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 Table of Contents  
CASE REPORT
Year : 2015  |  Volume : 27  |  Issue : 3  |  Page : 445-448

Unilateral condylar aplasia: A case report


1 Department of Oral Medicine and Radiology, Yogita Dental College and Hospital, Ratnagiri, Maharashtra, India
2 Department of Oral Medicine and Radiology, School of Dental Sciences, Krishna Institute of Medical Sciences Deemed University, Karad, Maharashtra, India
3 Department of Oral Medicine and Radiology, Guru Nanak Dev Dental College, Sunam, Punjab, India

Date of Submission01-Dec-2014
Date of Acceptance17-Nov-2015
Date of Web Publication25-Nov-2015

Correspondence Address:
Abhijeet Rajendra Sande
Department of Oral Medicine and Radiology, School of Dental Sciences, Krishna Institute of Medical Sciences Deemed University, Karad, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-1363.170491

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   Abstract 

Growth disturbances in the mandibular condyle region include aplasia, hypoplasia, and hyperplasia. Non-syndromic aplasia of the mandibular condyle is very rare, and may be due to congenital or acquired causes. The mandibular condyle is regarded as the center of mandibular growth. Therefore, regional disturbances during the growth phase may negatively impact mandibular growth. We present a case of unilateral condylar aplasia associated with severe hypoplasia of the posterior body and ascending ramus of the mandible.

Keywords: Aplasia, condyle, mandible


How to cite this article:
Joshi SS, Sande AR, Sunil MK, Suragimath A. Unilateral condylar aplasia: A case report. J Indian Acad Oral Med Radiol 2015;27:445-8

How to cite this URL:
Joshi SS, Sande AR, Sunil MK, Suragimath A. Unilateral condylar aplasia: A case report. J Indian Acad Oral Med Radiol [serial online] 2015 [cited 2021 Oct 16];27:445-8. Available from: https://www.jiaomr.in/text.asp?2015/27/3/445/170491


   Introduction Top


Condylar hypoplasia or underdevelopment of mandibular condyle can be either congenital or acquired. Congenital condylar hypoplasia is associated with head and neck syndromes, while acquired condylar hypoplasia results from disturbance of growth center of the developing condyle. The most common cause is mechanical trauma during active growth, though other causes may include inflammation in temporomandibular joint (TMJ) area, rheumatoid arthritis, and radiotherapy. [1] When not seen in concurrence with any other developmental anomalies, aplasia of mandible is an extremely rare condition. [2] The condyle is the primary growth center which is essential for mandibular development and any disturbance in this center leads to a corresponding inhibition of mandibular growth. [3],[4] This report presents a unique case of unilateral condylar aplasia with no association of any syndrome.


   Case Report Top


A 12-year-old female patient presented to the Department of Oral Medicine, Diagnosis and Radiology, with the chief complaint of facial asymmetry. The asymmetry was noticed by the parents when the patient was 5 years old. She had history of normal delivery with full-term birth. Past dental history revealed that the child had suffered from a dental infection and swelling along with pus discharge on the left side of the face when she was 4 years of age. Due to poor socioeconomic condition, the parents consulted a local physician who prescribed some medication at that time, but the specifics of the prescription were not available. A year after, her parents noted some amount of facial asymmetry, especially on the left side [Figure 1], which worsened with increasing age. General physical and systemic examinations were unremarkable. Extraoral examination revealed that the right side of the patient's face had less fullness as compared to the left. The malar prominence was decreased on the left side. The rest of the face and the auricles were normal. The mouth opening was within normal limits, but appeared to be deflecting toward the left side [Figure 2]. On intra-auricular palpation, no movement was felt in the left TMJ, with tenderness over the right side. Intraorally, cross-bite in relation to 23 and posterior open bite on the right side was present. On the basis of history and clinical findings, a provisional diagnosis of condylar aplasia of left side was made. Differential diagnoses of hemifacial microsomia, chronic osteomyelitis, and Gorham's disease were considered.
Figure 1: Extraoral photograph showing facial asymmetry and fullness on the left side

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Figure 2: Extraoral photograph showing deviation of the mandible to the left side on mouth opening

