CASE REPORT
Interdisciplinary treatment for an adult with a unilateral cleft lip and palate Qiao Ling Ma,a R. Scott Conley,b Tuojiang Wu,c and Huang Lid Nanjing, Jiangsu, People's Republic of China, and Ann Arbor, Mich
A young man, age 18 years 4 months, with a concave profile, a skeletal maxillary deficiency, and a severe alveolar cleft with an unesthetic appearance of the maxillary anterior teeth was referred for orthodontic treatment. After a detailed review of his pretreatment records, both surgical and nonsurgical treatment plans were presented to the patient, who opted for a nonsurgical interdisciplinary approach. His complex 3-dimensional malocclusion required palatal expansion, dental extractions, and periodontal and prosthodontic consultations and treatment, in addition to comprehensive orthodontic therapy. MBT (Xinya, HangZhou, China) 0.022 3 0.028in appliances combined with a mini-implant to enhance the orthodontic anchorage were used to level, align, and establish a Class I relationship. After the orthodontic treatment, a combined restorative and periodontal approach was used to enhance the patient's esthetic and functional outcomes. Both the final result and the 1year follow-up records demonstrate that the treatment goals of establishing proper occlusion, normal function, a balanced profile, better esthetics, and a stable outcome were achieved. The purpose of this case report is to demonstrate that an interdisciplinary treatment protocol can significantly improve the transverse discrepancies and achieve a satisfactory occlusion with a balanced profile in patients with cleft lip and palate. (Am J Orthod Dentofacial Orthop 2014;146:238-48)
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left lip and palate (CLP) is the most frequent congenital facial abnormality.1,2 Compared with the incidence rate reported from other countries, the frequency of all forms of clefts is highest among Asians.3 The etiology is related to both hereditary and environmental factors.4,5 Patients with CLP can also have other features such as deficient midface development resulting in a Class III tendency, severe maxillary transverse deficiency, alveolar cleft, and hypodontia.6-8 CLP patients might also have decreased facial and dental esthetics, resulting in low self-
a Attending orthodontist, Department of Orthodontics, Stomatological Hospital, Medical School of Nanjing University, Nanjing, Jiangsu, People's Republic of China. b Associate professor, Department of Orthodontics and Pediatric Dentistry, School of Dentistry, University of Michigan, Ann Arbor, Mich. c Associate professor, College of Stomatology, Nanjing Medical University, Nanjing, Jiangsu, People's Republic of China. d Associate professor, Department of Orthodontics, Stomatological Hospital, School of Medicine, Nanjing University, Nanjing, Jiangsu, People's Republic of China. All authors have completed and submitted the ICMJE Form for Disclosure of Potential Conflicts of Interest, and none were reported. Sponsored by the National Natural Science Foundation of China and Medical Science and Technology Development Foundation, Nanjing Department of Health, grant nos. 81070807 and YKK11038. Address correspondence to: Huang Li, Department of Orthodontics, Nanjing University, The Stomatological Hospital of Medical School, Nan Jing, Jiangsu, People's Republic of China; e-mail, [email protected]. Submitted, May 2013; revised and accepted, October 2013. 0889-5406/$36.00 Copyright Ó 2014 by the American Association of Orthodontists. http://dx.doi.org/10.1016/j.ajodo.2013.10.024
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confidence and difficulties in social interactions.9 Interdisciplinary treatment plans for patients with CLP frequently extend over many years, starting with primary surgeries during infancy, at least 1 stage of orthodontic treatment, and possible jaw surgery near the end of adolescence or early adulthood.10 Treatment frequently includes nasoalveolar molding, labial repair (about 36 months of age), palatoplasty (10-12 months of age), orthodontic expansion, primary or secondary alveolar bone graft, surgery, and comprehensive orthodontic treatment to reestablish facial esthetics and proper function.11-17 One important aspect of orthodontic treatment includes transverse expansion of the maxilla to correct the transverse deficiency of the palatal segments. This can be achieved using one of several different types of expanders, including traditional screw-type rapid palatal expansion, spring-type, magnetic, and shape memory alloy. The magnetic palatal expander (MPE) with repelling magnets to increase the width of maxilla has been previously reported by Darendeliler et al.18 Magnets produce a suitable force without requiring additional adjustment by the patient and are more rigid than spring-type expanders, potentially resulting in fewer undesirable effects; however, there are few reports of MPE treatment in CLP patients.19 This case report describes an interdisciplinary treatment approach for a young man with unilateral CLP. MPE, comprehensive orthodontics, periodontics, and nasoalveolar molding were used to obtain proper
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Fig 1. Pretreatment photographs.
occlusion and normal function; a balanced profile and better esthetics were achieved. DIAGNOSIS AND ETIOLOGY
A young Chinese man (age 18 years 4 months) came to the Orthodontic Department of Nanjing University for orthodontic evaluation of his severe crowding and posterior crossbite. His chief complaint was the unesthetic appearance of his face and maxillary anterior teeth. He was born with nonsyndromic unilateral left CLP. The lip was repaired at the age of 6 months (primary cheiloplasty), and the palate (palatoplasty) was repaired at 4 years of age, with no further orthodontic treatment or alveolar bone-grafting procedures. The patient had a concave profile with a retrusive upper lip and an everted lower lip. He showed good vertical balance among the facial thirds and good symmetry except for the nose and upper lip resulting from both the cleft and the scarring from the previous surgical repair. The maxillary and mandibular dental midlines were coincident but both were deviated to the left by 2 mm (Fig 1).
The maxillary arch was collapsed and constricted resulting from a combination of the initial cleft, the previous surgical scarring in the palate, the lack of previous expansion, and lack of alveolar bone graft, all of which contributed to the unilateral right posterior crossbite. The maxillary basal arch width was extremely narrow, as demonstrated by the width between the maxillary left and right first premolars (43 mm, measured with an electronic vernier caliper) compared with the 49.5mm Chinese mean value. The maxillary width between the first molars was also narrow at 42 mm. The maxillary left central incisor was hypomineralized, and the left lateral incisor was both undersized and erupting ectopically into the palate. The mandibular arch had a deep curve of Spee and severe crowding with both second premolars erupting in buccally ectopic positions. The molar relationships were a mild Class III on the right and Class I on the left side (Fig 2). Oral hygiene was poor, and bleeding was observed while brushing. The panoramic radiograph (Fig 3) showed a wide alveolar cleft between the maxillary left central incisor
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Fig 2. Pretreatment dental casts.
and the canine. The undersized maxillary lateral incisor was distal to the cleft, palatally ectopic, and severely malpositioned. The crown of the mandibular left third molar overlapped the distal aspect of the mandibular left second molar. The cephalometric analysis indicated a mild skeletal Class III tendency (ANB, 0.1 ; Wits appraisal, 2 mm) with a normal facial growth pattern (SN-MP, 33.6 ). The maxillary incisors were well positioned (1-NA, 25.7 ; 1-NA, 6.2 mm), whereas the mandibular incisors were slightly proclined (L1-MP, 99 ) (Fig 3, Table I). TREATMENT OBJECTIVES
The facial goals of the treatment were to obtain a more balanced profile. Dentally, the goals were to expand the maxillary arch to resolve the maxillary transverse deficiency and correct the unilateral right posterior crossbite, level and align the dental arches, and establish a Class I molar relationship with enhanced intercuspation. The maxilla was carefully observed during expansion because of the wide alveolar cleft and the previous soft-tissue palatal repair. Restoratively, the goal was to provide an ideal space to restore the hypomineralized maxillary left central incisor to enhance the patient's dental esthetics. To achieve these objectives, the following treatment plan was adopted. 1. 2.
