Fibrous Ankylosis of the Temporomandibular Joint in a Young Child ABSTRACT

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1 JDC CASE REPORT Fibrous Ankylosis of the Temporomandibular Joint in a Young Child Nigel R. Figueiredo, BDS, MDS 1 Manoj Meena, BDS, MDS 2 Ajit D. Dinkar, BDS, MDS 3 Manisha M. Khorate, BDS, MDS 4 ABSTRACT Temporomandibular joint (TMJ) ankylosis is an intracapsular union of the disccondyle complex to the temporal articular surface that restricts mandibular movements, including fibrous adhesions or bony fusion between the condyle, disc, glenoid fossa, and articular eminence. The leading causes include trauma and infection. It can be a serious and disabling condition that leads to difficulties in mastication, swallowing, speaking, esthetics and oral hygiene. Disturbances of facial and mandibular growth and acute compromise of the airway invariably result in physical and psychological disability. Treatment should be initiated as soon as the condition is recognized, with the main objective of re-establishing joint function and harmonious jaw function. The purpose of this report is to describe a case of unilateral fibrous ankylosis of the right TMJ in a three-year-old girl. (J Dent Child 2015;82(2):108-11) Received March 27, 2014; Last Revision May 17, 2014; Revision Accepted May 20, Keywords: ankylosis, fibrous, temporomandibular joint Temporomandibular joint (TMJ) ankylosis is a disorder that leads to restriction of mouth opening, ranging from partial reduction to complete immobility of the jaw. 1 The most common causes of TMJ ankylosis are trauma and local/systemic infection, which most commonly occurs secondary to contiguous spread from otitis media or mastoiditis but may also result from hematogenous spread of infectious conditions such as tuberculosis, gonorrhea, or scarlet fever. 2 Other etiologic factors implicated include rheumatoid arthritis, Paget s disease, ankylosing spondylitis, psoriasis, and burns. 3 1 Dr. Figueiredo is a senior resident; 3 Dr. Dinkar is professor and head, and 4 Dr. Khorate is an assistant professor, all in the Oral Medicine and Radiology Department, Goa Dental College and Hospital, Bambolim, Goa, India. 2 Dr. Meena is a senior lecturer, Rajasthan Dental College, Jaipur, Rajasthan, India. Correspond with Dr. Figueiredo at nigel_06@yahoo.co.in This disabling condition causes speech impairment, difficulty with mastication, poor oral hygiene, and abnormalities of facial growth, all of which generate significant psychological stress. 4 In children, TMJ ankylosis can result in mandibular retrognathism with concurrent esthetic and functional deficits. Therefore, treatment should be initiated as soon as the condition is recognized. The purpose of this paper is to describe a case of unilateral fibrous ankylosis of the right TMJ causing difficulty in mouth opening, and problems in esthetics and oral hygiene in a three-year-old girl. CASE DESCRIPTION A three-year-old girl was brought to the Oral Medicine and Radiology Department, Goa Dental College and Hospital, Bambolim, Goa, India, by her parents with a chief complaint of inability to open her mouth wide. The child had rheumatic fever 12 days after birth, with swelling over the right ankle and right TMJ, and behind 108 Figueiredo et al Fibrous ankylosis of temporomandibular joint

the right ear. There was no history of facial hollowness or feeding difficulty at birth, trauma or facial palsy.

mouth (Figure 1). The mouth opening was restricted to two mm (Figure 2).

Intra-oral examination revealed fair oral hygiene and a caries-free dentition.

2 the right ear. There was no history of facial hollowness or feeding difficulty at birth, trauma or facial palsy. Extraoral examination revealed facial asymmetry (bird-face deformity), with hollowness of the face, hypoplastic mandible, and deviation of the mandible to the right side on attempting to open the mouth (Figure 1). The mouth opening was restricted to two mm (Figure 2). A single café au lait macule measuring four by three cm was present over the abdomen, and a healed scar was observed over the dorsum of the right foot. The voice exhibited a nasal sound. Intra-oral examination revealed fair oral hygiene and a caries-free dentition. Computed tomography (CT) imaging of both TMJs revealed hypoplasia of the right mandibular ramus and a poorly developed right condylar fossa. There was deformity of the condylar process on the right side with broad articulation at the right TMJ and cortical irregularity. The left joint appeared normal (Figures 3 and 4). The findings of the CT scan confirmed the diagnosis of fibrous ankylosis of the right TMJ. Surgeons from the Oral and Maxillofacial Surgery Department of the Goa Dental College and Hospital performed a gap arthroplasty under general anesthesia at the university hospital. She was admitted to the oral surgery ward for 10 days. Following the surgery, the patient was prescribed amoxicillin and clavulanic acid (125mg/25mg IV every 8 hours) and acetaminophen (125mg suppositories twice daily) for seven days, and the post-operative course was uneventful. A liquid diet was advised for the first week, followed by a soft diet for the next three weeks. The patient s parents were also asked to meticulously maintain her oral hygiene. Oral physiotherapy was advised four times a day, for five-10 minutes each time, using a specially designed carrot-shaped key for six months. The patient was made to slowly bite on it, initially at the thin tip and gradually at the thicker portions. Two months later, the patient s interincisal opening had increased to 15 mm (Figure 5); The patient was followed up for another four months with no evidence of any complications. Dental appointments were scheduled in the department of Pedodontics and Preventive Dentistry at the same institution. The patient was lost to follow-up after six months because her father was transferred out of Goa. Figure 1. Extraoral view (preoperative). Figure 2. Preoperative mouth opening (severely restricted). Figure 3. Computed tomography scan (sagittal section) of: (A) right temporomandibular joint showing deformity and broad articulation of condyle; and (B) left temporomandibular joint showing a normal condyle. Fibrous ankylosis of temporomandibular joint Figueiredo et al 109

Figure 4. Computed tomography scan (3-D reconstruction) of: (A) right temporomandibular joint; and (B) left temporomandibular joint. Figure 5. Postoperative mouth opening.

