Simultaneous costochondral graft and distraction osteogenesis in unilateral TMJ ankylosis associated with mandibular retrognathia and asymmetry

The purpose of this study was to review the long-term effect of simultaneous costochondral graft (CCG) and distraction osteogenesis (DO) in the management of unilateral temporomandibular joint ankylosis associated with severe dentofacial deformities in our clinic. In addition, we sought to analyze the advantages and disadvantages of CCG and DO. Four patients were included in this clinical study during 2005 to 2007. The mean length of ankylosis history was 14.5 years. All patients had significant mandibular retrognathia and asymmetry histories and have been diagnosed with obstructive sleep apnea syndrome by a polysomnogram before surgery. A 1-stage surgery, with gap arthroplasty, CCG, and mandibular DO, was performed. The surgical plan and technique were reviewed. No severe complications were observed after surgery. Distraction was started on day 7 after surgery. The distance of distraction ranged from 20 to 25 mm (mean, 22.5 mm). Mouth opening was increased from 25 to 37 mm (mean, 33.5 mm) during the follow-up period (range of 3.5-5 y). No recurrence of joint ankylosis occurred based on the clinical and radiographic evaluations. All of the patients had significant improvement in obstructive sleep apnea syndrome after surgery. Mandibular asymmetry and retrognathia were well corrected in all of the patietns. In conclusion, a 1-stage surgical treatment with DO and CCG demonstrated its feasibility and effectiveness in management of temporomandibular joint ankylosis combined with severe dentofacial deformity. It is a safe and reliable method of treatment.     

牵张成骨术治疗颞下颌关节强直后睡眠呼吸暂停综合征

目的 评估牵张成骨术(DO)治疗颞颌关节强直后睡眠呼吸暂停综合征(OSAS)的效果。方法 对8例患有颞颌关节强直后OSAS的患者采用DO技术进行治疗。手术在全麻下分两期进行。第一期行关节成形、下颌体部截骨,安置牵引器,前徙下颌矫正小颌畸形及OSAS;第二期在拆除第一期牵引器的同时,进行下颌升支后缘L型截骨,安置牵引器,通过牵引延长下颌升支。结果 8例患有颞颌关节强直后OSAS的患者的症状均有不同程度的改善。术后张口度可迭3cm～4cm,小颌畸形得以矫治,AHI指数由术前的20～40降至5以下,最低血氧饱和度由术前的68％提高至术后的96％,OSAS得以治愈。术后半年～两年随访,未见复发。结论 DO技术是治疗颞颌关节强直后OSAS理想方法。     

Treatment of temporomandibular joint ankylosis and facial asymmetry with bidirectional transport distraction osteogenesis technique

Transport distraction technique is a good treatment modality for unilateral temporomandibular joint ankylosis. However, with a unidirectional distraction, it is not possible to correct facial asymmetry that results from mandibular hypoplasia associated with early-onset unilateral temporomandibular joint ankylosis. For this purpose, gap arthroplasty and simultaneous bidirectional transport distraction was used to correct these deformities. Although vertical distraction corrects vertical deficiency of the ramus and creates a neocondyle, the simultaneous anteroposterior distraction of the transport segment corrects facial asymmetry resulting from horizontal shortness of mandible. Three patients, whose mean mouth opening was 8.6 mm, were successfully treated with this technique. Mean advancements in vertical and anteroposterior direction were 14.7 and 7.7 mm, respectively. Mean maximal mouth opening was 29.7 mm postoperatively. The average follow-up period was 13 months (range, 12-15 mo). During this period, reankylosis was not observed, and the interincisal distance did not decrease. Gap arthroplasty and bidirectional transport distraction of the mandibular ramus is a good and effective therapeutic option in treatment.     

Mandibular distraction osteogenesis reconstructed the upper airway in a case of Melnick�CNeedles syndrome

Introduction

We report a 16-year-old female with Melnick?CNeedles syndrome complicated with severe obstructive sleep apnea?Chypoxia syndrome.

Clinical report

An overnight sleep study demonstrated that the patient had severe obstructive sleep apnea with an apnea?Chypopnea index of 95/h. The lowest oxygen saturation was only 34%. The distraction osteogenesis technique was used to lengthen her mandible for the reconstruction of her upper airway.

