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7. Pownell PH, Brown OE, Pransky SM, Manning SC: Congenital abnormalities of the submandibular duct. Int J Pediatr Otorhinolaryngol 24:161, 1992 8. Batsakis JG, Mackay B, Ryka AF, Seifert RW: Perinatal salivary gland tumours (embryomas). J Laryngol Otol 102:1007, 1988 9. Batsakis JG, Frankenthaler R: Embryoma (sialoblastoma) of salivary glands. Ann Otol Rhinol Laryngol 101:958, 1992 10. Hsueh C, Gonzalez-Crussi F: Sialoblastoma: A case report and review of the literature on congenital epithelial tumors of salivary gland origin. Pediatr Pathol 12:205, 1992 (published erratum in Pediatr Pathol 12:631, 1992) 11. Som PM, Brandwein M, Silvers AR, Rothschild MA: Sialoblastoma (embryoma): MR findings of a rare pediatric salivary gland tumor. Am J Neuroradiol 18:847, 1997
12. Brandwein M, Al-Naeif NS, Manwani D, et al: Sialoblastoma: Clinicopathological immunohistochemical study. Am J Surg Pathol 23:324, 1999 13. Vawter GF, Teftt M: Congenital tumors of the parotid gland. Arch Pathol 82:242, 1966 14. Taylor GP: Congenital epithelial tumor of the parotid sialoblastoma. Pediatr Pathol 8:447, 1988 15. Mostafapour SP, Folz B, Barlow D, Manning S: Sialoblastoma of the submandibular gland: Report of a case and review of the literature. Int J Pediatr Othorhinolaryngol 53:157, 2000 16. Scott JX, Krishnan S, Bourne AJ, Williams MP, Agzarian M, Revesz T: Treatment of metastatic sialoblastoma with chemotherapy and surgery. Pediatr Blood Cancer 2:March, 2006
J Oral Maxillofac Surg 66:126-132, 2008
Maxillary Anterior Segmental Advancement of Hypoplastic Maxilla in Cleft Patients by Distraction Osteogenesis: Report of 2 Cases Alper Alkan, DDS, PhD,* Burcu Bas¸, DDS,† Mete Özer, DDS, PhD,‡ Mehmet Bayram, DDS, PhD,§ and Emir Yüzbasıog˘lu, DDS, PhD储 Maxillary hypoplasia is a common deformity in patients with cleft lip and palate. Le Fort I osteotomy is one of the routine procedures for treatment of maxillary hypoplasia in cleft palate patients. In recent years, rigid external and internal distraction systems have gained popularity for improvement of severe maxillary hypoplasia. However, these techniques have the risk of velopharyngeal insufficiency by in-
Received from the Faculty of Dentistry, Ondokuz Mayıs University, Samsun, Turkey. *Formerly, Assistant Professor, Department of Oral and Maxillofacial Surgery; Currently, Assistant Professor, Faculty of Dentistry, Erciyes University, Kayseri, Turkey. †Research Assistant, Department of Oral and Maxillofacial Surgery. ‡Associate Professor, Department of Orthodontics. §Research Assistant, Department of Orthodontics. 储Former Research Assistant, Department of Prosthodontics. Address correspondence and reprint requests to Dr Bas¸: Department of Oral and Maxillofacial Surgery, Faculty of Dentistry, Ondokuz Mayıs University, 55139 Kurupelit, Samsun, Turkey; e-mail:
[email protected] © 2008 American Association of Oral and Maxillofacial Surgeons
0278-2391/08/6601-0020$34.00/0 doi:10.1016/j.joms.2006.10.033
creasing the nasopharyngeal distance. For this reason, advancement of anterior maxillary segment by distraction osteogenesis can be thought of as an alternative method that offers many advantages. In cleft lip and palate patients, early surgical corrections may result in poor skeletal and dental growth in the transverse and sagittal planes, especially in the maxilla.1 Maxillary advancement greater than 6 mm is often difficult to achieve in this group of patients because of maxillary scarring. A mean postoperative relapse of 20% to 25% has been documented with conventional orthognathic surgery.2 Distraction osteogenesis (DO) is a recent addition to the treatment modalities for reconstructing severe facial deformities. It was first used for correction of the craniofacial skeleton in the early 1990s.3 Rachmiel et al4 achieved maxillary advancement with DO in adult sheep, and an increasing number of studies have been reported about the advancement of the maxilla or midface region.4,5 Polley and Figueroa used adjustable rigid external distraction (RED) devices to advance the maxilla in children with cleft lip and palate and reported successful results.6 Today, RED systems and internal distractors are the most popular techniques for advancing the hypoplastic maxilla in cleft palate patients. Some limitations of the treatment are reported, such as exter-
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nal scarring and discomfort due to the head frame.7 In addition to these problems, sagittal advancement of the entire maxilla in cleft palate patients has the risk of shortening the soft palate and inducing velopharyngeal incompetence.8 In our experience, maxillary anterior segmental DO is used to treat cleft lip and palate patients with severe maxillary hypoplasia. It is proposed that maxillary anterior segmental DO is a useful alternative method to conventional osteotomy and RED systems for cleft lip and palate patients with severe velopharyngeal incompetence.
