Does distraction osteogenesis improve the airway volume and quality of life of obstructive sleep apnoea patients secondary to temporomandibular joint ankylosis?

Temporomandibular joint (TMJ) ankylosis leads to mandibular micrognathia that severely collapses the upper airway causing obstructive sleep apnoea (OSA), resulting in deterioration and compromise in the quality of life (QoL) of patients. In this study, we aimed to calculate airway volume changes, apnoea-hypopnoea index (AHI), and improvement in quality of life before and after distraction osteogenesis (DO). Fourteen Patients with OSA secondary to TMJ ankylosis at a mean (SD) age of 17.5 (5.43) years were enrolled in this prospective study. Multivector mandibular distractors were used in all patients following the standard Ilizarov distraction protocol with a mean (SD) anteroposterior distraction of 16.21 (4.37) mm and a consolidation period of 116.92 (14.35) days. The patients were followed up for six months. A polysomnography test (PSG) was done to quantify AHI and a low-dose computed tomographic scan was done to calculate airway volume using Dolphin medical imaging software pre and post-DO. The QoL of the patients was calculated using the OSA-18 questionnaire. Results analysis depicted that the mean (SD) preoperative AHI was 51.44 (37.99)/h which was improved to 9.57 (9.74)/h (p = 0.001) after DO. Airway volume was calculated on Dolphin software before and after DO showed a significant improvement in airway volume by 121.12% (98.30)%. Similarly, the OSA-18 questionnaire showed significant improvement in QoL from severe to normal. This study suggested that DO increases the corpus length of the mandible, leading to an increment in airway volume, which improves the QoL.

Surgical correction of neonatal obstructive sleep apnea due to a temporomandibular joint ankylosis.

In growing children, temporomandibular joint (TMJ) ankylosis and septic arthritis are uncommon. Retrognathia and micrognathia affect airway patency and can cause obstructive sleep apnea (OSA). No unified diagnostic criteria have been established for the management of this pathology. We describe the first case of treatment for pediatric TMJ ankylosis and severe OSA due to neonatal group B streptococcal septic TMJ arthritis. Untreated pathological changes in the TMJ will eventually lead to ankylosis. Among children, this will include facial growth disturbances leading to mandibular retrognathia, reduction in the oropharyngeal spaces, and OSA. Our patient had severe OSA with an apnea-hypopnea index of 24.9 events/h and oxygen saturation nadir of 73% as measured by polysomnography. She was treated successfully according to Andrade protocol. This is the first report of pediatric OSA due to TMJ ankylosis following neonatal group B streptococcal septic arthritis. CITATION: Pesis M, Goldbart A, Givol N. Surgical correction of neonatal obstructive sleep apnea due to a temporomandibular joint ankylosis. J Clin Sleep Med. 2024;20(1):173-179.

Post-traumatic spinal hematoma in diffuse idiopathic skeletal hyperostosis (DISH).

OBJECTIVES: To determine the incidence of spinal hematoma and its relation to neurological deficit after trauma in patients with spinal ankylosis from diffuse idiopathic skeletal hyperostosis (DISH). MATERIALS AND METHODS: A retrospective review of 2256 urgent or emergency MRI referrals over a period of 8 years and nine months revealed 70 DISH patients who underwent CT and MRI scans of the spine. Spinal hematoma was the primary outcome. Additional variables were spinal cord impingement, spinal cord injury (SCI), trauma mechanism, fracture type, spinal canal narrowing, treatment type, and Frankel grades during injury, before and after treatment. Two trauma radiologists reviewed MRI scans blinded to initial reports. RESULTS: Of 70 post-traumatic patients (54 men, median age 73, IQR 66-81) with ankylosis of the spine from DISH, 34 (49%) had spinal epidural hematoma (SEH) and 3 (4%) had spinal subdural hematoma, 47 (67%) had spinal cord impingement, and 43 (61%) had SCI. Ground-level fall (69%) was the most common trauma mechanism. A transverse, AO classification type B spine fracture (39%) through the vertebral body was the most common injury type. Spinal canal narrowing (p < .001) correlated and spinal cord impingement (p = .004) associated with Frankel grade before treatment. Of 34 patients with SEH, one, treated conservatively, developed SCI. CONCLUSIONS: SEH is a common complication after low-energy trauma in patients with spinal ankylosis from DISH. SEH causing spinal cord impingement may progress to SCI if not treated by decompression. CLINICAL RELEVANCE STATEMENT: Low-energy trauma may cause unstable spinal fractures in patients with spinal ankylosis caused by DISH. The diagnosis of spinal cord impingement or injury requires MRI, especially for the exclusion of spinal hematoma requiring surgical evacuation. KEY POINTS: • Spinal epidural hematoma is a common complication in post-traumatic patients with spinal ankylosis from DISH. • Most fractures and associated spinal hematomas in patients with spinal ankylosis from DISH result from low-energy trauma. • Spinal hematoma can cause spinal cord impingement, which may lead to SCI if not treated by decompression.