Result and Discussion

Postoperative panorex showed that the bilateral body of her mandible had been distracted about 16?mm singulorumly in the end of our distraction. Lateral radiograph revealed that the diameter of oral pharynx widen nearly 5?mm. The patient recovered after the upper airway reconstruction.     

正颌外科治疗颞下颌关节强直伴阻塞性睡眠呼吸暂停综合征

目的 评价正颌外科手术治疗颞下颌关节强直伴阻塞性睡眠呼吸暂停综合征(OSAS)的效果。方法 12例颞下颌关节强直伴OSAS患者(男4例，女8例，年龄10～25岁，平均18．4岁；双侧颞下颌关节强直8例，单侧颞下颌关节强直4例)，采用颞下颌关节成形术、下颌矢状劈开前徙术、颏前徙成形术、舌骨悬吊术以及牵张成骨术移动下颌骨和舌骨。术后随访3～36个月。结果 12例患者张口度由术前的0～2mm增大到术后25～40mm；术后患者颜面形态明显改善；其连续血氧饱和度最低值由术前的42％提高至术后的90％以上，睡眠呼吸障碍解除和睡眠质量获得提高。结论 在颞下颌关节强直伴OSAS患者的治疗中，行颞下颌关节成形术的同时，辅助正颌外科手术，不仅可以增大患者的张口度，而且还能解决患者下颌后缩的畸形，同时解除上气道狭窄，从而缓解或纠正患者的低氧血症。     

牵引成骨技术治疗小下颌畸形伴OSAS效果的初步报告

目的分析评价下颌骨牵引延长治疗小下颌畸形伴阻塞性睡眠呼吸暂停综合征后,患者上呼吸道、睡眠呼吸暂停指数及血的变化,方法 对５例小下颌畸形伴ＯＳＡＳ患者应用口内型牵引器行牵引颌骨牵引成骨管长下颌骨体、前伸下颌骨、扩大咽腔。结果 ５例下颌骨体最大牵引幅度为２４．４０ｍｍ最小牵引幅度８．５０ｍｍ。在小颌畸形得到明显改善的同时,患者咽腔扩大,睡眠呼吸暂停指数明显下降,血氧饱和度显著上升。最长随访时间１１个月     

Mandibular distraction osteogenesis in a patient with Melnick-Needles syndrome

Melnick-Needles syndrome is an X-inked-dominant skeletal dysplasia in which there is deficient osteoblastic activity. Patients present with craniofacial anomalies consisting of a prominent forehead, exorbitism, mandibular hypoplasia, cheek fullness, and class II malocclusion. Severe mandibular hypoplasia leads to upper airway restriction, an increased incidence of sleep apnea and pneumonias, and occasionally respiratory failure. This is a report of a patient with Melnick-Needles syndrome who presented to our unit after multiple bouts of respiratory failure and with a tracheostomy in whom mandibular distraction osteogenesis was used to retire her tracheostomy and to cure her sleep apnea. The patient underwent bilateral, external, unidirectional mandibular distraction with a vector parallel to the occlusal plane. After a latency period of 5 days, distraction was initiated at a rate of 1 mm/day for 34 days. At this point, the patient was able to breathe with the tracheostomy plugged, and her occlusion had changed from a class II to a class III relationship. She no longer snored, and pulse oximetry on room air was normal while standing or supine. Interestingly, the patient's consolidation phase was prolonged--255 days--possibly attributable to altered bony metabolism. To our knowledge, this is the first reported case of mandibular distraction osteogenesis used to cure obstructive sleep apnea and eliminate the need for tracheostomy in a patient with Melnick-Needles syndrome. In the future, prophylactic mandibular distraction may prevent the need for tracheostomy in this group of patients.     

