Tadashi Morishita

Clinical application of distraction osteogenesis for traumatic maxillofacial deformities.

Treatment including occlusal function is important for maxillofacial fractures. Because distraction osteogenesis is now used frequently for the treatment of various types of congenital maxillary hypoplasia, the authors applied this technique to six cases of traumatic maxillofacial deformity caused by old maxillofacial fractures. Several kinds of devices were used for the various deformities associated with severe malocclusion, resulting in extension of both bone and soft tissues. In each case, favorable facial aesthetics and occlusion were achieved. Distraction osteogenesis also appears to be an extremely effective method for repair of traumatic maxillofacial deformities.

A case of Crouzon syndrome treated by simultaneous bimaxillary distraction

In general, midfacial hypoplasia can be a cause of airway obstruction in patients with syndromic craniosynostosis, such as Apert syndrome and Crouzon syndrome. There have been recent reports indicating that Le Fort III midfacial distraction is effective in improvement of respiratory conditions. Our report describes a Crouzon syndrome case with repeated obstructive sleep apnea (OSA) and chronic respiratory disorder. The patient underwent not only midfacial distraction but also mandibular distraction to improve their respiratory conditions. The postoperative respiratory conditions markedly improved, and satisfactory improvements of the occlusal conditions and facial appearance were achieved. In recent years, there have been some reports in which maxillomandibular advancement or maxillomandibular distraction was performed on adults with severe OSA. However, no reports have been made on simultaneous distraction of the midface and mandible for patients with syndromic craniosynostosis. This report will present such a case in a child in whom good results were obtained. The patient was a ten-year-old boy with Crouzon syndrome, and his family history was non-contributory. During infancy, the patient underwent cranioplasty and ventriculo-peritoneal shunt surgery at the neurosurgery department. At age 2 years, he underwent Le Fort III midfacial distraction using an internal device. However, recurrence of OSA was observed. Preoperative polysomnography (PSG) showed an apnea-hypopnea index (AHI) of 29.6, and severe OSA was observed. The patient had a small mandible, narrow pharyngeal space, and severe glossoptosis during sleep. The respiratory disorder was determined to be caused by not only by maxillary hypoplasia but also by mandibular hypoplasia. Thus, Le Fort III distraction using a halo device (Blue Ddevice: W. Lorenz, Jacksonville, FL) and mandibular distraction using an internal device (W. Lorenz, Jacksonville, FL) were performed simultaneously (Figure 1). A few days after surgery, the maxilla and mandible were distracted at a rate of 1 mm per day. The amount of distraction was determined, taking into account the patient’s occlusal conditions, maxillary and mandibular solid models, and respiratory conditions which were checked on a regular basis. As a result, the midface was distracted 19 mm and the mandible 13 mm. Postoperatively, the hypoplastic midface and mandible were enlarged. The patient had an uneventful postoperative course and only slight pain with no major complications. Halo device removal and internal device removal were performed one month and six months after completion of distraction, respectively. Postoperatively, the treatment was continued with orthodontic treatment. Presently at 4 years after surgery, postoperative AHI is 4.2, indicating major improvement in the respiratory condition. Preand postoperative cephalograms were analyzed. Preoperatively, sella-nasion-subspinal (SNA)

Le Fort I Distraction Using Internal Devices for Maxillary Hypoplasia in Patients With Cleft Lip, Palate, and Alveolus: Complications and Their Prevention and Management

In recent years, advancement has been made in distraction osteogenesis in the facial area. It has been applied actively to treat maxillary hypoplasia in patients with cleft lip, palate, and alveolus. Le Fort I distraction using internal devices does not lead to disability in daily living, including during the retention period, and has low surgical invasiveness, facile management, and superior aesthetics. Thus, there have been occasional reports on this procedure in recent years. Because an internal device is unidirectional, the greatest disadvantage is the inability to adjust the direction of distraction after its placement. However, past reports have emphasized its advantages, and its complications have rarely been discussed. We performed Le Fort I distraction using internal devices on 15 young patients. Our results showed notable complications in a few cases. When serial cephalometric analysis was performed after maxillary distraction, it demonstrated that the maxilla assumes various three-dimensional distraction morphologies. In particular, cases with insufficient bone grafting of alveolar clefts developed collapse mainly in the alveolar cleft region during postoperative distraction. Mobility and deviation of the maxillary bone fragment occurred. We implemented preventative measures against complications such as using modifications to place the devices parallel on the left and right sides and using a bite splint for distraction. These measures produced improved outcomes, and we describe here the details.