TMJ Ankylosis Following Mandibular Distraction Osteogenesis: Management With Simultaneous Midface External Distraction and Bilateral Temporomandibular Joint Replacement.

Early mandibular distraction osteogenesis (MDO) can decrease upper airway and feeding complications in pediatric patients with micrognathia; however, temporomandibular joint (TMJ) complications like TMJ ankylosis (TMJA) may occur. TMJA can disturb pediatric patients' function and craniofacial growth, resulting in significant physical and psychosocial consequences. Additional surgical procedures may also be required, increasing the burden of care on patients and their families. CMF surgeons must discuss the potential complications of early MDO surgery with families as well as potential solutions should these problems occur. This report presents the case of a 17-year-old male with a severe craniofacial anomaly with features of Treacher-Collins syndrome (TCS) and a surgical history of tracheostomy, cleft palate repair, mandibular reconstruction with harvested costochondral grafts, and MDO with resultant bilateral TMJA and limited mouth opening. The patient Was treated with bilateral custom alloplastic TMJ replacements and simultaneous maxillary DO using a Rigid External Distraction (RED) device.

OUTCOMES OF AUTOTRANSPLANTED THIRD MOLARS WITH COMPLETE ROOT FORMATION: A SYSTEMIC REVIEW AND META-ANALYSIS.

OBJECTIVES: Clear evidence is lacking regarding the outcomes of autogenous tooth transplantation (ATT) of third molars with complete root formation. The current review aims to explore the long-term survival and complication rates. METHODS: A comprehensive search was performed in December 2022 of the PubMed, Scopus, Embase, EBSCO, Ovid, Science Direct, and Web of Science databases. The systematic review was conducted according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines and registered at the International Prospective Register of Systematic Reviews (CRD42022337659). The pooled survival, root resorption, and ankyloses rates were calculated. Subgroup analyses were performed to explore the effects of sample size and 3D techniques. RESULTS: Twelve studies from 5 countries fulfilled the eligibility, with 759 third molars transplanted in 723 patients. Five studies reported 100% survival at 1-year follow-up. After excluding these 5 studies, the pooled survival rate was 93.62% at 1 year. The survival rate of 1 large sample study was significantly higher than that of small ones at 5 years. The complications of studies using 3D techniques were: root resorption 2.06% (95% CI: 0.22, 7.50) and ankyloses 2.81% (95% CI: 0.16, 12.22), compared to those without 3D techniques: root resorption 10.18% (95% CI: 4.50, 17.80) and ankyloses 6.49% (95% CI: 3.45, 10.96). CONCLUSIONS: ATT of third molars with complete root formation is a reliable alternative for replacement of a missing tooth and has a promising survival rate. The use of 3D techniques can reduce complication rates and improve long-term survival.

Pediatric temporomandibular joint ankylosis and arthritis: Forgotten complications of acute otitis media.