A fixed MPE appliance would be used initially to expand the maxillary arch. The palatally ectopic and undersized maxillary lateral incisors and the buccally ectopic and infraoc-
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3.
4.
5. 6.
cluded mandibular second premolars would be extracted. A Class I molar relationship would be established, and the lower lip protrusion would be reduced by retracting the mandibular posterior teeth with mini-implants. Conventional fixed prostheses (2 splinted crowns) for the maxillary left central incisor and the canine would be placed after the orthodontic treatment. A revision rhinoplasty would be performed. Retention would be needed.
It was expected that both function and esthetics would improve, but less than with a combined orthognathic surgery and orthodontic treatment approach. TREATMENT ALTERNATIVES
The ideal treatment of resolving the maxillary transverse deficiency with skeletal expansion and an alveolar bone graft followed by orthognathic surgery to better address the skeletal imbalance was presented to the patient, but he declined. The risks of a nonsurgical treatment, particularly the increased risk of loss of teeth adjacent to the alveolar cleft (maxillary left lateral incisor and canine), potentially unstable dental expansion, and recurrence of the posterior crossbite, were discussed with the patient to provide complete informed consent. TREATMENT PROGRESS
Before we started the orthodontic treatment, the patient was referred for a prosthodontic consultation
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Table I. Cepholometric analysis before and after treat-
ment Measurement SNA ( ) SNB ( ) ANB ( ) Wits (mm) FH-NP ( ) SN-MP ( ) FMA ( ) U1 to SN ( ) U1 to NA ( ) U1 to NA (mm) L1 to NB ( ) L1 to NB (mm) L1 to MP ( ) U1/L1 ( ) Upper lip (mm) Lower lip (mm)
Chinese norm Pretreatment Posttreatment Difference 82.8 76.8 77.2 0.4 80.1 76.9 76.3 0.6 2.7 0.1 0.9 1.0 1 2 0 2 85.4 86.9 86.1 0.8 32.5 33.6 34.5 0.9 27.3 28.5 29.6 1.1 105.7 107.7 106.5 1.2 22.8 25.7 25.2 0.5 5.1 6.2 5.8 0.4 30.3 37.6 35.4 2.2 6.7 9.1 7.2 1.9 92.6 98.8 95.2 3.6 125.4 120.6 126.5 5.9 0.4 0.2 0.5 0.7 0.9 4.8 1.0 3.8
Fig 3. Pretreatment radiographs and computed tomography image.
regarding the possible approaches to restore the hypomineralized maxillary left central incisor and how to replace the lateral incisors by reshaping the maxillary canines. The patient was also referred to a periodontist to evaluate his periodontal health, particularly the teeth adjacent to the alveolar cleft. Finally, the patient was referred to the oral surgeon to evaluate the mesioangular and impacted maxillary right and the mandibular right and left third molars. After consultations with the other dental specialists, maxillary expansion, the first stage of orthodontic treatment, began. Because of the potential tissue damage that can result from rapid, heavy, intermittent forces produced by a screw type of rapid palatal expansion, a fixed MPE was constructed to create slow, suitable, continuous forces. The MPE appliance, consisting of right and left halves containing 8 3 5 3 3 mm3
Fig 4. Force produced between two 8 3 5 3 3 mm3 neodymium magnets.
neodymium (Nd2Fe14B) repelling magnets (Fig 4), was cemented to the maxillary first molars and the first premolars. The expansion force produced between the magnets is greatly affected by the distance separating them, with a maximum force of 550 g with a separation of 0.5 mm. As the distance increases, the expansive force declines, requiring periodic removal and adjustment by adding acrylic to reapproximate the repelling magnets.18 Pins and tubes were constructed to keep the magnets in line with one another and to guide the appliance separation. During expansion, the maxilla, palatal mucosa, and dentition were carefully observed for complications including possible fistula formation or dental tipping. After 3 months, 3 mm of expansion was achieved. Full
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Fig 5. Intraoral maxillary occlusal photographs. A magnetic expansion appliance was used to expand the maxillary arch bilaterally. The arch width of the posterior segment was gradually expanded.
Fig 6. Progress maxillary occlusal photograph and radiograph.
fixed 0.022-in MBT (Xinya, HangZhou, China) appliances were placed in the maxillary and mandible arches after 4 months of expansion to begin aligning the dental arches. After 10 months, the posterior crossbite was fully resolved, the palatal expander was removed (Fig 5), and an acrylic splint was placed to maintain the increased transverse dimension. After the initial leveling, alignment, and correction of rotations that were obtained in conjunction with the expansion, variable modulus orthodontic treatment was performed.20 An 0.020-in Australian wire was used as the main archwire to stabilize the maxillary arch, and an overlay 0.012-in nickel-titanium wire was used to erupt and align the maxillary left central incisor with a light continuous force. The movement of the central incisor was evaluated closely with periapical radiographs to monitor root proximity to the adjacent cleft and to ensure that a leading edge of alveolar bone was forming (Fig 6).21,22 To upright the mandible incisors and correct the mild Class III molar relationship on the left side, a miniimplant was placed between the mandible left first premolar and the first molar roots for 4 months. Class III elastics from the mandibular canine to the maxillary first
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molar were used to help seat the molar into a Class I relationship on the left side (Fig 7). The total active orthodontic treatment time was 22 months. After that, the patient was referred back to the periodontist to correct the gingival margin discrepancies and to the prosthodontist for fabrication of a 2-unit bridge. The final all-ceramic bridge was delivered after appropriate soft-tissue maturation as determined by the periodontist. Clear removable retainers were chosen, and the patient was instructed to wear them full time (24 hours per day except when eating) for 2 years and then nights thereafter because he had declined an alveolar bone graft. During the first year after debond, retainer checks were performed at 1, 2, 3, and 6 months. TREATMENT RESULTS
The posttreatment records demonstrate that the treatment objectives were achieved (Figs 8-10). The photographs show that improved profile and smiling esthetics (Fig 8) and Class I molar relationships were established. The maxillary first molars were expanded by 6 mm during treatment and remained stable during the observation period: pretreatment, 42 mm; posttreatment, 48 mm; retention, 47.9 mm (Table II). The dental midlines were coincident with the facial midline, ideal overbite and overjet were achieved, and the curve of Spee was successfully leveled. The occlusal relationship remained stable at both 1 and 2 years posttreatment (Figs 11 and 12). The posttreatment lateral cephalometric analysis and superimposition (Fig 13) showed mild skeletal changes, including a slight forward movement of the maxilla (ANB increased by 0.4 after treatment) and a mild increase in the mandibular plane angle (SN-MP increased by 0.9 ). The inclination of the maxillary incisors was stable (U1 to SN, 106.5 ). The mandibular incisors were uprighted and retracted slightly (Fig 10; Table I).