Neonatal distraction surgery for micrognathia reduces obstructive apnea and the need for tracheotomy

The objective of the study was to assess the effectiveness of neonatal mandibular distraction in treatment of obstructive sleep apnea in the perinatal period in preventing a tracheotomy. This was a prospective study of 17 infants at two centers with severe micrognathia who demonstrated obstructive sleep apnea refractory to conservative therapy. Age at surgery varied from 5 to 120 days. Distraction was performed at a rate of 2 mm/d. After distraction, callus consolidation was allowed for 4 to 6 weeks, and the device was then removed. Each child underwent a three-dimensional computed tomography scan before surgery and approximately 3 months after surgery. Of the 17 patients, 14 successfully underwent extubation and demonstrated significant improvement in the obstructive sleep apnea. Postoperative horizontal ramus length increased from 23.3 to 34.8 mm after surgery. Mean maxillary mandibular discrepancy was 8.28 mm before surgery and 2.2 mm after surgery. Ten infants who underwent pre- and postoperative polygraphic studies showed improvement in obstructive apnea. Three patients had postoperative polysomnographic studies only; the results were also within the normal range. The mean follow-up interval was 16.5 months (range: 8-48 months). Neonatal distraction is an effective method for treatment of micrognathia with obstructive sleep apnea in the perinatal period in preventing a tracheotomy.     

Rehabilitation of a hemophiliac with implants: a medical perspective and case report.

A patient suffering from classical hemophilia had previous surgery for ankylosis of the right temporomandibular joint. This was replaced by a costochondral graft and an overlay of temporalis muscle. A bilateral sagittal split was performed for a micrognathic mandible and a sleep apnea problem. That procedure solved the sleep apnea; however, it resulted in a prognathic mandible and an anterior open bite. The lower anterior teeth were periodontally involved with impaired alveolar support. The restricted opening of the oral cavity of 18 mm between maxillary and mandibular centrals and the potential danger of bleeding complicated the surgical and restorative procedures. The patient was prepared medically on each of 4 occasions with factor VIII replacement concentrate, and oral antifibrinolytic therapy (tranexamic acid). The treatment of choice was the extraction of the remaining lower incisors and their replacement with an implant-supported temporarily cemented retrievable fixed prosthesis. Serial extractions and chairside temporization provided the surgeon with precise guides for implant placement, and enabled the patient to enjoy unimpaired function through periods of healing and osseointegration.     

Distraction osteogenesis for management of obstructive sleep apnoea in temporomandibular joint ankylosis patients before the release of joint

AimTo evaluate the effects of distraction osteogenesis in management of obstructive sleep apnoea patients secondary to temporomandibular joints ankylosis.MethodsFifteen patients were included in study. Preoperatively the patients were worked up for polysomnography and CT scans. Only those patients with Apnoea–hypopnoea index >15 events/h denoting moderate to severe obstructive sleep apnoea were included in the study. Distraction osteogenesis was followed with 5 days latency period in adult patients and 0 days for children. Rate of distraction was 1 mm/day for adults and 2 mm/day for children till the mandibular incisors were in reverse overjet. After 3 months post distraction assessment was done using polysomnography and CT scan.TMJ ankylosis was released by doing gap arthroplasty after distraction osteogenesis.ResultsPost distraction improvement was seen in clinical features of OSA like daytime sleepiness and snoring. Epworth sleepiness scale improved from a mean of 10.25 to 2.25. Polysomnographic analysis also showed improvement in all cases with apnoea–hypopnoea index from 57.03 to 6.67 per hour. Lowest oxygen saturation improved from 64.47% to 81.20% and average minimum oxygen saturation improved from 92.17% to 98.19%. Body mass index improved from a mean of 18.26 to 21.39 kg/m².ConclusionDistraction osteogenesis is a stable and beneficial treatment option for temporomandibular joint ankylosis patients with obstructive sleep apnoea.     