OBJECTIVES: Temporomandibular joint (TMJ) arthritis and ankylosis represent unusual but potential complications of ear suppuration, especially in children. We performed a review of the literature of pediatric otogenic TMJ arthritis and ankylosis, discussing their clinical and radiological features, their mechanism of infection spread, and the importance of a prompt diagnosis and treatment. We additionally describe a case of TMJ ankylosis following acute mastoiditis in a 4-year-old female patient. METHODS: A search of English literature from January 1, 1980 to December 31, 2021 was performed on the electronic databases (PubMed, Web of Science and Scopus) in order to identify studies concerning TMJ complication after ear suppuration. RESULTS: Seventeen articles were considered eligible for the review. Eight and nine studies described otogenic TMJ ankylosis and arthritis, respectively. A total of 17 children affected by ankylosis consequent to ear infection and a total of 31 cases of TMJ arthritis concurrent to otomastoiditis were identified. Mean time elapsed between ear infection and diagnosis of TMJ ankylosis was 4.8 years (range 0.5-13). CONCLUSION: TMJ involvement during complicated otitis media should be kept in mind. Its prompt recognition is mandatory to set up appropriate treatment and follow-up and reduce the risk of ankylosis with its functional and psychological complications.

Pediatric temporomandibular joint (TMJ) arthritis, an elusive complication of acute mastoiditis.

INTRODUCTION: Septic arthritis of the Temporomandibular joint (TMJ) is a rare complication of acute middle ear infection. Presentation is elusive and could be easily missed. Often diagnosis is made only with consequential development of TMJ ankylosis. This study intends to characterize patients and course of disease and suggest a diagnostic and therapeutic strategy. METHODS: Retrospective review of all children diagnosed with TMJ arthritis and/or TMJ ankylosis secondary to acute middle ear infection, treated in a tertiary pediatric medical center between the years 2005 and 2021. RESULT: Seven patients were identified with otogenic TMJ arthritis. Median age at presentation was 1.14 years (IQ range 1.1-1.5). All seven were diagnosed with acute mastoiditis. CT scans demonstrated TMJ related collections in 5/7 and intracranial complications in 3/7. Treatment included cortical mastoidectomy for 5/7. One patient had a concomitant surgical washout of the TMJ. Two patients had drainage only of subperiosteal collections. Six of the seven patients went on to develop TMJ ankylosis that presented within a median of 2.8 years (IQ range 2.6-3.9) after the episode of acute mastoiditis. All six patients presented with trismus and facial growth anomalies, and all but one required surgery to release the ankylosis. CONCLUSION: Otogenic TMJ arthritis develops mostly in young children with acute mastoiditis and intratemproal and/or intracranial suppuration. Imaging is helpful in making the diagnosis, with TMJ related collections on CT being the most common finding. TMJ ankylosis can develop within a few years and present with trismus and abnormal facial growth. Cortical mastoidectomy does not seem to prevent ankylosis. It remains unclear whether focused treatment to the TMJ or physiotherapy could be beneficial in that.

Good result for twelve years after bilateral hip and knee arthroplasties for ankylosis with juvenile idiopathic arthritis: a case report.

Juvenile idiopathic arthritis (JIA) can lead to joint deformity and bone destruction, which can cause gait disturbances. To the best of our knowledge, there are no case reports with over 10 years of follow-up on quadruple joint arthroplasties (QJA) for bilateral hip and knee ankylosis associated with JIA. We present the case of a 29-year-old woman with JIA. The patient suffered from bilateral ankylosis of the hips and knees and developed a swing gait requiring double crutches. We performed staged QJA with careful attention to postoperative rehabilitation and her physical features, which included excessive pelvic anteversion, poor bone quality, and short statue of bones. Twelve years after surgery, the patient was able to walk without any support and showed good clinical functional scores. In addition, no radiological loosening following QJA was observed. We hereby introduce a surgical strategy for total hip arthroplasty for excessive pelvic anteversion, which involves two methods to calculate pelvic tilt on a pelvic anteroposterior radiograph. These methods were able to approximately predict postoperative pelvic changes.

Ankylosis of the cervical spine increases the incidence of blunt cerebrovascular injury (BCVI) in CTA screening after blunt trauma.

