CRANIOMAXILLOFACIAL DEFORMITIES/COSMETIC SURGERY

Le Fort IV + I Distraction Osteogenesis Using an Internal Device for Syndromic Craniosynostosis Yoshiaki Sakamoto, MD,* Hideo Nakajima, MD,y Ikkei Tamada, MD,z and Teruo Sakamoto, DDSx Monobloc and Le Fort III distractions can improve midfacial hypoplasia, a characteristic feature of syndromic craniosynostosis. The purpose of treating midfacial hypoplasia is to improve exophthalmos and dental occlusion. Typically, in Le Fort III or monobloc distractions, the midface is mobilized en bloc, and the extent and direction of the mobilization is determined according to the preferred intermaxillary occlusion. However, to obtain the preferred functional and esthetic results while correcting midface hypoplasia, the most sensible approach is the use of different degrees of mobilization and vectors for the upper and lower halves of the midface. This report describes the case of an adolescent with Crouzon syndrome showing frontal recession exophthalmos and an anterior crossbite. His condition was treated with monobloc minus Le Fort I and Le Fort I distraction using only internal devices, which the authors have designated Le Fort IV plus I distraction. Ó 2014 American Association of Oral and Maxillofacial Surgeons J Oral Maxillofac Surg 72:788-795, 2014 Monobloc and Le Fort III advancements can improve midfacial hypoplasia, a characteristic feature of syndromic craniosynostosis.1,2 The purpose of treating midfacial hypoplasia is to improve exophthalmos and dental occlusion. Typically, for Le Fort III or monobloc advancements, the disadvantages are significant blood loss, a long operation time, the need for a bone graft, postoperative relapse, and, occasionally, cerebrospinal fluid leaks caused by immediate advancement.3 Distraction osteogenesis can overcome such disadvantages by gradual advancement and the generation of new bone, and this procedure has become widely used for the craniofacial area.4 In conventional advancement and distraction osteogenesis, the midface is mobilized en bloc, and the amount and direction of the mobilization is determined according to the preferred intermaxillary occlusion. However, to obtain the preferred functional and

esthetic results while correcting midface hypoplasia, the most sensible approach involves the use of different degrees of mobilization and vectors for the upper and lower halves of the midface. This report describes the case of an adolescent with Crouzon syndrome showing frontal recession exophthalmos and an anterior crossbite. To treat his condition, the authors performed monobloc minus Le Fort I and Le Fort I distraction using only internal devices, which they have designated Le Fort IV plus I distraction.

Report of Case A 15-year-old patient with Crouzon syndrome refused to go to school because of severe midfacial hypoplasia. He had undergone fronto-orbital advancement and cranial reshaping at 2 years of age and subsequent Le Fort III distraction osteogenesis at 6 years of

*Assistant Professor, Department of Plastic and Reconstructive

Address correspondence and reprint requests to Dr Y. Sakamoto:

Surgery, Keio University School of Medicine, Tokyo, Japan. yAssistant Professor, Department of Plastic and Reconstructive

Department of Plastic and Reconstructive Surgery, Keio University School of Medicine, 35 Shinanomachi, Shinjuku Ward, Tokyo 160-

Surgery, Keio University School of Medicine, Tokyo, Japan.

8582, Japan; e-mail: [email protected]

zAssistant Professor, Department of Plastic and Reconstructive

Received July 22 2013

Surgery, Tokyo Metropolitan Children’s Medical Center, Tokyo,

Accepted September 24 2013

Japan.

Ó 2014 American Association of Oral and Maxillofacial Surgeons

xAssistant Professor, Department of Orthodontics, Tokyo Dental

0278-2391/13/01234-2$36.00/0

College, Chiba, Japan.

http://dx.doi.org/10.1016/j.joms.2013.09.041

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FIGURE 1. A, The patient immediately after Le Fort III advancement at 3 years of age. B, Lateral, C, frontal, and D, occlusal views at 15 years of age. (Fig 1 continued on next page.) Sakamoto et al. Distraction Osteogenesis for Craniosynostosis. J Oral Maxillofac Surg 2014.

