The Cleft Palate–Craniofacial Journal 52(4) pp. e95–e102 July 2015 Ó Copyright 2015 American Cleft Palate–Craniofacial Association

CASE REPORT Premaxillary Distraction Osteogenesis Using an Intraoral Appliance for Unilateral Cleft Lip and Palate: Case Report Munkhdulam Terbish, D.D.S., Hye-Young Choi, D.D.S., Young-Chel Park, D.D.S., Ph.D., Choong Kook Yi, D.D.S., Ph.D., Jung-Yul Cha, D.D.S., Ph.D. Objective: Premaxillary distraction osteogenesis was introduced using intraoral devices to correct maxillary hypoplasia and lengthen the alveolar bone horizontally in a patient with unilateral cleft lip and palate. Methods: For premaxillary distraction osteogenesis, Le Fort I osteotomy was performed. Vertical osteotomy lines were located distally of the upper right canine and left first premolar to separate the anterior segment of the maxilla. After a 7-day latency period, distraction was allowed to continue for 20 days at a rate of 0.5 mm/d, followed by a 3-month consolidation period. After consolidation, orthodontic treatment and bilateral intraoral vertical ramus osteotomy were performed for the mandibular setback. The implant and prosthodontic treatments were applied to the alveolar ridge area created by the distraction osteogenesis. Results: The A-point moved 8.0 mm forward during the distraction osteogenesis period, and the recurrence rate was 25% after the retention period. The transverse dimension of the upper arch was expanded during orthodontic treatment. The quality of the alveolar bone created by distraction osteogenesis was acceptable for the prosthodontic implant. Conclusions: Premaxillary distraction osteogenesis and arch expansion is an effective treatment strategy, improving function, aesthetics, and stability for cleft patients with multiple missing teeth. KEY WORDS:

cleft palate and lip, dental implants, distraction osteogenesis, skeletal class III

with cleft lip and palate (Figueroa and Polley, 1999; Tong et al., 2003; Karakasis and Hadjipetrou, 2004; Dolanmaz et al., 2003a). Premaxillary DO has been the alternative treatment option for patients with midfacial retrusion, crowding in the maxillary anterior area, and premolar lesions, including missing teeth and anterior crossbite (Liou et al., 2000; Tong et al., 2003). Therefore, it is essential to create an alveolar bone space for tooth alignment and simultaneously correct for maxillary hypoplasia. The premaxillary DO used in different types of extraoral and intraoral distractors has been established. A rigid external distraction device (RED) is fixed to the skull. Extraoral distractors have the volume for multidirectional maxillary advancement, and the vectors can be changed during the process (Meling et al., 2011). This allows for protection of the maxillary teeth, compared with other types of extraoral devices that are anchored to the cranium (Picard et al., 2011). However, many patients have difficulties with extraoral devices, primarily due to displeasure with the aesthetics. Choi et al. (2012) reported that downward movement of the premaxillary was observed with the RED, compared with an intraoral distractor appliance. Intraoral appliances are classified into tooth-borne and bone-borne distractors (Bengi et al., 2004; Iida et al., 2007). Tooth-borne appliances can be applied to distract alveolar

Simultaneous maxillary advancement by Le Fort I osteotomy is widely performed and is one of the most common treatment options for the correction of maxillary retrusion in cleft patients. However, this technique may cause velopharyngeal incompetence and deterioration of hypernasality, particularly for cleft palate patients, who already have disadvantages in function and elasticity of the soft tissues due to morphological changes in the nasopharynx (Watzke et al. 1990; Janulewicz et al., 2004). Other reports have indicated that patients with cleft lip and palate who have maxillary advancement greater than 10 mm are at increased risk of speech deterioration (Ko et al., 1999; Harada et al., 2002). Premaxillary and alveolar distraction osteogenesis (DO) are often used to correct maxillary hypoplasia in patients