5 TMJ ankylosis has been classified as either true or false.

3 Figure 4. Computed tomography scan (3-D reconstruction) of: (A) right temporomandibular joint; and (B) left temporomandibular joint. Figure 5. Postoperative mouth opening. DISCUSSION TMJ ankylosis is defined as the union of the condyle to the articular surface of the glenoid fossa, resulting in restriction of mandibular movements. 5 TMJ ankylosis has been classified as either true or false. True ankylosis is a condition that results in osseous or fibrous adhesion between the surfaces of the TMJ, within the limits of the articular capsule; false ankylosis results from diseases not directly related to the joint, such as muscle spasm or myositis ossificans. 1 True ankylosis may be further subdivided into bony (the condyle or ramus is attached to the temporal bone by an osseous bridge) or fibrous (soft tissue union of joint components). 6 TMJ ankylosis can also be classified according to the location (intra- articular versus extra-articular) and extent of fusion (complete versus incomplete). 7 TMJ ankylosis occurs predominantly in the first decade of life, and males and females are equally affected. 6 Children are more prone to ankylosis because of greater osteogenic potential and an incompletely formed disc. 1 Patients have a history of progressively restricted jaw opening or may have a longstanding history of limited opening. 6 Facial asymmetry is the classic feature in unilateral cases, 1 with the chin deviating toward the affected side. 3 Vertical height of the affected side is shorter when compared with the unaffected side. Pain and tenderness are unusual. 7 A complete absence of mouth opening and recession of the chin are typical characteristics of the bilateral ankylosis. 3 The severity of ankylosis is diagnosed by evaluating the degree to which the mouth opening is restricted. Radiographic assessment is essential and critical in evaluating and treating TMJ ankylosis patients. 7 Radiographic imaging, CT scans, and magnetic resonance images play an important role in determining any abnormality in the bone or soft tissue of the joints. 8 The treatment of TMJ ankylosis poses a significant challenge because of technical difficulties and a high incidence of recurrence. 5 There is no gold standard of treatment, and results have been variable and often less than satisfactory. The most frequently reported surgical interventions include gap arthroplasty, interpositional arthroplasty, and joint reconstruction. In gap arthroplasty, a resection of bone is created between the articular cavity and mandibular ramus without any interposition of material, while interpositional arthroplasty involves the addition of interpositional material between the new sculptured glenoid fossa and condyle. In joint reconstruction, the TMJ is reconstructed with autogenous/ alloplastic materials or total joint prosthesis. 4,9 Physiotherapy is also vital to further improve mouth opening and mainly to prevent reankylosis. Psychological rehabilitation for the affected child and parents before, during, and after surgical treatment is also important. 110 Figueiredo et al Fibrous ankylosis of temporomandibular joint

4 TMJ ankylosis is a serious and disabling condition that can result in structural and functional deformities. When it occurs during childhood, it can severely affect the growth and development of the jaws and teeth. In addition to that, it can exert a negative influence on the psychosocial development of the patient because of the obvious facial deformity. Early diagnosis and treatment are vital in re-establishing joint function and esthetics and preventing psychological/emotional or social problems. REFERENCES 1. Das UM, Keerthi R, Ashwin DP, Venkata Subramanian R, Reddy D, Shiggaon N. Ankylosis of temporomandibular joint in children. J Indian Soc Pedod Prevent Dent 2009;27: Rishiraj B, McFadden LR. Treatment of temporomandibular joint ankylosis: a case report. J Can Dent Assoc 2001;67: Guven O. A clinical study on temporomandibular joint ankylosis. Auris Nasus Larynx 2000;27: Bortoluzzi MC, Sheffer MA. Treatment of temporomandibular joint ankylosis with gap arthroplasty and temporal muscle/fascia graft: a case report with five-year follow-up. Rev Odonto Ciênc 2009;24: Chaturvedi A, Rawal M. A unique case of trauma from occlusion: TMJ fibrous ankylosis. Indian J Oral Sci 2012;3: Petrikowski CG. Diagnostic Imaging of the Temporomandibular Joint. In: White SC, Pharoah MJ, eds. Oral Radiology - Principles and Interpretation. 5th ed. St. Louis (Missouri): Mosby; 2004: Cunha CO, Pinto LM, Mendonca LM, Saldanha AD, Conti AC, Conti PC. Bilateral asymptomatic fibrous-ankylosis of the temporomandibular joint associated with rheumatoid arthritis: a case report. Braz Dent J 2012;23: Vibhute PJ, Bhola M, Borle RM. TMJ Ankylosis: multidisciplinary approach of treatment for dentofacial enhancement a case report. Case Rep Dent 2011;2011: doi: /2011/ Epub 2011 Sep Vasconcelos BC, Porto GG, Bessa-Nogueira RV, Nascimento NM. Surgical treatment of temporo mandibular joint ankylosis: follow-up of 15 cases and literature review. Med Oral Patol Oral Cir Bucal 2009;14:E34-8. Fibrous ankylosis of temporomandibular joint Figueiredo et al 111

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Report of Ankylosis of the Temporomandibular Joint: Treatment with a Temporalis Muscle Flap and Augmentation Genioplasty

Report of Ankylosis of the Temporomandibular Joint: Treatment with a Temporalis Muscle Flap and Augmentation Genioplasty Abstract A case of true bilateral ankylosis of the temporomandibular joint (TMJ)