Odontology (2010) 98:181184 DOI 10.

1007/s10266-010-0124-5

The Society of The Nippon Dental University 2010

CASE REPORT Akira Matsuo Michihide Kono Jun Toyoda Takayuki Nakai Masako Tsuzuki Hiroshige Chiba

Navigation surgery for Le Fort 1 osteotomy in a brous dysplasia patient

Received: August 25, 2009 / Accepted: February 7, 2010

Abstract Orthognathic surgery is sometimes performed for brous dysplasia to correct malocclusion or facial asymmetry. However, Le Fort 1 osteotomy for this disease is difcult because of severe anatomical abnormality. Computerassisted surgery is a rapidly developing technique in oral and maxillofacial surgery that is helping to ensure the safety of the surgery. We report a case of polyostotic craniofacial brous dysplasia in which two-jaw orthognathic surgery was performed using a navigation system with the Le Fort 1 osteotomy procedure. A 29-year-old woman presented with swelling and asymmetry on the right side of her face. Craniofacial brous dysplasia on the right side had been previously diagnosed, and she had undergone conservative surgery several times before. The disease extended to the right mandible, maxilla, and zygomatic, temporal frontal, and orbital areas, including the skull base. We rst performed conservative contouring around the frontal and orbital areas, and then Le Fort I osteotomy and sagittal split ramus osteotomy to correct the asymmetry and cant of the occlusal plane. A passive infrared navigation system (Vector Vision surgical navigation system) was used for the Le Fort I osteotomy. The postoperative course was stable, and the facial asymmetry and cant of the occlusal plane improved and remained suitable 2 years after surgery. Thus, Le Fort 1 osteotomy can be performed safely in brous dysplasia with the aid of a passive infrared navigation system. Key words Fibrous dysplasia Le Fort 1 osteotomy Navigation surgery

tive tissue, and it frequently affects craniofacial bone.1 In the treatment of craniofacial brous dysplasia, complete resection followed by immediate reconstruction or conservative surgery (curettage or contouring) is controversial.2 Although complete resection is difcult in polyostotic cases, severe facial asymmetry or malocclusion sometimes remains after conservative surgery only.3 Several reports have mentioned performing orthognathic surgery for brous dysplasia36 to correct these symptoms, but when Le Fort 1 osteotomy is performed to treat this disease, it is difcult to ensure safe surgery because of the severe anatomical abnormality.3 Computer-assisted surgery using images acquired by computed tomography (CT) or magnetic resonance imaging has recently been developed for cerebral, skull base, and sinus surgery.7 This procedure is more effective for patients in whom the normal anatomy has been altered, or for avoiding wide-open surgery.7 In craniofacial surgery, navigation surgery can also ensure safe surgery, even when the surgical site is in an unseen deep area.8 However, there are no reports of navigation surgery in a Le fort 1 osteotomy in a patient with brous dysplasia. We report a case of polyostotic craniofacial brous dysplasia in which two-jaw orthognathic surgery was performed using a passive infrared navigation system with Le Fort 1 osteotomy.

Case
A 29-year-old woman complaining of swelling and asymmetry on the right side of her face presented on 13 May 2005. She had received a diagnosis of brous dysplasia in childhood and had undergone conservative surgery several times. She had no relevant anamnesis or family history of disease. Her present status included marked bone swelling on the right side of her face and severe cant of the occlusal plane (Fig. 1a, c). Lateral cephalometric analysis showed SNA 86.2, SNB 79.2, and ANB 7.0. Severe asymmetry was observed on the frontal cephalogram (Fig. 1b). A bone scin-

Introduction
Fibrous dysplasia is a benign bone disease characterized by the replacement of normal bone with bro-osseous connecA. Matsuo (*) M. Kono J. Toyoda T. Nakai M. Tsuzuki H. Chiba Department of Oral and Maxillofacial Surgery, Tokyo Medical University, 6-7-1 Nishi-Shinjuku, Shinjuku-ku, Tokyo 160-0023, Japan Tel. +81-3-3342-6111 ext. 62725; Fax +81-3-3342-1723 e-mail matsuoam@tokyo-med.ac.jp