PURPOSE: To examine the incidence, location, and grade of blunt cerebrovascular injury (BCVI), as well as associated strokes in patients with ankylosis of the cervical spine, imaged with CT angiography (CTA) after blunt trauma. The related etiologies of ankylosis had an additional focus. MATERIALS AND METHODS: Altogether of 5867 CTAs of the craniocervical arteries imaged after blunt trauma between October 2011 and March 2020 were manually reviewed for a threshold value of ankylosis of at least three consecutive cervical vertebrae. BCVI was the primary outcome and associated stroke as the secondary outcome. Variables were craniofacial and cervical spine fractures, etiology and levels of ankylosis, traumatic brain injury, spinal hematoma, spinal cord injury, and spinal cord impingement, for which correlations with BCVI were examined. RESULTS: Of the 153 patients with ankylosis and blunt trauma of the cervical spine, 29 had a total of 36 BCVIs, of whom two had anterior and 4 posterior circulation strokes. Most of the BCVIs (n = 32) were in the vertebral arteries. Injuries were graded according to the Biffl scale: 17 grade II, 4 grade III, 14 grade IV, and 1 grade V. A ground-level fall was the most common trauma mechanism. Cervical spine fracture was the only statistically significant predictor for BCVI (OR 7.44). Degenerative spondylosis was the most prevalent etiology for ankylosis. CONCLUSION: Ankylosis of the cervical spine increases the incidence of BCVI up to sevenfold compared to general blunt trauma populations, affecting especially the vertebral arteries.

Does Evoked Electromyography Detect the Injured Facial Nerve Recovery Earlier Than Clinical Assessments?

PURPOSE: The key element in managing postoperative facial nerve (FN) injuries is timely diagnosis and intervention as indicated. The purpose of this study was to measure and compare evoked electromyography (EEMG) and clinical assessment in terms of the recovery of the injured FN in operated temporomandibular joint ankylosis (TMJA) cases. METHODS: The investigators designed a prospective cohort study in the primary operated TMJA patients. The primary predictor variable was the technique used to assess FN function, House-Brackmann Facial Nerve Grading System (HBFNGS) or EEMG. The primary outcome variable was time to FN recovery. The FN recovery was assessed in different time points (1 week, 1 month, 3 months, and 6 months). Age, gender, side (unilateral/bilateral), type of ankylosis (Sawhney's classification), and operating time were kept as covariates. Categorical variables were analyzed using Fisher's exact test. Multilevel survival analysis was performed considering the subject as cluster to perform Kaplan-Meier analysis and compute the hazards ratio using the Cox-regression method with adjustment for covariates. P <0.05 was set as statistically significant. RESULTS: The study sample composed of 43 (69 sides) TMJA cases who underwent surgery developed iatrogenic FN injury in 10 cases (14 sides [9 right; 5 left]). The incidence of FN injury was 20.3% (14/69). Sawhey's type III/type IV ankylosis and the operating time for more than 2 hours showed a statistically significant (p<0.05) increase in FN injury. The mean duration to detect FN recovery by EEMG was 9 days (95% confidence interval, 5 to 12 days), but the HBFNGS took 161 days (95% confidence interval, 141 to 181 days). The chance of early detection by EEMG was 18.6 times more than the chance by the HBFNGS (Cox-hazard ratio, 18.6). CONCLUSIONS: To conclude, EEMG is a noninvasive and reliable tool that detects FN recovery much earlier than the HBFNGS in the postoperative TMJA cases.

Severe Heterotopic Ossification After Revision Total Knee Arthroplasty: A Case Report and Review of the Literature.

The incidence of primary and revision total knee arthroplasty (TKA) is increasing worldwide. Heterotopic ossification is a common and concerning complication of TKA. There are few described cases of severe heterotopic ossification after revision TKA and no known cases of heterotopic ossification causing functional ankylosis after revision TKA. We describe a case of extensive heterotopic ossification in a patient who underwent right TKA for extensive adhesions and stiffness. After early range of motion improvement postoperatively, the patient discontinued a physical therapy regimen. The patient presented 13 years after revision TKA with radiographically evidenced severe heterotopic ossification resulting in a functional ankylosis. The patient elected for nonsurgical management. This case demonstrates a delayed finding of severe heterotopic ossification. The case prompted an applied literature review of several topics: heterotopic ossification as a complication of revision arthroplasty, the contribution of autoimmune and inflammatory conditions to heterotopic ossification; the use of medication, radiation, and physical therapy as prophylaxis against heterotopic ossification; and the range of treatment strategies for severe heterotopic ossification at the knee joint. Consent by the patient involved in this case report was obtained.