牵引成骨术治疗儿童单侧颞下颌关节强直伴OSAHS 4例报道

目的:评价牵引成骨术治疗儿童单侧颞下颌关节强直伴阻塞性睡眠呼吸暂停低通气综合征(OSAHS)的治疗效果。方法:4例儿童单侧颞下颌关节强直伴发OSAHS患者,男女各2例,年龄5～13岁(中位年龄6.5岁)。均采用颞下颌关节成形术以恢复开口功能,下颌体牵引成骨术治疗OSAHS;其中3例行同期手术,1例行分期手术;单侧和双侧下颌体牵引各2例。固定期约3个月时行呼吸监护仪监测(PSG)复查和牵引器拆除术。结果:4例患儿OSAHS症状均消失,平均AHI由术前的42.7降到4.9,平均最低血氧饱和度由术前的74.3%上升到89.8%;平均开口度由6.5mm增加至25.5mm;面部畸形得到满意矫正。经过平均38.1个月(13～58个月)的随访,无1例复发。结论:下颌骨牵引成骨术联合颞下颌关节成形术能够有效地治疗儿童单侧颞下颌关节强直及其伴发的OSAHS、面部不对称畸形,并且可以同期手术。     

下颌双侧体部牵引成骨联合上颌手术辅助快速扩弓治疗双侧髁突自溶性吸收继发重度骨性Ⅱ类错畸形伴上牙弓狭窄的效果

目的 介绍一种治疗双侧髁突自溶性吸收（ICR）继发重度骨性Ⅱ类错畸形伴上牙弓狭窄的方法。方法 纳入6例双侧ICR继发重度骨性Ⅱ类错畸形伴上牙弓狭窄的病例,进行下颌双侧体部牵引成骨（DO）联合上颌手术辅助快速扩弓（SARME）。依照计算机辅助手术模拟（CASS）技术标准流程进行下颌体部DO术模拟,根据临床检查及影像学检查,设计牵引方向及距离。在T0、T1、T2及T3分别进行头颅CT、颞下颌关节（TMJ）问卷、TMJ磁共振、多导睡眠监测（PSG）等检查,从面型、咬合关系、颞下颌关节情况及睡眠呼吸功能4个方面评估手术效果、二期手术必要性及关节稳定性等。结果 纳入病例中,3例已完成全部治疗。下颌体部分别牵引13.0 mm、6.7 mm和8.1 mm,二期颏成形分别前移7.2 mm、0（未做）和11 mm,上颌分别扩弓5.3 mm、7.3 mm和4.9 mm,PSG数据显示重度阻塞性睡眠呼吸暂停低通气综合征（OSAHS）痊愈。最终面型及咬合关系均达到预期,改建期无复发,TMJ亦无明显变化。结论 下颌体部DO联合SARME,可有效纠正双侧ICR继发重度骨性Ⅱ类错畸形伴上牙弓狭窄。运用CASS技术进行手术设计及模拟,可有效预计牵引方向及距离,使治疗效果及稳定性更加可靠。     

Smallest detectable difference of maximal mouth opening in patients with painfully restricted temporomandibular joint function

Changes in maximal mouth opening reflect the impact of temporomandibular disorders and the effect of a therapeutic intervention. No information about the amount of change in maximal mouth opening with regard to reasoned decision‐making is available. The smallest detectable difference, as a measure of reliability assessment, provides this information and is expressed in the unit of the measurement instrument. Twenty‐five consecutive patients (5 males, 20 females) with a painfully restricted temporomandibular joint participated in this study. Measurements of maximal mouth opening were performed by two well‐trained observers on two separate measurement days, one week apart. The maximal mouth opening measurements were repeated three times. Inter‐observer, intra‐observer, and test‐retest reliability varied between 0.90 and 0.96. Inconsistency in measurement results analyzed in terms of absolute error variance, i.e. the measurement facets plus all the interactions, represented 11% of total variance. The smallest detectable difference of maximal mouth opening varied from 9 to 6 mm. For being successful in painfully restricted temporomandibular joint patients, statistically as well as clinically, the clinician has to measure at least 9 mm of improvement in maximal mouth opening. To reduce the smallest detectable difference from 9 to 6 mm, repeated measurement is necessary.     

A case of improved obstructive sleep apnea by distraction osteogenesis for midface hypoplasia of an infantile Crouzon's syndrome

We performed Le Fort III midfacial advancement with gradual distraction using internal devices on a 2-year 5-month-old boy with Crouzon's syndrome with associated severe obstructive sleep apnea. The device was not activated until 7 days after surgery, after which the distraction was initiated, 1 mm per day, and the midface was advanced 4 mm intraoperatively and distracted 12 mm postoperatively. A total advancement of 16 mm was obtained. The obstructive sleep apnea improved remarkably after the distraction. In infants and younger children with associated severe obstructive sleep apnea, advancement by distraction osteogenesis of the midface in Le Fort III maxillary osteotomy will be initially indicated to obviate tracheostomy improving the upper airway obstruction.     