Report of Cases CASE 1 A 23-year-old woman who presented with severe maxillary hypoplasia, cleft lip and palate, was referred to our clinic. An 8.5-mm negative overjet was observed intraorally, and radiological evaluation showed that Class III malocclusion resulted from maxillary hypoplasia (Fig 1). She also had severe velopharyngeal incompetence that caused hypernasal speech. After lateral cephalometric analysis, segmental DO was planned to advance the anterior maxillary segment. The surgical procedure was performed under local anesthesia. The operation technique was similar to the Wasmund technique except for the palatal flap design. A horizontal incision was made from the vestibular sulcus, and a mucoperiosteal flap was dissected in a superior direction. Two mucoperiosteal tunnels were formed up to the alveolar crest on the location of vertical osteotomies. A horizontal osteotomy line, starting from the piriform aperture and passing approximately 4 to 5 mm above the dental root apices, and a vertical osteotomy line at the distal interdental region of the first premolars were outlined bilaterally with small burs and saws. At the palatal side only, a sulcular incision including premolar and molar teeth was made. Mucoperiosteal tunnels were formed up to the cleft, and osteotomy was then completed using fine osteotomes. Care was taken to keep palatal mucosa intact for vascularization. The mucoperiosteal flap was then closed primarily. After a 6-day latency period, a custom-made tooth-borne distractor (constructed with a Hyrax screw) was cemented to the teeth and then distraction was started. In order to prevent the increase of open bite, the distractor was designed with an angle of 10° to the occlusal plane for orientation of the maxilla in a forward and downward direction. The screw was activated 0.5 mm twice a day (1 mm/day) for 11 days. The procedure and the postoperative period were well tolerated by the patient. At the end of the distraction period, 11 mm bilaterally sagittal advancement of the anterior maxillary segments was achieved, and the anterior cross bite was eliminated (Fig 2). Three months later, the distractor was removed, and the fixed orthodontic treatment was applied in both arches. The alveolar cleft was closed with a tibial bone graft, and the occlusion was rehabilitated with prosthetic reconstruction at the end of the eighth month. The movement of the anterior segment also improved the upper lip and nasal profile. Satisfactory esthetic improvement was obtained both extraorally and intraorally (Fig 3). The patient last visited our clinic 2 years after the surgery without evidence of recurrence.
CASE 2 An 18-year-old woman was referred to our clinic for the evaluation of cleft lip and palate, severe open bite, and maxillary hypoplasia. Bilateral alveolar cleft continuity with a large palatal fistula was observed in the oral examination, and the premaxilla was mobile (Fig 4). Radiological examination showed that the retroposition of the upper jaw in relation to the mandible with a preoperative overjet of ⫺4.5 mm and open bite was more severe than that in case 1. The operation procedure and the distraction device used here are similar to those of the first patient (Fig 5). Unlike the first case, the distractor was cemented to all the teeth in the maxilla. At the end of the distraction period, 11-mm sagittal advancement of the anterior maxillary segment was achieved (Fig 6). Also, the large palatal fistula was reduced at the end of the distraction period due to the anteromedial movement of the segments. A second operation after the removal of the distractor was performed in order to close small residual fistula with adjacent mucosal flaps. Bilateral distraction spaces in the maxillary arch were closed with the anterior movement of the first and second molars by using reverse headgear (Fig 7). During the fixed orthodontic treatment, intraoral vertical elastics were used to reduce the open bite. Alveolar cleft surgery and prosthetic reconstruction were performed after the elimination of the open bite (Fig 8). Satisfactory esthetic improvement was obtained both extraorally and intraorally. Two-year follow-up showed no signs of recurrence.
