformity for whom complete correction was achieved by the Ponseti method. Exclusion criteria were patients with syndromic clubfoot deformity, correction achieved after the first year of life, and those who underwent surgical correction. Cases that met the inclusion criteria were collected using purposive non-probability sampling.
The Brace and Brace-Wearing Program
After the Ponseti method (serial manipulation and casting sessions) completely corrected the talipes equinovarus deformity, a foot abduction brace was applied to the patient’s feet. According to the Ponseti recommendations, the brace was in the form of high-top open-toe shoes connected by a bar. The family (parents/guardians) was given instructions on how to wear the brace on the patient’s feet.
The BWP was explained to the family, especially about the duration of wearing the brace per day until the end of the treatment program (wear the brace for 23 hours per day in the first 3 months and then for naps and night with an average of 12–14 hours per day in the remaining period until the age of 4 years according to the recommended brace wearing protocol).4,6) The importance of adherence to this protocol was also explained to the family to prevent relapse of the deformity.
Follow-up of the Patients
To conduct the current study, each patient was observed—as for all cases of clubfoot deformity corrected by the Ponseti method—until the end of BWP (at the end of the fourth year). Relevant data (like the patient’s age and adherence to the BWP) were retrieved through a series of appointments. The appointments were made either by a clinical interview during the patient’s routine visit to the mentioned medical center for follow-up (including foot examination and brace assessment) or via a phone call. Each patient in the present study had 18 follow-up appointments, 3 appointments in the first 3 months (every month), and 15 appointments (every 3 months) until the end of the fourth year. The investigator suggested using the terms “induction phase” for the first 3 months and “naps and night phase” for the remaining period of the BWP.
Sample Size
A sample size of approximately 30 cases with talipes equinovarus deformity was considered sufficient to provide satisfactory descriptive qualitative information about the problem for this study and to avoid a saturation point (repetitive data).13,14)
Data Collection
The data of each patient were recorded and saved in a separate file. Each file had 2 parts. Part 1 included general information about the patient, such as name, sex, age at the time the deformity was corrected and the brace was started, and the phone numbers of family members (parents or guardians). Part 2 included a follow-up chart that was used to record relevant data at each appointment. This part included the appointment date, which was at the end of the determined month, the age of the patient at the appointment, and adherence to the brace (compliance).
The patient was categorized at each appointment into one of the following categories: compliant if the patient wore the brace optimally or sub-optimally (more than 6 hours) as BWP recommends, or noncompliant if the patient wore or used the brace for a few hours but less than half the recommended time (less than 6 hours). These categories were subjective and suggested by the investigator of the current study. The objective measure of counting and recording the number of hours the patient wears the brace may be difficult and impractical for the family every day and for about 4 years. Grading suggested by Sætersdal et al.15) is not always applicable.
Data Analysis
The data were introduced into the computer via the SPSS software program version 23.0 (IBM Corp.) for further statistical analysis. The following variables were generated: sex, the patient’s age (in months) when the deformity was corrected and brace wear began, the patient’s age (in months) at each appointment, and compliance (yes or no) at each appointment.
Outcomes
The primary outcomes were the mean age of the patients at each appointment and the count (number and percentage) of compliant and noncompliant patients at each appointment. The objective was to determine the mean age of the patients at the appointment when noncompliance commonly occurred. The secondary outcomes were to assess a possible association between the appointment and patient age with the counts of noncompliant patients and the relation of sex to noncompliance.
Statistical Tests
Mean and standard deviation (SD) with a 95% CI were used to describe the descriptive data. For the primary outcome, the Kaplan-Meier survival analysis was used to estimate the probability of noncompliance (time-to-event outcome) over time. The Log Rank (Mantel-Cox) test was used to identify the statistically significant difference regarding the count of noncompliance (event). Pairwise comparisons over strata were used to compare the number of noncompliance instances during appointments. Results were considered statistically significant when the p-value was less than 0.05.
For the secondary outcome, Kendall’s Tau rank correlation test was used to identify a possible simple association (correlation) between the bracing phase appointments (ordinal variable) and the counts of noncompliant patients (scale variable). Pearson correlation test was used to identify a simple association (correlation) between the mean age of the patients at each appointment and the counts of noncompliants (both variables were of scale type). The correlation coefficient (CC) was also used to interpret the direction (positive or negative values) and the strength of the correlation according to Cohen’s recommended guidelines: a value less than 0.3 indicates a weak relationship, between 0.3 and 0.5 indicates a moderate relationship, and greater than 0.5 indicates a strong relationship.16) To test for a possible relationship between sex and the number of non-compliant patients, the chi-square test was used. The value of Fisher’s exact test was considered when one of the cells had a value less than 5.
