Stability of Surgical and Orthodontic Techniques for the Treatment of Patients with Cleft Palate: A Systematic Review A Review and a Proposal

By Elisa Darqué, Iván Nieto Sánchez, Inés Díaz Renovales, and Patricia Martín-Palomino Sahagún.

Abstract

Elisa Darqué is a doctor of dentistry (Alfonso X el Sabio University) and she’s a student in master’s degree in orthodontics (Alfonso X el Sabio University).

Iván Nieto Sánchez is a doctor of dentistry (Alfonso X el Sabio University); MS in Orthodontics (San Rafael Hospital); BS of Dentistry (Complutense University of Madrid); MS Biostatistics and Telemedicine for clinical practice and health management (UNED) Diploma in Lingual Orthodontics (University of Lyon I)

Introduction: Osteogenesis distraction is a medical technique in which surgery is performed to separate the bone, and a bone callus is created between the two segments that will ossify later. Unless stated otherwise, the process begins when incremental traction is applied to the repair callus joining the divided bone segments and continues for as long as this tissue is stretched. It is important to understand this concept to be able to compare it with orthognathic surgery in patients with cleft lip.

Objective: The main objective of this study is to examine how the cleft palate could be treated as efficiently as possible. We aimed to compare the stability of distraction osteogenesis versus orthognathic surgery in the treatment of cleft palate through a systematic review.

Patricia Martín-Palomino Sahagún

PhD in Dentistry (University Alfonso X el Sabio), Master in Orthodontics (University Alfonso X El Sabio), DDS (University Alfonso X El Sabio)

Materials and Methods: A systematic review was carried out on the PubMed and Medline, SciELO, ResearchGate, ScienceDirect, Dialnet, Web of Science, and Cochrane databases for articles published mainly in the last five years. We used the following keywords: “Distraction,” “cleft lip,” “orthognathic surgery,” “maxillary hypoplasia,” “Pierre Robin Syndrome,” “osteogenesis,” “cleft palate,” “cleft lip and palate treatment,” “distraction osteogenesis,” “maxillofacial surgery,” “callus distraction,” “callotasis,” “mandibula distraction,” “osteodistraction,” and “orthopedic surgery.” We added a filter to display articles from 2016 to May 2022.

Inés Díaz Renovales

PhD in Dentistry University Alfonso X el Sabio, Master in Orthodontics (University Alfonso X el Sabio), DDS (University Complutense Madrid)

Results: From the 67 articles included in the review, the authors observed a greater recurrence of point A (or decrease of ANB) in patients who had been treated with orthognathic surgery than those who had been treated with distraction.

Conclusion: According to the conclusions of several studies, it seems that distraction osteogenesis has better skeletal stability than orthognathic surgery in patients with cleft lip without growth. Moreover, orthognathic surgery has a much more important recurrence in the vertical plane than in the horizontal plane. In conclusion, with osteogenesis distraction, an overcorrection of 3040% must be made.

*This article has been peer reviewed

Introduction

Cleft lip and/or palate is one of the most serious congenital anomalies that affect the mouth and its surrounding structures. A cleft is a congenital abnormal space or gap in the upper lip, alveolus or palate. The term used for this condition is cleft lip. More specifically, the most appropriate terms are cleft lip, cleft palate, or cleft lip and palate. Malocclusions and cleft abnormalities have been described; however, few studies have associated malocclusions with oral clefts.1

Baek et al.2 reported similarities between types of malocclusions and the diverse classification of cleft mouths among Koreans. They also found that the type of cleft significantly influenced the development of a Class III malocclusion. Higher frequencies of crossbite and open bite were reported among patients with clefts. Chopra et al.3 reported that anterior open bite and high overjet were more common among children with clefts.2,3

A cleft lip with or without alveolar involvement was more frequently associated with Class I malocclusion, according to the study by Okoye et al.4 Angle Class I was significantly higher than other classes. According to their study, 20% of the patients had Class III malocclusion, while 12% had Class II.4 These results contrast with the those of Baeck et al.’s study,2 which found that the most frequent malocclusion was Class III, which was found in 72% of the participants.2 To treat this anomaly, two techniques were highlighted. The first of these is osteogenic distraction. It relies on the fact that new bone is formed with the preservation of its strength.5,6 Initially, the bone is transected surgically and allowed recovery time or latency time. This period varies from 0 to 7 days, depending on the surgical trauma. After this procedure, there is a period of activation in which the callus is stretched and new bone is created parallel to the traction vector. This period is also called distraction period. Once the elongation of the callus is sufficient, the activation stops and the consolidation time begins; the mineralization and ossification of the bone callus occurs.5,7

Another technique to treat these patients is orthognathic surgery. Such surgery for the correction of a malocclusion was performed in 1849. The Le Fort I osteotomy is a conventional and standard surgical procedure for the correction of maxillary