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FIGURE 1 (cont’d). (Fig 2 continued on next page.) Sakamoto et al. Distraction Osteogenesis for Craniosynostosis. J Oral Maxillofac Surg 2014.

age. After the latter procedure, his exophthalmos substantially improved, and good esthetic results were obtained. However, by the time he was 15 years of age, the frontal and midfacial hypoplasia had relapsed, and severe exophthalmos and malocclusion with an

open bite were noticed (Fig 1). Monobloc advancement was planned to treat his condition. However, the preoperative cephalometric analysis showed that a reasonable amount of horizontal advancement for the inferior orbital rim, nose, and malar complex was

FIGURE 2 (cont’d). Preoperative A, 3-dimensional computed tomogram and B, cephalogram. C, Three-dimensional computed tomogram and D, cephalogram during the consolidation period. Red and yellow arrows represent Le Fort IV, and blue arrows represent Le Fort I. E, Three-dimensional computed tomogram and F, cephalogram 2 years after removal of the devices. (Fig 2 continued on next page.) Sakamoto et al. Distraction Osteogenesis for Craniosynostosis. J Oral Maxillofac Surg 2014.

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FIGURE 2 (cont’d). (Fig 2 continued on next page.) Sakamoto et al. Distraction Osteogenesis for Craniosynostosis. J Oral Maxillofac Surg 2014.

10 mm. Meanwhile, the horizontal and vertical corrections needed for malocclusion were 16 and 6 mm, respectively. Thus, the amount of distraction was different for these 2 corrections. Hence, the authors decided to perform a distraction by Le Fort I for the lower part and by Le Fort III combined with the frontal bone for the upper part. Coronal and upper gingivobuccal sulcus incisions allowed access to the anterior cranial vault and midface. A bifrontal craniectomy was performed, and the frontal bone was removed. After orbital wall osteotomies, an additional pterygomaxillary disjunction was performed, and mobilization of the en bloc was confirmed. Then, a Le Fort I osteotomy with downfracture was performed. The removed frontal bone and the upper part were fixed with titanium plates and screws. Then, a pair of

internal distraction devices (NAVID System; Medical U&A, Osaka, Japan) was symmetrically positioned bilaterally at the supraorbital bandeau and zygoma. The maxilla also was attached using a pair of internal distraction devices. The route was from the temporal fossa, under the zygomatic arch, between the coronoid process and sphenoid bone, and then the zygomatic-alveolar crest.5 Distraction vectors for the upper and lower parts were set in an anteroinferior direction according to the preoperative cephalometric analysis. Gradual distraction was initiated 1 week after surgery at a rate of 1.0 mm/day. Lateral cephalographic and occlusal assessments were performed every week. With gradual Le Fort I advancement, the maxilla was rotated counterclockwise. Then, an elastic band for maxillary traction was applied. After the distraction

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FIGURE 2 (cont’d). (Fig 2 continued on next page.) Sakamoto et al. Distraction Osteogenesis for Craniosynostosis. J Oral Maxillofac Surg 2014.

was completed, the distraction shaft extending outside the scalp was cut as short as possible by a large pin cutter for orthopedic surgery, and the consolidation period was initiated. There were no special restrictions for the patient’s daily activity. The patient was allowed to bathe and wash his hair. At the end of the 1-year consolidation period, the distraction devices were removed under general anesthesia. The patient underwent a Le Fort IV plus I distraction without any intraoperative complications. After finishing the Le Fort IV distraction for 10 mm, the Le Fort I was continued to 18 mm (Fig 2). During the consolidation period, no portion of the distracters was visible; the appearance was cosmetically acceptable, such that the patient was able to attend school. At 2 years after removing the distractor, the relapse rate was only 6.7% at the nasion and 8.6% at point A,

and the open bite and Class III malocclusion continued to show improvement. The patient and his family were pleased with the functional and esthetic surgical results (Fig 3).

