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 Table of Contents  
CASE REPORT
Year : 2015  |  Volume : 3  |  Issue : 2  |  Page : 309-313

Neoplasms associated with dentigerous cyst: An insight into pathogenesis and clinicopathologic features


1 Department of Oral Pathology and Microbiology, Government Dental College and Hospital, Jamnagar, Gujarat, India
2 Department of Oral Pathology and Microbiology, Y. B. Chavan Dental College, Ahmednagar, Maharashtra, India
3 Department of Conservative Dentistry and Endodontics, Sidhapur Dental College and Hospital, Sidhapur, Patan, Gujarat, India
4 Department of Oral Pathology and Microbiology, Rural Dental College, Pravara Institute of Medical Sciences, Loni, Ahmednagar, Maharashtra, India

Date of Web Publication16-Dec-2015

Correspondence Address:
Jitendra V Kalburge
Department of Oral Pathology and Microbiology, Government Dental College and Hospital, Behind M. P. Shah Medical College, Jamnagar - 361 008, Gujrat
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2321-4848.171936

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  Abstract 

Odontogenic cysts may occur in association with odontogenic tumors. Because neoplastic and hamartomatous aberrations can occur at any stage of odontogenesis, combined features of odontogenic tumors with epithelial and mesenchymal components may arise within odontogenic cysts. One of the most common of these is dentigerous cyst (DC) which has neoplastic potential and shows associated pathologies such as ameloblastoma, squamous cell carcinoma, mucoepidermoid carcinoma (MEC), adenomatoid odontogenic tumor (AOT), and odontoma. The authors report four cases of DC and associated lesions exhibiting AOT in two cases while one case each of complex odontome and MEC. Emphasis is placed on pathogenesis and clinicopathologic features of these lesions.

Keywords: Adenomatoid odontogenic tumor, complex odontome, dentigerous cyst, mucoepidermoid carcinoma


How to cite this article:
Kalburge JV, Latti B, Kalburge V, Kulkarni M. Neoplasms associated with dentigerous cyst: An insight into pathogenesis and clinicopathologic features . Arch Med Health Sci 2015;3:309-13

How to cite this URL:
Kalburge JV, Latti B, Kalburge V, Kulkarni M. Neoplasms associated with dentigerous cyst: An insight into pathogenesis and clinicopathologic features . Arch Med Health Sci [serial online] 2015 [cited 2019 Aug 25];3:309-13. Available from: http://www.amhsjournal.org/text.asp?2015/3/2/309/171936


  Introduction Top


The odontogenic tissue surrounding an impacted tooth is prone to develop in to odontogenic cysts and tumors. The most common odontogenic cyst arising from pericoronal tissue is dentigerous cyst (DC). The DC is defined as, "developmental odontogenic cyst associated with the crown of an impacted tooth mostly a permanent tooth." The most common sites are the mandibular third molar and maxillary canine areas. It is asymptomatic in its early phase of development. A prominent radiographic feature of DC is a radiolucent area associated in some manner with an unerupted tooth. There are no characteristic microscopic features that reliably distinguish DC. They are composed of a connective tissue wall with a thin layer of stratified squamous epithelium lining the lumen. Rete peg formation is generally absent except in secondarily infected cases. [1],[2]

Although rare but secondary development of neoplastic lesions from DC such as adenomatoid odontogenic tumor (AOT), [3],[4],[5],[6],[7],[8],[9],[10] complex odontome (CO), [11],[12] ameloblastoma (AB), [13],[14] mucoepidermoid carcinoma (MEC), [15],[16],[17],[18] and squamous cell carcinoma (SCC) [2],[19],[20],[21] have been documented in various case reports and series of cases. In a retrospective clinicopathological analysis of 2082 DCs, Zhang et al. [14] reported that DC was co-existed with other pathologies: Five with odontomas, one with central giant cell granuloma, and one keratocystic odontogenic tumor. They also identified two rare DCs with unicystic AB derived from DC. This supports the notion that DC could evolve into more serious disease and shows the importance of submitting samples for pathology examination even when cysts present clinically as classical DC.

The purpose of this article was to discuss DC and its associated lesions with emphasis on their pathogenesis and clinicopathologic features.


