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Table of Contents   
CASE REPORT  
Year : 2021  |  Volume : 32  |  Issue : 4  |  Page : 533-536
Complete ankylosis of temporomandibular joint and cervical spine – A case report


Department of Oral and Maxillofacial Surgery, Balaji Dental and Craniofacial Hospital, Teynampet, Chennai, India

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Date of Submission04-Feb-2022
Date of Decision04-Mar-2022
Date of Acceptance05-Mar-2022
Date of Web Publication18-May-2022
 

   Abstract 


Ankylosing spondylitis (AS) is a chronic, multi-systemic inflammatory disorder. It rarely involves peripheral joints such as Temporomandibular joint (TMJ). Here, we present a case of a 44-year-old male, known case of AS for eight years who had TMJ-related complaints for two years. On examination, bony ankylosis of TMJ possibly due to pre-existing AS was made. A bilateral standard gap arthroplasty procedure was done. The challenges faced during the surgical procedure are discussed.

Keywords: Ankylosing spondylitis, gap arthroplasty, temporomandibular joint ankylosis

How to cite this article:
Balaji S M, Balaji P. Complete ankylosis of temporomandibular joint and cervical spine – A case report. Indian J Dent Res 2021;32:533-6

How to cite this URL:
Balaji S M, Balaji P. Complete ankylosis of temporomandibular joint and cervical spine – A case report. Indian J Dent Res [serial online] 2021 [cited 2022 Dec 7];32:533-6. Available from: https://www.ijdr.in/text.asp?2021/32/4/533/345416



   Background Top


Ankylosing spondylitis (AS) is a rare, multi-systemic, chronic inflammatory disorder that primarily involves axial skeleton. Disease process targets females, along the synovial and cartilaginous articulations as well as the sites of tendon and ligament attachment to bone. The current diagnostic criteria consider axial symptoms, limitation of spinal movement and radiological evidence of sacroiliitis. AS has a female predilection and most of the AS patients show excess of human leucocyte antigen (HLA-B27). AS is characterized by enthesopathy of the axial skeleton progressing to ossification and ankylosis of fibrocartilaginous joints.[1] Involvement of Temporomandibular Joint (TMJ) in AS are rare and estimated to range between 4 and 37%. The AS disease process destroys the joint capsule or components of the disc, leading to synovitis, breakdown of the articular surfaces. These changes lead to abnormal craniocervical postural changes. In spite of these changes, TMJ ankylosis is rare probably due to the presence of the intra-articular disc as a physical barrier. However, when involved, quality of life would be tremendously affected.[1],[2] The aim of the manuscript is to present successful surgical management of a male patient with AS and TMJ ankylosis.


   Case Report Top


A 44-year-old male, diagnosed 10 years back with AS and on regular treatment reported to the center with slowly progressive history of restriction in mouth opening and morning stiffness of jaw over the past two years and notably during the recent past. Patient had a bilateral hip replacement seven years back which till date posed no issues. At that time, he had been diagnosed with glenohumeral and acromioclavicular arthritis, calcification of supraspinatus and subscapularis muscle tendons. Patient had no relatable history of trauma or infection of the TMJ. Physical examination revealed severe limitation of spine mobility and Schober's test was positive. There was no neurological deficit. Vertical mouth opening was less than 1 mm and preauricular tenderness was present [Figure 1]. Investigations showed patient to be anemic, erythrocyte sedimentation rate 70 mm; C-reactive protein 35, and positive human leukocyte antigen-B27. Imaging revealed evidence of syndesmophytes bridging the vertebral bodies with a classic 'bamboo spine' appearance and sclerosis around the apophyseal joint. Orthopantomogram showed loss of joint space in bilateral TMJ [Figure 2]. Volume rendered CT images of TMJ revealed complete bony ankylosis of the TMJ. Based on the history, clinical and radiological evidence a diagnosis of TMJ ankylosis possibly due to pre-existing AS was made [Figure 3].
Figure 1: (a) Preoperative view showing a 44-year-old male with jaw stiffness and completely restricted mouth opening, (b) Complete inability to turn his head due to the fusion of the spinal column caused by ankylosing spondylitis

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Figure 2: (a) 3DCT scan showing the extent of cervical spondylitis and fusion of cervical bones. (b and c) CT image, 3D reconstruction showing the extent of cervical spondylitis, fusion of cervical bones and ankylosis of right and left temporomandibular joint

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Figure 3: (a-c) CT image, 3DCT scan of spine showing evidence of syndesmophytes bridging the vertebral bodies with a classical bamboo spine appearance with sclerosis around the apophyseal joint

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As the quality of life was drastically affected, surgical intervention was planned. The goal of the surgery was to ensure total removal of the ankylotic mass, restoring the form and function of the joint, increase the mouth opening and prevention of recurrence.

