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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 29  |  Issue : 4  |  Page : 314-316

An Unusual Association of Bifid Condyle, Eagle's Syndrome and Ely's Cyst: A New Kid on the Block


1 Department of Oral Medicine and Radiology, Haldia Institute of Dental Sciences and Research, Haldia, West Bengal, India
2 Department of Oral Medicine and Radiology, Murshidabad Medical College, Murshidabad, West Bengal, India

Date of Submission22-Jun-2017
Date of Acceptance10-Jan-2018
Date of Web Publication15-Feb-2018

Correspondence Address:
Dr. Biyas Bhowmik
Department of Oral Medicine and Radiology, Haldia Institute of Dental Sciences and Research, Balughata - 721 645, Haldia, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaomr.jiaomr_55_17

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   Abstract 


Bifid condyle is a rare anatomic modification of mandibular condyle caused by developmental or traumatic reasons, and often diagnosed incidentally on routine radiographic examination. Eagle's syndrome (ES) occurs when an elongated styloid process or calcified stylohyoid ligament causes recurrent throat pain or foreign body sensation, dysphagia, or orofacial pain. On the other hand, Ely's cysts are focal subchondral cysts resulting from bony remodelling and osteoarthritic changes. Here, we report a case depicting an interesting combination of all the abovementioned entities in a single patient.

Keywords: Bifid condyle, Eagle's syndrome (ES), Ely's cysts, syndrome


How to cite this article:
Sinha R, Bhowmik B, Sarkar S, Khaitan T. An Unusual Association of Bifid Condyle, Eagle's Syndrome and Ely's Cyst: A New Kid on the Block. J Indian Acad Oral Med Radiol 2017;29:314-6

How to cite this URL:
Sinha R, Bhowmik B, Sarkar S, Khaitan T. An Unusual Association of Bifid Condyle, Eagle's Syndrome and Ely's Cyst: A New Kid on the Block. J Indian Acad Oral Med Radiol [serial online] 2017 [cited 2019 Aug 21];29:314-6. Available from: http://www.jiaomr.in/text.asp?2017/29/4/314/225559




   Introduction Top


A rare condition of duplicated or lobulated mandibular condyle is known as bifid condyle characterized by partial or complete separation of condyle into lateral and medial halves. It was first reported by Hrdlicka in 1941 in a series of skull specimens.[1],[2] Eagle's syndrome (ES), or stylohyoid syndrome, is another commonly encountered painful condition caused by an abnormally elongated styloid process greater than 30 mm described by Dr. Watt Eagle in 1937.[3] Another common radiologic finding associated with temporomandibular joint (TMJ) disorders is Ely's cyst, which is nothing but rounded radiolucent areas just below the cortical plate or deep in the trabecular bone. Such cysts are also called subcortical or subchondral cysts.[4]


   Case Report Top


A 28-year-old male patient reported to the Department of Oral Medicine and Radiology with the chief complaint of pain and discomfort in TMJ, neck, and shoulder for 6 months. Episodic headache had become more frequent in the last 3 months. The neck pain was insidious in origin, moderate in intensity, dull, throbbing, and aggravated on looking upwards and flexing the neck to either side. His medical history was noncontributory, except an injury on the mandible at the age of 20 years, with a laceration scar on the soft tissue of the chin. Extraoral inspection revealed no facial asymmetry and deviation of the jaw to the left by 4 mm. Palpation revealed a small, circular, tender, bony hard projection with ill-defined margins, measuring approximately 4 mm in diameter in the right jugulodigastric region at the anterior border of the sternocleidomastoid muscle. Intraorally, tenderness on palpation could be elicited from the right lateral pterygoid muscle and paratonsillar fossa.