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Fig 7. Progress intraoral photographs and radiograph.
The posttreatment panoramic radiograph shows acceptable root parallelism with no signs of bone or root resorption (Fig 10). The patient's facial profile, particularly the protruded lower lip, was improved. Furthermore, his smile was improved by enhancing the interincisal angle (Fig 8). At the 1-year follow-up, the patient had a stable occlusion, with the results of the orthodontic treatment maintained (Fig 11). The radiographic examination showed fairly stable results (Fig 12). DISCUSSION
The treatment for patients with CLP is challenging because of the difficulties inherent in the skeletal
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discrepancy, bone deformity, multiple dental abnormalities, the necessity of interdisciplinary involvement, and the need for excellent patient cooperation. Even if solutions to all of these challenges are developed, some patients might see only partial correction. To maximize the treatment gains and obtain more consistent results, patients with CLP will typically undergo a specific treatment protocol that includes not only primary lip and palatal repairs, but also a primary or secondary alveolar bone graft surgery. The intent of the alveolar bone is to reestablish a continuous maxillary arch, stabilize the transverse dimension, provide adequate alveolar bone for the erupting canine (or lateral incisor when present), and provide a bony foundation for the nasal sill. The patient was informed that because the primary alveolar graft was never performed, for an ideal treatment to result, a secondary alveolar bone graft would be an important aspect of his care. The patient asked several questions pertaining to the graft procedure, including the success rate. He was told that the failure rate of late secondary alveolar cleft grafts is higher in adults than in children undergoing a primary alveolar bone graft.23,24 Potential secondary alveolar graft recipient-site risks include complete loss of the graft, partial integration resulting in an incomplete or poorly established bony bridge, and reduced nasal support. One must also consider the iliac crest donor-site risks, including pain, infection, and decreased mobility, although these can be ameliorated by a careful surgical technique and good postoperative care. Because of financial reasons, the higher failure rate, prior difficulties with access to care, fear of surgery, and other concerns, this patient elected not to undergo the alveolar graft. With a well-healed alveolar bone graft, expansion typically occurs at the midpalatal suture. Without the alveolar bone graft, expansion is more likely to occur at the cleft site, potentially resulting in a larger alveolar cleft.25 Although expansion in this patient would help to correct the posterior crossbite, an increase in cleft size might compromise the stability of the transverse correction and the alveolar support for the teeth adjacent to the cleft, and might be too large for prosthetic reconstruction. If during the expansion an increase in cleft size had been observed, the patient would have been told, and alternative treatment options would have been considered. Several active orthodontic approaches are available to expand the maxilla, including RME, semirapid and slow expansion, and passive expansion with functional appliances. Alternatively, several surgical approaches are possible to expand the maxilla, including
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Fig 8. Posttreatment facial and intraoral photographs.
Fig 9. Posttreatment dental casts.
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Table II. Width changes of the maxillary arch Measurement Pretreatment Posttreatment Difference Width between 22 22.5 0.5 canines (mm) Width between first 31 30.5 0.5 premolars (mm) Width between first 42 48 6.0 molars (mm) Alveolar width between 44 45.3 1.3 first premolars (mm) Basal bone width 43 43.6 0.6 between first premolars (mm)
Fig 10. Posttreatment radiographs and computed tomography image.
multisegment LeFort osteotomies or surgically assisted RME (although the patient declined surgery). Active expansion appliances can be separated into categories by activation method: screw type, spring type, magnetic, and shape memory alloy. For this patient, a fixed MPE appliance as described by Darendeliler et al18 was chosen because magnets can produce a light force without additional adjustments, and the patient's cooperation was expected to be poor. With magnets, the force produced is inversely proportional to the square of the distance between the magnets, meaning that as the magnets move apart, the force will rapidly decrease in magnitude. For this patient, the repelling force was kept between 250 and 350 g by maintaining the distance between the 2 magnets between 1.5 and 2.5 mm. To overcome the lateral deflection that the magnets exhibit when placed in a repelling configuration, guide pins and tubes were placed in the appliance.
Controlling all of these factors resulted in nearconstant force levels and slow expansion that aided in preventing undesirable tissue damage. The results from the cone-beam computed tomography showed that neither the cleft size nor the amount of buccal tipping of the posterior teeth changed significantly (Figs 10 and 14). Patients with CLP often have abnormal numbers, shapes, and sizes of teeth, which makes obtaining an ideal occlusion and restorative result more challenging. The incidence of missing and morphologic abnormalities (eg, enamel hypoplasia) is higher not only for the teeth adjacent to the cleft, but also for all the teeth including the opposing mandibular arch.26,27 In this patient, the maxillary left lateral incisor was extracted because of the undersized shape and the lack of bone and gingival support. Even though the maxillary left central incisor was hypomineralized, it was preserved to maintain as much alveolar support as possible. After the expansion that provided the necessary space, the maxillary left central incisor was slowly moved into the desired position without root resorption or apparent alveolar bone loss. The tooth was planned for restoration after the orthodontic treatment to enhance the esthetic results. From the prosthodontist's perspective, the hypomineralized maxillary left central incisor and the maxillary left canine had good root structure but only moderate alveolar support. As a result, he recommended a splinted 2-unit porcelain crown for these teeth. In this patient, because of the complexities, there were some additional treatment adjuncts to help achieve the desired result. To correct the Class III relationship of the left molars, the mandibular left posterior teeth were distalized using a miniscrew with Class III elastics. The miniscrew provided excellent skeletal anchorage to correct the asymmetric molar relationship and resulted in normal overjet and overbite and an Angle Class I molar
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Fig 11. Facial and intraoral photographs at 1 year posttreatment.
relationship. Adequate dental alignment and leveling, as well as maxillary and mandibular midline symmetry, were also established. This patient's successful treatment improved both his masticatory function and smile esthetics. Postorthodontic treatment can be as difficult as the therapeutic portion of the treatment in patients with CLP depending on the type of cleft. To date, the bridge has been able to bear the occlusal load and maintain the cleft size, as demonstrated in the 1-year followup records. From the periodontist's perspective, the risk of gingival and periodontal recession was increased in the cleft region because of the reduced alveolar support, so more frequent periodontal maintenance visits and excellent oral hygiene were recommended. The patient has been compliant with both. To further aid in retaining the result, lifetime retainers were recommended. For every patient, the care provider must be sure that he or she has received full informed consent from the
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patient to ensure that the patient understands both the benefits and the risks of treatment. Generally, after a complete presentation of their treatment options, patients will not only acknowledge their understanding, but also elect to proceed with the recommended treatment approach. On occasion, however, either ideal treatment options are unavailable, or patients are unwilling to proceed with them. In these situations, patients might refuse at least 1 aspect of care, as is their right. At this point, the practitioner must decide whether the portion of treatment that the patient has refused will adversely affect his or her health, the predictability of achieving the desired outcome, and the short- or long-term stability.28 If an appropriate treatment approach is possible and the practitioner is confident that the benefits of providing care exceed the risks, treatment can still proceed. If the practitioner is unwilling to assume the risks resulting from the patient's refusal, then he or she might elect not to proceed with treatment and should make further attempts to educate the patient about the ramifications of refusal.
