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Table of Contents   
CASE REPORT  
Year : 2015  |  Volume : 26  |  Issue : 2  |  Page : 210-213
Non-syndromic bilateral condylar aplasia: A rare case


Department of Oral Medicine and Radiology, Raja Rajeswari Dental College and Hospital, Ramholli Cross, Kumbalgodu, Bengaluru, Karnataka, India

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Date of Submission01-Aug-2013
Date of Decision02-Apr-2015
Date of Acceptance17-Apr-2015
Date of Web Publication22-Jun-2015
 

   Abstract 

The temporomandibular joint is one of the most complex joints of the human body. It consists of the mandibular condyle, glenoid fossa and the articular eminence of the temporal bone. Aplasia of the condyle is usually seen as part of a syndrome otherwise it is rare. We report a case of bilateral condylar aplasia in a 20-year-old male not associated with any syndrome. The patient reported to the department with a chief complaint of the underdeveloped lower jaw. Clinical examination, conventional radiographs, and 3D computer tomography images revealed a complete absence of condyle on the right and left sides.

Keywords: Condylar aplasia, condylar hypoplasia, mandibular condyle

How to cite this article:
Shivhare P, Lata S, Balaji P, Gupta A. Non-syndromic bilateral condylar aplasia: A rare case. Indian J Dent Res 2015;26:210-3

How to cite this URL:
Shivhare P, Lata S, Balaji P, Gupta A. Non-syndromic bilateral condylar aplasia: A rare case. Indian J Dent Res [serial online] 2015 [cited 2018 Oct 19];26:210-3. Available from: http://www.ijdr.in/text.asp?2015/26/2/210/159173
Temporomandibular joint (TMJ) is considered a ginglymous diarthrodial joint capable of both rotational and translatory movements. It consists of the mandibular condyle, glenoid fossa and the articular eminence of the temporal bone. The congenital deformities and developmental abnormalities of the mandibular condyle can be classified as hypoplasia or aplasia, hyperplasia, and bifidity. Hypoplasia or aplasia of the mandibular condyle indicates underdevelopment or nondevelopment associated mainly with various craniofacial abnormalities. These may be either congenital or acquired. [1]

Growth disturbances in the development of mandibular condyle may occur in utero late in the first trimester and may result in disorders such as aplasia or hypoplasia of the mandibular condyle. As compared to hypoplasia, hyperplasia of the mandibular condyle is not visible at birth and seems to be gradually acquired during growth. [1]


   Case Report Top


A 20-year-old male reported to the oral medicine and radiology department with a chief complaint of the underdeveloped lower jaw, which was first noticed during childhood and gradually progressed. There was no history of any trauma or any systemic diseases during birth. Patient's parents gave a history of consanguineous marriage. There was no family history of the present problem.

The general physical examination did not reveal any abnormalities. The vital signs were within normal limits. Extraoral examination revealed facial asymmetry with severe retruded mandible [Figure 1] and [Figure 2]. Mouth opening was restricted (25 mm) with no deviation or deflection [Figure 3]. Condyles were not palpable on both sides. Intraorally there was crowding in upper and lower anterior teeth with increased overjet and overbite. The molar relation was angles class II bilaterally. Deep palate was also seen. No other important clinical extraoral or intraoral findings were observed. Based on clinical findings, a provisional diagnosis of bilateral ankylosis and differential diagnosis of bilateral condylar hypoplasia or aplasia were given.
Figure 1: Frontal view of patient

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Figure 2: Lateral view of patient shows severe retruded mandible

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Figure 3: Restricted mouth opening (25 mm)

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After clinical examination, radiographic examinations were performed. Panoramic radiograph showed complete absence of condyle on the right side and left a side. Glenoid fossa was not developed on both sides. Ramus and body of the left side were comparatively smaller than the right side. Antigonial notch was prominent bilaterally [Figure 4]. Posterioanterior view showed facial asymmetry [Figure 5]. Lateral skull views showed severe retruded and micrognathic mandible [Figure 6]. Condylar aplasia on right and left sides was confirmed by reverse town and transpharngeal views. Computer tomography (CT) was advised for additional information. 3D CT showed complete condylar aplasia on the left side and condylar aplasia with two bony projections on right side [Figure 7], [Figure 8], [Figure 9], [Figure 10]. Magnetic resonance imaging showed complete absence of articular disc bilaterally [Figure 11]. After radiographic confirmation, patient was advised complete systemic evaluation and referred to General medicine, cardiology, ophthalmology, ear, nose, and throat and orthopedics to rule out any syndromes. The medical evaluation revealed no abnormalities. Based on the clinical and radiographic findings, a final diagnosis of nonsyndromic agenesis of right and left condyle was given. Patient was referred to oral surgeon and orthodontist for the best possible treatment.
Figure 4: Orthopantogram shows condylar aplasia on right side and left side, prominent anti-gonial notch and hypoplasia of mandible

