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 Table of Contents  
CASE REPORT
Year : 2014  |  Volume : 26  |  Issue : 3  |  Page : 323-326

Right zygomatico coronoid ankylosis: A rare clinical entity


1 Departments of Oral Medicine and Radiology, Narsinhbhai Patel Dental College and Hospital, Visnagar, Gujarat, India
2 Departments of Oral and Maxillofacial Surgery, Narsinhbhai Patel Dental College and Hospital, Visnagar, Gujarat, India

Date of Submission13-Oct-2014
Date of Acceptance09-Nov-2014
Date of Web Publication19-Nov-2014

Correspondence Address:
Kamala Rawson
Professor, Department of Oral Medicine and Radiology, Narsinhbhai Patel Dental College and Hospital, Visnagar - 384 315, Gujarat
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-1363.145019

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   Abstract 

Trismus is defined as a limitation in the mouth- or jaw-opening ability due to reduced mandibular mobility. Limited mouth opening can be caused by several disorders affecting the masticatory system. Most of them involve the temporomandibular joint (TMJ) and the masticatory muscles. Ankylosis of the TMJ is a common cause for the occurrence of trismus. Ankylosis of the TMJ may be extra- or intra-articular. Zygomatico coronoid ankylosis is a rare type of extra-articular ankylosis. In case of limitation of mouth opening, coronoid process locking may be overlooked, because attention is generally focused on the TMJ. The union between the coronoid process and zygomatic arch may be due to bony ankylosis or fibrous ankylosis. It may follow facial fractures caused by gunshots, treated and untreated fractures of the zygomatic complex, with or without a concomitant fracture of the coronoid process, mandibular fractures, extension of the intracapsular ankylosis or due to the enlargement of the coronoid process. A review of literature has disclosed 15 reported cases of this disorder. The true incidence of this condition may be even higher. Zygomatico coronoid ankylosis may have been encountered by many clinicians, and yet may have been misdiagnosed or overlooked. The present article reports a rare entity of a post-traumatic case of trismus, for a duration of 18 years, diagnosed as right zygomatico coronoid ankylosis.

Keywords: CBCT, coronoid process, coronoidectomy, TMJ, zygomatico coronoid ankylosis


How to cite this article:
Kallalli BN, Rawson K, Manugutti A, Sulaga S. Right zygomatico coronoid ankylosis: A rare clinical entity . J Indian Acad Oral Med Radiol 2014;26:323-6

How to cite this URL:
Kallalli BN, Rawson K, Manugutti A, Sulaga S. Right zygomatico coronoid ankylosis: A rare clinical entity . J Indian Acad Oral Med Radiol [serial online] 2014 [cited 2022 May 22];26:323-6. Available from: https://www.jiaomr.in/text.asp?2014/26/3/323/145019


   Introduction Top


Temporomandibular joint (TMJ) ankylosis is classified into two types, intra-articular or true ankylosis and extra-articular or false ankylosis. Fibrous or bony adhesion between the coronoid process and the zygomatic arch is a rare cause of extra-capsular/extra-articular ankylosis. [1] In case there is a limitation of mouth opening, coronoid process locking may be overlooked, because attention is generally focused on the TMJ. [2] This condition may occur due to facial fractures caused by gunshots, treated and untreated fractures of the zygomatic complex, with or without a concomitant fracture of the coronoid process, mandibular fractures, extension of the intracapsular ankylosis or due to the enlargement of the coronoid process. [1],[3]

Enlargement of the coronoid process of the mandible was first described by Langenbeck in 1853, and joint formation between the coronoid process and the zygoma was first described by Jacob in 1899. [1] Zygomatico coronoid ankylosis was first described by Brown and Peterson. A review of English literature has reported 15 cases of this entity. Two cases were caused by infection, two cases occurred subsequent to surgical interventions, and eleven cases were caused by trauma. Among these eleven cases, eight cases were caused by trauma to the coronoid process and three cases were caused by trauma to the zygoma. The treatment consists of surgical excision of the coronoid process followed by vigorous jaw opening exercises. [2]


   Case Report Top


A 42-year-old male patient reported to the Department of Oral Medicine and Radiology with a history of difficulty in opening his mouth, since 18 years. The patient had a history of trauma to the face due to a road traffic accident and underwent surgery for the same, 18 years ago. One month postoperatively he started noticing a gradual decrease in the mouth opening, which had led to a lock jaw within three months of surgery. Since then the patient was on a liquid diet. He also had difficulty in speech. On clinical examination, the face was bilaterally asymmetrical due to the depression on the right zygomatic process of the maxilla, with complete trismus [Figure 1]. A healed surgical scar was present on the right temporal region. On condylar palpation, no rotatory or translatory movement was discernible on the right side and limited movement was discernible on the left side. A detailed intraoral examination could not be performed due to complete trismus [Figure 2]. On the basis of these findings a provisional diagnosis of post-traumatic right TMJ ankylosis was made. With these clinical findings, differential diagnoses of right coronoid hyperplasia, right zygomatico coronoid ankylosis, and tetany were included.
Figure 1: Frontal view of the patientt

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Figure 2: Photograph showing complete trismus

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The patient was subjected to further investigations. An orthopantomograph (OPG) revealed probable re-ankylosis of the right joint and an irregular shaped radio-opaque mass was noticed over the right zygoma. There was alteration in the shape of the right coronoid process, which was enlarged, deformed, and was in continuation with the bony mass over the right zygoma. The OPG further revealed the presence of bone plates in the floor of the maxillary sinus, right zygomatic arch, right coronoid process, infraorbital margin, and the lateral orbital margin, and wires over the floor of the maxillary sinus and infraorbital margin. An enlargement of the left coronoid process was also noticed, although it was not very apparent [Figure 3]. A cone-beam computerized tomography (CBCT) scan revealed a unified and ankylosed zygomatic arch and coronoid process on the right side, and enlargement of the left coronoid process [Figure 4]a and b.
Figure 3: OPG of the patient