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Fig 14. Comparison of cleft size; left, pretreatment; right, posttreatment. Fig 12. Radiographs at 1 year posttreatment. CONCLUSIONS
Our report describes the challenging treatment of an adult with unilateral CLP. Without surgery, he was successfully treated by multidisciplinary cooperative work. In orthodontic camouflage treatment, magnetic expansion has a favorable expansion effect in the cleft site; this helped to correct the posterior crossbite. The big issue in this patient was how to move the teeth adjacent to cleft site without secondary bone grafting. Although the maxillary left central incisor showed amelogenesis imperfecta before treatment, the good result was achieved by the labial orthodontic movement and the prosthetic crown of restorative treatment. The posttreatment and 1-year follow-up records showed good stability for the teeth in the cleft site. The retraction of mandibular incisors and the use of a miniscrew helped to obtain a proper occlusion and a balanced profile. The proposed objectives of normal function and better esthetics were achieved by multidisciplinary treatment. REFERENCES
Fig 13. Superimposed tracings. Black, pretreatment; red, posttreatment.
For this patient, the team thought that care could still proceed, and the results demonstrate that this was an acceptable treatment option.
1. Arosarena OA. Cleft lip and palate. Otolaryngol Clin North Am 2007;40:27-60:vi. 2. Mossey PA, Little J, Munger RG, Dixon MJ, Shaw WC. Cleft lip and palate. Lancet 2009;374(9703):1773-85. 3. Ching GH, Chung CS. A genetic study of cleft lip and palate in Hawaii. I. Interracial crosses. Am J Hum Genet 1974;26:162-76. 4. Srzentic M, Handzic J, Trotic R. The developmental characteristics of mastoid pneumatisation in cleft palate children: the genetic influence. Coll Antropol 2012;36:885-91. 5. Jia ZL, Shi B, Chen CH, Shi JY, Wu J, Xu X. Maternal malnutrition, environmental exposure during pregnancy and the risk of nonsyndromic orofacial clefts. Oral Dis 2011;17:584-9.
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6. Lai MC, King NM, Wong HM. Abnormalities of maxillary anterior teeth in Chinese children with cleft lip and palate. Cleft Palate Craniofac J 2009;46:58-64. 7. Ranta R, Tulensalo T. Symmetry and combinations of hypodontia in non-cleft and cleft palate children. Scand J Dent Res 1988;96:1-8. 8. Qureshi WA, Beiraghi S, Leon-Salazar V. Dental anomalies associated with unilateral and bilateral cleft lip and palate. J Dent Child (Chic) 2012;79:69-73. 9. Meyer-Marcotty P, Stellzig-Eisenhauer A. Dentofacial selfperception and social perception of adults with unilateral cleft lip and palate. J Orofac Orthop 2009;70:224-36. 10. Aljohar A, Ravichandran K, Subhani S. Pattern of cleft lip and palate in hospital-based population in Saudi Arabia: retrospective study. Cleft Palate Craniofac J 2008;45:592-6. 11. David DJ, Smith I, Nugent M, Richards C, Anderson PJ. From birth to maturity: a group of patients who have completed their protocol management. Part III. Bilateral cleft lip-cleft palate. Plast Reconstr Surg 2011;128:475-84. 12. Boyne PJ, Sands NR. Combined orthodontic-surgical management of residual palato-alveolar cleft defects. Am J Orthod 1976;70:20-37. 13. Bertz JE. Bone grafting of alveolar clefts. J Oral Surg 1981;39:874-7. 14. Bergland O, Semb G, Abyholm F, Borchgrevink H, Eskeland G. Secondary bone grafting and orthodontic treatment in patients with bilateral complete clefts of the lip and palate. Ann Plast Surg 1986;17:460-74. 15. Suzuki EY, Watanabe M, Buranastidporn B, Baba Y, Ohyama K, Ishii M. Simultaneous maxillary distraction osteogenesis using a twin-track distraction device combined with alveolar bone grafting in cleft patients: preliminary report of a technique. Angle Orthod 2006;76:164-72. 16. Carlini JL, Biron C, Gomes KU, Da Silva RM. Surgical repositioning of the premaxilla with bone graft in 50 bilateral cleft lip and palate patients. J Oral Maxillofac Surg 2009;67:760-6.
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17. Geraedts CT, Borstlap WA, Groenewoud JM, Borstlap-Engels VM, Stoelinga PJ. Long-term evaluation of bilateral cleft lip and palate patients after early secondary closure and premaxilla repositioning. Int J Oral Maxillofac Surg 2007;36:788-96. 18. Darendeliler MA, Strahm C, Joho JP. Light maxillary expansion forces with the magnetic expansion device. A preliminary investigation. Eur J Orthod 1994;16:479-90. 19. Romanyk DL, Lagravere MO, Toogood RW, Major PW, Carey JP. Review of maxillary expansion appliance activation methods: engineering and clinical perspectives. J Dent Biomech 2010;2010. 20. Burstone CJ. Variable-modulus orthodontics. Am J Orthod 1981; 80:1-16. 21. Roberts WE, Marshall KJ, Mozsary PG. Rigid endosseous implant utilized as anchorage to protract molars and close an atrophic extraction site. Angle Orthod 1990;60:135-52. 22. Graber TM, Swain BF. Orthodontics, current principles and techniques. St Louis: Mosby; 1985. p. 111-229. 23. Enemark H, Sindet-Pedersen S, Bundgaard M. Long-term results after secondary bone grafting of alveolar clefts. J Oral Maxillofac Surg 1987;45:913-9. 24. Toscano D, Baciliero U, Gracco A, Siciliani G. Long-term stability of alveolar bone grafts in cleft palate patients. Am J Orthod Dentofacial Orthop 2012;142:289-99. 25. Long RE Jr, Spangler BE, Yow M. Cleft width and secondary alveolar bone graft success. Cleft Palate Craniofac J 1995;32:420-7. 26. Ranta R. Comparison of tooth formation in noncleft and cleftaffected children with and without hypodontia. ASDC J Dent Child 1982;49:197-9. 27. Kraus BS, Jordan RE, Pruzansky S. Dental abnormalities in the Deciduous and Permanent Dentition of Individuals with Cleft Lip and Palate. J Dent Res 1966;45:1736-46. 28. Greco PM. Informed consent or informed refusal? Am J Orthod Dentofacial Orthop 2013;143:598.