Discussion In conventional procedures for improving midface hypoplasia, Le Fort I, II, and III and monobloc advancement are the commonly used surgical procedures. Separate advancement of the upper and lower parts of the midface has conventionally been emphasized as the best means to obtain not only the most desirable esthetic results, but also functional intermaxillary occlusion.6 Based on this concept, several reports have described the use of dual midfacial osteotomies.7,8 However, no study has been published on monobloc

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FIGURE 2 (cont’d). Sakamoto et al. Distraction Osteogenesis for Craniosynostosis. J Oral Maxillofac Surg 2014.

minus Le Fort I plus Le Fort I osteotomies. In the present procedure, the midface did not undergo monobloc distraction but was separated, and the osteotomy line was an upper one rather than Le Fort III. To distinguish the conventional monobloc osteotomy from the present dual osteotomy, the authors have called this procedure Le Fort IV. The use of distraction osteogenesis for midfacial hypoplasia has been an important tool for dual osteotomy. In addition, the dual distraction procedure uses an external distraction device with or without internal distraction devices.7,8 However, to the best of the authors’ knowledge, no report has described dual midfacial distraction using only internal distraction devices. Previous internal devices have not allowed for 3-dimensional control during distraction. Hence, halo distraction has been widely performed because the external device permits vector changes during the advancement phase. The NAVID System used for the present procedure is an internal device that permits 3-dimensional control. The maxillary distractor in the NAVID System uses a route-and-tilt method. Patients with craniofacial dysostosis have

not only a retrusive but also a vertically short maxilla, resulting in an anterior open bite. Therefore, the direction of the distraction in the maxilla is naturally suitable for treating craniofacial dysostosis. Of course, with distraction, counterclockwise rotation was observed, but the NAVID System has a 3-dimensional joint and can be corrected by elastic intermaxillary fixation.5 Furthermore, the result is cosmetically acceptable because the distractor rods can be cut after the distraction period, and no part is visible during the consolidation period. Therefore, patients can tolerate a long consolidation period more easily, thereby preventing relapse. The relapse ratio reported for the halo and other distraction devices ranges from 15% to 25%.9,10 Hence, the present 8.6% relapse rate was much lower than for previous devices. In conclusion, Le Fort IV plus I for dual distraction using angle-variable internal devices is a useful procedure because of the 3-dimensional control of the external device, the esthetic and functional outcomes, the lack of impact on the patient’s social life during the consolidation period, and the low relapse rate.

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FIGURE 3. A, Lateral, B, frontal, and C, occlusal views 2 years after removal of the devices. Sakamoto et al. Distraction Osteogenesis for Craniosynostosis. J Oral Maxillofac Surg 2014.

References 1. Tessier P: The definitive plastic surgical treatment of the severe facial deformities of craniofacial dysostosis: Crouzon’s and Apert’s diseases. Plast Reconstr Surg 48:419, 1971 2. Ortiz-Monasterio F, del Campo AF, Carrillo A: Advancements of the orbits and the midface in one piece, combined with frontal repositioning, for the correction of Crouzon’s deformities. Plast Reconstr Surg 61:507, 1978

3. Fearon JA, Whitaker LA: Complications with facial advancement: A comparison between the Le Fort III and monobloc advancements. Plast Reconstr Surg 91:990, 1993 4. Fearon JA: The Le Fort III osteotomy: To distract or not to distract? Plast Reconstr Surg 107:1091, 2001 5. Nakajima H, Sakamoto Y, Tamada I, et al: An internal distraction device for Le Fort distraction osteogenesis: The NAVID system. J Plast Reconstr Aesthet Surg 65:61, 2012 6. Obwegeser HL: Surgical correction of small or retrodisplaced maxillae. The ‘ dish-face’’ deformity. Plast Reconstr Surg 43: 351, 1969

SAKAMOTO ET AL 7. Satoh K, Mitsukawa N, Hosaka Y: Dual midfacial distraction osteogenesis: Le Fort III minus I and Le Fort I for syndromic craniosynostosis. Plast Reconstr Surg 111:1019, 2003 8. Paliga JT, Goldstein JA, Storm PB, et al: Monobloc minus Le Fort II for single-stage treatment of the Apert phenotype. J Craniofac Surg 24:596, 2013

795 9. Cho BC, Kyung HM: Distraction osteogenesis of the hypoplastic midface using a rigid external distraction system: The results of a one- to six-year follow-up. Plast Reconstr Surg 118:1201, 2006 10. Saltaji H, Major MP, Altalibi M, et al: Long-term skeletal stability after maxillary advancement with distraction osteogenesis in cleft lip and palate patients. Angle Orthod 82:1115, 2012

Le Fort IV + I distraction osteogenesis using an internal device for syndromic craniosynostosis.

Monobloc and Le Fort III distractions can improve midfacial hypoplasia, a characteristic feature of syndromic craniosynostosis. The purpose of treatin...
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