RESULTS
Thirty-six cases of talipes equinovarus deformity, 23 males and 13 females, were included in this study. Two of them (both males) were lost to follow-up after the sixth and eighth appointments, and they did not complete the follow-up chart; therefore, they were excluded from the statistical analysis. The final number of cases who completed the BWP (follow-up for 4 years) and were included in the statistical analysis was 34 (21 males and 13 females). The deformity was bilateral in 24 cases (48 feet) and unilateral in 10 cases (4 on the right and 6 on the left), for a total of 58 feet.
The mean of their age at the time of deformity correction and commencement of the brace was 2.3 months (SD, 0.6), and at the final follow-up appointment was 50.6 months (SD, 0.9). Noncompliance occurred in 11 of 34 cases (32.4%). There were 7 male and 4 female patients. All patients were considered optimally compliant with the brace until the fifth appointment, at which point the mean age was 11.5 months (SD, 0.8 months) (Table 1). After that, compliance started diminishing, and noncompliance increased. However, only 2 of 34 cases became noncompliant at the ninth appointment, at which point the mean age was 23.6 months (SD, 0.8 months). The number of noncompliant cases increased rapidly at the tenth appointment when the mean age of the patients was 26.6 months (SD, 0.9; 95% CI, 26.3–26.9). This increase was statistically significantly different from the other appointments when pairwise over-strata comparison was done between the appointments by the Log-Rank (Mantel-Cox) test (p = 0.008). Most of the children (23 / 25) who remained compliant at the tenth appointment (mean age, 26.6 months; SD, 0.9 months) remained compliant until the 18th appointment (mean age, 50.6 months; SD, 0.9 months) when the BWP finished.
Table 1. Mean Age of the Patients and Compliance Counts at Each Appointment.
| Appointment | Patients’ age (mo) | Compliance | |
|---|---|---|---|
| Compliant | Noncompliant | ||
| Beginning | 2.3 ± 0.6 | - | - |
| 1 | 3.4 ± 0.6 | 34 (100) | 0 |
| 2 | 4.4 ± 0.7 | 34 (100) | 0 |
| 3 | 5.4 ± 0.7 | 34 (100) | 0 |
| 4 | 8.4 ± 0.7 | 34 (100) | 0 |
| 5 | 11.5 ± 0.8 | 34 (100) | 0 |
| 6 | 14.6 ± 0.8 | 33 (97.1) | 1 (2.9) |
| 7 | 17.6 ± 0.8 | 33 (97.1) | 1 (2.9) |
| 8 | 20.6 ± 0.8 | 32 (94.1) | 2 (5.9) |
| 9 | 23.6 ± 0.8 | 32 (94.1) | 2 (5.9) |
| 10 | 26.6 ± 0.9 | 25 (73.5) | 9 (26.5) |
| 11 | 29.6 ± 0.9 | 25 (73.5) | 9 (26.5) |
| 12 | 32.6 ± 0.9 | 25 (73.5) | 9 (26.5) |
| 13 | 35.6 ± 0.9 | 24 (70.6) | 10 (29.4) |
| 14 | 38.6 ± 0.9 | 24 (70.6) | 10 (29.4) |
| 15 | 41.6 ± 0.9 | 24 (70.6) | 10 (29.4) |
| 16 | 44.6 ± 0.9 | 24 (70.6) | 10 (29.4) |
| 17 | 47.6 ± 0.9 | 23 (67.6) | 11 (32.4) |
| 18 | 50.6 ± 0.9 | 23 (67.6) | 11 (32.4) |
Values are presented as mean ± standard deviation or number (%).
There was a significant monotonic positive correlation between the number of noncompliant patients and both the number of appointments (CC, 0.925; p < 0.001) and the mean age of the patient (CC, 0.937; p < 0.001) (Figs. 1 and 2). Conversely, the correlation was negative between the percentage of complaints and the patients’ mean age (Fig. 3). The difference between the noncompliant patients regarding their sex was not statistically significant (p = 0.591) (Table 2).
Fig. 1. Relationship between appointments and the number of noncompliant patients.
Fig. 2. Relationship of mean patient age with the number of noncompliant patients.
Fig. 3. Relationship of mean patient age with the percentage of complaints.
Table 2. Relationship between Sex and Noncompliance.
| Sex | Compliance | Total | Chi-square value | p-value | |
|---|---|---|---|---|---|
| Compliant | Noncompliant | ||||
| Male | 14 (66.7) | 7 (33.3) | 21 (100) | 0.024 | 0.591 |
| Female | 9 (69.2) | 4 (30.8) | 13 (100) | ||
Values are presented as number (%).
