Ankyloblepharon-ectodermal dysplasia-clefting (AEC) syndrome is an ectodermal dysplasia presenting with cleft lip or palate and congenital filiform eyelid fusion. This is a case report of a 1-year and 6-month-old girl with AEC syndrome presenting with temporomandibular joint ankylosis who underwent release of temporomandibular joint ankylosis, coronoidectomy, bilateral cheiloplasty, Tajima rhinoplasty, and repair of lower lip pits under general anesthesia. Fiberoptic nasotracheal intubation was done successfully using a two-stage technique originally described by Stiles. As necessitated by the surgical plan, video laryngoscope guidance was subsequently used to facilitate the conversion from nasotracheal to orotracheal intubation.

Objective: To report a case of isolated ankyloglossia superior in a one-month-old boy.

Methods:

Design: Case Report

Setting: Tertiary Government Training Hospital 

Patient: One

Results: A one-month-old boy with failure to thrive, feeding difficulties and a palatoglossal band that limited mouth opening and anterior posturing of the tongue was diagnosed to have non-syndromic ankyloglossia superior and underwent surgical transection of the fibrous ankylosis under intravenous sedation due to difficulty of insinuating an endotracheal tube orally. Direct latch breastfeeding was successfully adapted from the second to tenth postoperative day before they were lost to follow up.

Conclusion: Despite a complicated pediatric airway, coordinated surgical and anesthesia management successfully restored tongue function and mouth opening to allow effective feeding.

The human homologue of mouse progressive ankylosis protein(ANKH)is an inorganic pyrophosphate transport regulator,which regulates tissue mineralization by controlling the level of inorganic pyrophosphate.It plays an important role in the pathogenesis and development of bone and joint diseases,such as ankylosing spondylitis,craniometaphyseal dysplasia,and articular cartilage calcification.This review summarizes the progress of research on ANKH and the above-mentioned diseases.
Ankylosis ; Humans ; Hyperostosis ; Hypertelorism ; Joint Diseases ; Mice ; Mutation

OBJECTIVE: Mandibular condyle injury usually results in malocclusion and disharmony of facial growth in growing children. This study aimed to evaluate the long-term effects of autogenous coronoid grafts on the facial growth of children with unilateral temporomandibular joint (TMJ) ankylosis who underwent mandibular condyle reconstruction. METHODS: Ten growing patients with unilateral bony TMJ ankylosis admitted in West China Hospital of Stomatology, Sichuan University between January 1st, 2008 and December 31st, 2016 were followed up and evaluated. These patients include three males and seven females with ages ranging from 5 years to 12 years at the time of surgery. Each patient underwent gap arthroplasty, condyle reconstruction with ipsilateral coronoid, and interposition of the pedicled temporalis fascial flap in a single operation. The postoperative follow-up ranged from 3 years to 8 years with an average of 4.9 years. Postoperative panoramic radiographs determined the growth of the mandibular height and length on the affected side and compared it with those of the healthy side. RESULTS: All patients recovered uneventfully after surgery. At the end of follow-up period, the maximal mouth opening ranged from 32 mm to 41 mm with an average of 35.6 mm. Mandibular height and length continued to grow after the successful treatment of ankylosis using autogenous coronoid grafts for condyle reconstruction. However, growth deficit still existed. The final ramus height and mandibular length of the affected side at the end of follow-up increased by 25.3% (P<0.05) and 26.1% (P<0.05), respectively, compared with the initial values measured immediately after surgery. Growth rates of ramus height and mandibular length of the affected side were 47.1% and 27.2% lower (P<0.05) than those of the healthy side, respectively. CONCLUSIONS Mandibular height and length continued to grow after the successful treatment of ankylosis by using autogenous coronoid grafts for condyle reconstruction. However, growth deficit still existed. The growth rate of the affected mandible was reduced compared with that of the undisturbed side even after treatment of ankylosis.
Ankylosis ; Bone Transplantation ; Child ; Child, Preschool ; China ; Female ; Humans ; Male ; Mandibular Condyle ; Temporomandibular Joint ; Temporomandibular Joint Disorders