Treatment of Dentofacial Deformity Associated with Temporomandibular Joint Ankylosis Through Vertical and Sagittal Osteotomies of the Mandibular Ramus.

ABSTRACT: Ankylosis of the temporomandibular joint is a disorder resulting from fibrous, osseous, or fibro-osseous adhesion that directly affects the quality of life of the individual. The authors present a case of unilateral temporomandibular joint ankylosis treated by condilectomy and ipsilateral sliding vertical ramus osteotomy associated with contralateral sagittal osteotomy aiming to restore function and to correct dentofacial deformity class II. A 31-year-old female patient presented with a history of facial trauma and major complaint of oral opening limitation. Physical examination revealed hypoplasia of the lower third of the face, facial pattern type II, anterior open bite, and maximum mouth opening of 22.5 mm. Computed tomography showed an ankylotic mass in the right mandibular condyle with deformity of the condylar structure and fusion to the right zygomatic arch and a contralateral condylar fracture sequel. It was proposed to perform a condilectomy of the right mandibular condyle for the removal of the bone mass concomitant to the sliding vertical ramus osteotomy of the mandibular ramus for condylar reconstruction by rhytidectomy approach and the sagittal osteotomy of the left mandibular aiming the reestablishment of occlusion and the correction of dentofacial deformity. The condylar fracture was not operated because the condyle was remodeled. The patient is in her fourth year postoperative presenting satisfactory esthetic-functional re-stabilization, without clinical signs of recurrence. In conclusion, the authors believe that combined sliding vertical ramus osteotomy and sagittal osteotomy can bring satisfactory results in complex cases.

Treatment of severe mandibular deficiency following TMJ ankyloses by distraction osteogenesis and orthodontic treatment with microimplants.

Severe mandibular deficiency caused by temporomandibular joint (TMJ) ankyloses produces functional and aesthetic problems that require complicated long-term treatment. In this case report, we describe the benefits of using microimplant mechanics for controlling the direction of distraction during distraction osteogenesis and for performing the movement of teeth. We also present its remarkable results and long-term stability. A 20-year-old girl presented with a convex profile due to severe mandibular retrognathia after a history of several TMJ surgeries for bilateral TMJ ankyloses. Mandibular distraction osteogenesis (MDO) was performed, and elastics were placed between the microimplants to control the direction of distraction. Subsequently, after retraction of the maxillary anterior teeth and distalisation of the whole mandibular dentition, the facial profile was markedly improved, and good interdigitation was obtained. The six-year follow-up retention and overall stability were satisfactory with good interdigitation and jaw function.

Prospective study of the long-term outcomes and complications after total temporomandibular joint replacement: analysis at 10 years.

This prospective analysis was performed to assess the long-term benefits of the TMJ Concepts joint replacement system in the UK. All patients who had replacement temporomandibular joints (TMJ) with at least 10 years of follow-up were included. The most common primary diagnoses were trauma, multiple previous operations, psoriatic arthritis, rheumatoid arthritis, degenerative disease, and ankylosis. A total of 43 patients (62 joints) were followed up for 10 years (mean age 45, range 22-70 years); 39 were female and four were male. The mean number of previous TMJ procedures was 2.5 (range 0-10). Over the 10 years of follow-up, there were significant improvements in pain score (10-point scale; decreased from 7.4 to 1.7), maximum mouth opening (increased from 21.0 mm to 34.7 mm), and dietary score (10-point scale; increased from 4.1 to 9.5). Joints in two patients failed, one secondary to a local dental infection and one due to reankylosis. None failed due to wear of the prosthesis, whether the prosthesis was standard cobalt-chrome or all-titanium. Total TMJ replacement gives good long-term improvements, both lessening pain and improving function, and is an effective form of management for irreparably damaged joints.

Characteristics, natural evolution and surgical treatment outcomes of unilateral laryngeal paralysis versus ankylosis: A longitudinal cohort study.