Dr. Terbish is graduate student; Dr. Choi is graduate student; and Dr. Park is Professor, Department of Orthodontics, Institute of Craniofacial Deformity; Dr. Yi is Emeritus Professor, Department of Oral and Maxillofacial Surgery; and Dr. Cha is Associate Professor, Department of Orthodontics, Institute of Craniofacial Deformity, College of Dentistry, Yonsei University, Seoul, South Korea. Submitted April 2014; Revised July 2014; Accepted July 2014. Address correspondence to: Dr. Jung-Yul Cha, Department of Orthodontics, Yonsei University College of Dentistry, 50 Yonseiro, Seongsanno, Seodaemun-gu, Seoul 120–752, South Korea. E-mail [email protected]. DOI: 10.1597/14-105 95

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TABLE 1

Distraction Protocols and Stability of Alveolar or Premaxillary Distraction Osteogenesis for Distraction Appliances

Author

Appliances

Dolanmaz et al. (2003b) Erverdi et al. (2012) Karakasis and Hadjipetrou (2004) Iida et al. (2005) Choi et al. (2012) Meling et al. (2011) Picard et al. (2011)

Intraoral, Intraoral, Intraoral, Intraoral, Intraoral, RED RED RED

tooth-borne tooth-borne bone-borne bone-borne bone-borne

Amount of Distraction (mm)

Latency Period (d)

Consolidation Period (wk)

Follow-up Period (y)

Relapse (mm)

5–11.5 14 8 11 10.1 7.4 14–28 8.5–24

3 4 4 7 5–7 5–7 5 3

2 8 10 12 4 4 3–31 20

1 5 – – 1 1 – 1

No relapse – – – 2.6 2.3 – –

Type of Study Original article Case report Case report Original article Original article Original article Original article

* RED ¼ rigid external distraction device; – indicates no quantitative information.

bone 8 to 10 mm (Table 1). However, the appliance is expected to increase the load onto the anterior teeth and may exert a significant dental effect (Dolanmaz et al., 2003b; Iida et al., 2007). Therefore, a bone-borne intraoral appliance was introduced to provide effective distraction without rotation of the maxillary segment (Karakasis and Hadjipetrou, 2004; Iida et al., 2007). This intraoral appliance can be maintained during the consolidation period to prevent relapse because patients tolerate it with fewer psychosocial problems than the RED (Mitsukawa et al., 2010). In some cases, anterior movement of the premaxilla segment has been reported to relapse after distraction due to uncontrolled tipping of the anterior segment (Choi et al., 2012). This problem can be prevented by stabilizing the distractor with skeletal anchorage (Niederhagen et al., 1999) or controlled using a rigid external device (Table 1) (Posnick and Ewing, 1990; Ko et al., 1999; Yun and Park, 2011). However, whether premaxillary DO is useful for creating sufficient alveolar bone space with new bone formation and whether the quality of the newly formed bone is acceptable for implantation in patients with cleft lip and palate are uncertain (Nosaka et al., 2000). Previous reports have not shown the long-term stability of boneborne appliances. In addition, the final prosthetic treatment of the distracted area has not been reported well (Table 1). Even when alveolar distraction is performed, the transverse discrepancy of the maxilla remains. Surgical expansion can be considered in this situation but is limited due to the fibrous scars left from the previous reconstruction of the alveolar cleft. Relapse increases due to heavy scarring, which also limits the anterior movement of maxilla. This case report describes a patient with cleft lip and palate treated with alveolar DO and transverse control with transpalatal arch, followed by implantation in the regenerated alveolar bone. CASE REPORT Patient and Methods A 25-year-old man presented with history of a repaired unilateral left cleft lip and palate. The upper