182 Fig. 1af. Clinical and roentgenological ndings before and after surgery. a Frontal view before surgery. b Frontal cephalogram. Arrows indicate the affected area. c Intraoral view before surgery. d Frontal view after surgery. e Frontal cephalogram after surgery. f Intraoral view after surgery

tigram (99mTc-methyl diphosphonate) showed high uptake in the right mandible, maxilla, and zygomatic temporal, frontal, and orbital areas, including the skull base. The same areas were occupied by brous bone, and the contours of the affected areas were expanded on the CT images. The right maxillary sinus cavity had disappeared and the orbital canal was narrowed as a result of bone growth, but there was no optical or nasal functional disorder. All clinical laboratory data were normal. The isozyme ratios of alkaline phosphatase (ALP) were 46% ALP 2, 45% ALP 3, and 9% ALP 5. The diagnosis was polyostotic craniofacial brous dysplasia on the right side. The patient wanted the swelling around the orbit zygomatic arch as well as the asymmetry to be reduced, especially the cant of the occlusal plane. We performed a three-dimensional (3D) computer simulation using stereolithograph and SimPlant CMF software (Materialize Dental, Leuven, Belgium) to select the best surgical approach, and concluded that radical surgery was impossible because of the large affected area, including the skull base, but it was also impossible to correct the occlusal plane by conventional surgery. Finally, we decided to perform conservative contouring around the orbital area rst, and then to perform a Le Fort I osteotomy and sagittal split ramus osteotomy (SSRO) to correct the severe cant of the occlusal plane. Because of the severe morphological abnormality, we decided to use a navigation system to ensure the safety of the Le Fort 1 osteotomy. Immediately before surgery, we obtained CT scans with 2-mm slices of the whole face with a LightSpeed Ultra CT scanner (General Electric Company, Faireld, CT, USA). The CT scans were loaded into the navigation software, and image-guided surgery was performed using the passive infrared Vector Vision surgical navigation system (BrainLAB, Heimstetten, Germany). All surgical procedures were performed with the patient under general anesthesia in

supine position. Registration was performed on the patient directly after intubation. First, a headband localizer with a marker star was attached to enable the head to be moved freely during surgery. The Vector Vision Compact system is a two-camera system that works passively. Infrared light is emitted by transmitters installed near the receiving cameras and is reected by suitable objects (localizers or markers). These reections are detected and used by the computer to establish both the positional coordinates of the patients head and also the position of the surgical instruments. After the navigation system was suitably positioned, surface registration of the patient was carried out. The laser beam was moved over the face of the patient, and reections from the surface of the patients face were detected by the cameras to match the patient with the 3D data record (Fig. 2a). Primary surgery was performed in November 2006 under general anesthesia. We contoured the expanded bone of the right frontal orbital and zygomatic area by using a bicoronal ap. The second surgery was performed in May 2007 under general anesthesia. The navigation system was used with a Le Fort I osteotomy. Besides the use of a pointer instrument, this navigation system also allows registration of other instruments such as chisels or saws (Fig. 2b). As the invisible deep areas were approached using the navigation system, the positions of the tips of the instruments were visualized on the monitor, and all maneuvers were performed safely (Fig. 2ce). The maxillary segments were xed with four 1.5-mm titanium plates using four screws each. Bilateral SSRO was performed, and xation was done with a single 2.0-mm plate xed with four screws and with one bicortical screw additionally on each side used to reinforce the xation. We were able to x the screws with appropriate torque even in the affected bone. The operation time was 6 h 43 min, and total blood loss was 864 ml. We gave 400 ml of autologous blood transfusion. Plate removal and shaving of the right lower border of the mandible was performed under general anesthesia in February 2009.

183

(a)

(c)

(b)

(d)

(e)
images on the monitor. d Macroscopic ndings of the osteotomized surface by Le Fort 1 osteotomy. e Coronal slice CT image of the osteotomized surface. d, e The maxillary sinus can be seen on the left side (asterisk) but not on the right side (star), and the maxilla on the right side has expanded deep inside (arrow)

Fig. 2ae. Procedure and the results of navigation. a Preparation of the navigation system. Star, special marker pointer instrument; arrow, headband localizer with a marker star. b Attachment of the registration star to the chisel. c Monitor display. The position of the apex of the special marker pointer instrument was visualized as a multiple plane reconstruction and three-dimensional computed tomography (CT)

The postoperative course was stable 2 years after orthognathic surgery. The patient was pleased with the decreased swelling around the orbit and zygomatic arch, and with the lessened asymmetry, especially the lessened cant of the occlusal plane (Fig. 1df).