Distraction osteogenesis in adult obstructive sleep apnea surgery: a preliminary report.

PURPOSE: This study evaluated the potential application of distraction osteogenesis (DO) for skeletal expansion in the management of adult patients with obstructive sleep apnea syndrome (OSAS). PATIENTS AND METHODS: Five consecutive adult patients (3 male, 2 female) underwent a DO procedure with intraoral distraction devices for the management of OSAS. The surgical procedures were mandibular advancement (n = 4, with 3 bilateral advancement and 1 unilateral advancement) and simultaneous maxillomandibular advancement (n = 1). Starting 7 days after surgery, the distraction devices were activated 4 times daily at a rate of 1.0 mm/d until the desired skeletal expansion was achieved. All of the patients underwent pre- and postoperative radiographic analysis and polysomnography. The distraction devices were removed 3 months after the completion of the distraction process. RESULTS: The amount of skeletal advancement by distraction ranged from 5.5 to 12.5 mm, with the mean distance being 8.1 mm. The postoperative polysomnographic results showed improvement of OSAS in all 5 patients with an improvement of the mean RDI from 49.3 events per hour to 6.6 events per hour. The lowest oxygen saturation improved from 79.8% to 85.8%. The mean follow-up period was 12 months. Complications included localized inflammation at the distraction device site (n = 2), temporary temporomandibular joint (TMJ) discomfort during distraction process (n = 1), and temporary paresthesia of the inferior alveolar nerve distribution (n = 2). A moderate amount of pain during the activation of the distraction devices was reported (n = 3) and was adequately treated with analgesics. None of the complications led to permanent problems. CONCLUSION: This limited case series shows that DO is applicable in selected adult patients for skeletal advancement in the treatment of OSAS. However, there are disadvantages with the use of DO, and further investigations are necessary to determine the potential of this technique.     

同期关节重建及下颌体牵张成骨治疗颞下颌关节强直3例报道

目的:对伴有严重牙颌面畸形的颞下颌关节强直患者,采用自体肋骨软骨移植重建关节,同期运用牵张成骨术行下颌骨牵张成骨延长下颌体长度,治疗下颌后缩和阻塞性睡眠呼吸暂停低通气综合征(OSAHS),评价治疗计划的可行性及短期效果。方法:3例颞下颌关节强直患者,平均年龄17.5岁,发生关节强直的平均年龄为3.6岁,病程平均为13.9a,开口度均为0,均伴有严重牙颌面畸形和OSAHS。根据头影测量结果,预先设计患侧下颌支下降的长度和下颌体延长长度;采用关节成形术加双侧冠突切除,术中取模制备板,进行同期自体肋骨软骨移植重建颞下颌关节和双侧下颌体牵张成骨术。术后第7天开始牵引,每天2次,牵引速率为0.8mm/d。结果:3例患者均顺利完成手术,术后未出现感染等严重并发症。顺利完成牵张成骨。下颌骨牵引长度平均为22.5mm(20.5～25mm)。术后3个月开口度平均为28mm(26～32mm),患者面形及OSAHS获得良好改善。结论:肋骨软骨移植关节重建同期进行下颌体牵张成骨具有良好的稳定性,该设计有利于缩短治疗周期和治疗费用,在短期内可同时解决开口、面形和OSAHS等问题,为后续矫正咬合关系奠定了基础。     

Effects of a mandibular repositioner on obstructive sleep apnea.