DISCUSSION
The current study showed that noncompliance with the feet abduction brace started after the end of the first year of life when the mean age of the patients was 14.6 months (SD, 0.8 months). The prominent rise of the noncompliant count was after the end of the second year of life (mean age, 26.6 months; SD, 0.9 months; 95% CI, 26.3–26.9). Although the brace was worn during naps and nighttime, some children experienced discomfort. The exact cause is not clear. One possible explanation is the increase in the child’s physical activity during this developmental period; however, such activity typically occurs during the daytime, when the child is out of bed. The current study could not confirm or reject this. A new study that monitors the child’s behavior during sleep while wearing the brace via a video camera, may clarify this issue.
During the first year of life, all the children complied with the brace but exhibited different behaviors. Some began to become noncompliant earlier, while others remained compliant until BWP ended. Family may have an essential role in helping the child to accept the brace. Although family education and economic level have not yet been proven to play a role, perhaps an overcrowded household with many children might.11,12) Noncompliance with the brace increases the risk of deformity relapse. Researchers have invented different options to improve patient adherence to the brace.5,17,18) Available comparative studies, however, showed that these orthoses were inferior to the bilateral limb orthosis in preventing recurrences in clubfoot treatment.19,20) Nevertheless, they can be used as a salvage option to improve compliance and may diminish the relapse rate.21,22)
In summary, and based on the results of the current study, it can be concluded that a foot abduction brace can be used until the end of the second year of life as 94.1% of the patients were compliant. After that, if the child starts to become noncompliant, modification of the BWP by changing the orthotic with one of the alternative substitutes may salvage the situation and improve compliance.
The association between the brace phase appointment and/or the patient's age with noncompliance was positive and monotonic. Therefore, with further appointments and/or a gradual increase in the patient’s age, a greater number of patients were suspected to be noncompliant. The author suspected this association represented a simple correlation rather than a causal (cause-effect) relationship.
As a limitation of the study, sampling was done using a purposive (judgment) non-probability method. This approach was chosen to select the most valuable samples for the research’s purpose. The sampling had no random criteria because the study was exploratory and qualitative. It had been conducted to develop information about the problem under study in this research (when most children start to become noncompliant with the brace) within this limited population (patients with idiopathic talipes equinovarus deformity) rather than to test a hypothesis or make a statistical inference. Different methods were used for data collection to minimize the risk of observer bias.
The sample size was small because the study focused on a rare medical condition (limited population) with a global prevalence of approximately 1.18 cases per 1,000 live births.23) The research population was homogeneous and a non-probability sampling method was used. The research had a specific objective, and the number of variables to be estimated was small.
In conclusion, noncompliance with the foot abduction brace started at the end of the first year and peaked after the end of the second year when the mean age of the patients was 26.6 months (SD, 0.9; 95% CI, 26.3–26.9).
Footnotes
CONFLICT OF INTEREST: No potential conflict of interest relevant to this article was reported.
References
- 1.Rastogi A, Agarwal A. Long-term outcomes of the Ponseti method for treatment of clubfoot: a systematic review. Int Orthop. 2021;45(10):2599–2608. doi: 10.1007/s00264-021-05189-w. [DOI] [PubMed] [Google Scholar]
- 2.López-Carrero E, Castillo-López JM, Medina-Alcantara M, Domínguez-Maldonado G, Garcia-Paya I, Jiménez-Cebrián AM. Effectiveness of the ponseti method in the treatment of clubfoot: a systematic review. Int J Environ Res Public Health. 2023;20(4):3714. doi: 10.3390/ijerph20043714. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 3.Agarwal A, Rastogi A, Rastogi P. Relapses in clubfoot treated with Ponseti technique and standard bracing protocol- a systematic analysis. J Clin Orthop Trauma. 2021;18:199–204. doi: 10.1016/j.jcot.2021.04.029. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 4.Desai L, Oprescu F, DiMeo A, Morcuende JA. Bracing in the treatment of children with clubfoot: past, present, and future. Iowa Orthop J. 2010;30:15–23. [PMC free article] [PubMed] [Google Scholar]