Discussion Le Fort I osteotomy has become a routine procedure over the last half of the 20th century as a definite treatment for midface hypoplasia in cleft patients.9 The surgical difficulties of using conventional osteotomy to transpose the maxillary segments in cleft lip and palate patients are related to the severity of scarring from cleft palate repair, the less predictable vascular supply, the extent of advancement, the fixation of the transposed segments, and the possibility of postsurgical relapse.10 Alterations in velopharyngeal function, especially in borderline cases, is another disadvantage of conventional Le Fort I osteotomies. Several articles have described deterioration in both velopharyngeal closure and speech after maxillary advancement with conventional osteotomies.11,12 Alternatively, Posnick and Tompson performed maxillary segmental osteotomies for maxillary hypoplasia in cleft patients.13 However, literature has pointed out the possible complications of segmental maxillary osteotomies in cleft patients due to blood supply problems.14 In the present case reports, flap design that needs minimal incision and flap reflection overcome this problem by keeping and expanding an adequate intact mucosa for vascularization. DO in the maxilla has several advantages when compared with the conventional Le Fort I and segmental osteotomy. In this method, osteotomized bone segments are distracted gradually at a regular interval of 0.5 to 1 mm/day after surgery, and the
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FIGURE 1. Preoperative intraoral and extraoral photographs of case 1. Alkan et al. Maxillary Hypoplasia in Cleft Patients. J Oral Maxillofac Surg 2008.
FIGURE 2. Intraoral and lateral cephalometric views, before and after distraction. Alkan et al. Maxillary Hypoplasia in Cleft Patients. J Oral Maxillofac Surg 2008.
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FIGURE 3. Final radiographic, occlusal, and extraoral views of case 1. Alkan et al. Maxillary Hypoplasia in Cleft Patients. J Oral Maxillofac Surg 2008.
FIGURE 4. Preoperative intraoral and extraoral views of case 2. Alkan et al. Maxillary Hypoplasia in Cleft Patients. J Oral Maxillofac Surg 2008.
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FIGURE 5. Schematic view of incision and bone cuts of the operation in case 2. Alkan et al. Maxillary Hypoplasia in Cleft Patients. J Oral Maxillofac Surg 2008.
callus is stretched with a distraction device to generate new bone formation.15 DO allows not only the lengthening of bone but also lengthening of the soft tissue over the bone. Cheung and Chua16 concluded that DO tends to be preferred to conventional osteotomy for younger cleft lip and palate patients with more severe deformities. In such cases, it was feasible to use distraction for advancement of the maxilla by either complete or incomplete Le Fort I osteotomy, and a concurrent mandibular osteotomy was less frequently required. Recently, adjustable rigid external distraction devices have been used for maxillary advancement, which was designed and reported by Polley and Figueroa in the 1990s.6 However, a major problem with current external devices is that they can create significant physical and social inconvenience to the patient. For this reason, various internal devices,
which are easily tolerated by patients, were developed by various companies.2 To date, clinical applications included total advancement of the maxilla or midface. Altuna et al5,15 first examined maxillary anterior segmental DO in cynomologus primates experimentally, and Dolanmaz et al17 used a tooth-borne device for maxillary anterior segmental advancement of a 42-year-old man with a Class III pattern. To the best of our knowledge, maxillary anterior segmental DO was first used by Karakasis and Hadjipetrou in a cleft patient to advance the hypoplastic maxilla.18 The investigators performed a 2-stage procedure. At first, they osteotomized the palate and closed the palatal fistula with adjacent mucosal flaps. After a recovery period of 3 weeks, they performed the vestibular osteotomies and used 2 intraoral bonesupported distractors to advance the maxilla. In contrast, we performed both palatal and vestibular osteot-
FIGURE 6. Intraoral and lateral radiographic views before and after distraction. Alkan et al. Maxillary Hypoplasia in Cleft Patients. J Oral Maxillofac Surg 2008.
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FIGURE 7. Preoperative and postoperative radiographs of case 2. Alkan et al. Maxillary Hypoplasia in Cleft Patients. J Oral Maxillofac Surg 2008.
omies in a single operation and advanced the maxilla with a tooth-borne-type distractor. We preferred to perform anterior segmental distraction before closing the palatal fistula and alveolar cleft. In this way, exposure of the surgical site could be achieved by minimal flap reflection during the osteotomy operation. Reflection of the whole palatal mucosa has the risk of losing blood supply at the operation site due to the scarred palatal tissue in the cleft area. Therefore, care
was taken to keep palatal mucosa intact for vascularization. Maxillary DO was shown to be an effective method for improving the soft tissue profile in cleft lip and palate patients.19 In the first case, movement of the anterior segment not only improved the upper lip but also affected the nasal profile by forward movement of the anterior nasal spine. However, in the second case, although the anterior advancement of lateral segments
FIGURE 8. Final intraoral and extraoral appearance of case 2. Alkan et al. Maxillary Hypoplasia in Cleft Patients. J Oral Maxillofac Surg 2008.
