Background. Cleft lip and palate (CLP) cause severe malocclusion, which requires numerous orthodontic interventions in specialized centers. There is little literature regarding the overall orthodontic burden of care for these patients.
Objectives. The aim of the study was the evaluation of orthodontic-care burden for patients treated in the Division of Facial Abnormalities at the Department of Dentofacial Orthopedics and Orthodontics of Wroclaw Medical University in Poland.
Material and methods. The medical data of patients with complete unilateral and bilateral cleft lip and palate (ULCP and BLCP) who finished orthodontic treatment between 2012 and 2019 was evaluated. The duration of orthodontic treatment, the number of removable appliances, the number of kilometers traveled as well as the number of visits and surgical procedures performed were recorded. The sample was divided into 2 groups according to the World Health Organization (WHO) International Classification of Diseases (ICD-10) diagnosis codes. All data was subjected to statistical analysis.
Results. For the UCLP patients (n = 54), the mean time of orthodontic treatment was 9.24 years, the mean number of orthodontic appointments was 62.91, the mean number of removable appliances was 4.12, the mean number of surgical procedures was 3.35, and the mean distance traveled to visit the center for orthodontic appointments was 5,466.95 km. For the BCLP patients (n = 19), the mean time of orthodontic treatment was 10.16 years, the mean number of orthodontic appointments was 66.26, the mean number of removable appliances was 4.12, the mean number of surgical procedures was 4.05, and the mean distance traveled to visit the center for orthodontic appointments was 3,758.23 km.
Conclusions. The orthodontic treatment of patients with UCLP and BCLP is very burdensome for the patients. However, the burden of care in the Division of Facial Abnormalities at the Department of Dentofacial Orthopedics and Orthodontics of Wroclaw Medical University in Poland is not greater than in other European countries.
Key words: orthodontics, cleft lip, cleft palate, burden of care
The main purpose of cleft palate orthodontic treatment is to minimize craniofacial growth dysfunction and to achieve correct pronunciation in the patient. The achievement of this goal requires interdisciplinary treatment by specialists of many fields, including neonatologists, pediatricians, orthodontists, maxillofacial surgeons, plastic surgeons, laryngologists, phoniatricians, speech therapists, pediatric dentists, prosthetists, and psychologists. This results in treatment lasting from childhood to adulthood and is very burdensome for the patient and their parents.
The term ‘burden of care’ is commonly used nowadays in medical care. It refers to a balance between the compromises the patient and their family need to make and the benefits the child receives. The burden of care in this specific case refers to the total number of surgeries, treatment episodes, appointments, and procedures during the patient’s whole multidisciplinary rehabilitation.1 Many centers where children with similar defects are treated have analyzed orthodontic-care burden for their patients and they strive to minimize it, while maintaining the best treatment results.2
The importance of the topic is emphasized by the fact that it has been discussed at World Health Organization (WHO) Meetings on International Collaborative Research on Craniofacial Anomalies. The WHO has even created strategies to reduce the health-care burden of craniofacial anomalies.1 However, despite the systematic introduction of these strategies, the burden of treatment remains a huge problem for patients and their families. In 2002, a study comparing the treatment burden of unilateral cleft lip and palate (UCLP) in 5 European centers was carried out.3 The average length of orthodontic treatment varied up to 5 years between the centers. At this time, there is only 1 report (from the UK) taking into account the orthodontic-care burden for patients with bilateral cleft lip and palate (BCLP).4 Bearing in mind that BCLP causes greater impairment than UCLP,5 it can be expected that its treatment might be longer and more demanding, and therefore more burdensome for the patient.
This study aimed to assess the orthodontic-care burden for patients with UCLP and BCLP from a single center in Poland.
Material and methods
All the data necessary to carry out the analysis was obtained by reviewing the existing medical records at the Academic Dental Policlinic in Wrocław, Poland. For the purposes of the research, all patients of both sexes who met the following inclusion criteria were enrolled: UCLP or BCLP (according to the WHO International Classification of Diseases (ICD-10); Q37.1 and Q37.0, respectively), patients who underwent orthodontic treatment in 1 center – the Division of Facial Abnormalities, Department of Dentofacial Orthopedics and Orthodontics, Wroclaw Medical University, Poland – and finished the therapy between 2012 and 2019. The following information was retrospectively obtained from the patients’ existing records: sex, date of birth, place of residence, cleft side (in the case of UCLP), age at registration at the Academic Dental Policlinic, age at the beginning and ending of orthodontic treatment, number of all visits at the clinic, number of visits forced by the damage of the appliance, age at the beginning and ending of treatment with a mobile and permanent appliance with the number of the related follow-up visits, number of removable appliances in use, age during the lip and palate surgery, age during bone grafting to the alveolar process, number of all operations related to the defect, and total number of days spent in the hospital. The distance traveled for treatment at the clinic was calculated by multiplying the number of all visits by the distance from the patient’s place of residence to the clinic based on the fastest suggested route according to Google Maps.