A clinical report on distraction osteogenesis applied for Apert syndrome

Abstract This case report describes the orthodontic treatment done to an 11-year-old female patient having Apert syndrome, a genetic disorder manifested by craniofacial and limb anomalies. The orthodontic treatment was accompanied with distraction osteogenesis for forward advancement of the correction of midfacial hypoplasia. After the active treatment, a better facial profile and occlusion were achieved. The occlusion remained stable during the 2 years and 8 months follow-up.

Special distraction osteogenesis before bone grafting for alveolar cleft defects to correct maxillary deformities in patients with bilateral cleft lips and palates: Distraction osteogenesis performed separately for each bone segment

INTRODUCTION Patients with bilateral cleft lips and palates have premaxillary protrusion and characteristic jaw deformities involving three-dimensional malposition of the premaxilla and bilateral maxillary bone segments. This study examined patients with bilateral cleft lips and palates who had deviation and hypoplasia of the premaxillas and bilateral maxillary segments. Before bone grafting, the patients were treated with special distraction performed separately for each bone segment using a halo-type external device. This report describes this novel treatment method which produced good results. MATERIAL AND METHODS The subjects were five patients with severe jaw deformities due to bilateral cleft lip and palate. They were treated with maxillary Le Fort I osteotomy and subsequent distraction performed separately for each bone segment using a halo device. In three of five patients, premaxillary osteotomy was not performed, and osteotomy and distraction were performed only for the right and left lateral segments with severe hypoplasia. RESULTS All patients achieved distraction close to the desired amount. The widths of the alveolar clefts were narrowed, and satisfactory occlusion and maxillary arch form were achieved. After the surgery, three of five patients underwent bone grafting for bilateral alveolar cleft defects and the bone graft survival was satisfactory. CONCLUSIONS This method had many benefits, including narrowing of alveolar clefts, improvement of maxillary hypoplasia, and achievement of a good maxillary arch form. In addition, subsequent bone grafting for alveolar cleft defects was beneficial, dental prostheses were unnecessary, and frequency of surgery and surgical invasiveness were reduced. This method is a good surgical procedure that should be considered for patients with bilateral cleft lips and palates who have premaxillary protrusion and hypoplasia of the right and left lateral segments.

Backward distraction osteogenesis in a patient with severe mandibular micrognathia.

Abstract Maxillary skeletal prognathism can involve severe mandibular micrognathia with marked mandibular retrognathism or hypoplasia. For patients with such a condition, a conventional treatment is mandibular advancement by sagittal split ramus osteotomy (SSRO). This procedure has problems such as insufficient advancement, instability of jaw position, and postoperative relapse. Thus, in recent years, mandibular distraction osteogenesis has been used in some patients. Mandibular distraction has many advantages, but an ideal occlusion is difficult to achieve using this procedure. That is, 3-dimensional control cannot be attained using an internal device that is unidirectional. This report describes a case of severe mandibular micrognathia in a 14-year-old girl treated using backward distraction osteogenesis. This procedure was first reported by Ishii et al (Jpn J Jaw Deform 2004; 14:49) and involves a combination of SSRO and ramus distraction osteogenesis. In the present study, intermaxillary fixation in centric occlusion was performed after osteotomy, and proximal bone segments were distracted in a posterosuperior direction. This procedure is a superior surgical technique that avoids the drawbacks of SSRO and conventional mandibular distraction. However, it applies a large load to the temporomandibular joints and requires thorough management. Thus, careful evaluation needs to be made of the indication for backward distraction osteogenesis.

Atypical Apert syndrome: Sequential and segmental distraction osteogenesis of the skull, midface, and mandible

We describe a boy with Apert syndrome, including cranial deformities and syndactyly (acrocephalosyndactyly), though intracranial hypertension, exophthalmos, and midfacial hypoplasia were mild. We treated him by mandibular distraction, in addition to fronto-orbital distraction, and Le Fort III midfacial distraction, with good results.