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Figure 4: CBCT image of (a) right side (b) left side

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Bilateral surgical resection of the coronoid processes was performed under general anesthesia, with an intraoral approach. During the operative procedure, even after right coronoidectomy, adequate mouth opening could not be achieved (perhaps due to the enlargement of the left coronoid process), and hence, bilateral coronoidectomy was performed. The ankylosed structures were also resected, and the inner aspect of the right zygomatic arch was smoothened to prevent re-ankylosis. Increased mouth opening of 30 mm was achieved immediately after the surgery [Figure 5]a. The patient was instructed to perform mouth opening exercises postoperatively and follow-up was done after one week, wherein the mouth opening increased to 33 mm [Figure 5]b and [Figure 6].
Figure 5: Postoperative photograph of the patient showing increased mouth opening (a) immediately after surgery (b) after one week

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Figure 6: Postoperative OPG of the patient

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   Discussion Top


Ankylosis is the stiffening or immobility and fixation of a joint. Temporomandibular joint ankylosis is a structural disease that produces functional and esthetic disability in the form of limited mouth opening and facial deformity. [4] It is most commonly associated with trauma (13 to 100%), local or systemic infection (0 to 53%), or systemic diseases, such as ankylosing spondylitis, rheumatoid arthritis or psoriasis. [5],[6] Temporomandibular joint ankylosis may be classified on the basis of location (intra- or extra-articular), type of tissue involved (bony, fibrous, or fibro-osseous), and extent of fusion (complete or incomplete). Ankylosis may be true or false. Any condition that gives rise to an osseous or fibrous adhesion between the surfaces of the TMJ is true ankylosis. False ankylosis results from pathological conditions not directly related to the joint. [5] Fibrous or bony adhesion between the coronoid process and the zygoma is a rare cause of extracapsular ankylosis. [1],[3],[6]

The pathogenesis of ankylosis is not clear. Furthermore, heterotopic ossification is rarely encountered in the maxillofacial region. As a sequel of trauma, infection or surgery, metaplastic changes in connective tissue elements, which do not normally have an osteogenic potential, may cause heterotopic ossification. [1] Histologically, the proliferating connective tissue with fibroblasts in transition to osteoblasts, and areas of cartilage, osteoid, and bone are seen. [3]

Although zygomatico coronoid ankylosis is a rare clinical condition, in cases of limitation of mouth opening, the coronoid process also must be evaluated. Only 15 cases have been reported in literature, the details of which are mentioned in [Table 1]. Eleven of the 15 cases have been caused by trauma, which were similar to the present case. To be more specific, the etiologic factor in this case was trauma due to an accident. Cases of direct trauma to the zygoma were first reported by Findlay in 1972. Kellner et al. (1979) and Rikalainen et al. (1981) reported similar cases. Eight cases were caused by direct trauma to the coronoid process. In the present case, there were no jaw movements suggestive of bony ankylosis. According to Cooper and Finch, the lesion arises following the formation of a hematoma at the time of the original trauma, with subsequent fibrosis and differentiation of cartilage-forming cells. The production of cartilage rather than bone might be due to the stress of continued mandibular movement. The treatment is obviously surgical. [2] In the present case, bilateral surgical resection of the coronoid process was performed under general anesthesia, with an intraoral approach. The ankylosed structures were also resected, and the inner aspect of the right zygomatic arch was smoothened for preventing recurrence.
Table 1: Details of the 15 cases reported in literature[2]

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   Conclusion Top


To conclude, zygomatico coronoid ankylosis is a very rare entity. Only few cases have been reported so far; hence, the oral physicians should have a thorough knowledge of the clinical and radiographic features of this rare entity, and should correlate the clinical and radiographic findings in cases of ankylosis, for appropriate diagnosis. Also in cases of ankylosis, instead of concentrating only on the TMJ, the coronoid process should also be evaluated. Furthermore, follow-up of the lesion is necessary, as it has a high recurrence rate, and in order to prevent this, postoperative physiotherapy is required.

 
   References Top

1.Tippu SR, Rahman F. Heterotopic calcification: A cause for zygomatico-coronoid ankylosis. Biomedical Res 2011;22:211-4.  Back to cited text no. 1
    
2.Güven O. Zygomatico coronoid ankylosis: A rare clinical condition leading to limitation of mouth opening. J Craniofac Surg 2012;23:829-30.  Back to cited text no. 2
    
3.Agarwal M, Gupta DK, Tiwari AD, Jakhar SK. Extra-articular ankylosis after zygoma fracture: A case report and review of literature. J Oral Biol Craniofac Res 2013;3:105-7.  Back to cited text no. 3
    
4.Murad N, Rasool G. Trauma as a most frequent cause of TMJ ankylosis. Pakistan Oral and Dental Journal 2011;31:45-7.  Back to cited text no. 4
    
5.Vasconcelos BC, Bessa-Nogueira RV, Cypriano RV. Treatment of temporomandibular joint ankylosis by gap arthroplasty. Med Oral Patol Oral Cir Bucal 2006;11:E66-9.  Back to cited text no. 5
    
6.Vanhove F, Dom M. Zygomatico-coronoid ankylosis: A case report. Int J Oral Maxillofac Surg 1999;28:258-9.  Back to cited text no. 6
    


    Figures

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

  [Table 1]


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