right lateral incisor and first premolar and the upper left lateral incisor, canine, and second premolars were missing in the panoramic radiograph. At the referring hospital, soft palate surgery and cheilorrhaphy were performed to correct the cleft lip deformity. Bone graft surgery at the cleft site failed. A massive scar formed in the palatal area as a result of the previous surgical approach. The patient had an edentulous space of about 4.2 mm in the narrow V-shaped dental arch. The patient presented with a severe class III malocclusion with an orthodontist judgment of 13 mm (Fig. 1) and sagittal and vertical maxillary deficiencies. The maxilla was retruded, the upper incisors were uprighted, and the lower incisors were tipped lingually (Table 2). The patient was diagnosed with skeletal class III with unilateral cleft lip and palate (Fig. 1). Treatment Progress Surgery was performed under oral endotracheal anesthesia. A skin incision was made along the old scar of the exposed alveolar cleft site with a curette and a periosteal elevator. Dissection was performed to free the nasal and oral mucosa from the muscle layer, and the nasal floor was reconstructed. After freeing the mucoperiosteal flap, the periosteum was sutured with 4-0 Vicryl (Ethicon, Belgium). At the same time, the other surgical team obtained iliac bone for grafting. When the iliac bone crest was exposed, a 1.5 3 1 3 1-cm trapezoid-shaped block bone was harvested with a reciprocating saw. The block was inserted in the nasal floor and packed around the alveolar cleft site. The labial vestibule was sutured around the alveolar cleft site and upper lip area with 5-0 Vicryl (Ethicon), and the periosteum and cartilage layer were sutured with 2-0 Vicryl (Ethicon). Osteotomy After a 1-year healing period, a surgical operation for premaxillary DO was planned. The vestibular incision was made horizontally in the upper labial vestibule above the mucogingival junction that extended from the upper

Terbish et al., PREMAXILLARY DISTRACTION OSTEOGENESIS FOR UNILATERAL CLEFT LIP AND PALATE

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FIGURE 1 Pretreatment facial and intraoral photographs and lateral cephalometric radiograph. The upper right lateral incisor and first premolar and the upper left lateral incisor, canine, and second premolar were missing, and cleft lip and palate were observed in the photographs. The maxilla was retruded, the upper incisors were uprighted, and the lower incisors were inclined lingually.

right first premolar to the second premolar. The margins of the superior flap were raised to expose the lateral and anterior walls of the maxilla. A horizontal bone incision approximately 1 cm in length was made using a saw blade with a malleable retractor in place to protect the nasal mucoperiosteum. A Le Fort I osteotomy and vertical osteotomy were performed to separate the anterior segment of the maxilla, and a vertical osteotomy was performed perpendicular to the horizontal line on the distal sides of the upper right canine and upper left first premolar with a fissure bur and straight osteotomy. We noticed free movement of the premaxilla, and the flap was sutured with 3-0 and 4-0 Vicryl (Ethicon).

We fixed the ready-made intraoral device with eight selftapping screws (diameter ¼ 1.8 mm; length ¼ 8 mm; Orlus, Seoul, South Korea) to the palate. Distraction force was applied according to the following protocol (Fig. 2g). Distraction Procedure After a 7-day latency period, distraction was allowed to continue for 20 days at a rate of 0.5 mm/d followed by a 3-month consolidation period (Figs. 2 and 3, postDO). When the premaxilla was distracted about 10.0 mm, distraction was stopped. A space maintainer was delivered to the upper arch after removing the appliance to prevent immediate dental relapse.

Distractor Design Using the dental cast model, the location of the distractor was determined and the jackscrew (Dentarum Co., Ispringen, Germany) was placed parallel to the occlusal plane and positioned at the center of the palate. TABLE 2

Orthodontic Treatment After DO and Orthognathic Surgery After consolidation, orthodontic treatment began with a self-ligating bracket (0.022-inch slots, Damon-3,