The data was described using individual descriptive statistics, such as central tendency (mean – M and median – Me) and measures of variability (standard deviation – SD, the minimum and maximum values of the variables).
The sample consisted of 73 patients, including 54 with one-sided cleft (18 women and 36 men) and 19 patients with bilateral cleft (10 women and 9 men). For UCLP, no statistically significant difference was observed in the mean treatment duration for both sexes. Women’s therapy was 51.1 days longer. This is a relatively small difference as compared to the total length of treatment. However, in the case of BCLP, the difference was significant. The average length of treatment for women was 11.6 years, and for men only 8.56 years.
All data was statistically analyzed with the determination of a 95% confidence interval (CI). Table 1 shows the results for the patients with UCLP. Complete orthodontic treatment took a mean time of 9.24 years. The mean number of orthodontic appointments was 62.91. The mean total distance traveled for the patient/family to attend orthodontic appointments at the clinic was 5,466.95 kilometers. Table 2 shows the results for the patients with DCLP. Complete orthodontic treatment took a mean time of 10.16 years. The mean number of orthodontic appointments was 66.26. The mean total distance traveled for the patient/family to attend orthodontic appointments at the clinic was 3,758.23 km.
From among all the analyzed cases with UCLP, 9% were treated without a mobile appliance, and 4% without a fixed appliance. In BCLP, these results were 11% and 16%, respectively. The lack of treatment with a mobile appliance was in most cases caused by the late beginning of treatment. The average age at registration at the clinic was around 4 years in both groups; however, the maximum age was as much as 18 years old.
The treatment of patients with CLP is long-lasting and requires a lot of commitment from both parents and patients. The initial malformation as well as the subsequent surgical procedures – the stitching of the fissure within the lip and the palate – lead to scars that cause three-dimensional disorders in the development of the maxilla, which causes complex malocclusion. The treatment of patients with clefts consists of surgical and orthodontic procedures. The most commonly used surgical protocol is two-stage treatment – the suturing of cleft lip at around 6 months of age and of cleft palate at around 12–18 months of age. There is also a single-stage treatment protocol, in which the suturing of the lip and the palate is performed simultaneously. In Poland, both surgical treatment protocols are used, depending on the center where the treatment is carried out. Orthodontic treatment can be divided into early orthopedic treatment before cleft lip correction, orthodontic treatment during the period of primary dentition and treatment with mixed and permanent dentition. Early pre-surgical treatment aims to bring the split tissues closer together and to minimize the extent of the subsequent surgery. Taping, McNeil’s plates and nasoalveolar molding (NAM) plates are used at this stage. After the treatment, the child is under the constant care of the orthodontist, who monitors the eruption of the teeth in order to intervene at the right time. The postoperative scar of the jaw often disturbs the growth of the maxilla, which makes it necessary to use rapid maxillary expansion and a facial mask. This usually takes place during the period of mixed dentition. After obtaining the correct bite, we align the teeth with a fixed appliance. Meanwhile, surgical and prosthetic interventions due to hypodontia, hyperdontia and the deformation of individual teeth proceed.6, 7 The same protocol of treatment is followed in Australia.8
Many centers around the world have analyzed the effects of the orthodontic treatment of people with CLP in terms of treatment burden for patients. In the comparative study of 5 treatment centers from Northern Europe mentioned in the introduction, the length of orthodontic treatment with the correlated total number of appointments as well as the number of days spent in the hospital were analyzed.3 Their results were very divergent. The length of treatment varied even 5.2 years between the centers, and the number of visits varied from 49 to even 96. The number of days spent in the hospital varied from 24 to 60. In another study, conducted in Oslo, patients with ULCP spent an average of 24.1 days in the hospital; orthodontic treatment with permanent dentition lasted 2.4 years.9 Outside Europe, a similar analysis was carried out in Brazil, where orthodontic treatment lasted on average 11.68 years and required 61.89 visits.10 The data which we analyzed was age at the time of the surgeries, the total number of surgeries and the total number of days spent in the hospital. Patients suffering from UCLP spent fewer days in the hospital (M: 18.24 days) than patients with BCLP (M: 30.42 days). This was due to the fact that UCLP required fewer surgical procedures as compared to BCLP (M: 3.35 and M: 4.05, respectively).
