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Journal of Plastic, Reconstructive & Aesthetic Surgery (2015) xx, 1e6
A novel active intraoral appliance for presurgical orthopaedic treatment in patients with complete bilateral cleft lip and palate Koichiro Kiya a,*, Tomoki Oyama a, Yumiko Sone b, Nobuyuki Ishii c, Ko Hosokawa d a
Department of Plastic Surgery, Kobe Children’s Hospital, 1-1-1 Takakuradai Suma-Ku, Kobe, Japan Department of Dentistry, Kobe Children’s Hospital, 1-1-1 Takakuradai Suma-Ku, Kobe, Japan c Kanomi Dental Clinic, 30-1 Minamiekimaecho, Himeji, Hyogo, Japan d Department of Plastic Surgery, Osaka University Graduate School of Medicine, 2-2-C11 Yamadaoka, Suita, Osaka, Japan b
Received 24 April 2014; accepted 13 December 2014
KEYWORDS Complete bilateral cleft lip and palate; Presurgical orthopaedic treatment; Active appliance; Intraoral appliance; Protruding premaxilla
Summary Background: Management of the protruding/deviated premaxilla in patients with complete bilateral cleft lip and palate is a challenging problem for surgeons and orthodontists. Various passive and active methods have been developed for the presurgical orthopaedic treatment. However, most of these treatments are complicated and laborious for the patient’s parents and clinicians. Here, we describe our original active intraoral appliance comprising two components, that is, the premaxillary and palatine process plates, connected with two elastic chains, and we assess its therapeutic efficacy. Patients and methods: We retrospectively evaluated 15 patients treated using this appliance during 2006e2012, followed up for an average of 60.3 months (range, 18e97 months). We analysed the cleft widths and maxillary size, obtained pretreatment, post-treatment and pre-palatoplasty. Results: Cleft widths and premaxillary protrusion were significantly decreased post treatment; however, the transverse dimensions were not significantly altered. In all cases, the protruding/ deviated premaxilla was set into a suitable position within 1 month, and we could perform onestage cheiloplasty using the modified Mulliken method with low tension. Conclusion: Our appliance is technically simple to use, less invasive to the skin and bone and cost-effective, with reliable and predictable outcomes. In the follow-up period, we observed
* Corresponding author. 1-1-1 Takakuradai Suma-Ku, Kobe, Japan. Tel.: þ81 787326961; fax: þ81 787350910. E-mail address: [email protected] (K. Kiya). http://dx.doi.org/10.1016/j.bjps.2014.12.022 1748-6815/ª 2014 British Association of Plastic, Reconstructive and Aesthetic Surgeons. Published by Elsevier Ltd. All rights reserved.
Please cite this article in press as: Kiya K, et al., A novel active intraoral appliance for presurgical orthopaedic treatment in patients with complete bilateral cleft lip and palate, Journal of Plastic, Reconstructive & Aesthetic Surgery (2015), http://dx.doi.org/10.1016/ j.bjps.2014.12.022
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K. Kiya et al. no detrimental growth of the maxilla or dentition. Therefore, we consider our appliance to be useful for application in presurgical orthopaedic treatments of complete bilateral cleft lip and palate. ª 2014 British Association of Plastic, Reconstructive and Aesthetic Surgeons. Published by Elsevier Ltd. All rights reserved.
In cheiloplasty for complete bilateral cleft lip and palate with protruding/deviated premaxilla, it is difficult to approximate the skin, mucosa and muscle symmetrically with low tension, resulting in functional and cosmetic problems postoperatively.1e3 Therefore, various methods, including active/passive and intraoral/extraoral appliances, have been described for the presurgical orthopaedic treatment of complete bilateral cleft lip and palate. Most extraoral appliances e that consist of elastic traction to the premaxilla through a bonnet or facial taping e are easy to set up but they are typically troublesome for the patient’s parents and clinicians. By contrast, passive intraoral appliances are meant to guide skeletal growth in the desired direction or even stimulate growth.4 However, in certain cases, passive appliances cannot provide the ideal result, particularly in patients with a severely protruding/deviated premaxilla. In such patients, active intraoral appliances can direct premaxillary growth in the downward and backward direction by the application of force to the premaxilla, with highly predictable outcomes.4 We have developed an original active intraoral appliance e comprising two components, that is, premaxillary and palatine process plates, connected with two elastic chains e for presurgical orthopaedic treatment in patients with complete bilateral cleft lip and palate with a severely protruding/deviated premaxilla. The purpose of this study is to introduce our original appliance, which was successfully used to shift the protruding/deviated premaxilla into a suitable position, with low invasiveness to the skin of the prolabium and the bone of the premaxilla. We also discuss its therapeutic efficacy based on maxillary impressions.
