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 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 7  |  Issue : 2  |  Page : 114-117

Nonsyndromic multiple dentigerous cyst: A rare clinical presentation


Department of Dental Surgery, KAPV Government Medical College and Hospitals, Trichy, Tamil Nadu, India

Date of Web Publication19-May-2016

Correspondence Address:
D Kamatchi
B4, 172, BHEL Township, Kailasapuram, Trichy - 620 014, Tamil Nadu
India
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DOI: 10.4103/0976-433X.182661

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  Abstract 

Odontogenic developmental cysts of jaws usually present as asymptomatic lesions. Dentigerous cyst is one variety of these cysts, which may grow to a large size within the jaws before it manifests clinically. This aggressive behavior can be attributed to its potential to transform to ameloblastoma and squamous cell carcinoma. We present a case of nonsyndromic multiple dentigerous cysts associated with multiple impacted maxillary and mandibular teeth manifesting as diffuse swelling of the mid-face and lower face region.

Keywords: Dental follicle, dentigerous cyst, enucleation, impacted teeth


How to cite this article:
Gnanaselvi U P, Kamatchi D, Sekar K, Narayanan S. Nonsyndromic multiple dentigerous cyst: A rare clinical presentation. SRM J Res Dent Sci 2016;7:114-7

How to cite this URL:
Gnanaselvi U P, Kamatchi D, Sekar K, Narayanan S. Nonsyndromic multiple dentigerous cyst: A rare clinical presentation. SRM J Res Dent Sci [serial online] 2016 [cited 2020 Oct 25];7:114-7. Available from: https://www.srmjrds.in/text.asp?2016/7/2/114/182661


  Introduction Top


Cysts of the jaws usually present as asymptomatic swellings of the mandible and mid-face region. Most dentigerous cysts are asymptomatic, and their discovery is usually an incidental finding on radiography. The cyst being asymptomatic may attain a large size with resorption of the roots of teeth until they manifest clinically or become evident radiographically. Usually, dentigerous cysts occur around the crown of an impacted tooth, and multiple dentigerous cysts are rare and occur concurrently with syndromes. This case presentation is unique while considering the age of the patient and the massive bone destruction associated with the lesion, the multiple locations of the cyst, and unassociation with any syndrome.


  Case Report Top


A 14-year-old patient presented to the Department of Dental Surgery, KAPV Government Medical College and Hospitals with a chief complaint of pain and swelling in the lower front tooth region for the past 2 months. Past medical history revealed that patient had a history of frontoparietal abscess for which craniotomy was performed before 1 year. History revealed a progressive increase in the swelling for past 3 months, painful for past 2 weeks. Pain was dull, continuous, nonradiating in character, and gets aggravated on chewing. Extraoral examination revealed a swelling involving the bilateral parasymphysis region, tender on palpation, soft, and fluctuant in nature. Intraorally multiple retained deciduous teeth 52, 53, 54, 55, 62, 63, 64, 65,73, 74, 83, 84 were seen and clinically missing multiple teeth 12, 13, 14, 15, 22, 23, 24, 25, 33, 34, 43, 44, 45 were seen. Diffuse swelling obliterating the buccal sulcus extending from 35 to 45 regions in the mandible was evident. On further intraoral examination, pus discharge is seen from the gingival sulcus with tenderness on palpation in relation to 44 region [Figure 1]. Distally tilted 32 and 42, mesially tilted 35, and Grade II mobility in relation to 31, 32, 41, 42 and Grade I mobility with 35 were observed. Diagnostic orthopantomograph [Figure 2] showed a multiple unilocular radiolucency associated with 33, 34 and 43, 44 was seen impacted and enclosed within the lesion. Computed tomography scan of facial skeleton [Figure 3] revealed the presence of unilocular osteolytic lesion present in both sides of mandible in parasymphsis region and bilateral maxilla with the evidence of eccentrically placed tooth inside.
Figure 1: (a) Intraoral preoperative view (b) extraoral preoperative view

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Figure 2: Panoramic radiograph

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Figure 3: Computed tomography scan

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Routine laboratory parameters were normal. Aspiration of the lesion was performed, and purulent fluid was obtained. An incisional biopsy was taken from both right and left mandibular lesions for histopathological examination (HPE). HPE showed cyst lined by hyperplastic benign squamous lining and congested inflamed fibrous stroma with inflammatory cell infiltrate [Figure 4].
Figure 4: Incisional biopsy showing cyst lined by stratified squamous epithelium admixed with fibrous tissue (H and E, ×10)

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Under general anesthesia, an intraoral vestibular incision was given, and cyst enucleation was performed with the extraction of involved teeth [Figure 5]. The enucleacted cyst contents were sent for HPE. It revealed cystic lining with hyperplastic benign squamous lining, irregular rete pegs and congested inflamed stroma with mixed inflammatory cell infiltration [Figure 6]. Radiographic findings, surgical resection, and HPE confirmed the diagnosis of multiple infected dentigerous cysts. The patient was kept under observation for periodic clinical reviews and follow-up.
Figure 5: Intraoperative image after removal of the cyst

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Figure 6: Excisional biopsy showing cystic lining with hyperplastic benign squamous lining, irregular rete pegs, and congested inflamed stroma with mixed inflammatory cell infiltration picture (H and E, ×10)

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  Discussion Top


Dentigerous cyst is one that encloses the crown of an unerupted tooth by the expansion of the follicle and is attached to its neck. The term “dentigerous” is preferred, the literal meaning being “tooth bearing.”[1] Dentigerous cyst was earlier termed as “follicular cyst” but as the latter implies to be derived from the tooth follicle which is mesodermal structure, the term was discontinued.

