Keywords
clubfoot - foot deformities - foot orthoses - orthopedic devices
Introduction
Congenital clubfoot (CC) is a complex three-dimensional deformity including pes equinus,
varus, adductus, and cavus.[1] CC results from ligamentous and myotendinous abnormalities and occurs in 1/1,000
live births.[2]
The Ponseti method is the main form of treatment and consists of joint manipulations
of the connective tissue using serial casts. After correction, the patient must use
a Dennis-Brown (DB) orthosis for 2 to 4 years.[1]
Many parents of children diagnosed with CC are afraid of the disease and its treatment.
CC causes stress, anxiety, and depression in parents.[3] Therefore, it is critical to clarify all doubts regarding the therapeutic process.[4] The Ponseti method requires a lot of commitment from the medical team and the patient's
family to be successful.[5]
[6]
Considering the significance of full adherence to treatment, this study aimed to identify
the main difficulties encountered in DB use by caregivers of CC patients.
Method
This descriptive exploratory study relied on a quantitative approach to the difficulties
of consistently using DB orthosis by children undergoing conservative CC treatment
using the Ponseti method in a reference hospital in Curitiba, PR, Brazil, from 2015
to 2018. The Committee of Research Ethics from our institution approved the study
under number CAAE 56086821.0.0000.5580. To collect the required information, we sent
a 16-item questionnaire addressing the child, their caregivers, and difficulties in
orthosis use to the caregivers. First, we contacted the caregivers (parents, grandparents,
or others) by cell phone to inform them that they would receive a questionnaire via
Google Forms and its purpose. The questionnaire could be answered by email or cell
phone. This study included caregivers of children over 4 years old diagnosed with
CC at birth and treated on an outpatient basis at the reference hospital.
Statistical Analysis
We created an Excel spreadsheet with all the answers and inserted it into the Sphinx
IQ2 software to analyze profile variables. Next, we proceeded to bivariate analyses
to relate the data obtained. We performed three statistical tests (p-value, chi-square,
and degree of freedom) concerning the variables looking for any statistical influence
on the difficulty of using the orthosis.
Results
Our sample consisted of 176 patients, and we managed to contact 114 of their caregivers.
However, only 36 caregivers answered the form, with a 31.6% response rate. Most (88.9%)
patients underwent treatment at the Brazilian Unified Health System (SUS, for its
acronym in Portuguese), 8.3% received treatment through an insurance plan, and 2.8%
had private treatment. The gender distribution revealed 63.9% of patients were males
and 36.1% were female. In 55.6% of subjects, CC was bilateral. CC affected the right
foot of 33.3% of patients and the left foot of 11.1% of subjects.
There was no significant correlation between gender and the side affected by CC ([Fig. 1]), meaning the reported statistical tests revealed no direct relationship between
gender and deformity. Most (80.6%) children required a calcaneus tenotomy. Orthosis
use after conservative treatment started when the child was less than 12 months old
in 66.7% of patients and continued until 48 months old in 38.9% of children. Most
(58.3%) caregivers denied any difficulty and 41.7% reported difficulties. There was
no association between difficulty in orthosis use and CC side or patient's gender
and age, demonstrating that difficulty is independent of these factors.
Fig. 1 Types of difficulty faced by the patient. Source: Authors (2022).
Of the 41.7% of caregivers reporting some orthosis-related difficulty, the main one
was the child's irritation during its use (93.3%). Other issues highlighted included
long using time (33.3%), orthosis cost (26.7%), difficulty in putting the orthosis
on (26.7%), and feeling sorry for the child (26.7%). Caregivers also cited difficulty
in changing the orthosis when it became too small (13.3%), the child not accepting
its use (6.7%), failure to obtain orthosis from SUS promptly (6.7%), and pressure
from family members to stop using the orthosis (6.7%). Discontinuation in orthosis
treatment occurred in 27.8% of patients; of these, none reported CC recurrence but
30% reported the need to resume treatment.