Material and methods Treatment procedure In our hospital, a passive intraoral plate was constructed and inserted for patients with complete bilateral cleft lip and palate as early as possible. Subsequently, a maxillary impression was obtained from each patient at the age of 2 months without general anaesthesia to fabricate our active appliance, which comprised two acrylic resin components e a premaxillary plate (PM plate) fitting the anterior surface of the premaxillary form with the wire twisted twice at the each lateral end and a palatine process plate (PP plate) with two hooks attached to the midline (Figure 1). The appliance was fitted at the age of approximately 3 months under general anaesthesia. The PP plate was fixed onto both palatine processes with four titanium screws, taking care not to damage the tooth buds or vascular bundles. The PM plate was fixed onto the anterior surface of the premaxilla using a 25-gauge wire with a needle. The wire penetrated the premaxillary bone through the twisted wire hole and it was ligated after encompassing the premaxilla. The hooks of the PP plate and the twisted wire of the PM plate were connected with two elastic chains in the appropriate strength. Most procedures were completed within 20 min. All patients could be fed orally a few hours postoperatively and they were discharged on postoperative day 1. Subsequently, the premaxilla was set centrally and backwards by the elastic force. After a 1-month consolidation period, at the age of 4 months, the appliance was removed during cheiloplasty according to the modified Mulliken method.
Figure 1 Our original active intraoral appliance (left) and its intraoral view (right). This comprises two acrylic resin components e a premaxillary plate (PM plate) and a palatine process plate (PP plate), connected with two elastic chains. Please cite this article in press as: Kiya K, et al., A novel active intraoral appliance for presurgical orthopaedic treatment in patients with complete bilateral cleft lip and palate, Journal of Plastic, Reconstructive & Aesthetic Surgery (2015), http://dx.doi.org/10.1016/ j.bjps.2014.12.022
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A novel active intraoral appliance for presurgical orthopaedic treatment
Patients and measurements We retrospectively evaluated 15 consecutively treated patients (eight boys and seven girls) born between 2006 and 2012, followed up for an average of 60.3 months (range, 18e97 months). Intraoral maxillary impressions were obtained at pretreatment, post-treatment during cheiloplasty, and pre-palatoplasty (a few weeks before palatoplasty). Measurements of maxillary dimensions in these impressions were performed using a digital calliper from the reference points shown in Figure 2. The means and standard deviations of all dimensions were calculated at the three different stages (pretreatment, post-treatment and pre-palatoplasty). The changes during the periods were analysed by two-tailed paired ttest, and p < 0.05 was considered statistically significant.
Results Table 1 presents the detailed results. The mean age for inserting this appliance was 101.4 33.6 days. After the orthopaedic treatment, the right and left cleft widths (PeL and P0 eL0 ) and the premaxillary protrusion (IeTT0 ) were both significantly decreased. By contrast, the differences in the transverse dimensions (CeC0 and TeT0 ) were not significantly changed. From post-treatment to pre-palatoplasty, both the right and left cleft widths were additionally decreased, whereas the premaxillary protrusion was not significantly changed. Concerning the transverse dimensions, no significant differences were found during this period. The appliances were inserted and removed safely in all the patients, without complications. Neither detachment of the appliance nor loss of screws was observed during treatment. In all cases, the protruding/deviated premaxilla could be set into a suitable position within 1 month;
Figure 2 The reference points of the maxillary impressions. I, the incisal point; P, P0, the right and left cleft edges of the premaxilla; L, L0 , the right and left cleft edges of the lateral segments; C, C0 , the cuspid points; T, T0, the tuberosity points; IeTT0, the length of the perpendicular line from I to TT0.
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further, the premaxillary vestibule was deepened by the PM plate’s pressure. Subsequently, we could perform onestage cheiloplasty using the modified Mulliken method with low tension.
Clinical cases Case 1 A female infant with a complete bilateral cleft lip and palate with a markedly protruding and right-deviating premaxilla was treated using our appliance. Appliance placement was performed at the age of 102 days, reducing both cleft widths (PeL: from 11.92 to 4.58 mm, P0 eL0 : from 9.13 to 2.72 mm) and the premaxillary protrusion (IeTT0: from 33.36 to 25.64 mm) at 1 month postoperatively. The premaxillary vestibule was observed to have deepened sufficiently. She underwent cheiloplasty according to the modified Mulliken method with low tension at the age of 137 days. We considered that the appliance contributed to the satisfactory results obtained at 69 months after cheiloplasty (Figures 3 and 4). Case 2 A male infant with a complete bilateral cleft lip and palate with a severely protruding and left-deviating premaxilla was treated using our appliance. Appliance placement was performed at the age of 83 days, with successful reduction in the cleft widths (PeL: from 9.81 to 4.52 mm, P0 eL0 : from 10.54 to 5.77 mm) and the premaxillary protrusion (IeTT0: from 33.31 to 26.62 mm) at 1 month postoperatively. The premaxillary vestibule was noted to be deepened sufficiently. At the age of 111 days, the patient underwent cheiloplasty according to the modified Mulliken method with low tension. We considered that our appliance contributed to the satisfactory results obtained at 52 months after cheiloplasty (Figures 5 and 6).