Cephalometric Measurements*†

Measurement

Mean

Pre–DO

Post–DO

Consolidation

Postsurgery

Deband

1 y Posttreatment

SNA SNB ANB Wits MP angle N-A N-ANS U1 to SN SPACE IMPA E-Upper lip E-Lower lip

82.4 80.4 2.0 –2.0 32.0 –1.0 60.0 107.0 – 95.0 1.0 2.0

71.5 86.6 –15.1 –16.7 16.4 –12.4 58.2 96.6 4.2 68.7 –5.3 1.4

78.5 83.2 –4.7 –7.8 19.2 –4.4 54.4 116.2 12.2 69.9 –4.0 –2.6

77.6 82.9 –5.2 –9.4 22.1 –5.4 54.7 100.2 10.2 71.1 –1.2 –9.4

77.1 76.9 0.3 –5.0 26.9 –6.1 56.1 96.4 10.5 77.8 –4.8 –3.8

76.8 76.7 0.2 –4.4 30.9 –6.6 56.6 97.5 – 75.9 –3.3 –1.0

77.5 77.3 0.2 –6.6 32.0 –5.7 56.0 99.0 – 78.6 –3.0 –1.1

* DO ¼ distraction osteogenesis. † – indicates no quantitative information. SNA, Sella-Nasion-A point angle; SNB, Sella-Nasion-B point angle; ANB, A point-nasion-B point angle; MP angle, mandibular plane angle; N-A, horizontal distance of A point from perpendicular line of nasion, in mm; N-ANS, Vertical distance of anterior nasal spine from nasion, in mm; U1 to SN, upper incisor angulation to sella to nasion plane; IMPA, incisor mandibular plane angle; E-Upper lip, upper lip protrusion from Rickett s E line; E-Lower, lower lip protrusion from Rickett’s E line.

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FIGURE 2 Facial and intraoral photographs of treatment progress. In the post-DO photograph, a jackscrew was placed parallel to the occlusal plane and positioned at the center of the palate (g). After a 7-day latency period, distraction was allowed for 20 days at the rate of 0.5 mm/d, followed by a 3month consolidation period.

FIGURE 3 Cephalometric and panoramic x-ray of treatment progress. Missing space was regained during DO procedure and alveolar bone was leveled for aligning the teeth.

Terbish et al., PREMAXILLARY DISTRACTION OSTEOGENESIS FOR UNILATERAL CLEFT LIP AND PALATE

TABLE 3

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Analysis of Arch Width During Treatment Progress and Retention Period (unit, mm) Pre-DO*

Post-DO

Deband

1 y Posttreatment

Measurement

Mx

Mn

Mx

Mn

Mx

Mn

Mx

Mn

Inter–first premolar width Inter–first molar width

26.3 33.2

31.8 43.4

29.0 34.7

32.8 42.1

28.0 40.8

32.4 42.5

26.8 41.2

33.3 42.4

* DO ¼ distraction osteogenesis.

Ormco, Glendora, CA). The leveling and alignment initiated by 0.014 copper-nickel-titanium (CuNiTi, Ormco) was followed by 0.014 3 0.025 CuNiTi, 0.019 3 0.025 titanium molybdenum alloy, and stainless steel in arch wires. Closed coil springs were delivered to consolidate the space between the maxillary right second premolar and the central incisor and between the left second premolar and the lateral incisor by DO. A fullsized arch wire was inserted to control the torque of the upper incisors, and a transpalatal arch (TPA) was placed in the upper molars for arch expansion. After the preoperative orthodontic treatment was achieved, mandibular setback was performed by intraoral vertical ramus osteotomy. Implantation and Prosthodontic Treatment Cone-beam computed tomography (CT) imaging (Ray, Kyunggi Province, South Korea) was performed to evaluate bone quantity and the quality of the newly formed alveolar maxillary ridge areas. The maxillary right first premolar area distracted by DO was not appropriate for dental implantation due to distal curvature of the maxillary left canine root, and the maxillary left lateral incisor area was not appropriate for the dental implant due to lack of space and a vertical defect resulting from incomplete bone grafting. A threedimensional guided splint (OnDemand 3D, Cybermed, Seoul, South Korea) was used to implant the left second premolar area of the maxilla. Finally, fixed partial dentures were installed from the right lateral incisor to the first premolar and from the left central incisor to the