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Interdisciplinary treatment for an adult with a unilateral cleft lip and palate Qiao Ling Ma,a R. Scott Conley,b Tuojiang Wu,c and Huang Lid Nanjing, Jiangsu, People's Republic of China, and Ann Arbor, Mich
A young man, age 18 years 4 months, with a concave profile, a skeletal maxillary deficiency, and a severe alveolar cleft with an unesthetic appearance of the maxillary anterior teeth was referred for orthodontic treatment. After a detailed review of his pretreatment records, both surgical and nonsurgical treatment plans were presented to the patient, who opted for a nonsurgical interdisciplinary approach. His complex 3-dimensional malocclusion required palatal expansion, dental extractions, and periodontal and prosthodontic consultations and treatment, in addition to comprehensive orthodontic therapy. MBT (Xinya, HangZhou, China) 0.022 3 0.028in appliances combined with a mini-implant to enhance the orthodontic anchorage were used to level, align, and establish a Class I relationship. After the orthodontic treatment, a combined restorative and periodontal approach was used to enhance the patient's esthetic and functional outcomes. Both the final result and the 1year follow-up records demonstrate that the treatment goals of establishing proper occlusion, normal function, a balanced profile, better esthetics, and a stable outcome were achieved. The purpose of this case report is to demonstrate that an interdisciplinary treatment protocol can significantly improve the transverse discrepancies and achieve a satisfactory occlusion with a balanced profile in patients with cleft lip and palate. (Am J Orthod Dentofacial Orthop 2014;146:238-48)
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left lip and palate (CLP) is the most frequent congenital facial abnormality.1,2 Compared with the incidence rate reported from other countries, the frequency of all forms of clefts is highest among Asians.3 The etiology is related to both hereditary and environmental factors.4,5 Patients with CLP can also have other features such as deficient midface development resulting in a Class III tendency, severe maxillary transverse deficiency, alveolar cleft, and hypodontia.6-8 CLP patients might also have decreased facial and dental esthetics, resulting in low self-
a Attending orthodontist, Department of Orthodontics, Stomatological Hospital, Medical School of Nanjing University, Nanjing, Jiangsu, People's Republic of China. b Associate professor, Department of Orthodontics and Pediatric Dentistry, School of Dentistry, University of Michigan, Ann Arbor, Mich. c Associate professor, College of Stomatology, Nanjing Medical University, Nanjing, Jiangsu, People's Republic of China. d Associate professor, Department of Orthodontics, Stomatological Hospital, School of Medicine, Nanjing University, Nanjing, Jiangsu, People's Republic of China. All authors have completed and submitted the ICMJE Form for Disclosure of Potential Conflicts of Interest, and none were reported. Sponsored by the National Natural Science Foundation of China and Medical Science and Technology Development Foundation, Nanjing Department of Health, grant nos. 81070807 and YKK11038. Address correspondence to: Huang Li, Department of Orthodontics, Nanjing University, The Stomatological Hospital of Medical School, Nan Jing, Jiangsu, People's Republic of China; e-mail, [email protected]. Submitted, May 2013; revised and accepted, October 2013. 0889-5406/$36.00 Copyright Ó 2014 by the American Association of Orthodontists. http://dx.doi.org/10.1016/j.ajodo.2013.10.024
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confidence and difficulties in social interactions.9 Interdisciplinary treatment plans for patients with CLP frequently extend over many years, starting with primary surgeries during infancy, at least 1 stage of orthodontic treatment, and possible jaw surgery near the end of adolescence or early adulthood.10 Treatment frequently includes nasoalveolar molding, labial repair (about 36 months of age), palatoplasty (10-12 months of age), orthodontic expansion, primary or secondary alveolar bone graft, surgery, and comprehensive orthodontic treatment to reestablish facial esthetics and proper function.11-17 One important aspect of orthodontic treatment includes transverse expansion of the maxilla to correct the transverse deficiency of the palatal segments. This can be achieved using one of several different types of expanders, including traditional screw-type rapid palatal expansion, spring-type, magnetic, and shape memory alloy. The magnetic palatal expander (MPE) with repelling magnets to increase the width of maxilla has been previously reported by Darendeliler et al.18 Magnets produce a suitable force without requiring additional adjustment by the patient and are more rigid than spring-type expanders, potentially resulting in fewer undesirable effects; however, there are few reports of MPE treatment in CLP patients.19 This case report describes an interdisciplinary treatment approach for a young man with unilateral CLP. MPE, comprehensive orthodontics, periodontics, and nasoalveolar molding were used to obtain proper
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Fig 1. Pretreatment photographs.
occlusion and normal function; a balanced profile and better esthetics were achieved. DIAGNOSIS AND ETIOLOGY
A young Chinese man (age 18 years 4 months) came to the Orthodontic Department of Nanjing University for orthodontic evaluation of his severe crowding and posterior crossbite. His chief complaint was the unesthetic appearance of his face and maxillary anterior teeth. He was born with nonsyndromic unilateral left CLP. The lip was repaired at the age of 6 months (primary cheiloplasty), and the palate (palatoplasty) was repaired at 4 years of age, with no further orthodontic treatment or alveolar bone-grafting procedures. The patient had a concave profile with a retrusive upper lip and an everted lower lip. He showed good vertical balance among the facial thirds and good symmetry except for the nose and upper lip resulting from both the cleft and the scarring from the previous surgical repair. The maxillary and mandibular dental midlines were coincident but both were deviated to the left by 2 mm (Fig 1).