Discussion Although several studies have examined the long-term effects of presurgical orthopaedic treatment in patients with unilateral cleft lip and palate,5,6 there are few systematic reviews or randomised clinical trials concerning patients with complete bilateral cleft lip and palate. Therefore, we consider that this remains a controversial subject for surgeons and orthodontists, especially in the case of patients with complete bilateral cleft lip and palate with a severely protruding/deviated premaxilla and wide cleft. Various passive and active methods have been developed for the presurgical orthopaedic treatment of complete bilateral cleft lip and palate.1e4,7e13 Extraoral appliances, comprising elastic labial taping applied on the infant’s face or attached with a band covering the infant’s head,7,8 are easy to set up, but they are typically troublesome for the patient’s parents and clinicians because of the skin injuries caused by the tape and the stuffy environment caused by the headband. Furthermore, the results are not highly predictable. Active intraoral appliances are expensive and require additional general anaesthesia for their insertion. Nevertheless, these are relatively easy to manage and have high
Please cite this article in press as: Kiya K, et al., A novel active intraoral appliance for presurgical orthopaedic treatment in patients with complete bilateral cleft lip and palate, Journal of Plastic, Reconstructive & Aesthetic Surgery (2015), http://dx.doi.org/10.1016/ j.bjps.2014.12.022
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K. Kiya et al. Table 1
Results. Pretreatment
Age (days)
76.6 34.3
Measurements (mm) PeL 9.19 P0 eL0 9.94 CeC0 31.60 34.41 TeT0 31.43 IeTT0
3.67 2.24 2.73 2.20 3.20
Post-treatment
Pre-palatoplasty
132.5 38.6
417.2 82.8
3.96 4.31 31.86 34.90 26.03
2.44 2.50 2.73 2.51 3.36
1.96 1.54 30.96 34.88 26.83
2.32 1.94 3.84 3.47 2.56
outcome predictability. Several intraoral appliances using elastic chains employ different approaches for the retraction of the premaxillary segment, such as using a transpremaxillary pin,1,2,9,10 a latex rubber strip,3 or an elastic chain covered with soft denture liner.11 A unique characteristic of our appliance is the pressure exerted by the PM plate on the anterior surface of the premaxilla rather than on the prolabial skin. This makes our technique less invasive
p-value between pretreatment and post-treatment
p-value between post-treatment and pre-palatoplasty
p < 0.001 p < 0.001 0.61 0.31 p < 0.001
0.004 p < 0.001 0.2 0.98 0.33
to the prolabial skin and premaxillary bone. Moreover, the premaxillary vermilion mucosa was expanded by the PM plate and its traction force. Accordingly, during cheiloplasty, the premaxillary mucosa could be utilised not only for the vestibule but also for reinforcing the nasal floor. In our study, the maxillary transverse widths at pretreatment were greater than those in healthy Japanese infants but without severe collapse of the lateral
Figure 3 Case 1. A female infant with a complete bilateral cleft lip and palate with a markedly protruding and right-deviating premaxilla was treated using our appliance. (Upper left) Pretreatment view. (Upper right) Intraoral view just after setting. (Middle left) Post-treatment view. (Middle right) Post-treatment intraoral view. (Lower left) Postoperative view just after cheiloplasty according to the modified Mulliken method. (Lower right) Postoperative view at 69 months after cheiloplasty.
Please cite this article in press as: Kiya K, et al., A novel active intraoral appliance for presurgical orthopaedic treatment in patients with complete bilateral cleft lip and palate, Journal of Plastic, Reconstructive & Aesthetic Surgery (2015), http://dx.doi.org/10.1016/ j.bjps.2014.12.022
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A novel active intraoral appliance for presurgical orthopaedic treatment
Figure 4 The maxillary impression from case 1, obtained at pretreatment (left) and post-treatment (right). Both cleft widths (PeL: from 11.92 to 4.58 mm, P0 eL0 : from 9.13 to 2.72 mm) and the premaxillary protrusion (IeTT0: from 33.36 to 25.64 mm) were reduced at 1 month postoperatively. The photograph was taken with the two models next to one another to show the actual changes in size and shape.