Discussion
The incidence of CC has had a similar pattern over the years: 64.5% in male patients
and 35.5% in female patients in 1951,[2] and a predominance of male patients (68%) in 2020.[4] In our study, 63.9% of patients were male. Regarding the affected side, our sample
showed a higher CC incidence on the right foot and a difference in frequency, with
75% affecting the right side and 25%, on the left side. In 2011, the incidence of
right foot deformities was 56%.[7] In 2020, the same pattern remained, with a 52.1% incidence on the right foot.[4] As for bilateral (55.6%) and unilateral (44.4%) CC, our data are consistent with
the literature. Stewart reported an equal distribution between bilaterality and unilaterality.[2]
In our study, 66.7% of patients started CC treatment using the Ponseti method at less
than 12 months old, and most (38.9%) completed it by 48 months old. This indicates
that most patients underwent 4 years of treatment, consistent with the literature
reporting an average follow-up period of 4.6 years.[6] Most (80.6%) of our patients required a percutaneous tenotomy of the calcaneus tendon,
consistent with reports from Ponseti[8] and, more recently, Bhaskar and Patni.[9]
In 2004, the lack of commitment to using the abduction orthosis was the main factor
associated with CC recurrence, with these patients being 183 times more susceptible
to relapses.[10] In 2011, researchers stated that the appropriate orthosis use directly relates to
the treatment success.[11] A report from 2022 found that 46% of patients with relapses were incorrectly using
the DB orthosis.[12] Of the 36 caregivers participating in our research, 10 (27.8%) reported having stopped
using the orthosis before the stipulated treatment period. Among these ten caregivers,
three (30%) had to restart treatment and resume orthosis use.
As mentioned before, almost 30% of our patients presented an increased risk of CC
recurrence and potential surgical intervention. A report from 2015 identified that
this inappropriate use increases the chance of needing a surgical procedure by 7.9-fold.[13] Furthermore, in 2013, researchers highlighted that treatment for recurrent CC is
not simple and does not result in the same outcome of improving foot mobility and
anatomy compared with conservative treatment.[14]
A study from 2013 described that parents of patients who received greater focus on
the need for adherence and clarification during classes about the disease had a lower
recurrence and higher therapeutic success when compared with the group that did not
receive these instructions[5] Similarly, in 2016, researchers stated the importance of commitment from the doctor
and the patient's family from changing casts to orthosis use.[6] A report from 2011 identified some factors specific to parents that influence orthosis
use, stating that parents with education levels of up to high school or less had a
greater chance of their children developing relapses.[11]
In our study, 41.7% of the sample presented some difficulty using the DB orthosis,
consistent with a 2022 study on the Mitchell-Ponseti orthosis stating that 46.7% of
families from CC patients reported issues with the orthosis.[15] Therefore, by integrating parents into the treatment, teaching them, and reinforcing
the need for orthosis use, it may be possible to reduce orthosis use discontinuation
or its irregular use since they would understand the direct relationship between success
and proper use.
In 2022, authors identified two main factors for the decrease in compliance with orthosis
use: first, after 18 months old, children sleep less, using the orthosis for less
time, as they cannot tolerate it when awake.[16] The second point reported by these authors is that when faced with this difficulty,
parents do not use the orthosis correctly or even discontinue its use.[16] Our research identified that the most important factor for irregular orthosis use
is child irritation, reported by 93% of study participants, consistent with descriptions
from previous authors.
Given the above, there is a need to address the reported factors to reduce CC recurrences.
Sheta and El-Sayed, in 2020, did not use orthosis, teaching parents stretching exercises
for daily performance, and giving them classes three times a week about the disease
and its treatment.[4] The authors reported that the children of these parents had above-average results
concerning functionality, pain, and other factors. However, the recurrence rate was
21%, consistent with the literature.[4] Therefore, despite removing the concern about inappropriate orthosis use, this method
is not more efficient than the traditional one to the point of replacing orthosis
completely.
Just as described in a 2022 study, the difficulties encountered by family members
in using the orthosis did not influence therapeutic success. In our study, despite
27.8% having discontinued orthosis use, none reported CC relapse.[15] Therefore, although the literature states that inappropriate orthosis use is the
main factor in CC recurrence, our study cannot state that orthosis-related difficulties
are direct factors for its inappropriate use.
Conclusion
Few studies specifically seek to understand why there are difficulties in using the
DB orthosis, even though it is the main orthosis used in SUS. The 41.7% rate of DB
orthosis use difficulty is unprecedented, and fundamental to creating strategies to
reduce its inappropriate use. Furthermore, our data allows us to understand that despite
the issues during orthosis use, they were not factors in CC recurrence. In addition,
we noted that the difficulty is independent of the patient's gender, age, and affected
side. However, the literature suggests that the socioeconomic condition of the parents
impacts recurrence. Finally, the limitation of this study was the low questionnaire
response rate.