132 was achieved in the sagittal plane, the nasal spine did not advance proportionally as it was expected. Therefore, alar cartilages were not supported by the anterior nasal spine. As a result, the upper lip and nasal profile was not improved as in the first case, and open bite was increased. By this technique, anteroposterior movement of the segments is possible, but the main problem is how to prevent the increase of the open bite. In our opinion, bone-supported distractors could be more effective to achieve a symmetric movement of the anterior nasal spine. The patients described in this article had severe maxillary hypoplasia. To the best of our knowledge, this was the first use of maxillary anterior segmental DO with a tooth-borne distractor in cleft patients for advancement of hypoplastic maxilla. Treating this situation by routine orthognathic surgery or rigid external DO would increase the velopharyngeal insufficiency. Case 1 had velopharyngeal insufficiency from the outset, and the technique did not obviate the need for palatoplasty. However, total advancement of the maxilla could have made the velopharyngeal incompetence more severe, resulting in more complicated and advanced future surgical procedures. The technique was minimally traumatic when compared with other surgical alternative treatment methods. In conclusion, maxillary anterior segmental DO is an alternative technique to correct maxillary hypoplasia in cleft patients.
References 1. Tae KC, Gong SG, Min SK, et al: Use of distraction osteogenesis in cleft palate patient. Angle Orthod 73:602, 2003 2. Gateno J, Engel ER, Teichgraeber JF, et al: A new Le Fort I internal distraction device in the treatment of severe maxillary hypoplasia. J Oral Maxillofac Surg 63:148, 2005
MAXILLARY HYPOPLASIA IN CLEFT PATIENTS 3. McCarthy JG, Schreiber J, Karp N, et al: Lengthening the human mandible by gradual distraction. Plast Reconstr Surg 89:1, 1992 4. Rachmiel A, Potparic Z, Jackson IT, et al: Midface advancement by gradual distraction. Br Plast Surg 46:201, 1993 5. Block MS, Brister GD: Use of distraction osteogenesis for maxillary advancement: Preliminary results. J Oral Maxillofac Surg 52:282, 1994 6. Polley JW, Figueroa AA: Management of severe maxillary deficiency in childhood and adolescence through distraction osteogenesis with an external, adjustable, rigid distraction device. J Craniofac Surg 8:181, 1997 7. Kalk WW, de Lange J, Jansma J, et al: Distraction osteogenesis of the maxilla in cleft patients. Worth considering. Ned Tijdschr Tandheelkd 111:496, 2004 8. Spiessl B, Tschopp HM: Surgery of the jaws, in Naumann HH (ed): Head and Neck Surgery (vol 2). Stuttgart, Germany: Thieme, 1980, p 282-288. 9. Janulewics J, Costello BJ, Buckley MJ, et al: The effects of Le Fort I osteotomies on velopharyngeal and speech functions in cleft patients. J Oral Maxillofac Surg 62:308, 2004 10. Welch TB: Stability in the correction of dentofacial deformities: A comprehensive review. J Oral Maxillofac Surg 47:1142, 1989 11. Sugano K, Tsuneda K, Tanaka N: Velopharyngeal incompetence following maxillary advancement: A case report. Jpn J Plast Reconstr Surg 8:154, 1988 12. Witzel MA, Munro IR: Velopharyngeal insufficiency after maxillary advancement. Cleft Palate J 14:1761, 1977 13. Posnick JC, Tompson B: Cleft-orthognathic surgery: Complications and long-term results. Plast Reconstr Surg 96:255, 1995 14. Willmar K: On Le Fort I osteotomy: A follow-up of 106 operated patient with maxillofacial deformity. Scand J Plast Reconstr Surg Suppl 12:1, 1974 15. Altuna G, Walker DA, Freeman E: Surgically assisted rapid orthodontic lengthening of the maxilla in primates: A pilot study. Am J Orthod Dentofacial Orthop 107:531, 1995 16. Cheung KL, Chua HDP: A meta-analysis of cleft maxillary osteotomy and distraction osteogenesis. Int J Oral Maxillofac Surg 35:14, 2006 17. Dolanmaz D, Karaman AI˙, Durmus E, et al: Management of alveolar clefts using dento-osseous transport DO. Angle Orthod 73:723, 2003 18. Karakasis D, Hadjipetrou L: Advancement of the anterior maxilla by distraction (case report). J Craniomaxillofac Surg 32:150, 2004 19. Harada K, Baba Y, Ohyama K, et al: Soft tissue profile changes of the midface in patients with cleft lip and palate following maxillary distraction osteogenesis: A preliminary study. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 94:673, 2002