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Figure 6 The maxillary impression from case 2, obtained at pretreatment (left) and post-treatment (right). Both cleft widths (PeL: from 9.81 to 4.52 mm, P0 eL0 : from 10.54 to 5.77 mm) and the premaxillary protrusion (IeTT0: from 33.31 to 26.62 mm) were reduced at 1 month postoperatively. The photograph was taken with the two models next to one another to show the actual changes in size and shape.
Figure 5 Case 2. A male infant with a complete bilateral cleft lip and palate with a severely protruding and left-deviating premaxilla was treated using our appliance. (Upper left) Pretreatment view. (Upper right) Intraoral view just before setting. (Middle left) Post-treatment view. (Middle right) Post-treatment intraoral view. (Lower left) Postoperative view just after cheiloplasty according to the modified Mulliken method. (Lower right) Postoperative view at 52 months after cheiloplasty.
Please cite this article in press as: Kiya K, et al., A novel active intraoral appliance for presurgical orthopaedic treatment in patients with complete bilateral cleft lip and palate, Journal of Plastic, Reconstructive & Aesthetic Surgery (2015), http://dx.doi.org/10.1016/ j.bjps.2014.12.022
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6 segment.14 Therefore, we considered it unnecessary to expand the palatal shelf transversely. Consequently, our appliance is simple, lightweight and cost-effective as compared with Latham’s appliance or other available modalities. None of our patients experienced the detachment of the appliance or loss of screws during treatment. We consider that these complications are less likely to occur using this appliance because of the dual directions of traction by the elastic chains and the perpendicular direction of screw insertion. However, as our sample size is relatively small, it is necessary to examine the morbidity and complications associated with this device in a larger number of patients in the future. In the follow-up period (maximum, 97 months), we observed no detrimental growth of the maxilla or dentition. However, further follow-up and investigations are needed to determine the long-term outcomes of treatment using this novel appliance, including its effects on maxillary growth and tooth formation.
Conclusion Our original appliance is technically easy and simple to fabricate and use, less invasive to the prolabial skin and premaxillary bone as compared to other available modalities and cost-effective, with reliable and predictable outcomes. In our case series, it provided ideal reduction of the cleft widths and premaxillary protrusion within 1 month after the treatment. In the follow-up period, we observed no detrimental growth of the maxilla or dentition. Therefore, the application of our appliance may be considered a useful presurgical orthodontic treatment method for patients with complete bilateral cleft lip and palate with protruding/deviated premaxilla.
Funding None declared.
Conflicts of interest None declared.
Ethical approval
K. Kiya et al.
References 1. Bitter K. Latham’s appliance for presurgical repositioning of the protruded premaxilla in bilateral cleft lip and palate. J Craniomaxillofac Surg 1992;20:99e110. 2. Berkowitz S, Mejia M, Bystrik A. A comparison of the effects of the Latham-Millard procedure with those of a conservative treatment approach for dental occlusion and facial aesthetics in unilateral and bilateral complete cleft lip and palate: part I. Dental occlusion. Plast Reconstr Surg 2004;113:1e18. 3. Reisberg DJ, Figueroa AA, Gold HO. An intraoral appliance for management of the protrusive premaxilla in bilateral cleft lip. Cleft Palate J 1988;25:53e7. 4. Oosterkamp BC, van Oort RP, Dijkstra PU, Stellingsma K, Bierman MW, de Bont LG. Effect of an intraoral retrusion plate on maxillary arch dimensions in complete bilateral cleft lip and palate patients. Cleft Palate Craniofac J 2005;42:239e44. 5. Uzel A, Alparslan ZN. Long-term effects of presurgical infant orthopedics in patients with cleft lip and palate: a systematic review. Cleft Palate Craniofac J 2011;48:587e95. 6. Konst EM, Rietveld T, Peters HF, Prahl-Andersen B. Phonological development of toddlers with unilateral cleft lip and palate who were treated with and without infant orthopedics: a randomized clinical trial. Cleft Palate Craniofac J 2003;40: 32e9. 7. Robertson N, Shaw W, Volp C. The changes produced by presurgical orthopedic treatment of bilateral cleft lip and palate. Plast Reconstr Surg 1977;59:87e93. 8. Mishima K, Sugahara T, Mori Y, Minami K, Sakuda M. Effects of presurgical orthopedic treatment in infants with complete bilateral cleft lip and palate. Cleft Palate Craniofac J 1998;35: 227e32. 9. Georgiade NG, Mason R, Riefkohl R, Georgiade G, Barwick W. Preoperative positioning of the protruding premaxilla in the bilateral cleft lip patient. Plast Reconstr Surg 1989;83:32e40. 10. Millard DR, Latham R, Huifen X, Spiro S, Morovic C. Cleft lip and palate treated by presurgical orthopedics, gingivoperiosteoplasty, and lip adhesion (POPLA) compared with previous lip adhesion method: a preliminary study of serial dental casts. Plast Reconstr Surg 1999;103:1630e44. 11. Figueroa AA, Reisberg DJ, Polley JW, Cohen M. Intraoralappliance modification to retract the premaxilla in patients with bilateral cleft lip. Cleft Palate Craniofac J 1996;33: 497e500. 12. Papay FA, Morales Jr L, Motoki DS, Yamashiro DK. Presurgical orthopedic premaxillary alignment in cleft lip and palate reconstruction. Cleft Palate Craniofac J 1994;31:494e7. 13. Choo H, Maguire M, Low DW. Modified technique of presurgical infant maxillary orthopedics for complete unilateral cleft lip and palate. Plast Reconstr Surg 2012;129:244e8. 14. Kojo H. Growth and development of the dentition during the first one year of life: three dimension measurement on maxillary and mandibular alveolar arch morphological palate. Shoni Shikagaku Zasshi 1988;26:112e30.