FIGURE 4

canine. The total active treatment time was 3.9 years (Figs. 2 and 3). RESULTS The skeletal measurements are shown in Table 2. After the 3-month consolidation period, the A-point moved forward 7.0 mm during the premaxillary DO. However, it also moved backward about 0.3 mm between the post DO and the 1-year follow-up period. The vertical height of the A-point (N-ANS) decreased 3.8 mm during DO and moved downward about 1.6 mm by the 1-year follow-up (Figs. 2 and 3). For the dental measurements, the U1 to SN increased about 19.68 during the DO procedure, then recovered during the retention period (Fig. 1; Table 2). The alveolar space significantly increased during the distraction period by 8 mm but relapsed during the retention period by about 2 mm. At the end of the orthodontic treatment, a proper overjet (2 mm) and overbite (2.2 mm) were achieved. Before treatment, the inter–first premolar width and inter–first molar width were 26.3 and 33.2 mm, respectively. After DO, they had increased by 29.0 and 34.7 mm, respectively. By expanding the upper arch with a self-ligation bracket and precision TPA, the inter–first premolar width and inter–first molar width of the maxilla had changed to 28.0 and 40.8 mm, respectively. One year posttreatment, they had changed to 26.8 and 41.2 mm, respectively (Table 3). The posttreatment facial intraoral and panoramic photographs (Fig. 4) show an improved facial profile, especially midface, and occlusion after prosthodontic

Posttreatment facial and intraoral photographs and lateral cephalometric radiograph.

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FIGURE 5 Computed tomographic images for evaluating bone continuity and quality of distracted alveolar ridge and locating the implant. After the distraction procedure, alveolar ridges on maxillary first left premolar area were used for the implantation.

treatment. In the CT scan, alveolar bone ridges were created for prosthetic replacement of the upper right lateral incisor and left first premolar. In the upper left first premolar area, an implant fixture was placed by a guided implant system (Fig. 5). Facial and intraoral photographs 1 year posttreatment show good stability of the treatment outcome. The occlusion was also maintained and there were few dental or skeletal changes. Superimposition of the lateral

cephalograms of the overall treatment and retention periods shows stable positioning of the premaxillary area (Fig. 6). DISCUSSION In this case, the A-point of the maxilla moved forward 7.0 mm during the DO period. This result was improved from that of a previous report of premaxillary DO. Bengi et

FIGURE 6 Superimposition of a: pre-DO and consolidation and b: deband and one-year posttreatment.

Terbish et al., PREMAXILLARY DISTRACTION OSTEOGENESIS FOR UNILATERAL CLEFT LIP AND PALATE

al. (2004) and Dolanmaz et al. (2003a) reported that the ANS moved forward 4 to 6 mm after premaxillary DO using the tooth-borne type. Iida et al. (2007) reported that the ANS moved anteriorly about 8 mm using a bone-borne type distractor, but they did not show long-term stability and final occlusion (Dolanmaz et al., 2003b; Tong et al., 2003; Bengi et al., 2004). Consequently, the extent of premaxillary movement was larger in the bone-borne type than in the tooth-borne type, and the present case also achieved a higher skeletal effect in the bone-borne–type distractor. To increase skeletal change, a bone-borne type of distractor was used (Dolanmaz et al., 2003a; Karakasis and Hadjipetrou, 2004; Iida et al., 2007), which is favorable when applied to the maxillary palate where fibrous scars remain. This also reduces the tendency for relapse associated with soft tissue regeneration over the long term (Liou et al., 2000; Bengi et al., 2004; Karakasis and Hadjipetrou, 2004). Despite the skeletal anchorage, rotation of the anterior segment was observed during the DO procedure. The U1 to SN increased 19.68 from the pre-DO to post-DO period in this patient. The patient’s profile had worsened due to excessive proclination of the upper anterior teeth during the distraction. The maxillary anterior segment was rotated counterclockwise: therefore, point A moved less than the anterior dentition. The rotation of the anterior segment was determined from the relationship between the line of the distraction force and the segmental center of mass (Dolanmaz et al., 2003b; Bengi et al., 2004; Karakasis and Hadjipetrou, 2004). The direction of force applied can be controlled by combining the application of a rigid external distractor appliance and an intraoral device (Ahn et al., 1999; Karakasis and Hadjipetrou, 2004). An alveolar space with new bone was created, and the bone quality and space was acceptable for implantation. As in previously reported cases, the distracted alveolar bone was confirmed as being of good quantity by histological and radiographic analyses, and proper osseointegration could be achieved with implants placed into the regenerated bone (Nosaka et al., 2000). In this case, the width of the alveolar bone and sufficient bone quality was also confirmed with three-dimensional CT (Fig. 5). Significant expansion of the intermolar width on the upper was observed during orthodontic treatment with the help of TPA and a self-ligating bracket system (Block and Brister, 1994; Damon, 1998; Kessler et al., 2001). The amount of expansion of the upper arch was about 1.7 mm in the premolar area and about 7.6 mm in the molar area. Patients with cleft lip and palate normally require a quad helix or rapid palatal expansion in order to expand the maxillary arch (Tong et al., 2003; Bengi et al., 2004). Fibrous scarring on the hard palate induced by the surgical approach for the expansion of the maxilla was a limitation in this case (Tong et al., 2003).