The maxillary arch was collapsed and constricted resulting from a combination of the initial cleft, the previous surgical scarring in the palate, the lack of previous expansion, and lack of alveolar bone graft, all of which contributed to the unilateral right posterior crossbite. The maxillary basal arch width was extremely narrow, as demonstrated by the width between the maxillary left and right first premolars (43 mm, measured with an electronic vernier caliper) compared with the 49.5mm Chinese mean value. The maxillary width between the first molars was also narrow at 42 mm. The maxillary left central incisor was hypomineralized, and the left lateral incisor was both undersized and erupting ectopically into the palate. The mandibular arch had a deep curve of Spee and severe crowding with both second premolars erupting in buccally ectopic positions. The molar relationships were a mild Class III on the right and Class I on the left side (Fig 2). Oral hygiene was poor, and bleeding was observed while brushing. The panoramic radiograph (Fig 3) showed a wide alveolar cleft between the maxillary left central incisor
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Fig 2. Pretreatment dental casts.
and the canine. The undersized maxillary lateral incisor was distal to the cleft, palatally ectopic, and severely malpositioned. The crown of the mandibular left third molar overlapped the distal aspect of the mandibular left second molar. The cephalometric analysis indicated a mild skeletal Class III tendency (ANB, 0.1 ; Wits appraisal, 2 mm) with a normal facial growth pattern (SN-MP, 33.6 ). The maxillary incisors were well positioned (1-NA, 25.7 ; 1-NA, 6.2 mm), whereas the mandibular incisors were slightly proclined (L1-MP, 99 ) (Fig 3, Table I). TREATMENT OBJECTIVES
The facial goals of the treatment were to obtain a more balanced profile. Dentally, the goals were to expand the maxillary arch to resolve the maxillary transverse deficiency and correct the unilateral right posterior crossbite, level and align the dental arches, and establish a Class I molar relationship with enhanced intercuspation. The maxilla was carefully observed during expansion because of the wide alveolar cleft and the previous soft-tissue palatal repair. Restoratively, the goal was to provide an ideal space to restore the hypomineralized maxillary left central incisor to enhance the patient's dental esthetics. To achieve these objectives, the following treatment plan was adopted. 1. 2.
A fixed MPE appliance would be used initially to expand the maxillary arch. The palatally ectopic and undersized maxillary lateral incisors and the buccally ectopic and infraoc-
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3.
4.
5. 6.
cluded mandibular second premolars would be extracted. A Class I molar relationship would be established, and the lower lip protrusion would be reduced by retracting the mandibular posterior teeth with mini-implants. Conventional fixed prostheses (2 splinted crowns) for the maxillary left central incisor and the canine would be placed after the orthodontic treatment. A revision rhinoplasty would be performed. Retention would be needed.
It was expected that both function and esthetics would improve, but less than with a combined orthognathic surgery and orthodontic treatment approach. TREATMENT ALTERNATIVES
The ideal treatment of resolving the maxillary transverse deficiency with skeletal expansion and an alveolar bone graft followed by orthognathic surgery to better address the skeletal imbalance was presented to the patient, but he declined. The risks of a nonsurgical treatment, particularly the increased risk of loss of teeth adjacent to the alveolar cleft (maxillary left lateral incisor and canine), potentially unstable dental expansion, and recurrence of the posterior crossbite, were discussed with the patient to provide complete informed consent. TREATMENT PROGRESS
Before we started the orthodontic treatment, the patient was referred for a prosthodontic consultation
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Table I. Cepholometric analysis before and after treat-
ment Measurement SNA ( ) SNB ( ) ANB ( ) Wits (mm) FH-NP ( ) SN-MP ( ) FMA ( ) U1 to SN ( ) U1 to NA ( ) U1 to NA (mm) L1 to NB ( ) L1 to NB (mm) L1 to MP ( ) U1/L1 ( ) Upper lip (mm) Lower lip (mm)
Chinese norm Pretreatment Posttreatment Difference 82.8 76.8 77.2 0.4 80.1 76.9 76.3 0.6 2.7 0.1 0.9 1.0 1 2 0 2 85.4 86.9 86.1 0.8 32.5 33.6 34.5 0.9 27.3 28.5 29.6 1.1 105.7 107.7 106.5 1.2 22.8 25.7 25.2 0.5 5.1 6.2 5.8 0.4 30.3 37.6 35.4 2.2 6.7 9.1 7.2 1.9 92.6 98.8 95.2 3.6 125.4 120.6 126.5 5.9 0.4 0.2 0.5 0.7 0.9 4.8 1.0 3.8
Fig 3. Pretreatment radiographs and computed tomography image.
regarding the possible approaches to restore the hypomineralized maxillary left central incisor and how to replace the lateral incisors by reshaping the maxillary canines. The patient was also referred to a periodontist to evaluate his periodontal health, particularly the teeth adjacent to the alveolar cleft. Finally, the patient was referred to the oral surgeon to evaluate the mesioangular and impacted maxillary right and the mandibular right and left third molars. After consultations with the other dental specialists, maxillary expansion, the first stage of orthodontic treatment, began. Because of the potential tissue damage that can result from rapid, heavy, intermittent forces produced by a screw type of rapid palatal expansion, a fixed MPE was constructed to create slow, suitable, continuous forces. The MPE appliance, consisting of right and left halves containing 8 3 5 3 3 mm3
Fig 4. Force produced between two 8 3 5 3 3 mm3 neodymium magnets.
neodymium (Nd2Fe14B) repelling magnets (Fig 4), was cemented to the maxillary first molars and the first premolars. The expansion force produced between the magnets is greatly affected by the distance separating them, with a maximum force of 550 g with a separation of 0.5 mm. As the distance increases, the expansive force declines, requiring periodic removal and adjustment by adding acrylic to reapproximate the repelling magnets.18 Pins and tubes were constructed to keep the magnets in line with one another and to guide the appliance separation. During expansion, the maxilla, palatal mucosa, and dentition were carefully observed for complications including possible fistula formation or dental tipping. After 3 months, 3 mm of expansion was achieved. Full
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Fig 5. Intraoral maxillary occlusal photographs. A magnetic expansion appliance was used to expand the maxillary arch bilaterally. The arch width of the posterior segment was gradually expanded.
Fig 6. Progress maxillary occlusal photograph and radiograph.
fixed 0.022-in MBT (Xinya, HangZhou, China) appliances were placed in the maxillary and mandible arches after 4 months of expansion to begin aligning the dental arches. After 10 months, the posterior crossbite was fully resolved, the palatal expander was removed (Fig 5), and an acrylic splint was placed to maintain the increased transverse dimension. After the initial leveling, alignment, and correction of rotations that were obtained in conjunction with the expansion, variable modulus orthodontic treatment was performed.20 An 0.020-in Australian wire was used as the main archwire to stabilize the maxillary arch, and an overlay 0.012-in nickel-titanium wire was used to erupt and align the maxillary left central incisor with a light continuous force. The movement of the central incisor was evaluated closely with periapical radiographs to monitor root proximity to the adjacent cleft and to ensure that a leading edge of alveolar bone was forming (Fig 6).21,22 To upright the mandible incisors and correct the mild Class III molar relationship on the left side, a miniimplant was placed between the mandible left first premolar and the first molar roots for 4 months. Class III elastics from the mandibular canine to the maxillary first
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molar were used to help seat the molar into a Class I relationship on the left side (Fig 7). The total active orthodontic treatment time was 22 months. After that, the patient was referred back to the periodontist to correct the gingival margin discrepancies and to the prosthodontist for fabrication of a 2-unit bridge. The final all-ceramic bridge was delivered after appropriate soft-tissue maturation as determined by the periodontist. Clear removable retainers were chosen, and the patient was instructed to wear them full time (24 hours per day except when eating) for 2 years and then nights thereafter because he had declined an alveolar bone graft. During the first year after debond, retainer checks were performed at 1, 2, 3, and 6 months. TREATMENT RESULTS
The posttreatment records demonstrate that the treatment objectives were achieved (Figs 8-10). The photographs show that improved profile and smiling esthetics (Fig 8) and Class I molar relationships were established. The maxillary first molars were expanded by 6 mm during treatment and remained stable during the observation period: pretreatment, 42 mm; posttreatment, 48 mm; retention, 47.9 mm (Table II). The dental midlines were coincident with the facial midline, ideal overbite and overjet were achieved, and the curve of Spee was successfully leveled. The occlusal relationship remained stable at both 1 and 2 years posttreatment (Figs 11 and 12). The posttreatment lateral cephalometric analysis and superimposition (Fig 13) showed mild skeletal changes, including a slight forward movement of the maxilla (ANB increased by 0.4 after treatment) and a mild increase in the mandibular plane angle (SN-MP increased by 0.9 ). The inclination of the maxillary incisors was stable (U1 to SN, 106.5 ). The mandibular incisors were uprighted and retracted slightly (Fig 10; Table I).