A Survey of Orthodontic Treatment in Team Care for Patients With Syndromic Craniosynostosis in Japan

Objective: To understand the actual condition of orthodontic treatment in team care for patients with syndromic craniosynostosis (SCS) in Japan. Design: A nationwide collaborative survey. Setting: Twenty-four orthodontic clinics in Japan. Patients: A total of 246 patients with SCS. Main Outcome Measure: Treatment history was examined based on orthodontic records using common survey sheets. Results: Most patients first visited the orthodontic clinic in the deciduous or mixed dentition phase. Midface advancement was performed without visiting the orthodontic clinic in about a quarter of the patients, and more than a half of the patients underwent “surgery-first” midface advancement. First-phase orthodontic treatment was carried out in about a half of the patients, and maxillary expansion and protraction were performed. Tooth extraction was required in about two-thirds of patients, and the extraction of maxillary teeth was required in most patients. Tooth abnormalities were found in 37.8% of patients, and abnormalities of maxillary molars were frequently (58.3%) found in patients who had undergone midface surgery below the age of 6 years. Conclusions: Many patients underwent “surgery-first” midface advancement, and visiting the orthodontic clinic at least before advancement was considered desirable. First-phase orthodontic treatment should be performed considering the burden of care. Midface advancement below the age of 6 years had a high risk of injury to the maxillary molars. This survey is considered useful for improving orthodontic treatment in team care of patients with SCS.

A collaborative survey on occlusion after orthodontic treatment in patients with unilateral cleft lip and palate in Japan

Abstract Purpose A nationwide collaborative survey was performed in Japan to evaluate the recent treatment outcomes on occlusion after orthodontic/orthognathic treatment for patients with unilateral cleft lip and palate (UCLP). Methods A retrospective collaborative survey was performed. Twenty-five facilities, including 14 university hospitals, examined their orthodontic records and cast models of patients with UCLP. Using common format, information of 492 non-syndromic UCLP cases were reported. Patients with syndromic UCLP or congenital missing maxillary central incisors were excluded. Basically, each facility reported their most recent 30 cases. Results Of 492 cases, 330 cases (67.1%) were reported from 11 facilities, suggesting that orthodontic treatment for patients with CL/P was not fully centralized in Japan. Secondary alveolar bone grafting and orthognathic surgery were performed in 92.7% and 22.5% of cases, respectively. A significantly higher rate of orthognathic surgery was seen in cases reported from university hospitals (27.5%) than other facilities (14.8%). Average age at the end of active orthodontic/orthognathic treatment was 18.5 years. The lateral incisors on cleft sides were congenitally missing in 50.2% and displayed microdontia in 42.9% of cases. After orthodontic treatment, the central incisors and the canines were aligned without interdental spaces in 55.3% of cases. The positive anterior overjet was seen in 88.2%, and the centerline discrepancies between maxillary and mandibular incisors within 1/4 of mandibular incisor width was in 74.8% of cases. Conclusions The final occlusion after orthodontic treatment was satisfactory in most cases. Findings of this study are referable for improving standard Japanese orthodontic care for patients with CL/P.

Clinical Experience of Treatment of Facial Malformations in Oto-Palato-Digital Syndrome: A Familial Patient

Abstract Oto-palato-digital syndrome type 1 (OPD1) is an X-linked recessive disorder comprising characteristic facial appearances and skeletal alterations. The authors report OPD1 in a mother and her 2 sons who had multiple common congenital anomalies. Both of the brothers were born with mild hearing impairment, frontal bossing with prominent supraorbital ridges, downslanting palpebral fissures, dental malocclusion, and palatal clefts. They underwent a series of aesthetic surgeries for their facial malformations with good cosmetic results. The mother had a milder phenotype with less prominent craniofacial defects that did not require surgical correction. The older brother required a 2-jaw surgery whereas the younger brother required a surgically assisted rapid palatal expansion. In the second series of operations, both brothers underwent scraping of their prominent supraorbital ridge. They have been free of complications throughout their 16-year follow-up. The authors also screened the family for possible genetic etiologies and identified mutations in the causative gene of OPD1 on Xq28 in all 3 patients. In conclusion, the authors have identified 3 patients with OPD1, performed surgical treatments on the affected brothers and have obtained good reconstructive results. There are no reports involving OPD1 patients who have received good surgical treatment. With careful examination and identification of patients with characteristic facies and skeletal abnormalities, it is our opinion that the authors can help more patients with this disease through surgical management.