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For the dental expansion, a light force can be applied with a self-ligation bracket, which exerts a lateral expansion force until the dentition reaches its new equilibrium position (Cordasco et al., 2012). There are reports showing stable premolar and molar widths after expansion of both arches in nonextraction cases, even 6 to 8 years postretention (BeGole et al., 1998). However, arch expansion by the self-ligation bracket system is still controversial for both the stability and bone support of the buccal alveolus (Basciftci et al., 2014). Our results showed as light constriction of the maxillary premolar width after treatment, which resulted from relapse related to fibrous scars on the palate. The relapse of the upper expansion was minimal due to compliance of the retainer for a long period (Cattaneo et al., 2011). The reason for retardation of maxillary growth in patients with cleft lip and palate is unknown. The reason may be related to external factors such as timing and the method of operation for the organization of the alveolar cleft defect. In this case, severe deficiency of the maxilla with class III malocclusion resulted from fibrous scars on the palate as well as the cleft lip (Ross, 1970). For this reason, mandibular setback was surgically treated to correct the remaining jaw discrepancy. This combination of premaxillary DO and mandibular setback is the treatment of choice for a severe cleft deformity instead of total advancement of the maxilla only. The A-point moved back after surgery, as seen in 25% of the recurrences within 6 months after DO. The outcome of this case remained stable after 1 year, although more follow-up is necessary to determine longterm stability. A rigid retainer and a sufficient retention period are recommended for stability of distracted premaxilla. CONCLUSIONS Upper molar width expanded significantly during orthodontic treatment with TPA and a self-ligating bracket system. New bone created by alveolar distraction was acceptable for a prosthodontic implant. Therefore, premaxillary DO and arch expansion is an effective treatment strategy to improve function, aesthetics, and stability in cleft patients with multiple missing teeth. REFERENCES Ahn JG, Figueroa AA, Braun S, Polley JW. Biomechanical considerations in distraction of the osteotomized dentomaxillary complex. Am J Orthod Dentofacial Orthop. 1999;116:264–270. Basciftci FA, Akin M, Ileri Z, Bayram S. Long-term stability of dentoalveolar, skeletal, and soft tissue changes after non-extraction treatment with a self-ligating system. Korean J Orthod. 2014;44:119–127. BeGole EA, Fox DL, Sadowsky C. Analysis of change in arch form with premolar expansion. Am J Orthod Dentofacial Orthop. 1998;113:307–315.

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Premaxillary Distraction Osteogenesis Using an Intraoral Appliance for Unilateral Cleft Lip and Palate: Case Report.

Premaxillary distraction osteogenesis was introduced using intraoral devices to correct maxillary hypoplasia and lengthen the alveolar bone horizontal...
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