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Fig 7. Progress intraoral photographs and radiograph.
The posttreatment panoramic radiograph shows acceptable root parallelism with no signs of bone or root resorption (Fig 10). The patient's facial profile, particularly the protruded lower lip, was improved. Furthermore, his smile was improved by enhancing the interincisal angle (Fig 8). At the 1-year follow-up, the patient had a stable occlusion, with the results of the orthodontic treatment maintained (Fig 11). The radiographic examination showed fairly stable results (Fig 12). DISCUSSION
The treatment for patients with CLP is challenging because of the difficulties inherent in the skeletal
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discrepancy, bone deformity, multiple dental abnormalities, the necessity of interdisciplinary involvement, and the need for excellent patient cooperation. Even if solutions to all of these challenges are developed, some patients might see only partial correction. To maximize the treatment gains and obtain more consistent results, patients with CLP will typically undergo a specific treatment protocol that includes not only primary lip and palatal repairs, but also a primary or secondary alveolar bone graft surgery. The intent of the alveolar bone is to reestablish a continuous maxillary arch, stabilize the transverse dimension, provide adequate alveolar bone for the erupting canine (or lateral incisor when present), and provide a bony foundation for the nasal sill. The patient was informed that because the primary alveolar graft was never performed, for an ideal treatment to result, a secondary alveolar bone graft would be an important aspect of his care. The patient asked several questions pertaining to the graft procedure, including the success rate. He was told that the failure rate of late secondary alveolar cleft grafts is higher in adults than in children undergoing a primary alveolar bone graft.23,24 Potential secondary alveolar graft recipient-site risks include complete loss of the graft, partial integration resulting in an incomplete or poorly established bony bridge, and reduced nasal support. One must also consider the iliac crest donor-site risks, including pain, infection, and decreased mobility, although these can be ameliorated by a careful surgical technique and good postoperative care. Because of financial reasons, the higher failure rate, prior difficulties with access to care, fear of surgery, and other concerns, this patient elected not to undergo the alveolar graft. With a well-healed alveolar bone graft, expansion typically occurs at the midpalatal suture. Without the alveolar bone graft, expansion is more likely to occur at the cleft site, potentially resulting in a larger alveolar cleft.25 Although expansion in this patient would help to correct the posterior crossbite, an increase in cleft size might compromise the stability of the transverse correction and the alveolar support for the teeth adjacent to the cleft, and might be too large for prosthetic reconstruction. If during the expansion an increase in cleft size had been observed, the patient would have been told, and alternative treatment options would have been considered. Several active orthodontic approaches are available to expand the maxilla, including RME, semirapid and slow expansion, and passive expansion with functional appliances. Alternatively, several surgical approaches are possible to expand the maxilla, including
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Fig 8. Posttreatment facial and intraoral photographs.
Fig 9. Posttreatment dental casts.
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Table II. Width changes of the maxillary arch Measurement Pretreatment Posttreatment Difference Width between 22 22.5 0.5 canines (mm) Width between first 31 30.5 0.5 premolars (mm) Width between first 42 48 6.0 molars (mm) Alveolar width between 44 45.3 1.3 first premolars (mm) Basal bone width 43 43.6 0.6 between first premolars (mm)
Fig 10. Posttreatment radiographs and computed tomography image.
multisegment LeFort osteotomies or surgically assisted RME (although the patient declined surgery). Active expansion appliances can be separated into categories by activation method: screw type, spring type, magnetic, and shape memory alloy. For this patient, a fixed MPE appliance as described by Darendeliler et al18 was chosen because magnets can produce a light force without additional adjustments, and the patient's cooperation was expected to be poor. With magnets, the force produced is inversely proportional to the square of the distance between the magnets, meaning that as the magnets move apart, the force will rapidly decrease in magnitude. For this patient, the repelling force was kept between 250 and 350 g by maintaining the distance between the 2 magnets between 1.5 and 2.5 mm. To overcome the lateral deflection that the magnets exhibit when placed in a repelling configuration, guide pins and tubes were placed in the appliance.
Controlling all of these factors resulted in nearconstant force levels and slow expansion that aided in preventing undesirable tissue damage. The results from the cone-beam computed tomography showed that neither the cleft size nor the amount of buccal tipping of the posterior teeth changed significantly (Figs 10 and 14). Patients with CLP often have abnormal numbers, shapes, and sizes of teeth, which makes obtaining an ideal occlusion and restorative result more challenging. The incidence of missing and morphologic abnormalities (eg, enamel hypoplasia) is higher not only for the teeth adjacent to the cleft, but also for all the teeth including the opposing mandibular arch.26,27 In this patient, the maxillary left lateral incisor was extracted because of the undersized shape and the lack of bone and gingival support. Even though the maxillary left central incisor was hypomineralized, it was preserved to maintain as much alveolar support as possible. After the expansion that provided the necessary space, the maxillary left central incisor was slowly moved into the desired position without root resorption or apparent alveolar bone loss. The tooth was planned for restoration after the orthodontic treatment to enhance the esthetic results. From the prosthodontist's perspective, the hypomineralized maxillary left central incisor and the maxillary left canine had good root structure but only moderate alveolar support. As a result, he recommended a splinted 2-unit porcelain crown for these teeth. In this patient, because of the complexities, there were some additional treatment adjuncts to help achieve the desired result. To correct the Class III relationship of the left molars, the mandibular left posterior teeth were distalized using a miniscrew with Class III elastics. The miniscrew provided excellent skeletal anchorage to correct the asymmetric molar relationship and resulted in normal overjet and overbite and an Angle Class I molar
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Fig 11. Facial and intraoral photographs at 1 year posttreatment.