  Case Report Top


Four cases of DC are presented in this case series with co-existence of odontogenic tumors (AOT and CO) in three patients and MEC in one patient. The clinicopathologic features are discussed in [Table 1].
Table 1: Clinicopathologic features of dentigerous cyst and its associated lesions

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Histopathology

Case 1

Hematoxylin and eosin (H and E) stained section shows lining epithelium of DC and AOT in the connective tissue wall. AOT shows tumor epithelial cells arranged in the form of ducts, whorls, sheets, and rossett's with scanty connective tissue [Figure 1].
Figure 1: Photomicrograph showing adenomatoid odontogenic tumor located in the connective tissue wall of dentigerous cyst (H and E, ×10)

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Case 2

Hematoxylin and eosin section shows lining epithelium of DC showing mural proliferation of AOT with tumor epithelial cells arranged in the form of ducts and sheets [Figure 2].
Figure 2: Photomicrograph showing mural nodules of adenomatoid odontogenic tumor developing from epithelial lining of dentigerous cyst. Continuity between the cystic lining and the tumor area are seen (H and E, ×20)

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Case 3

Hematoxylin and eosin stained section of DC shows typical features of lining epithelium and connective tissue. There are numerous odontogenic epithelial islands in the connective tissue [Figure 3]a]. Decalcified and H and E stained section of odontoma show haphazardly arranged dental hard tissues suggestive of CO [Figure 3]b].
Figure 3: (a) Photomicrograph showing lining epithelium and connective tissue of dentigerous cyst and (b) features of complex odontome (H and E, ×20)

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Case 4

Hematoxylin and eosin stained section shows lining epithelium of DC showing mucous cell metaplasia and transition into MEC [Figure 4]a]. The tumor shows epidermoid cells, mucous cells, and intermediate cells arranged in the form of islands with areas of cyst formation [Figure 4]b].
Figure 4: (a) Photomicrograph showing mucous cell metaplasia and neoplastic transformation in lining epithelium of dentigerous cyst and (b) tumor epithelial cells arranged in the form of islands and invading the connective tissue, the features of mucoepidermoid carcinoma (H and E, ×20)

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  Discussion Top


Odontogenic tissue surrounding impacted teeth has the potential to differentiate into a wide variety of tissue types, and the potential for cyst and tumor development exists. Odontogenic cysts and tumors vary in their pathologic potential. DCs can destroy a significant amount of bone; odontogenic keratocyst has the potential for recurrence and may be associated with nevoid basal cell carcinoma syndrome. AB and calcifying epithelial odontogenic tumor cause widespread tissue destruction, tend to recur, cause facial deformity, and have the potential for malignant transformation. [1]

The epithelial lining of an odontogenic cyst may transform into an odontogenic neoplasm or malignant epithelial tumor. The most common neoplasms that originate in the lining of cysts are benign odontogenic tumors. Nonodontogenic tumors that arise in conjunction with odontogenic cysts are usually malignant: Epidermoid carcinoma and MEC. [18]

The present case series deals with four separate cases in which DC is the primary pathology while amongst associated lesions two are AOT's and one case each of CO and MEC.

In our case series, we found that both the cases of DC-AOT were histologically distinct. First case showed development of AOT in the connective tissue wall of DC [Figure 1] while second case presented with mural nodules of AOT developing from the epithelial lining of DC [Figure 2]. Both these cases were clinically and radiographically diagnosed as DC. The true nature of these lesions was only identified after thorough histopathologic evaluation of tissue. Both affected patients were teenaged females with involvement of maxillary canines. There is an uncertainty whether the lining of an associated cyst represents a true DC, cystic change within an AOT or may represent a distinct entity. Furthermore, it is unclear whether this entity has a more aggressive potential.

The hypothesis that follicular AOTs arise from the reduced enamel epithelium (REE) that lines the follicles of unerupted teeth is fairly conclusive. They surround the crowns and are attached to the necks of unerupted teeth in a true follicular relationship. Many present as cystic lesions with only mural nodules of AOT lesional tissue and in some instances, origin of the lesional tissue from the REE can be demonstrated histologically. Whether the origin of the follicular variant occurs before or after cystic expansion has taken place is open to conjecture. If it occurs after cystic expansion, then this effectively means origin from a DC, as of our case number 2 and several such case reports have been published. [3],[4],[10] If it occurs before cystic expansion, then the tumor tissue will fill the follicular space and the AOT will present as a solid tumor. It is reasonable to assume that given enough time, even those originating from a cyst may grow and fill the lumen completely. It cannot be ruled out that the DC with an impacted tooth developed first followed by development of AOT in the cyst wall. [6]