A large number of surgical procedures results from the fact that some of them must be repeated. Scientific research shows that in the case of the split lip surgery, the procedure must be repeated within a year in up to 36% of patients.11 But according to the results of the Americleft study project, the use of secondary surgery did not lead to a significantly better nasolabial appearance than what was achieved in children who underwent only primary surgery.12, 13 The Americleft study project also showed that children with UCLP had fewer surgeries in the United States than in Europe; the mean number varied from 1 to 4.14 In Europe, the average number of days spent in the hospital by children with UCLP varied from 24 to 60, depending on the center.3, 15 Our results also show that the procedures were performed earlier in children with UCLP than in those with BCLP. The mean age at lip repair for UCLP was 6.96 months old, and for BCLP 9.42 months old. The mean age at palate repair was 18.09 and 23.37 months old, respectively.
The orthodontic treatment protocol used at the Division of Facial Abnormalities, Wroclaw Medical University, includes treatment with a removable appliance, maxillary expansion if the patient requires it (according to other authors, up to 70% of patients need it16), and then treatment with a fixed appliance. If bone grafting to the alveolar process is needed, it is most often performed before or after maxillary expansion, considering that the appliance can be fixed only after about 3 months post bone grafting, but not later than 6 months.17 The average length of treatment with fixed appliances in patients with unilateral cleft was 3.1 years, which does not differ from the results from other centers. For example, in Oslo, the average duration of treatment with permanent dentition was 2.4 years.9 One of the hospitals in the UK has an average score of 3.0 years.4 For comparison, the treatment of people without cleft with full dentition lasts on average 19.9 months.18
In a study whose goal was to calculate the amount of treatment and the associated travel experienced by 5 groups of patients treated at different centers, the average duration of the entire orthodontic treatment ranged from 3.3 to 8.5 years.15 With these results, the average of 9.24 years obtained in our results does not look satisfactory. One of the reasons may be the fact that patients treated at the center are included in the Program of Orthodontic Care for Children with Facial Abnormalities. Due to the program, parents do not bear the costs of treatment. However, in the years 2002–2004, the program was not implemented, which raises the presumption that some parents could not afford to continue treatment. This is only the authors’ supposition, but taking this thesis into account would explain the extension of the treatment time.
Outside Europe, a similar retrospective study was conducted in Brazil. There, the average duration of the entire orthodontic treatment was even longer – 11.68 years.10 The comparison of the average values obtained in our research vs other countries’ research is presented in Table 3. On the issue of BCLP, we can only compare our results with a London hospital. As initially assumed, in both studies, the results showed that the treatment of bilateral cleft was longer than in the case of UCLP. According to the data from the present study, the average duration of treatment with a removable appliance was 5.65 years, and with a fixed appliance 3.5 years. The total length of treatment was 10.16 years. In the study presented by Hameed et al., the mean time length values were 1 year for a removable appliance, 3 years for a fixed appliance and 3.5 years for total treatment.4 It is easy to notice that treatment with a removable appliance lasted much longer in Wrocław than in London. This data is not sufficient to draw conclusions, but it is alarming information supporting the need to carry out analyses in other centers. Conducting tests in several centers on larger groups of patients would make it possible to assess whether the problem really exists and to verify the possible causes of the prolonged treatment with removable appliances. Some authors think that there is an association between the treatment outcome and intensity; thus, a simple protocol with minimum economic burden can provide better or equally good outcomes with less burden of care.19
The analysis of the number of visits and the distance traveled by the patient for treatment at the clinic shows that for patients with BCLP, the number of visits was higher in proportion to the duration f treatment (M: 66.26). However, this does not coincide at all with the distance traveled, with its average being 1,708.72 km higher for UCLP, despite a smaller number of visits (M: 62.91). The distance the patient had to travel was very small as compared to patients from Brazil (M: 38,978.58 km).10 This is due to the high availability in Poland of clinics covered by the Program of Orthodontic Care for Children with Facial Abnormalities – there are 16 such clinics (data from the National Health Fund).
The orthodontic treatment of patients with ULCP and BLCP is long-lasting and very burdensome for the patients. The treatment of BLCP lasts longer and requires more procedures than in the case of ULCP. The orthodontic-care burden of patients ULCP and BLCP treated at the Division of Facial Abnormalities, Wroclaw Medical University, does not differ from that in other European countries. Therefore, considering that the estimation of the burden of care depends on the results obtained, it should be remembered that a positive objective assessment of treatment may not be synonymous with a subjective assessment by the patient,20 which requires further research.