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Figure 5: Posterioanterior view reveals asymmetry

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Figure 6: Lateral skull. Radiograph shows severe retruded mandible

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Figure 7: 3D computed tomography right lateral-shows absence of glenoid fossa and complete absence of the condyle

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Figure 8: 3D computed tomography left lateral-shows absence of glenoid fossa and complete absence of the condyle

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Figure 9: 3D computed tomography reconstruction of mandible showing condylar aplasia on right side

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Figure 10: 3D computed tomography reconstruction of mandible showing condylar aplasia on left side

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Figure 11: Magnetic resonance imaging showed complete absence of articular disc bilaterally

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   Discussion Top


The TMJ initially develops as two separate areas of mesenchymal blastemas that are, condylar blastema and temporal blastema near the eventual location of the mandibular condyle and glenoid fossa in the 8 th week of gestation. [2],[3] The mandibular condyle and temporal blastemas move towards each other as the joint develops by 12 th week. At birth, the articular surfaces of both the mandibular condyle and temporal bones are covered with fibrous connective tissue. Later, this tissue is slowly converted to fibrocartilage as the fossa deepens, and the mandibular condyle develops under functional influences. [4],[5]

Congenital (primary) condylar hypoplasia is characterized by unilateral or bilateral underdevelopment of the mandibular condyle and usually occurs as part of some systemic condition originating in the first and second branchial arches [Table 1]. [1],[6],[7],[8]
Table 1: Syndromes and their skeletal and general manifestations[14]


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Acquired (secondary) condylar hypoplasia takes place if the condyle is injured during active growth because of which development may be arrested. The most common causes are mechanical injury, such as trauma (before the age of 2), infection of the joint itself or the middle ear, childhood rheumatoid arthritis, radiotherapy and Parathyroid hormone-related protein deficiency, which affect bone formation and chondrocyte differentiation. [1],[9],[10]

In a newborn child, the TMJ is mobile. After the eruption of the first permanent dentition at the age of 7 years, the articular eminence begins to be prominent, and its development continues at an accelerated pace until 12 years of age. If a condyle is present, an adaptive well-defined fossa and articular eminence should be present. Hence, in the case of the absence of a condyle, those coherent structures should also be absent. [11],[12] In our case, the complete absence of the right and left condyle and glenoid fossa were observed.

Aplasia of the mandibular condyle without any other facial malformations is an extremely rare condition. [8] There are very less cases of nonsyndromic mandibular condyle aplasia reported. [9],[13],[14],[15],[16],[17],[18] Our case also presented condylar aplasia without any other features suggestive of any syndrome.

Various treatment approaches have been proposed for treating condylar aplasia and possibilities for influencing mandibular growth. Most of the time, it is treated by multimode with the help of an oral surgeon, general surgeon, plastic surgeon, and orthodontics. [9],[17],[19]

The treatment could then be a costochondral graft transplant, preferably before the growth spurt, orthognathic surgery at the end of the growth period, or both. [19] Surgery is often required, but the timing and regimen of this choice are still an issue to be resolved. [17]


   Conclusion Top


Nonsyndromic condylar hypoplasia and aplasia are exceedingly rare conditions. We report a rare case of bilateral total condylar aplasia, not related to any pathological disorder. This condition should be separated from different associated syndromes based on different clinical features and investigations. A very few case reports are published in the literature, to date. Hence, our case is very special in this aspect.