Radiological investigations were performed. Panoramic radiograph (OPG) and TMJ open and closed views revealed an elongated styloid process measuring 40.6 mm on the left side and 41.5 mm on the right [Figure 1] and [Figure 2]. The right condylar head orientation was in the mediolateral direction and revealed a prominent vertical notch anteroposteriorly on the articulating surface, dividing the condyle into medial and lateral heads. Morphologically, the left condylar head appeared to have a lobulated surface whereas the right condyle appeared to have a heart-like morphology. Both the condylar heads showed well-defined radiolucency on the medial aspect – oval-to circular in shape, circumscribed on the periphery by a well-corticated margin. Based on the clinical and radiological features, the case was diagnosed as of ES, bilateral bifid condyle, and Ely's cyst. Therapeutic approach was conservative, with the patient being prescribed analgesic, anti-inflammatory, and muscle relaxants. Occlusal splint was fabricated and physiotherapy was started to relax the muscles, correct the occlusion, and provide symptomatic relief.
Figure 1: Elongated styloid process present bilaterally as first detected on panoramic radiograph (OPG), which is the screening radiograph

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Figure 2: TMJ open and closed views revealed elongated styloid processes measuring 40.6 mm on the left side and 41.5 mm on the right

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   Discussion Top


A bifid condyle may be discovered incidentally on routine screening radiographic examinations such as OPG. Its appearance results from a vertical depression, notch, or deep cleft in the centre of the condylar head or an actual duplication of the condylar head. Coronal or sagittal planes of three-dimensional imaging such as cone beam computed tomography (CBCT) or computed tomography (CT) scan is ideal for the visualization of bifid condyle.[3],[4],[5] Proposed etiology is the appearance of a well-vascularized fibrous septa in the condylar cartilage at approximately 20 weeks of intrauterine life, and extending into the cavity of the developing ascending ramus, resulting in bifid condyle.[5] Mostly asymptomatic, bifidism occasionally exhibits clinical symptoms such as pain, swelling, restriction of mandibular movement, trismus, ankylosis, and facial asymmetries along with TMJ disorders. Osteoarthrosis might develop in cases resulting from trauma.[1],[3] Radiological investigation begins with OPG, followed by open and closed TMJ views and CBCT/CT.[3],[4] Treatment modalities include conservative approach, consisting of analgesics and anti-inflammatory agents, muscle relaxants, physiotherapy and occlusal splint, and surgical intervention.[1],[2]

ES is characterized by elongated styloid process through ossification of the stylohyoid ligament. Ossification can be simply an “anatomical variation” or results through activation of the remnants of the original connective and fibrocartilaginous cells secondary to trauma leading to “reactive hyperplasia,” or “reactive metaplasia.”[6] The styloid process, derived from the second branchial arch of Reichert's cartilage, is an elongated tapered projection originating in the petrous portion of the temporal bone and usually measures 20–30 mm.[3] Protracted styloid process longer than 30 mm in an adult is considered to be abnormal, and shows a female predilection.[6]

Two forms of ES are recognized: the “classic syndrome” can develop when there is compression of cranial nerves V, VII, IX, and X by the scar tissue under the tonsillar fossa after tonsillectomy. “Foreign body” sensation in the throat, pharyngodynia, dysphagia, tinnitus, and referred otalgia are the commonly encountered symptoms. In the “styloid-carotid artery syndrome,” additional symptoms of headache and pain occur due to irritation of the sympathetic nerve plexus by the elongated styloid process, which compresses the internal carotid and external carotid arteries.[7],[8]

The diagnosis of ES must be based on medical history and physical examination. Careful intraoral palpation by placing the index finger in the tonsillar fossa and applying gentle pressure should confirm the tip of an elongated styloid process.[8] If pain is reproduced by palpation and either referred to the ear, face, or head, the diagnosis of an elongated styloid process is very likely done. Injection of local anesthetic into tonsillar fossa relieves pain and can be used as a diagnostic tool.[8]