In order of its frequency, they are associated with third molars, maxillary canines, mandibular second premolars, and maxillary third molars.[2] They may also occur around supernumerary teeth; however, they are only rarely associated with primary teeth.[2],[3] Our case was related to permanent maxillary canine. These cysts can grow to very large size and can cause displacement of teeth or in few cases, it may remain relatively small although some may grow to considerable size causing bony expansion that is usually painless until secondary infection occurs. The age range varies widely from 5 years to 57 years.[4] Radiographically, the dentigerous cyst presents as a well-defined unilocular radiolucency, often with a sclerotic border. Since the epithelial lining is derived from the reduced enamel epithelium, this radiolucency typically and preferentially surrounds the crown of the tooth. A large dentigerous cyst may give the impression of a multilocular process because of the persistence of bone trabeculae within the radiolucency. However, dentigerous cysts are grossly, and histopathologically, unilocular processes and probably are never truly multilocular lesions.[4] The histological features of dentigerous cysts may vary greatly depending mainly on whether or not the cyst is inflamed. In the noninflamed dentigerous cyst, a thin epithelial lining may be present with the fibrous connective tissue wall loosely arranged with inflammatory cells. As the lining is derived from reduced enamel epithelium, it is 2–4 cell layer thick primitive type. The cells are cuboidal or low columnar, and rete pegs formation is absent except in cases that are secondarily infected. As the connective tissue wall is derived from the dental follicle of developing enamel organ, it is a loose connective tissue stroma, which is rich in acid mucopolysaccharides. In the inflamed dentigerous cyst, the epithelium commonly demonstrates hyperplastic rete ridges and the fibrous cyst wall shows an inflammatory infiltrate. Rarely, sebaceous glands in the walls are observed. The content of the cystic lumen is usually thin watery yellow fluid and is occasionally blood tinged. The osmolality of the cyst fluid is modified by increased permeability to glycosaminoglycans such as hyaluronic acid, heparin and chondroitin sulfate, which causes expansile growth rapid.[5] Edamatsu et al. examined the expression of Fas, bcl-2, and single-stranded deoxyribonucleic acid in dental follicles to classify the possible role of these apopstosis-related factors in the pathogenesis of dentigerous cyst.[6] Fas is a cell surface glycoprotein that transmits apoptotic signals from the cell surface to the cytoplasm while bcl-2 proto-oncogene encodes a protein that inhibits apoptosis. Most dentigerous cysts are treated with enucleation of the cyst and removal of the associated tooth. Large dentigerous cysts may be treated with marsupialization when enucleation and curettage might otherwise result in neurosensory dysfunction or predispose the patient to an increased chance of pathological fracture. Occasionally, it transforms to squamous cell carcinoma, mucoepidermoid carcinoma or ameloblastoma from or in association with a dentigerous cyst.[7],[8],[9] The prognosis for most histopathologically diagnosed dentigerous cysts is excellent, recurrence being a rare finding.[2],[4]

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Browne RM. The pathogenesis of odontogenic cysts: A review. J Oral Pathol 1975;4:31-46.  Back to cited text no. 1
    
2.
Neville BW, Damm DD, Allen CM, Bouquot JE, editors. Odontogenic cysts and tumors. In: Oral and Maxillofacial Pathology. 2nd ed. Philadelphia: WB Saunders; 2002. p. 589-642.  Back to cited text no. 2
    
3.
Kusukawa J, Irie K, Morimatsu M, Koyanagi S, Kameyama T. Dentigerous cyst associated with a deciduous tooth. A case report. Oral Surg Oral Med Oral Pathol 1992;73:415-8.  Back to cited text no. 3
    
4.
Shear M, Speight P. Cysts of the Oral and Maxillofacial Regions. 4th ed., Blackwell Publishing Ltd., Munksgaard; 2007. p. 5978  Back to cited text no. 4
    
5.
Browne R. The pathogenesis of odontogenic cysts: A review. J Oral Pathol Med 1975;4:31-46.  Back to cited text no. 5
    
6.
Edamatsu M, Kumamoto H, Ooya K, Echigo S. Apoptosis-related factors in the epithelial components of dental follicles and dentigerous cysts associated with impacted third molars of the mandible. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2005;99:17-23.  Back to cited text no. 6
    
7.
Eversole LR, Sabes WR, Rovin S. Aggressive growth and neoplastic potential of odontogenic cysts: With special reference to central epidermoid and mucoepidermoid carcinomas. Cancer 1975;35:270-82.  Back to cited text no. 7
    
8.
Johnson LM, Sapp JP, McIntire DN. Squamous cell carcinoma arising in a dentigerous cyst. J Oral Maxillofac Surg 1994;52:987-90.  Back to cited text no. 8
    
9.
Leider AS, Eversole LR, Barkin ME. Cystic ameloblastoma. A clinicopathologic analysis. Oral Surg Oral Med Oral Pathol 1985;60:624-30.  Back to cited text no. 9
    


    Figures

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


This article has been cited by
1 Bilateral dentigerous cysts: an updated literature review and report of a case with associated root resorption
Zehra Yonel,Arathi Papineni McIntosh,Neil Donaldson,Michael Murphy,Pankaj Taneja
Dental Update. 2018; 45(11): 1063
[Pubmed] | [DOI]



 

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