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On radiographic examination, panoramic radiograph (OPG) revealed complete absence of the head and neck of condyle on the left side with a prominent and large articular eminence. The coronoid process was elongated and larger in size, and the sigmoid notch was shallow and smaller in size on the left side. Also, the left articular fossa was ill defined. There was severe constriction in the region of the angle of the mandible causing reduced height and width of the ramus and posterior body of mandible, with a concavity along the inferior border of the mandibular angle. Prominent antegonial notching was seen on the left side along with complete absence of the gonial angle. The right side of the mandible appeared to be normal [Figure 3]. To rule out condylar aplasia subsequent to middle ear infection, the patient was referred to an otorhinolaryngologist, but the otoscopic findings were not significant. Due to unavailability of Cone Beam Computed Tomography (CBCT) center, the patient was referred for a computed tomography (CT) scan accompanied by three-dimensional (3D) reconstruction. Findings on the CT scan (coronal views) confirmed those on the panoramic radiograph [Figure 4]a, b and [Figure 5]. As noted in the 3D reconstruction, there was absence of the head and neck of the condyle on the left side [Figure 6] and [Figure 7]. Based on history, clinical examination, and radiographic investigation, final diagnosis of unilateral condylar aplasia on the left side was made. No treatment was given at the time of presentation. Follow-up and corrective surgery was planned for the patient at a later date after completion of mandibular growth.
Figure 3: OPG revealing absence of the left mandibular condyle (arrow) with ill-defined articular fossa

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Figure 4: (a) Coronal section CT revealing absence of head and neck of the condyle on the left side. (b) Coronal section CT revealing absence of head and neck of the condyle on the left side at a different level

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Figure 5: Coronal section CT revealing deviation of the mandible to the left side

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Figure 6: 3D CT reconstruction revealing absence of head and neck of the condyle on the left side

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Figure 7: 3D CT reconstruction revealing lower border of the mandible

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   Discussion Top


This case demonstrates non-syndromic, isolated unilateral condylar aplasia, with marked underdevelopment of the left posterior body and ramus of mandible. The growth of the mandibular condyle and its related structures may be affected due to many causes. Any alteration in the condyle's size or shape affects the TMJ and results in facial abnormalities. According to developmental circumstances, mandibular hypoplasia can be classified into three groups: Congenital, developmental, and acquired. From a morphological and clinical standpoint, hypoplasia or aplasia can be (i) primary or (ii) secondary. [5] Alterations, including condylar aplasia, are present in several syndromes like hemifacial microsomia, Goldenhar syndrome, Treacher Collins syndrome, Proteus syndrome, Morquio syndrome, and auriculocondylar syndrome. [6] The present case lacked other clinical features of these syndromes. Also, aplasia is commonly associated with an associated failure of development of some part of the adjacent auditory apparatus. In our case, no deformity of the auditory apparatus was noted. [3]

The development of the condyle may be arrested due to mechanical injury such as trauma, infection of the joint itself or the middle ear, and childhood rheumatoid arthritis. This may result in facial deformity which may be evident after months of injury. In unilateral condylar hypoplasia, the continued growth of the contralateral side causes deviation toward the affected side and results in a cross-bite relationship of the teeth since the ramus and body of the mandible remain underdeveloped, [5] which was evident in our case. Osteomyelitis involving the TMJ is relatively rare. Bony ankylosis may follow suppuration of the joint, especially in children. In some cases, there may be deformity of the condyle and limitation of movement. Very rarely, osteomyelitis was found to result in a loss of a portion of the mandible. [3]

El-Mofty [4] reported a case wherein there was unilateral loss of the ramus of mandible with no obvious facial asymmetry. He opined that compensatory activity of the suprahyoid muscles could have masked the decreased pterygoid muscle function. Kanemoto et al. [7] reported a case of osteomyelitis of mandibular condyle in which the specific cause could not be determined as there was no dental focus of infection or history of trauma. In our case, the mouth opening was normal, but there was a marked deviation to the affected side. There was a history of dental infection which was treated by a non-specialist medical professional, albeit many years ago, and the resulting deformity might be a fallout of the same.