Not required.
Please cite this article in press as: Kiya K, et al., A novel active intraoral appliance for presurgical orthopaedic treatment in patients with complete bilateral cleft lip and palate, Journal of Plastic, Reconstructive & Aesthetic Surgery (2015), http://dx.doi.org/10.1016/ j.bjps.2014.12.022
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Journal of Plastic, Reconstructive & Aesthetic Surgery (2015) xx, 1e6
A novel active intraoral appliance for presurgical orthopaedic treatment in patients with complete bilateral cleft lip and palate Koichiro Kiya a,*, Tomoki Oyama a, Yumiko Sone b, Nobuyuki Ishii c, Ko Hosokawa d a
Department of Plastic Surgery, Kobe Children’s Hospital, 1-1-1 Takakuradai Suma-Ku, Kobe, Japan Department of Dentistry, Kobe Children’s Hospital, 1-1-1 Takakuradai Suma-Ku, Kobe, Japan c Kanomi Dental Clinic, 30-1 Minamiekimaecho, Himeji, Hyogo, Japan d Department of Plastic Surgery, Osaka University Graduate School of Medicine, 2-2-C11 Yamadaoka, Suita, Osaka, Japan b
Received 24 April 2014; accepted 13 December 2014
KEYWORDS Complete bilateral cleft lip and palate; Presurgical orthopaedic treatment; Active appliance; Intraoral appliance; Protruding premaxilla
Summary Background: Management of the protruding/deviated premaxilla in patients with complete bilateral cleft lip and palate is a challenging problem for surgeons and orthodontists. Various passive and active methods have been developed for the presurgical orthopaedic treatment. However, most of these treatments are complicated and laborious for the patient’s parents and clinicians. Here, we describe our original active intraoral appliance comprising two components, that is, the premaxillary and palatine process plates, connected with two elastic chains, and we assess its therapeutic efficacy. Patients and methods: We retrospectively evaluated 15 patients treated using this appliance during 2006e2012, followed up for an average of 60.3 months (range, 18e97 months). We analysed the cleft widths and maxillary size, obtained pretreatment, post-treatment and pre-palatoplasty. Results: Cleft widths and premaxillary protrusion were significantly decreased post treatment; however, the transverse dimensions were not significantly altered. In all cases, the protruding/ deviated premaxilla was set into a suitable position within 1 month, and we could perform onestage cheiloplasty using the modified Mulliken method with low tension. Conclusion: Our appliance is technically simple to use, less invasive to the skin and bone and cost-effective, with reliable and predictable outcomes. In the follow-up period, we observed
* Corresponding author. 1-1-1 Takakuradai Suma-Ku, Kobe, Japan. Tel.: þ81 787326961; fax: þ81 787350910. E-mail address: [email protected] (K. Kiya). http://dx.doi.org/10.1016/j.bjps.2014.12.022 1748-6815/ª 2014 British Association of Plastic, Reconstructive and Aesthetic Surgeons. Published by Elsevier Ltd. All rights reserved.
Please cite this article in press as: Kiya K, et al., A novel active intraoral appliance for presurgical orthopaedic treatment in patients with complete bilateral cleft lip and palate, Journal of Plastic, Reconstructive & Aesthetic Surgery (2015), http://dx.doi.org/10.1016/ j.bjps.2014.12.022
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K. Kiya et al. no detrimental growth of the maxilla or dentition. Therefore, we consider our appliance to be useful for application in presurgical orthopaedic treatments of complete bilateral cleft lip and palate. ª 2014 British Association of Plastic, Reconstructive and Aesthetic Surgeons. Published by Elsevier Ltd. All rights reserved.