AIMS: Although unilateral laryngeal immobility (ULI) can results from paralysis or ankylosis of the cricoarytenoid joint, no comparative study exists to date. Aim of this study was to compare clinical features, aetiologies, spontaneous recovery and evolution after surgical treatment of ULI according to its mechanism. METHODOLOGY: Longitudinal observational cohort study between 1992 and 2017 in a tertiary care referral centre and university teaching hospital. All adult patients with isolated ULI were included. Presenting symptoms and demographic data were recorded at baseline. During follow-up, natural recovery and, if a surgical treatment was performed, treatment failure rate were noted. RESULTS: 994 patients were included, 56.4% of male and with a mean age of 58 years. Overall, 91% had paralysis and 9% had ankylosis. Dysphonia was the main symptom in both groups (>96%). Dyspnoea was more frequent in patients with ankylosis (26.1% vs 4.2% in those with paralysis) whereas dysphagia was more frequent in those with paralysis (31.1% vs 20% in those with ankylosis). With a mean follow-up of 2.3 years (±5.1), spontaneous recovery did not differ according to ULI's aetiology (hazard ratio 1.43, 95% confidence interval 0.85-2.40). Overall, 37.1% underwent a surgical treatment, and paralysis was associated with a lower odd of treatment failure (hazard ratio 0.27, 95% confidence interval 0.10-0.70) over a mean follow-up of 3.1 years (±4.1). CONCLUSION: ULI resulting from paralysis or ankylosis differ in their symptoms and responses to surgical treatment, whereas natural evolution was similar.

Bow hunter's syndrome after cervical laminoplasty in a patient with rheumatoid arthritis with bony ankylosis in the cervical spine: a case report.

Bow hunter's syndrome, or rotational vertebral artery (VA) occlusion, refers to vertebrobasilar insufficiency due to mechanical occlusion of the VA. We present a case of surgical treatment for bow hunter's syndrome that occurred after cervical laminoplasty in a patient with rheumatoid arthritis with bony ankylosis of the facet joints. A 59-year-old female with rheumatoid arthritis experienced sudden incomplete left hemiplegia. Fifteen months earlier, the patient had undergone cervical decompression surgery between C3 and C7. MRI of the head showed cerebral infarction in the right VA area, while vertebral angiography with the head rotated to the right revealed that the right VA was occluded at the level of C3-C4. The patient was successfully treated via posterior cervical fusion from C2 to C7. Patients with rheumatoid arthritis have a potential risk of cervical bony ankyloses. Cervical laminoplasty for patients with cervical bony ankyloses can induce rotational VA occlusion due to spinal rotational instability.

Multiple synostoses syndrome: Clinical report and retrospective analysis.

Multiple synostoses syndrome (SYNS1; OMIM# 186500) is a rare autosomal dominant disorder reported in a few cases worldwide. We report a Chinese pedigree characterized by proximal symphalangism, conductive hearing loss, and distinctive facies. We examined the genetic cause and reviewed the literature to discuss the pathogeny, treatment, and prevention of SYNS1. Audiological, ophthalmological, and radiological examinations were evaluated. Whole-exome sequencing (WES) was performed to identify mutations in the proband and her parents. Sanger sequencing was used to verify the results for the proband, parents, and grandmother. The literature on the genotype-phenotype correlation was reviewed. The patient was diagnosed with multiple synostoses syndrome clinically. WES and bioinformatic analysis revealed a novel missense mutation in the NOG gene, c.554C>G (p.Ser185Cys), cosegregated in this family. The literature review showed that the phenotype varies widely, but the typical facies, conductive hearing loss, and proximal symphalangism occurred frequently. All reported mutations are highly conserved in mammals based on conservation analysis, and there are regional hot spots for these mutations. However, no distinct genotype-phenotype correlations have been identified for mutations in NOG in different races. Regular systematic examinations and hearing aids are beneficial for this syndrome. However, the outcomes of otomicrosurgery are not encouraging owing to the regrowth of bone. This study expanded the mutation spectrum of NOG and is the first report of SYNS1 in a Chinese family. Genetic testing is recommended as part of the diagnosis of syndromic deafness. A clinical genetic evaluation is essential to guide prevention, such as preimplantation genetic diagnosis.