relationship. Adequate dental alignment and leveling, as well as maxillary and mandibular midline symmetry, were also established. This patient's successful treatment improved both his masticatory function and smile esthetics. Postorthodontic treatment can be as difficult as the therapeutic portion of the treatment in patients with CLP depending on the type of cleft. To date, the bridge has been able to bear the occlusal load and maintain the cleft size, as demonstrated in the 1-year followup records. From the periodontist's perspective, the risk of gingival and periodontal recession was increased in the cleft region because of the reduced alveolar support, so more frequent periodontal maintenance visits and excellent oral hygiene were recommended. The patient has been compliant with both. To further aid in retaining the result, lifetime retainers were recommended. For every patient, the care provider must be sure that he or she has received full informed consent from the
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patient to ensure that the patient understands both the benefits and the risks of treatment. Generally, after a complete presentation of their treatment options, patients will not only acknowledge their understanding, but also elect to proceed with the recommended treatment approach. On occasion, however, either ideal treatment options are unavailable, or patients are unwilling to proceed with them. In these situations, patients might refuse at least 1 aspect of care, as is their right. At this point, the practitioner must decide whether the portion of treatment that the patient has refused will adversely affect his or her health, the predictability of achieving the desired outcome, and the short- or long-term stability.28 If an appropriate treatment approach is possible and the practitioner is confident that the benefits of providing care exceed the risks, treatment can still proceed. If the practitioner is unwilling to assume the risks resulting from the patient's refusal, then he or she might elect not to proceed with treatment and should make further attempts to educate the patient about the ramifications of refusal.
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Fig 14. Comparison of cleft size; left, pretreatment; right, posttreatment. Fig 12. Radiographs at 1 year posttreatment. CONCLUSIONS
Our report describes the challenging treatment of an adult with unilateral CLP. Without surgery, he was successfully treated by multidisciplinary cooperative work. In orthodontic camouflage treatment, magnetic expansion has a favorable expansion effect in the cleft site; this helped to correct the posterior crossbite. The big issue in this patient was how to move the teeth adjacent to cleft site without secondary bone grafting. Although the maxillary left central incisor showed amelogenesis imperfecta before treatment, the good result was achieved by the labial orthodontic movement and the prosthetic crown of restorative treatment. The posttreatment and 1-year follow-up records showed good stability for the teeth in the cleft site. The retraction of mandibular incisors and the use of a miniscrew helped to obtain a proper occlusion and a balanced profile. The proposed objectives of normal function and better esthetics were achieved by multidisciplinary treatment. REFERENCES
Fig 13. Superimposed tracings. Black, pretreatment; red, posttreatment.
For this patient, the team thought that care could still proceed, and the results demonstrate that this was an acceptable treatment option.
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6. Lai MC, King NM, Wong HM. Abnormalities of maxillary anterior teeth in Chinese children with cleft lip and palate. Cleft Palate Craniofac J 2009;46:58-64. 7. Ranta R, Tulensalo T. Symmetry and combinations of hypodontia in non-cleft and cleft palate children. Scand J Dent Res 1988;96:1-8. 8. Qureshi WA, Beiraghi S, Leon-Salazar V. Dental anomalies associated with unilateral and bilateral cleft lip and palate. J Dent Child (Chic) 2012;79:69-73. 9. Meyer-Marcotty P, Stellzig-Eisenhauer A. Dentofacial selfperception and social perception of adults with unilateral cleft lip and palate. J Orofac Orthop 2009;70:224-36. 10. Aljohar A, Ravichandran K, Subhani S. Pattern of cleft lip and palate in hospital-based population in Saudi Arabia: retrospective study. Cleft Palate Craniofac J 2008;45:592-6. 11. David DJ, Smith I, Nugent M, Richards C, Anderson PJ. From birth to maturity: a group of patients who have completed their protocol management. Part III. Bilateral cleft lip-cleft palate. Plast Reconstr Surg 2011;128:475-84. 12. Boyne PJ, Sands NR. Combined orthodontic-surgical management of residual palato-alveolar cleft defects. Am J Orthod 1976;70:20-37. 13. Bertz JE. Bone grafting of alveolar clefts. J Oral Surg 1981;39:874-7. 14. Bergland O, Semb G, Abyholm F, Borchgrevink H, Eskeland G. Secondary bone grafting and orthodontic treatment in patients with bilateral complete clefts of the lip and palate. Ann Plast Surg 1986;17:460-74. 15. Suzuki EY, Watanabe M, Buranastidporn B, Baba Y, Ohyama K, Ishii M. Simultaneous maxillary distraction osteogenesis using a twin-track distraction device combined with alveolar bone grafting in cleft patients: preliminary report of a technique. Angle Orthod 2006;76:164-72. 16. Carlini JL, Biron C, Gomes KU, Da Silva RM. Surgical repositioning of the premaxilla with bone graft in 50 bilateral cleft lip and palate patients. J Oral Maxillofac Surg 2009;67:760-6.
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17. Geraedts CT, Borstlap WA, Groenewoud JM, Borstlap-Engels VM, Stoelinga PJ. Long-term evaluation of bilateral cleft lip and palate patients after early secondary closure and premaxilla repositioning. Int J Oral Maxillofac Surg 2007;36:788-96. 18. Darendeliler MA, Strahm C, Joho JP. Light maxillary expansion forces with the magnetic expansion device. A preliminary investigation. Eur J Orthod 1994;16:479-90. 19. Romanyk DL, Lagravere MO, Toogood RW, Major PW, Carey JP. Review of maxillary expansion appliance activation methods: engineering and clinical perspectives. J Dent Biomech 2010;2010. 20. Burstone CJ. Variable-modulus orthodontics. Am J Orthod 1981; 80:1-16. 21. Roberts WE, Marshall KJ, Mozsary PG. Rigid endosseous implant utilized as anchorage to protract molars and close an atrophic extraction site. Angle Orthod 1990;60:135-52. 22. Graber TM, Swain BF. Orthodontics, current principles and techniques. St Louis: Mosby; 1985. p. 111-229. 23. Enemark H, Sindet-Pedersen S, Bundgaard M. Long-term results after secondary bone grafting of alveolar clefts. J Oral Maxillofac Surg 1987;45:913-9. 24. Toscano D, Baciliero U, Gracco A, Siciliani G. Long-term stability of alveolar bone grafts in cleft palate patients. Am J Orthod Dentofacial Orthop 2012;142:289-99. 25. Long RE Jr, Spangler BE, Yow M. Cleft width and secondary alveolar bone graft success. Cleft Palate Craniofac J 1995;32:420-7. 26. Ranta R. Comparison of tooth formation in noncleft and cleftaffected children with and without hypodontia. ASDC J Dent Child 1982;49:197-9. 27. Kraus BS, Jordan RE, Pruzansky S. Dental abnormalities in the Deciduous and Permanent Dentition of Individuals with Cleft Lip and Palate. J Dent Res 1966;45:1736-46. 28. Greco PM. Informed consent or informed refusal? Am J Orthod Dentofacial Orthop 2013;143:598.
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