Surgery

After standard preparation, general anesthesia was administered. It was challenging as laryngoscopy and patient positioning was difficult due to complete trismus of the temporomandibular joint and fixed flexion deformity of the cervical spine. Additionally challenges were in relation to intubation and securing the airway, awake extubation, and postoperative maintenance of a patent airway. To make procedure comfortable and predictable, fibreoptically assisted nasotracheal intubation via bronchoscope-nasotracheal intubation were carried out. Using a standard Al-Kayat-Bramley incision and full thickness flap elevation, dissection was done to reach the right TMJ [Figure 4]. Ankylosed was identified and standard gap arthroplasty done using autogenous temporalis muscle as a myofascial flap to prevent reankylosis [Figure 5].[3] Hemostasis was achieved, suction drain placed and sutured using silk, ovicryl™ and ethilon™ in layers. Then left TMJ was similarly corrected. Appropriate antibiotics, painkillers and post-operative maintenance instructions were provided. Healing was uneventful. Mouth opening at end of two weeks was 38 mm and end of four months was 37 mm [Figure 6]. Patient was followed up for 24 months with no reduction or physical complaints involving TMJ.
Figure 4: (a and b) Al-Kayat-Bramley incision placed and ankylosed TMJ identified bilaterally

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Figure 5: (a and b) Gap arthroplasty done bilaterally

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Figure 6: (a) Immediate post-operative view. (b) Post-operative view after three months showing complete recovery of facial muscles with good motor function and adequate mouth opening

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   Discussion Top


Patients with AS with a bilateral TMJ involvement, often have severe degenerative form. This causes chronic pain and a negative impact on quality of life. It drastically alters psychological status and functional capacities. Treating such patient poses anesthetical and surgical challenge.

The pathology of AS primarily affects enthuses (place where ligaments, tendons and capsules are attached to bone) by immune mechanism causing inflammation of bone and bone related tissues, bone erosion and syndesmophyte (spur) formation. The disease is attributed to genetics with HLA-B27; far are ERAP1, IL-23R, ANTXR2 and IL-1R2 genes.[4] These genes interact possibly with immune reaction, microbial interleukin, environmental agents and endocrinal abnormity. They stimulate the interlukin 23/17 (IL-23/IL-17) pathway, triggering lymphocyte activation and differentiation as well as innate cytokines.[5] The mechanism of initiation of AS from HLA-B27 have been postulated by unfolded-protein-response hypothesis, arthritogenic-peptide hypothesis and free-heavy-chain hypothesis.[4] The gene and signaling products cause rapid, abnormal remodeling of bone joints leading to painful conditions. Most of such damage is restricted to the axial skeleton. There is very less incidence of the peripheral joints such as TMJs have been reported. The etiopathogenesis of TMJ involvement in SA is severely debated with several theories being postulated and incidence disputed. Earlier studies of 1970s have remarkably concluded that TMJ ankylosis like symptoms may be the result of abnormal postural effects of the ankylosing spondylitis and not the disease itself. In later studies from the 1990s, 50 AS patient's clinical and radiographical features were studied.[6] Of this, 10 of them showed radiological evidence while only one had clinical features. This study highlighted that TMJ pathology is rare among AS patients and symptoms are often secondary to muscle spasm, occlusal factors, and postural imbalance.[7] Few early authors reported that the restricted mouth opening in the patients with AS could be attributed to the proximity of the chin to the neck, the area where AS invariably affects severely. Later on, a few authors attributed the difficulty in opening the mouth to flattening and erosions of the mandibular condyle and elongation of the mandibular coronoid process. In these reports, AS patients uniformly reported of experiencing pain, stiffness, headache and restricted movements in TMJ. These were later believed to emanate from the compression of the retrodiscal TMJ tissues.[6],[7]

Clinically, the severe cervical spine stiffness and associated deformities render clinical, dental and radiological examination uncomfortable, or even painful, to sit in the desired position. In the operation theatre, the stiffness poses extreme difficulty while patient is under general anaesthesia as the patient feedback would be missing. Hence the patient/surgeon's position is challenging. This would mean that patient cannot provide feedback and surgeon-anesthetist needs to take a call blindly based on their experience, expertise and knowledge. If associated inflammation of the costovertebral joints of the chest wall occur, limitation of chest expansion could complicate issues.