In cheiloplasty for complete bilateral cleft lip and palate with protruding/deviated premaxilla, it is difficult to approximate the skin, mucosa and muscle symmetrically with low tension, resulting in functional and cosmetic problems postoperatively.1e3 Therefore, various methods, including active/passive and intraoral/extraoral appliances, have been described for the presurgical orthopaedic treatment of complete bilateral cleft lip and palate. Most extraoral appliances e that consist of elastic traction to the premaxilla through a bonnet or facial taping e are easy to set up but they are typically troublesome for the patient’s parents and clinicians. By contrast, passive intraoral appliances are meant to guide skeletal growth in the desired direction or even stimulate growth.4 However, in certain cases, passive appliances cannot provide the ideal result, particularly in patients with a severely protruding/deviated premaxilla. In such patients, active intraoral appliances can direct premaxillary growth in the downward and backward direction by the application of force to the premaxilla, with highly predictable outcomes.4 We have developed an original active intraoral appliance e comprising two components, that is, premaxillary and palatine process plates, connected with two elastic chains e for presurgical orthopaedic treatment in patients with complete bilateral cleft lip and palate with a severely protruding/deviated premaxilla. The purpose of this study is to introduce our original appliance, which was successfully used to shift the protruding/deviated premaxilla into a suitable position, with low invasiveness to the skin of the prolabium and the bone of the premaxilla. We also discuss its therapeutic efficacy based on maxillary impressions.
Material and methods Treatment procedure In our hospital, a passive intraoral plate was constructed and inserted for patients with complete bilateral cleft lip and palate as early as possible. Subsequently, a maxillary impression was obtained from each patient at the age of 2 months without general anaesthesia to fabricate our active appliance, which comprised two acrylic resin components e a premaxillary plate (PM plate) fitting the anterior surface of the premaxillary form with the wire twisted twice at the each lateral end and a palatine process plate (PP plate) with two hooks attached to the midline (Figure 1). The appliance was fitted at the age of approximately 3 months under general anaesthesia. The PP plate was fixed onto both palatine processes with four titanium screws, taking care not to damage the tooth buds or vascular bundles. The PM plate was fixed onto the anterior surface of the premaxilla using a 25-gauge wire with a needle. The wire penetrated the premaxillary bone through the twisted wire hole and it was ligated after encompassing the premaxilla. The hooks of the PP plate and the twisted wire of the PM plate were connected with two elastic chains in the appropriate strength. Most procedures were completed within 20 min. All patients could be fed orally a few hours postoperatively and they were discharged on postoperative day 1. Subsequently, the premaxilla was set centrally and backwards by the elastic force. After a 1-month consolidation period, at the age of 4 months, the appliance was removed during cheiloplasty according to the modified Mulliken method.
Figure 1 Our original active intraoral appliance (left) and its intraoral view (right). This comprises two acrylic resin components e a premaxillary plate (PM plate) and a palatine process plate (PP plate), connected with two elastic chains. Please cite this article in press as: Kiya K, et al., A novel active intraoral appliance for presurgical orthopaedic treatment in patients with complete bilateral cleft lip and palate, Journal of Plastic, Reconstructive & Aesthetic Surgery (2015), http://dx.doi.org/10.1016/ j.bjps.2014.12.022
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A novel active intraoral appliance for presurgical orthopaedic treatment
Patients and measurements We retrospectively evaluated 15 consecutively treated patients (eight boys and seven girls) born between 2006 and 2012, followed up for an average of 60.3 months (range, 18e97 months). Intraoral maxillary impressions were obtained at pretreatment, post-treatment during cheiloplasty, and pre-palatoplasty (a few weeks before palatoplasty). Measurements of maxillary dimensions in these impressions were performed using a digital calliper from the reference points shown in Figure 2. The means and standard deviations of all dimensions were calculated at the three different stages (pretreatment, post-treatment and pre-palatoplasty). The changes during the periods were analysed by two-tailed paired ttest, and p < 0.05 was considered statistically significant.