Another odontogenic tumor associated with DC in this case series is CO. As a result of their odontogenic nature, including epithelial and mesenchymal tissue odontomas can develop cystic transformation into DC. This cyst results from the cystic degeneration of enamel organ after partial or total development of the crown, cystic transformation of the follicle associated with the unerupted tooth may also occur when its eruption is impeded by the odontoma. The case discussed here was 21-year-old male with impacted 23 and DC was found to be associated with it while CO was seen overlapping the cystic lesion. It was unknown whether odontome led to impaction of 23 and development of cyst or trauma led to the development of both the lesions simultaneously. After surgical exploration, both adjacent lesions came out separately. Histologically it was confirmed as DC associated with 23 and CO [Figure 3]a and b].

The AOT/CO and DC are benign, encapsulated lesions, and conservative surgical enucleation or curettage is the treatment of choice. The prognosis is good and recurrences are very rare after complete removal of the lesion.

The fourth case in this case series is a case of MEC arising from DC, and the diagnosis is based on the criteria given by Gardener. The criteria include:

  1. A microscopic transition area from benign cystic epithelial lining to invasive malignant SCC;
  2. No carcinoma changes in the overlying epithelium;
  3. No source of carcinoma in the adjacent structures. [19],[20]
An excellent review of epidermoid carcinoma that arose from cystic linings was published by Gardner [19],[20] and Uchida et al. [21] but little is known about MEC arising within the jaws. Brookstone and Huvos [16] described central MECs in association with DCs, radicular cysts, and AB. In addition, as in odontogenic cysts and tumors, the most common aspect of central MEC is a uni/multi-locular radiolucency in the posterior mandible, frequently associated with impacted teeth. The possible relationship of central MECs with odontogenic lesions is also reinforced by the fact that central MECs outnumber other intraosseous salivary tumors such as pleomorphic adenoma and adenoid cystic carcinoma. Several sources of histogenic origin have been proposed for central MEC, including:

  1. Mucous metaplasia of odontogenic cyst epithelium;
  2. Entrapment of salivary tissues from the submandibular, sublingual, or minor salivary glands from the retromolar area during embryonic development;
  3. Maxillary sinus epithelium;
  4. Iatrogenic entrapment of minor salivary glands (e.g., chronic osteomyelitis and sinusitis);
  5. Remnants of the dental lamina.
The ability of the linings of benign odontogenic cysts to undergo mucous metaplasia is well-documented. [17],[18],[19],[20],[21] Their relation to the unerupted molar raises the suspicion that they may have arisen from the lining of a DC. Although the clear mechanism of malignant transformation in the lining epithelium of the odontogenic cyst remains unknown, long-term chronic inflammation may stimulate the lining epithelium, resulting in this transformation. [21] The significance of mucous cell prosoplasia is unknown. We theorize that the luminal mucus alters the osmotic pressure gradient, leading to cyst enlargement. In addition, many believe intraosseous MECs arise from the odontogenic cysts with mucous cell prosoplasia. [15]

However, a wide variety of pathologically significant pericoronal lesions occur and that those lesions, especially carcinomas, tend to occur with increasing age. The patient described here is 55-year-old male with impacted 48 and histological confirmatory diagnosis of MEC [Figure 4]a and b]. This serves to increase the oral surgeon's index of suspicion when treating adult patients with impacted teeth. The clinical significance of malignant tumors arising from odontogenic cysts or de novo should never be underestimated as illustrated by the present case. This re-emphasizes the importance of careful histopathological evaluation of all excised tissue so that such neoplastic transformation may be identified and treated effectively. Radical resection offers the best chance of tumor eradication and prevention of local recurrence and late distant metastasis.


  Conclusion Top


A DC is one of the commonly encountered odontogenic cysts in routine practice. However, it is noteworthy that neoplastic transformation, benign or malignant is possible. Thus, there is a need of a thorough evaluation of impacted teeth and its removal should remain as acceptable therapy. Otherwise, lifelong follow-up of retained impacted teeth seems prudent. We also would like to stress the complete histopathological evaluation of excised specimen of DC to rule out its complications.

 
  References Top

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Tajima Y, Sakamoto E, Yamamoto Y. Odontogenic cyst giving rise to an adenomatoid odontogenic tumor: Report of a case with peculiar features. J Oral Maxillofac Surg 1992;50:190-3.  Back to cited text no. 3
    
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    Figures

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

  [Table 1]



 

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