The purpose of this study was to investigate the effects of a mandibular repositioner on airway, sleep, and respiratory variables in patients with obstructive sleep apnea. Twenty-two patients selected for this study were confirmed with a diagnosis of obstructive sleep apnea based on initial nocturnal polysomnography. The patients were fitted with a mandibular repositioner designed to hold the mandible anteroinferiorly. Six months later, an outcome polysomnographic study was undertaken for each patient with the appliance in place. Lateral cephalometric radiographs in the upright position were also obtained before and after 6 months of treatment. The respiratory disturbance index decreased in 21 of the 22 patients with the appliance in place. The mean respiratory disturbance index of the 22 patients decreased significantly from 40.3 to 11.7 events per hour (P <.01). Some 59.1% of subjects were considered a treatment success with follow-up respiratory disturbance index < 10 events per hour. The mean minimum blood oxygen saturation level during sleep also improved significantly from 73.4% to 81.3% (P <. 01). The mandibular repositioner was constructed to position the mandible at 75% of the maximal mandibular advancement and with a 7 mm opening between the upper and lower incisors, and no aberrant effect on temporomandibular joint was noted. The retropalatal airway space increased and the cross-sectional area of the soft palate and the vertical distance of the hyoid bone to the mandibular plane decreased significantly. The tongue posture became significantly flatter. A significant linear correlation was found between the reduction in apnea index and specific craniofacial skeletal structures (length of anterior cranial base, mandibular plane angle, and upper to lower facial height ratios, P <.05). Subjects with a smaller reduction in apnea index tended to have shorter anterior cranial bases, steeper mandibular planes, and smaller upper to lower facial height ratios. We conclude that a mandibular repositioner may be an effective treatment alternative for obstructive sleep apnea and that a reduction in the frequency of apneic episodes is mainly attributed to the effects of the appliance on oropharyngeal structures.     

Fibrous ankylosis after distraction osteogenesis of a costochondral neomandible in a patient with grade III hemifacial microsomia.

Distraction osteogenesis has recently become a mainstay for treatment of craniofacial syndromes with mandibular hypoplasia. This article presents the difficult case of a patient with a previous costochondral rib graft who underwent mandibular distraction and developed a fibrous pseudoarthrosis at the distraction site. This was attributed in part to an associated temporomandibular joint ankylosis. Resorption of the pseudoarthrosis occurred once the distractor was removed. It appears that distraction osteogenesis of a mandible with an ankylosed temporomandibular joint can result in healing with a fibrous union, presumably because of movement at the distraction site when masticating. This can result in a pseudo "temporomandibular joint" at the distraction site. A temporomandibular joint arthroplasty was performed, followed by repeat distraction. We conclude that if there is an ankylosed temporomandibular joint or a stiff temporomandibular joint that may ankylose during the course of the distraction process, then a temporomandibular joint arthroplasty should be performed before or at the time the distractor is placed.     

Smallest detectable difference in outcome variables related to painful restriction of the temporomandibular joint

The smallest detectable difference is the smallest statistically significant change in measurement results. In the field of temporomandibular disorders, the smallest detectable difference is not a commonly used concept. Most outcome studies are based on comparisons of group means, although this does not provide information about individual changes or about the clinical relevance thereof. The smallest detectable difference for maximal mouth opening was calculated from previously published reliability coefficients and the standard deviations of different samples of healthy subjects and patients with complaints of the temporomandibular joint. The smallest detectable difference of pain intensity measured with different visual analogue scales was calculated from the reliability coefficients and standard deviations of a heterogeneous group of pain patients. The smallest detectable difference of function impairment was calculated for a group of patients with complaints of the temporomandibular joint. For maximal mouth opening in healthy subjects, the smallest detectable difference was 5 mm. Repeated measurements improved it to 3 mm. The smallest detectable difference on a visual analogue scale was 28 mm for actual pain intensity and 22 mm for minimal pain as well as for maximal pain intensity. For total function impairment of patients with complaints of the temporomandibular joint, the smallest detectable difference was 8 units on a 0 to 68 scale.     

Temporomandibular disorder pain and dental treatment of obstructive sleep apnea

Treatment of sleep apnea with mandibular advancement devices (MADs) may be associated with the development of symptoms of temporomandibular disorder (TMD). This article discusses the different types of TMD and orofacial pain problems that may occur during treatment of obstructive sleep apnea (OSA) with a MAD. It is critical that the general dentist who is providing dental devices for OSA perform a thorough physical and neurologic assessment of the temporomandibular joint and associated structures before providing such a device so that preexisting problems are identified and discussed with the patient.