Figure 1. Biological effect of distraction. (a) Formation of a hematoma after osteotomy with infiltration of capillaries and granulation tissue. Distraction osteogenesis activation begins with the separation of bone fragments. (b) Strain-stress force elicited by the cellular event cascade. (c) A fibrous hypoplasia in patients with cleft lip palate. However, the risk of recurrence is high in patients with scar contractions. Therefore, maxillary advancement by distraction osteogenesis (DO) in the anteroposterior direction has become more popular. DO is now frequently recommended for the correction of maxillary hypoplasia in patients with a cleft lip palate (CLP). Several studies have shown that there is much less recurrence with DO than with surgery (8.2% vs. 37%, respectively).8

The objective of this study is to evaluate the stability of osteogenic distraction and orthognathic surgery in patients with cleft lip and palate.

Materials and Methods

This study was conducted and reported according to the preferred reporting items for systematic reviews and meta-analyses (PRISMA) guidelines.

Eligibility Criteria

This systematic review was conducted to observe which of the two techniques has the best stability with patients with a cleft lip. Randomised controlled trials, prospective studies, systematic reviews, case-control studies and cohort studies that compared the effects of an orthognathic surgery and osteogenesis distraction in subjects with a cleft lip were included.

Studies published before 2008, those not in Spanish, English, or French, duplicate articles, publications without full text and studies with patients who have had their arms or legs distracted were not included. In addition, studies that did not evaluate patients with unilateral or bilateral clefts were excluded. Case reports, case series and expert opinions were not included in this systematic review.

According to the Participants-InterventionsComparisons-Outcome-Study (PICOS) strategy, randomised controlled clinical trials on human patients were included if they met the following selection criteria:

• Participants (P): Adult patients or adolescents who have completed their growth with cleft lip and/ or palate.

• Intervention (I): Osteogenic distraction.

• Comparison (C): Group of patients who have undergone orthognathic surgery.

• Outcomes (O): Recurrence (movement of A point in mm) after the intervention and in the long term.

Information Sources, Search Strategy and Selection of Studies

To carry out an information search on osteogenic distraction and orthognathic surgery in patients with cleft palate, we used PubMed, Web of Science, SciELO, and Cochrane.

To find articles that match our search, a search strategy was used. The MeSH terms were elaborated to develop the following equation: (osteogenesis distraction OR maxillary distraction OR osteodistraction) AND (cleft lip AND palate) AND (othognathic surgery OR orthognathic surgeries OR orthopedic surgery).

To begin with, the titles and abstracts were evaluated by two evaluators (ED and MC) to reduce errors and choose all the articles that met the search requirements. When the information from the abstract was not sufficient, full texts were read for a complete analysis. The decision to include the articles was made after full texts were analyzed by the two experts. In case of discrepancy, a third reviewer (IN) was called to either accept or reject the article. The search included articles from 2008 to September 2022. We applied filters for languages (French, English, and Spanish), year of publication (last five years), and types of studies.

The following keywords were used:

“Distraction,” “cleft lip,” “orthognathic surgery,” “hypoplastic maxilla,” “osteogenesis,” “cleft palate,” “cleft lip and palate treatment,” “distraction osteogenesis,” “maxillofacial surgery,” “callus distraction,” “callotasis,” “maxillary distraction,” “osteodistraction,” and “orthopedic surgery.”

Results Study Selection

The flowchart of the research selection procedure, in accordance with the PRISMA guidelines, is presented in Figure 2.

The electronic-database search identified a total of 174 articles. After eliminating 61 duplicates, 113 studies were screened based on title and abstract to identify potentially eligible articles. This led to the exclusion of 94 publications, and full texts of the remaining 13 articles were retrieved and analysed according to the eligibility criteria. Subsequently, four articles were excluded after full-text assessment because they were retrospective cohort studies.9-12 Finally, eight clinical trials and five systematic reviews were considered eligible for this systematic review.13-25

Discussion

Summary of Evidence

This systematic review elucidates on which treatment has greater stability: orthognathic surgery or osteogenesis distraction. Few articles demonstrate which technique would be the most appropriate to treat a patient with cleft palate. The literature is conflicting regarding intraoperative skeletal movement and higher relapse rates. Our analysis suggests that for every 1 mm of maxillary advancement achieved with surgery, an average of 0.23 mm of recurrence is expected (p 1⁄4 0.007), and for every 1 mm of maxillary vertical descent, a mean recurrence of 0.13 mm is expected (p 1⁄4 0.039). Therefore, DO has been proposed when a greater range of movement is required since it is described as a more stable method.13,14,15

The study by Kloukos et al.16 included 47 participants with a minimum age of 13 years with a mature skeletal growth that required a maxillary advancement of 4 to 10 mm. They used internal mechanism osteogenic distraction with a buccal cut. The maxilla was fully mobile but was not brought into the final occlusal position. The internal devices were placed and activated a few millimeters apart to gradually position the maxilla. After a three-day wait, activation of 1 mm every day begun until an incisal Class I was achieved. As a control group study, it had patients who had undergone a Le Fort I osteotomy. Surgery was performed with fragmentation of the maxilla. Here, the maxilla was placed in the final occlusal position. One of the conclusions of this article was that, from the patients’ point of view, it was evident that aesthetics would improve regardless of the technique. Patients must be prepared for any recurrence to occur.