 
   References Top

1.
Kaneyama K, Segami N, Hatta T. Congenital deformities and developmental abnormalities of the mandibular condyle in the temporomandibular joint. Congenit Anom (Kyoto) 2008;48:118-25.  Back to cited text no. 1
    
2.
Buchbinder D, Kaplan AS. Biology. In: Kaplan AS, Assael LA, editors. Temporomandibular Disorders Diagnosis and Treatment. Philadelphia, PA: Saunders; 1991. p. 11-23.  Back to cited text no. 2
    
3.
Choi JW, Kim JT, Park JH, Park EK, Kim SY, Kwon TG, et al. Gp130 is important for the normal morphogenesis of Meckel′s cartilage and subsequent mandibular development. Exp Mol Med 2007;39:295-303.  Back to cited text no. 3
    
4.
Cleall JF. Postnatal craniofacial growth and development. In: Laskin DM, editor. Oral and Maxillofacial Surgery. Vol. 1. St. Louis: Mosby; 1980. p. 70-107.  Back to cited text no. 4
    
5.
Pruzansky S. Postnatal development of craniofacial malformations. J Dent Res 1968;47:936.  Back to cited text no. 5
    
6.
DeLone DR, Brown WD, Gentry LR. Proteus syndrome: Craniofacial and cerebral MRI. Neuroradiology 1999;41:840-3.  Back to cited text no. 6
    
7.
Morgan KA, Rehman MA, Schwartz RE. Morquio′s syndrome and its anaesthetic considerations. Paediatr Anaesth 2002;12:641-4.  Back to cited text no. 7
    
8.
Ozturk S, Sengezer M, Isik S, Gul D, Zor F. The correction of auricular and mandibular deformities in auriculo-condylar syndrome. J Craniofac Surg 2005;16:489-92.  Back to cited text no. 8
    
9.
Krogstad O. Aplasia of the mandibular condyle. Eur J Orthod 1997;19:483-9.  Back to cited text no. 9
    
10.
Shibata S, Suda N, Fukada K, Ohyama K, Yamashita Y, Hammond VE. Mandibular coronoid process in parathyroid hormone-related protein-deficient mice shows ectopic cartilage formation accompanied by abnormal bone modeling. Anat Embryol (Berl) 2003;207:35-44.  Back to cited text no. 10
    
11.
Gorlin RJ, Cohen MM, Hennekan RC. Syndromes of Head and Neck. 4 th ed. New York: Oxford University Press; 2001.  Back to cited text no. 11
    
12.
Sperber GH. Temporomandibular Joint. In Craniofacial Development Hamilton. Ontariop: BC Decker Inc.; 2001. p. 17-8.  Back to cited text no. 12
    
13.
Prowler JR, Glassman S. Agenesis of the mandibular condyles; diagnostic findings and treatment of deformity by polyethylene implant. Oral Surg Oral Med Oral Pathol 1954;7:133-9.  Back to cited text no. 13
    
14.
Akihiko I, Ritsuo T, Junichi F, Nobuyuki I, Yutaka T. Agenesis of bilateral mandibular condyle-report of a case. J Jpn Stomatol Soc 2006;55:187-92.  Back to cited text no. 14
    
15.
Santos KC, Dutra ME, Costa C, Lascala CA, Lascala CE, de Oliveira JX. Aplasia of the mandibular condyle. Dentomaxillofac Radiol 2007;36:420-2.  Back to cited text no. 15
    
16.
Bowden CM Jr, Kohn MW. Mandibular deformity associated with unilateral absence of the condyle. J Oral Surg 1973;31:469-72.  Back to cited text no. 16
    
17.
Canger EM, Celenk P. Aplasia of the mandibular condyle associated with some orthopaedic abnormalities. Dentomaxillofac Radiol 2012;41:259-63.  Back to cited text no. 17
    
18.
Shivhare P, Shankarnarayan L, Jambunath U, Kumar M, Sowbhagya MB. Condylar aplasia and hypoplasia: A rare case. Case Rep Dent 2013;2013:745602.  Back to cited text no. 18
    
19.
Vargervik K, Kaban LB. Hemifacial microstomia diagnosis and management. In: Bell WH, editor. Modern Practice in Orthognathic and Reconstructive Surgery. Vol. 2. Philadelphia: W.B. Saunders Company; 1992. p. 1533-60.  Back to cited text no. 19
    

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Correspondence Address:
Dr. Peeyush Shivhare
Department of Oral Medicine and Radiology, Raja Rajeswari Dental College and Hospital, Ramholli Cross, Kumbalgodu, Bengaluru, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-9290.159173

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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11]
 
 
    Tables

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