Calcium hydroxide (CH) is the gold-standard intracanal dressing for teeth subjected to traumatic avulsion. A common complication after the replantation of avulsed teeth is root resorption (RR). The current review was conducted to compare the effect of CH with that of other intracanal medications and filling materials on inflammatory RR and replacement RR (ankylosis) in replanted teeth. The PubMed and Scopus databases were searched through June 2018 using specific keywords related to the title of the present article. The materials that were compared to CH were in 2 categories: 1) mineral trioxide aggregate (MTA) and endodontic sealers as permanent filling materials for single-visit treatment, and 2) Ledermix, bisphosphonates, acetazolamide, indomethacin, gallium nitrate, and enamel matrix-derived protein (Emdogain) as intracanal medicaments for multiple-visit management of avulsed teeth prior to the final obturation. MTA can be used as a single-visit root filling material; however, there are limited data on its efficacy due to a lack of clinical trials. Ledermix and acetazolamide were comparable to CH in reducing RR. Emdogain seems to be an interesting material, but the data supporting its use as an intracanal medication remain very limited. The conclusions drawn in this study were limited by the insufficiency of clinical trials.
Acetazolamide ; Ankylosis ; Bandages ; Calcium Hydroxide ; Calcium ; Dental Enamel ; Diphosphonates ; Gallium ; Indomethacin ; Miners ; Pemetrexed ; Replantation ; Root Resorption ; Tooth Ankylosis ; Tooth Avulsion ; Tooth Replantation ; Tooth

The incidence of heterotopic ossification in adolescents appears to be lower than in adults. There exist very few reports of heterotopic ossification with total bony ankylosis in child or adolescent populations. We describe a case of total bony ankylosis of the elbow secondary to heterotopic ossification, in a 14-year-old female. Total ankylosis of the elbow at 45 degrees of flexion was noted 6 months post-surgery, and complete surgical excision of the heterotopic mass was performed. After an additional one-time dose of radiation therapy and nonsteroidal anti-inflammatory drug medication, full range of motion was obtained without any recurrence or other complications, up to the last follow-up of 30 months.
Adolescent ; Adult ; Ankylosis ; Child ; Dislocations ; Elbow ; Female ; Follow-Up Studies ; Fractures, Bone ; Humans ; Incidence ; Ossification, Heterotopic ; Range of Motion, Articular ; Recurrence

OBJECTIVES: The objective of this study was to highlight the role of lateral arthroplasty along with interposition of the buccal fat pad (BFP) in the management of Sawhney type III temporomandibular joint (TMJ) ankylosis. MATERIALS AND METHODS: Ten patients with TMJ ankylosis (7 unilateral and 3 bilateral, total of 13 joints) were treated with lateral arthroplasty and BFP interposition. The bony bridge of the ankylotic mass on the lateral aspect was resected, leaving a distance of 1.5 to 2.0 cm from the base of the skull to the neck of the condyle. The condyle was left intact. Coronoidectomy was performed on the ipsilateral side via the same approach in all cases. The inter-incisal opening was measured at that time, and if it was less than 35 mm, contralateral coronoidectomy was performed by using the intra-oral approach. After satisfactory inter-incisal mouth opening (≥35 mm) was achieved, the TMJ surgical site was revisited, and BFP was retrieved and used to cover the lateral aspect of the medially placed condyle. RESULTS: With lateral arthroplasty, the medially displaced condyle can be left in-situ to maintain the mandibular ramal height and function and to act as a growth center in children. Interposition of the BFP prevents reformation of the lateral bony bridge that was removed. CONCLUSION: Lateral arthroplasty along with interpositioning of the BFP is a novel technique for managing Sawhney type III ankylosis that achieves management goals while avoiding complex and advanced reconstructive surgical procedures.
Adipose Tissue ; Ankylosis ; Arthroplasty ; Child ; Humans ; Mouth ; Neck ; Reconstructive Surgical Procedures ; Skull ; Temporomandibular Joint