Results Table 1 presents the detailed results. The mean age for inserting this appliance was 101.4 33.6 days. After the orthopaedic treatment, the right and left cleft widths (PeL and P0 eL0 ) and the premaxillary protrusion (IeTT0 ) were both significantly decreased. By contrast, the differences in the transverse dimensions (CeC0 and TeT0 ) were not significantly changed. From post-treatment to pre-palatoplasty, both the right and left cleft widths were additionally decreased, whereas the premaxillary protrusion was not significantly changed. Concerning the transverse dimensions, no significant differences were found during this period. The appliances were inserted and removed safely in all the patients, without complications. Neither detachment of the appliance nor loss of screws was observed during treatment. In all cases, the protruding/deviated premaxilla could be set into a suitable position within 1 month;
Figure 2 The reference points of the maxillary impressions. I, the incisal point; P, P0, the right and left cleft edges of the premaxilla; L, L0 , the right and left cleft edges of the lateral segments; C, C0 , the cuspid points; T, T0, the tuberosity points; IeTT0, the length of the perpendicular line from I to TT0.
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further, the premaxillary vestibule was deepened by the PM plate’s pressure. Subsequently, we could perform onestage cheiloplasty using the modified Mulliken method with low tension.
Clinical cases Case 1 A female infant with a complete bilateral cleft lip and palate with a markedly protruding and right-deviating premaxilla was treated using our appliance. Appliance placement was performed at the age of 102 days, reducing both cleft widths (PeL: from 11.92 to 4.58 mm, P0 eL0 : from 9.13 to 2.72 mm) and the premaxillary protrusion (IeTT0: from 33.36 to 25.64 mm) at 1 month postoperatively. The premaxillary vestibule was observed to have deepened sufficiently. She underwent cheiloplasty according to the modified Mulliken method with low tension at the age of 137 days. We considered that the appliance contributed to the satisfactory results obtained at 69 months after cheiloplasty (Figures 3 and 4). Case 2 A male infant with a complete bilateral cleft lip and palate with a severely protruding and left-deviating premaxilla was treated using our appliance. Appliance placement was performed at the age of 83 days, with successful reduction in the cleft widths (PeL: from 9.81 to 4.52 mm, P0 eL0 : from 10.54 to 5.77 mm) and the premaxillary protrusion (IeTT0: from 33.31 to 26.62 mm) at 1 month postoperatively. The premaxillary vestibule was noted to be deepened sufficiently. At the age of 111 days, the patient underwent cheiloplasty according to the modified Mulliken method with low tension. We considered that our appliance contributed to the satisfactory results obtained at 52 months after cheiloplasty (Figures 5 and 6).
Discussion Although several studies have examined the long-term effects of presurgical orthopaedic treatment in patients with unilateral cleft lip and palate,5,6 there are few systematic reviews or randomised clinical trials concerning patients with complete bilateral cleft lip and palate. Therefore, we consider that this remains a controversial subject for surgeons and orthodontists, especially in the case of patients with complete bilateral cleft lip and palate with a severely protruding/deviated premaxilla and wide cleft. Various passive and active methods have been developed for the presurgical orthopaedic treatment of complete bilateral cleft lip and palate.1e4,7e13 Extraoral appliances, comprising elastic labial taping applied on the infant’s face or attached with a band covering the infant’s head,7,8 are easy to set up, but they are typically troublesome for the patient’s parents and clinicians because of the skin injuries caused by the tape and the stuffy environment caused by the headband. Furthermore, the results are not highly predictable. Active intraoral appliances are expensive and require additional general anaesthesia for their insertion. Nevertheless, these are relatively easy to manage and have high
Please cite this article in press as: Kiya K, et al., A novel active intraoral appliance for presurgical orthopaedic treatment in patients with complete bilateral cleft lip and palate, Journal of Plastic, Reconstructive & Aesthetic Surgery (2015), http://dx.doi.org/10.1016/ j.bjps.2014.12.022
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K. Kiya et al. Table 1
Results. Pretreatment
Age (days)
76.6 34.3
Measurements (mm) PeL 9.19 P0 eL0 9.94 CeC0 31.60 34.41 TeT0 31.43 IeTT0
3.67 2.24 2.73 2.20 3.20
Post-treatment
Pre-palatoplasty
132.5 38.6
417.2 82.8
3.96 4.31 31.86 34.90 26.03
2.44 2.50 2.73 2.51 3.36
1.96 1.54 30.96 34.88 26.83
2.32 1.94 3.84 3.47 2.56
outcome predictability. Several intraoral appliances using elastic chains employ different approaches for the retraction of the premaxillary segment, such as using a transpremaxillary pin,1,2,9,10 a latex rubber strip,3 or an elastic chain covered with soft denture liner.11 A unique characteristic of our appliance is the pressure exerted by the PM plate on the anterior surface of the premaxilla rather than on the prolabial skin. This makes our technique less invasive
p-value between pretreatment and post-treatment
p-value between post-treatment and pre-palatoplasty
p < 0.001 p < 0.001 0.61 0.31 p < 0.001
0.004 p < 0.001 0.2 0.98 0.33
to the prolabial skin and premaxillary bone. Moreover, the premaxillary vermilion mucosa was expanded by the PM plate and its traction force. Accordingly, during cheiloplasty, the premaxillary mucosa could be utilised not only for the vestibule but also for reinforcing the nasal floor. In our study, the maxillary transverse widths at pretreatment were greater than those in healthy Japanese infants but without severe collapse of the lateral
Figure 3 Case 1. A female infant with a complete bilateral cleft lip and palate with a markedly protruding and right-deviating premaxilla was treated using our appliance. (Upper left) Pretreatment view. (Upper right) Intraoral view just after setting. (Middle left) Post-treatment view. (Middle right) Post-treatment intraoral view. (Lower left) Postoperative view just after cheiloplasty according to the modified Mulliken method. (Lower right) Postoperative view at 69 months after cheiloplasty.