Our present case is a male which by itself is quite uncommon. The slow progression of the TMJ pain and reduced mouth opening is indicative of the worsening disease process. It could have begun as a destruction of the capsular or disc attachment leading to an internal derangement and subsequent degenerative joint diseases. Alternatively, it could have started as a primary synovitis with direct breakdown of the articular surfaces. In such an instance, the derangement would be a result of articular surface changes and not cause these changes. There may be hypermobility due to destruction of capsular attachment. Disc derangement or fibrosis of the capsule could lead to hypomobility, as seen in our present case. Also, there is a possibility of a combined presentation that had finally led to the total ankylosis.

The possibility of early or late involvement of TMJ in AS patients should not be ignored. Examination of TMJ should be made a part of regular clinical examination to identify, manage and if required intervene surgically earlier for better quality of life. The rarity of the condition and multitude of spectrum of the condition prevents the surgical fraternity to draw effective treatment algorithm for appropriate time of surgery and nature of intervention. Till such a protocol is developed, the surgeon's expertise and skill needed to be entrusted. Within the limited evidence, gap arthroplasty is relied to provide best relief.[8] In our present case too, the same has been employed with satisfactory result.


   Conclusion Top


Patients with AS may have TMJ involvement. Clinicians, particularly dentists should look for progressive signs and symptoms of TMJ ankylosis. When conservative treatment fails, surgical therapy would be helpful. The cervical AS poses surgical and anesthetic challenges during the surgery. The options and precautions need to be listed and discussed.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Arora P, Amarnath J, Ravindra SV, Rallan M. Temporomandibular joint involvement in ankylosing spondylitis. BMJ Case Rep 2013;2013:bcr2013009386. doi: 10.1136/bcr-2013-009386.  Back to cited text no. 1
    
2.
Souza RC, de Sousa ET, Sousa D, Sales M, Dos Santos Oliveira R, Mariano MH, et al. Prevalence of temporomandibular joint disorders in patients with ankylosing spondylitis: A cross-sectional study. Clin Cosmet Investig Dent 2021;13:469-78.  Back to cited text no. 2
    
3.
Balaji SM. Modified temporalis anchorage in craniomandibular reankylosis. Int J Oral Maxillofac Surg 2003;32:480-5.  Back to cited text no. 3
    
4.
Tam LS, Gu J, Yu D. Pathogenesis of ankylosing spondylitis. Nat Rev Rheumatol 2010;6:399-405.  Back to cited text no. 4
    
5.
Zhu W, He X, Cheng K, Zhang L, Chen D, Wang X, et al. Ankylosing spondylitis: Etiology, pathogenesis, and treatments. Bone Res 20195;7:1-6. doi: 10.1038/s41413-019-0057-8.  Back to cited text no. 5
    
6.
Souza RC, de Sousa ET, Sousa D, Sales M, dos Santos Oliveira R, Mariano MH, et al. Prevalence of temporomandibular joint disorders in patients with ankylosing spondylitis: A cross-sectional study. Clin Cosmetic Investig Dent 2021;13:469-78.  Back to cited text no. 6
    
7.
Locher MC, Felder M, Sailer HF. Involvement of the temporomandibular joints in ankylosing spondylitis (Bechterew's disease). J Cranio-Maxillofac Surg 1996;24:205-13.  Back to cited text no. 7
    
8.
Katsnelson A, Markiewicz MR, Keith DA, Dodson TB. Operative management of temporomandibular joint ankylosis: A systematic review and meta-analysis. J Oral Maxillofac Surg 2012;70:531-6.  Back to cited text no. 8
    

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Correspondence Address:
Dr. S M Balaji
Director and Consultant Oral and Maxillofacial Surgeon, Balaji Dental and Craniofacial Hospital, 30, KB Dasan Road, Teynampet Chennai – 600 018, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijdr.ijdr_117_22

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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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