Regarding stability, osteogenic distraction might be preferable.16,17 Quality-of-life results during treatment suggest that osteogenic surgery might be preferable.16 Ganoo et al.17 found no significant difference between the functional results offered by the conventional orthognathic surgery and those offered by distraction osteogenesis. Similarly, Cheung18 found that distraction provided better skeletal stability, while there was a significant amount of skeletal relapse in the first 12 weeks after a conventional maxillary osteotomy. In the skeletal recurrences of the osteotomy group, a statistically significant vertical recurrence of point A was observed during the second to the twelfth week as compared with that in the distraction group. Statistically significant horizontal recurrence of point A was observed during the eighth to twelfth weeks when the osteotomy group was compared with the distraction group.

Anderson et al.19 observed that DO results in a stable position of the maxilla. Conventional orthognathic treatment resulted in recurrence in the sagittal direction and continued downward movement of the maxilla postoperatively in a Le Fort I surgery. Le Fort I with osteogenic distraction and osteogenic distraction of the anterior maxilla have been evaluated for the treatment of maxillary hypoplasia in patients with clefts. Relapse rates were 20%, 12%, and 12%, respectively.20 So, the study by Jiang20 has the same results in terms of DO stability as the previous studies. There were significant improvements immediately after distraction, but during the one-year follow-up, some differences were observed.20 This emphasised the need for an overcorrection of around 35% to 40% for adult patients with CLP.21,22 In a study by Aksu et al.,21 in which 7 adult patients with PLC were treated with OD, after three years, a relapse rate of 22% was observed in the maxilla. Cho and Kyung12 found a relapse rate of approximately 23% during the one- to six-year period after distraction, Baek et al.22 found 21%, while Kanno et al.9 found fewer relapses (approximately 8%) during a follow-up period of 2.8 years. In total, 70% of improvements were stable at long-term follow-up.11

Regarding comorbidities, there were no important differences in clinical morbidities between the osteotomy and distraction groups.18

Quality of life is one of the most important aspects when choosing between the two techniques. DO caused increased anxiety and distress as compared with orthognathic surgery. This is caused by the device itself and the pressure on patients and their families to correctly handle the device, as they were responsible for part of the treatment. However, in long-term follow-up, patients who had received DO were more satisfied. Therefore, orthognathic surgery offered a better quality of life during treatment for patients; however, DO offered a better quality of life after treatment.17 So, patients with a cleft lip who had undergone DO looked worse during treatment, but after treatment (in the long term), they had higher satisfaction.16,17

Other authors also concluded that DO has no advantage over orthognathic surgery in preventing velopharyngeal incompetence and speech impairment in moderate cleft maxillary advancement.16,23,24 However, Ramanathan25 finds that DO offers an advantage, in that velopharyngeal incompetence can be monitored during the activation phase. Distraction of the anterior part of the maxilla is another alternative to alleviate the disadvantage of worsening speech.

There is a lack of agreement among researchers regarding the best time to treat these patients. If early distraction treatment is performed on cleft lip patients before their facial skeletal growth is complete, orthognathic surgery or further distraction may be needed later. Therefore, there are only two options available to treat maxillary hypoplasia in patients aged 11 to 13 years: total or anterior maxillary distraction. Orthognathic surgery can be performed only after complete growth.25

Limitations

Choosing articles that concord with the eligibility criteria was difficult. This systematic review did not make a distinction between the use of intern or extern distraction devices. Finally, the follow-up duration was not defined precisely.

Conclusion

According to the results of this systematic review, in terms of stability, distraction is the treatment of choice in patients with cleft palate without growth. Osteogenesis distraction has better stability than orthognathic surgery. In addition, orthognathic surgery has a much more significant recurrence at the vertical level than at the horizontal level. The best treatment option for children with cleft lip is DO and, more precisely, distraction of the anterior maxilla, as it does not compromise the muscles of the velopharynx. However, it may not prevent future surgery or further distraction.

During treatment, orthognathic surgery is preferred by patients, but in the long term, patients are more satisfied with DO. Regarding speech, many researchers agree that neither orthognathic surgery nor distraction seems to bring advantages for velopharyngeal incompetence. However, the distraction would have the advantage of controlling and monitoring the incompetence during the activation of the device.

Disclosure of Interest

The authors declare that they have no competing interest.

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