Please cite this article in press as: Kiya K, et al., A novel active intraoral appliance for presurgical orthopaedic treatment in patients with complete bilateral cleft lip and palate, Journal of Plastic, Reconstructive & Aesthetic Surgery (2015), http://dx.doi.org/10.1016/ j.bjps.2014.12.022
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MODEL
A novel active intraoral appliance for presurgical orthopaedic treatment
Figure 4 The maxillary impression from case 1, obtained at pretreatment (left) and post-treatment (right). Both cleft widths (PeL: from 11.92 to 4.58 mm, P0 eL0 : from 9.13 to 2.72 mm) and the premaxillary protrusion (IeTT0: from 33.36 to 25.64 mm) were reduced at 1 month postoperatively. The photograph was taken with the two models next to one another to show the actual changes in size and shape.
5
Figure 6 The maxillary impression from case 2, obtained at pretreatment (left) and post-treatment (right). Both cleft widths (PeL: from 9.81 to 4.52 mm, P0 eL0 : from 10.54 to 5.77 mm) and the premaxillary protrusion (IeTT0: from 33.31 to 26.62 mm) were reduced at 1 month postoperatively. The photograph was taken with the two models next to one another to show the actual changes in size and shape.
Figure 5 Case 2. A male infant with a complete bilateral cleft lip and palate with a severely protruding and left-deviating premaxilla was treated using our appliance. (Upper left) Pretreatment view. (Upper right) Intraoral view just before setting. (Middle left) Post-treatment view. (Middle right) Post-treatment intraoral view. (Lower left) Postoperative view just after cheiloplasty according to the modified Mulliken method. (Lower right) Postoperative view at 52 months after cheiloplasty.
Please cite this article in press as: Kiya K, et al., A novel active intraoral appliance for presurgical orthopaedic treatment in patients with complete bilateral cleft lip and palate, Journal of Plastic, Reconstructive & Aesthetic Surgery (2015), http://dx.doi.org/10.1016/ j.bjps.2014.12.022
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MODEL
6 segment.14 Therefore, we considered it unnecessary to expand the palatal shelf transversely. Consequently, our appliance is simple, lightweight and cost-effective as compared with Latham’s appliance or other available modalities. None of our patients experienced the detachment of the appliance or loss of screws during treatment. We consider that these complications are less likely to occur using this appliance because of the dual directions of traction by the elastic chains and the perpendicular direction of screw insertion. However, as our sample size is relatively small, it is necessary to examine the morbidity and complications associated with this device in a larger number of patients in the future. In the follow-up period (maximum, 97 months), we observed no detrimental growth of the maxilla or dentition. However, further follow-up and investigations are needed to determine the long-term outcomes of treatment using this novel appliance, including its effects on maxillary growth and tooth formation.
Conclusion Our original appliance is technically easy and simple to fabricate and use, less invasive to the prolabial skin and premaxillary bone as compared to other available modalities and cost-effective, with reliable and predictable outcomes. In our case series, it provided ideal reduction of the cleft widths and premaxillary protrusion within 1 month after the treatment. In the follow-up period, we observed no detrimental growth of the maxilla or dentition. Therefore, the application of our appliance may be considered a useful presurgical orthodontic treatment method for patients with complete bilateral cleft lip and palate with protruding/deviated premaxilla.
Funding None declared.
Conflicts of interest None declared.
Ethical approval
K. Kiya et al.
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Please cite this article in press as: Kiya K, et al., A novel active intraoral appliance for presurgical orthopaedic treatment in patients with complete bilateral cleft lip and palate, Journal of Plastic, Reconstructive & Aesthetic Surgery (2015), http://dx.doi.org/10.1016/ j.bjps.2014.12.022