- 5.Alves C. Bracing in clubfoot: do we know enough? J Child Orthop. 2019;13(3):258–264. doi: 10.1302/1863-2548.13.190069. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 6.Cady R, Hennessey TA, Schwend RM. Diagnosis and treatment of idiopathic congenital clubfoot. Pediatrics. 2022;149(2):e2021055555. doi: 10.1542/peds.2021-055555. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 7.Zionts LE, Dietz FR. Bracing following correction of idiopathic clubfoot using the Ponseti method. J Am Acad Orthop Surg. 2010;18(8):486–493. doi: 10.5435/00124635-201008000-00005. [DOI] [PubMed] [Google Scholar]
- 8.Zhao D, Liu J, Zhao L, Wu Z. Relapse of clubfoot after treatment with the Ponseti method and the function of the foot abduction orthosis. Clin Orthop Surg. 2014;6(3):245–252. doi: 10.4055/cios.2014.6.3.245. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 9.Hallaj-Moghaddam M, Moradi A, Ebrahimzadeh MH, Habibzadeh Shojaie SR. Ponseti casting for severe club foot deformity: are clinical outcomes promising? Adv Orthop. 2015;2015:821690. doi: 10.1155/2015/821690. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 10.Hu W, Ke B, Niansu X, et al. Factors associated with the relapse in ponseti treated congenital clubfoot. BMC Musculoskelet Disord. 2022;23(1):88. doi: 10.1186/s12891-022-05039-9. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 11.Jawadi AH, Al-Abbasi EM, Tamim HA. Factors predicting brace noncompliance among idiopathic clubfoot patients treated with the Ponseti method. J Taibah Univ Med Sci. 2015;10(4):444–448. [Google Scholar]
- 12.Ramírez N, Flynn JM, Fernández S, Seda W, Macchiavelli RE. Orthosis noncompliance after the Ponseti method for the treatment of idiopathic clubfeet: a relevant problem that needs reevaluation. J Pediatr Orthop. 2011;31(6):710–715. doi: 10.1097/BPO.0b013e318221eaa1. [DOI] [PubMed] [Google Scholar]
- 13.Marshall B, Cardon P, Poddar A, Fontenot R. Does sample size matter in qualitative research? A review of qualitative interviews in is research. J Comput Inf Syst. 2013;54:11–22. [Google Scholar]
- 14.Boddy CR. Sample size for qualitative research. Qual Mark Res. 2016;19(4):426432 [Google Scholar]
- 15.Sætersdal C, Fevang JM, Fosse L, Engesæter LB. Good results with the ponseti method: a multicenter study of 162 clubfeet followed for 2–5 years. Acta Orthop. 2012;83(3):288–293. doi: 10.3109/17453674.2012.693015. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 16.Schober P, Boer C, Schwarte LA. Correlation coefficients: appropriate use and interpretation. Anesth Analg. 2018;126(5):1763–1768. doi: 10.1213/ANE.0000000000002864. [DOI] [PubMed] [Google Scholar]
- 17.Kessler JI. A new flexible brace used in the Ponseti treatment of talipes equinovarus. J Pediatr Orthop B. 2008;17(5):247–250. doi: 10.1097/BPB.0b013e32830cc3e5. [DOI] [PubMed] [Google Scholar]
- 18.McCartney S, Turner S, Davies K, Morris J, Sproston C, Kiely N. A new unilateral abduction orthosis for Ponsetitreated clubfoot: A cohort study to assess compliance. Prosthet Orthot Int. 2019;43(3):325–330. doi: 10.1177/0309364618814866. [DOI] [PubMed] [Google Scholar]
- 19.Sætersdal C, Fevang JM, Engesæter LB. Inferior results with unilateral compared with bilateral brace in Ponseti-treated clubfeet. J Child Orthop. 2017;11(3):216–222. doi: 10.1302/1863-2548.11.160279. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 20.Agarwal A, Rastogi A, Talwar J, Deo NB, Rastogi P. Unilateral limb orthosis for maintenance of deformity correction following treatment of clubfoot with Ponseti technique: a systematic review. J Pediatr Orthop B. 2022;31(2):e195–e201. doi: 10.1097/BPB.0000000000000897. [DOI] [PubMed] [Google Scholar]
- 21.Abdi R, Hajzargarbashi R, Baghdadi T, Shirazi MR, Omidnia B, Tajik H. Effect of a modified dynamic accordion hinge Denis Browne brace on the success rate of the Ponseti method in idiopathic clubfoot: a preliminary study. J Pediatr Orthop B. 2017;26(2):112–115. doi: 10.1097/BPB.0000000000000357. [DOI] [PubMed] [Google Scholar]
- 22.Chen RC, Gordon JE, Luhmann SJ, Schoenecker PL, Dobbs MB. A new dynamic foot abduction orthosis for clubfoot treatment. J Pediatr Orthop. 2007;27(5):522–528. doi: 10.1097/bpo.0b013e318070cc19. [DOI] [PubMed] [Google Scholar]
- 23.Smythe T, Rotenberg S, Lavy C. The global birth prevalence of clubfoot: a systematic review and meta-analysis. EClinicalMedicine. 2023;63:102178. doi: 10.1016/j.eclinm.2023.102178. [DOI] [PMC free article] [PubMed] [Google Scholar]