Discussion
Denitive treatment methods for craniofacial brous dysplasia have not been established. Chan and Noordhoff1 classied the sites of occurrence of this disease into four zones, and recommended that the treatment be performed according to the zone. For the maxilla and mandible areas (zone 4), they recommended treating in a conservative manner. On the other hand, Valentini et al.2 reported that radical treatment could be performed in most cases even if the maxilla and mandible were involved. However, they also pointed out that polyostotic cases should be approached conservatively. Corrections of severe deformity, especially facial asymmetry or malocclusion, are very difcult by con-

servative surgery in cases of polyostotic brous dysplasia. Orthognathic surgery is suitable for reducing the asymmetry in these cases,3 and several cases treated by orthognathic surgery have been reported.36 However, Le Fort 1 osteotomy was performed in only four of these cases.3,4,6 Yeow and Chan3 reported that it was difcult to ensure the safety of a Le Fort 1 osteotomy in brous dysplasia because of the severe anatomical abnormality. In Le Fort 1 osteotomy, the posterior maxillary area is the most difcult area to approach because the maneuver must be performed blindly and the area shows a tendency to bleed.9 Recently, navigation systems have been used with craniomaxillofacial surgery to improve access to deep-seated locations8 and in abnormal anatomy cases with lost landmarks.10 In the present case, surgical simulation by computer software and stereolithography before surgery revealed a severe anatomical abnormality in the right maxilla. However, these methods could not ensure safe surgery. With the navigation system, we were able to visualize the position of the unseen deep area close to the pterygoid process and abnormal sinus cavity during the procedure. We believe that only a navigation system can provide a sufcient level of safety during this

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surgery because the position of the instruments can be visualized on site. Some limitations of this system were revealed. First, although the navigation is supposed to shorten operation time in complex cases,8 in fact, the registration process and attachment of the registration star to the various instruments were time consuming. The headband was the easiest and most minimally invasive method of attaching the marker star to the head but it occupied a lot of space in the surgical eld and frequently hampered the operation. We used the navigation system only in the maxilla. Additional registration methods are necessary for applications to the mandible because of its mobility.8 The operation would have been more complex if navigation had been performed for the mandible in our case. We need to carefully choose the most effective way of using a navigation system. In conclusion, Le Fort 1 osteotomy in brous dysplasia can be performed safely by using a navigation system.
Acknowledgments We are indebted to Prof. J. Patrick Barron and Roderick J. Turner of the Department of International Medical Communications of Tokyo Medical University for their review of this manuscript.

2. Valentini V, Cassoni A, Marianetti TM, Terenzi V, Fadda MT, Iannetti G. Craniofacial brous dysplasia: conservative treatment of radical surgery? A retrospective study on 68 patients. Plast Reconstr Surg 2009;123:65360. 3. Yeow VKL, Chan Y-R. Orthognathic surgery in craniofacial brous dysplasia. J Craniofac Surg 1999;10:1559. 4. Sachs SA, Kleiman M, Pasternak R. Surgical management of a facial deformity secondary to craniofacial brous dysplasia. J Oral Maxillofac Surg 1984;42:1926. 5. Samman N, Piette E, Cheung LK, Tideman AH. Feasibility of osteotomies in brous dysplasia of the jaws. Int J Oral Maxillofac Surg 1991;20:3536. 6. Cheung LK, Samman N, Pang H, Tiedman AH. Titanium miniplate xation for osteotomies in facial brous dysplasia. A histological study of the screw/bone interface. Int J Oral Maxillofac Surg 1995;24:4015. 7. Caversaccio M, Langlotz F, Nolte L-P, Hausler R. Impact of a selfdeveloped planning and self-constructed navigation system on skull base surgery: 10 years experience. Acta Otolaryngol 2007;127: 4037. 8. Widmann G, Stoffner R, Bale R. Errors and error management in image-guided craniofacial surgery. Oral Surg Oral Med Oral Pathol Oral Rad Endod 2009;107:70115. 9. Cheung LK, Fung SC, Samman N. Posterior maxillary anatomy: implication for Le Fort 1 osteotomy. Int J Oral Maxillofac Surg 1998;27:34651. 10. Heiland M, Habermann CR, Schmelzle. Indication and limitation of intraoperative navigation in maxillofacial surgery. J Oral Maxillofac Surg 2004;62:105963.

References
1. Chan Y-R, Noordhoff S. Treatment of craniomaxillofacial brous dysplasia: how early and how extensive? Plast Reconstr Surg 1989;86:83542.

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