Initial radiological examination calls for an OPG (screening radiograph), followed by CT, which is the best modality for ascertaining the length and angulation of the styloid process and for evaluating anatomical relationships between the stylohyoid complex and adjacent structures. According to few authors, panoramic or lateral radiographs are sufficient for diagnosing the disease.[3],[6],[9]

Treatment for ES can be nonsurgical or surgical. Pain control may be achieved by oral analgesics or transpharyngeal infiltration of steroids and local anesthetics in the tonsillar fossa. The treatment of choice is styloidectomy, which can be performed transorally or extraorally.[10],[11]

Ely's cysts are small, round, radiolucent areas with irregular margins surrounded by areas of variable increased density deep to the articulating surfaces. These lesions are not true cysts, but are areas of degeneration that contain fibrous tissue, granulation tissue, and osteoid.[4] These pseudocysts result from constant remodeling of the subchondral trabecular bone, phases of bony erosion, followed by accelerated bone turnover.[3],[12]


   Conclusion Top


Bifid condyle, ES, and Ely's cyst may be prompted by trauma and may be contemporaneously present in a single individual. The importance of diagnosing this uncommon, yet unsuspected disease comprising these three entities should be emphasized. Depending on the patient's clinical presentation, the righteous therapeutic approach, consisting of a blend of conservative, pharmacological, and/or surgical treatment should be undertaken. Can this interesting clinical combination of these three entities be called a syndrome?

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.
de Moraes Ramos FM, de Vasconcelos Filho JO, Manzi FR, Boscolo FN, de Almeida SM. Bifid mandibular condyle: A case report. J Oral Sci 2006;48:35-7.  Back to cited text no. 1
    
2.
Lubambo de Melo S, Novais Barbosa JM, Carvalho Peixoto A, de Santana Santos T, Gerbi M. Bilateral bifid mandibular condyle: A case report. Int J Morphol 2011;29:922-6.  Back to cited text no. 2
    
3.
Som PM, Curtin HD. Head and Neck Imaging. 4th Ed. St Louis: Mosby; 2003.  Back to cited text no. 3
    
4.
White SC, Pharoah MJ. Oral Radiology: Principles and Interpretation. 7th ed. St Louis: Elsevier Mosby; 2014.  Back to cited text no. 4
    
5.
Blackwood HJ. The double-headed mandibular condyle. Am J Phys Anthropol 1957;15:1-8.  Back to cited text no. 5
[PUBMED]    
6.
Piagkou M, Anagnostopoulou S, Kouladouros K, Piagkos G. Eagle's syndrome: A review of the literature. Clin Anat 2009;22:545-58.  Back to cited text no. 6
[PUBMED]    
7.
Pinheiro TG, Soares VY, Ferreira DB, Raymundo IT, Nascimento LA, Oliveira CA. Eagle's Syndrome. Int Arch Otorhinolaryngol 2013;17:347-50.  Back to cited text no. 7
[PUBMED]    
8.
Montalbetti L, Ferrandi D, Pergami P, Savoldi F. Elongated styloid process and Eagle's syndrome. Cephalalgia 1995;15:80-93.  Back to cited text no. 8
[PUBMED]    
9.
Murtagh RD, Caracciolo JT, Fernandez G. CT findings associated with Eagle's syndrome. Am J Neuroradiol 2001;22:1401-2.  Back to cited text no. 9
[PUBMED]    
10.
Khandelwal S, Hada YS, Harsh A. Eagle's syndrome – A case report and review of the literature. Saudi Dent J 2011;23:211-5.  Back to cited text no. 10
[PUBMED]    
11.
Ceylan A, Koybasioglu A, Celenk.F, Yilmaz O, Uslu S. Surgical treatment of elongated styloid process: Experience of 61 cases. Skull Base 2008;18:289-95.  Back to cited text no. 11
    
12.
Ferrazzo KL, Osório LB, Ferrazzo VA. CT images of a severe TMJ osteoarthritis and differential diagnosis with other joint disorders. Case Rep Dent 2013;2013:242685.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2]



 

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