Proliferation of the proximal zone of the condylar process contributes to the vertical growth of the ramus during 1-3 years of postnatal life. Since the left condyle was affected during this period, there was arrested growth of the ramus and body of the mandible on the ipsilateral side. [8] At the age of 7 years, the articular fossa begins to become prominent and its development accelerates until the 12th year of life. When the condyle is absent, there is no well-defined fossa. [2] The articular fossa was ill defined in our case suggesting that these changes had taken place before the eruption of the permanent dentition.

Frederiksen et al. [9] found that all reported cases of massive osteolysis (Gorham's disease) of the maxillofacial region initially involved the mandible. Coalescence of scattered radiolucency over time, porosity of trabecular structure, disappearance of lamina dura, and atrophy of bony contour are characteristic radiographic features in Gorham's disease. These features were not observed in our case and there could have been an infectious etiology for the loss of mandibular condyle. [10] Cases have also been reported with condylar aplasia with contralateral TMJ ankyloysis. [11]

Various treatment approaches have been proposed for treating condylar aplasia and possibilities have been suggested for influencing mandibular growth. Treatment could then be a costochondral graft transplant, preferably before the growth spurt, orthognathic surgery at the end of the growth period, or both. [12]


   Conclusion Top


To conclude, we presented here a rare case of condylar aplasia on the left side, associated with severe hypoplasia of the posterior body and ascending ramus of the mandible with no underlying pathological disorder or in conjunction with any syndrome. The case also serves as a reminder to promptly treat any dental infections in children, as in the developmental stages, it can result in significant functional impairment and esthetic disfigurement.

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.
Neville BW, Damm DD, Allen CM, Bouquot JE. Developmental defects of the oral and maxillofacial region. In: Neville BW, Damm DD, Allen CM, Bouquot JE, editors. Oral and Maxillofacial Pathology. 2 nd ed. Philadelphia, PA: WB Saunders; 2002. p. 17-8.  Back to cited text no. 1
    
2.
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. 2
    
3.
Worth HM. Infections of the jaws. In: Worth HM, editor. Principles and Practice of Oral Radiologic Interpretation. Chicago: Year Book Medical Publishers Inc.; 1963. p. 215-32.  Back to cited text no. 3
    
4.
El-Mofty S. Unilateral loss of the ramus of the mandible. Oral Surg Oral Med Oral Pathol 1981;52:372-4.  Back to cited text no. 4
    
5.
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. 5
    
6.
Nallamothu R, Kodali RM, Koteswara Rao N, Guttikonda LK, Vijayalakshmi U. Nonsyndromic facial asymmetry with unilateral condylar aplasia. Case Rep Dent 2013;2013:631284.  Back to cited text no. 6
    
7.
Kanemoto K, Suzuki R, Okano T, Nagumo M. Osteomyelitis of the mandibular condyle: Report of a case. J Oral Maxillofac Surg 1992;50:1337-9.  Back to cited text no. 7
    
8.
Soames RW. Skeletal system. In: Williams PL, editor. Gray's Anatomy: The Anatomical Basis of Medicine and Surgery. 38 th ed. Edinburgh: Churchill Livingstone; p. 578.  Back to cited text no. 8
    
9.
Frederiksen NL, Wesley RK, Sciubba JJ, Helfrick J. Massive osteolysis of the maxillofacial skeleton: A clinical, radiographic, histologic, and ultrastructural study. Oral Surg Oral Med Oral Pathol 1983;55:470-80.  Back to cited text no. 9
    
10.
Tsang WM, Tong AC, Chow LT, Ng IO. Massive osteolysis (Gorham disease) of the maxillofacial skeleton: Report of 2 cases. J Oral Maxillofac Surg 2004;62:225-30.  Back to cited text no. 10
    
11.
Singh AI, Singh PM, Dey N, Singh NS. Condylar aplasia with contralateral temperomandibular joint ankyloses. J Med Soc 2014;28:57-9.  Back to cited text no. 11
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12.
Vargervik K, Kaban LB. Hemifacial microstomia diagnosis and management. In: Bell WH, editor. Modern Practice in Orthognathic and Reconstructive Surgery. Philadelphia, PA: Saunders; 1992. p. 1533-60.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]



 

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