OBJECTIVE: To evaluate the feasibility of computer-aided design virtual mandibular position in the simultaneous treatment of children with temporomandibular joint ankylosis (TMJA) and jaw deformity. METHODS: The children with unilateral TMJA were surgically treated from January 2016 to June 2017 in the Department of Oral and Maxillofacial Surgery. The image data of jaw and dentition were imported into the computer software to simulate the surgical procedure. An osteotomy of the affected side was performed to remove ankylosed bone mass. Then the mandible was rotated around the unaffected condyle to eliminate the chin deviation by the virtual plan. An open bite was thus created on the affected side to provide space for vertical midfacial growth. In the virtual mandibular position, the affected ramus was reconstructed with costochondral grafting. Finally, digital occlusal splint was designed and prefabricated by 3D printing. During surgery, temporomandibular joint ankylosis was released and mandibular position was guided in place by the digital occlusal splint. In the new mandibular position, costochondral grafting was completed. After the surgery, the occlusal splint was wired to the mandibular dentition and would be gradually adjusted by grinding off the maxillary side to promote downward growth of the maxilla until the open bite was eliminated. The ramus height and chin deviation were measured before and one week after the surgery to validate the method. Comparisons of the measurements were made by means of a repeatedmeasures analysis of variance (ANOVA) (P=0.05). Pairwise multiple comparisons were conducted using the Bonferroni correction (P=0.05). RESULTS: Five patients were included in this study. Under the guidance of the digital occlusal splint, the mandible could reach the preoperative designed position smoothly in all the cases. The chin deviations were (0.58±0.20) mm in the virtual plan before surgery, and (0.70±0.27) mm after surgery, which were not significant statistically (P>0.05). The ramus heights on the affected side were (48.19±3.20) mm in the virtual plan before surgery, and (48.17±3.62) mm after surgery, which were not significant statistically (P>0.05). CONCLUSION It is feasible and reliable in the simultaneous treatment of TMJA with jaw deformity under the guidance of virtual mandibular position.
Ankylosis ; Child ; Humans ; Mandible ; Mandibular Condyle ; Temporomandibular Joint ; Temporomandibular Joint Disorders

The aim of this study was to present a case series of the orthognathic treatment of facial asymmetry due to temporomandibular joint (TMJ) ankylosis and to characterize the current treatment modalities through a literature review. Four patients who presented with facial asymmetry due to TMJ ankylosis between 2010 and 2014 were included in this study. TMJ ankylosis was surgically treated before bimaxillary surgery with advancement genioplasty in some of the cases. In 2 cases, 3-dimensional (3D) models were used for diagnosis and treatment planning, as 3D models are very important tools for planning surgical maneuvers. Aesthetically pleasant facial symmetry and a good facial profile were obtained in all the cases.
Ankylosis ; Cephalometry ; Diagnosis ; Facial Asymmetry ; Genioplasty ; Humans ; Maxillofacial Development ; Orthognathic Surgery ; Temporomandibular Joint

PURPOSE: Neurogenic myositis ossificans (NMO) in patients with traumatic spinal cord or brain injuries can cause severe joint ankylosis or compromise neurovascularture. The purpose of this study was to evaluate the clinical and radiological outcomes of and review considerations relevant to surgical resection of NMO of the hip joint. MATERIALS AND METHODS: Six patients (9 hips) underwent periarticular NMO resection between 2015 and 2017. The medical records of these patients were retrospectively reviewed. Preoperative computed tomography including angiography was performed to determine osteoma location and size. Improvement in hip motion allowing sitting was considered the sole indicator of a successful surgery. The anterior approach was used in all patients. The ranges of motion (ROM) before and after surgery were compared. RESULTS: The mean time from accident to surgery was 3.6 years. Average ROM improved from 24.3°(flexion and extension) to 98.5°(flexion and extension) after surgery, and improvement was maintained at the last follow-up. No commom complications (e.g., deep infection, severe hematoma, deep vein thrombosis) occurred in any patient. Improvement in ROM in one hip in which surgical resection was performed 10 years after the accident was not satisfactory owing to the pathologic changes in the joint. CONCLUSION: Surgical excision of periarticular NMO of the hip joint can yield satisfactory results, provided that appropriate preoperative evaluation is performed. Early surgical intervention yields satisfactory results and may prevent the development of intra-articular pathology.
Angiography ; Ankylosis ; Brain Injuries ; Follow-Up Studies ; Hematoma ; Hip Joint ; Hip ; Humans ; Joints ; Medical Records ; Myositis Ossificans ; Myositis ; Osteoma ; Pathology ; Retrospective Studies ; Spinal Cord ; Veins