Glandular odontogenic cyst mimickers: A review and report of two cases

Shruti Narendra Vichare; Srivalli Natarajan; Padmakar Sudhakar Baviskar; Suraj Arjun Ahuja; Pradeep Pandurang Vathare

doi:10.4103/jmgims.jmgims_14_21

CASE REPORT

Year : 2021 | Volume : 26 | Issue : 2 | Page : 124-128

Glandular odontogenic cyst mimickers: A review and report of two cases

Shruti Narendra Vichare, Srivalli Natarajan, Padmakar Sudhakar Baviskar, Suraj Arjun Ahuja, Pradeep Pandurang Vathare
Department of Oral and Maxillofacial Surgery, MGM Dental College and Hospital, Navi Mumbai, Maharashtra, India

Date of Submission	12-Feb-2021
Date of Acceptance	30-Jun-2021
Date of Web Publication	10-Feb-2022

Correspondence Address:
Dr. Srivalli Natarajan
Department of Oral and Maxillofacial Surgery, MGM Dental College and Hospital, Sector 1, Kamothe, Navi Mumbai - 410 209, Maharashtra
India

Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/jmgims.jmgims_14_21

Abstract

Glandular odontogenic cyst (GOC) is a rare odontogenic cyst with a high recurrence rate. It is also associated with other odontogenic cysts and tumours. Odontogenic cysts with some histopathological features of GOC are termed as GOC mimickers (GOC-M). The diagnosis of a mimicker is challenging due to the lack of distinctive clinical and radiological features. Diagnosis is confirmed exclusively on histopathology. Specific guidelines based on histopathological features exist to recognize and delineate these mimickers from GOC. These features may be evident on incisional biopsy presenting an incorrect diagnosis of GOC leading to an erroneous overtreatment. On the contrary, overlooking these features in an incisional biopsy may result in misdiagnosis and under-treatment which increases the patient susceptibility to recurrence. This article reviews the enigmatic nature of GOC-M and presents two rare cases of the same in dentigerous cysts of the impacted supernumerary teeth in the anterior maxilla.

Keywords: Dentigerous cyst, glandular odontogenic cyst mimickers, glandular odontogenic cyst, histopathology, impacted teeth

How to cite this article:
Vichare SN, Natarajan S, Baviskar PS, Ahuja SA, Vathare PP. Glandular odontogenic cyst mimickers: A review and report of two cases. J Mahatma Gandhi Inst Med Sci 2021;26:124-8

How to cite this URL:
Vichare SN, Natarajan S, Baviskar PS, Ahuja SA, Vathare PP. Glandular odontogenic cyst mimickers: A review and report of two cases. J Mahatma Gandhi Inst Med Sci [serial online] 2021 [cited 2023 Jun 4];26:124-8. Available from: https://www.jmgims.co.in/text.asp?2021/26/2/124/337429

Introduction

The odontogenic tissue surrounding an impacted tooth is associated with odontogenic cysts and tumors due to the presence of various dental structural remnants which have the propensity to undergo changes on the application of the stimulus. Glandular odontogenic cyst (GOC) is a rare developmental cyst of the jaw of odontogenic origin arising from the cell rests of Serres (remnant of the dental lamina).^[1] Shear stated that GOCs account for 0.2% of all jaw cysts and their diagnosis can be entirely by histopathological examination (HPE).^[2],[3] This paper aims to present the enigmatic nature of GOC and glandular odontogenic cyst mimickers (GOC-M) to facilitate precise diagnosis and implement necessary treatment.

Case Reports

Case 1

A 26-year-old female patient reported to the Department of Oral and Maxillofacial Surgery (OMFS) with chief complaints of swelling in the right maxillary anterior region of 1 month duration, and pain on percussion associated with teeth #5, #6, #7, #8 and #9. The patient did not report any history of previous trauma to the teeth. Intraorally, a single soft, fluctuant, and tender swelling with bluish hue was seen in the labial vestibule with respect to the noncarious, nonmobile #5, #6, #7, #8 and #9. On pulp vitality testing, no response was seen with #9; while the remaining showed delayed response. Cone-beam computed tomography (CBCT) demonstrated a localized, ellipsoid, well-demarcated, expansile, hypodense lesion with the scalloped periphery of the size 27 mm × 20.5 mm with external root resorption of involved teeth and a partially formed supernumerary tooth situated palatal to #8 and #9 [Figure 1]. Thinning of the labial cortex and nasal floor was noted. The aspirate was a straw-colored fluid. Following root canal treatment (RCT), enucleation of the lesion, extraction of the supernumerary tooth and apicoectomy of the involved teeth was performed under local anesthesia. HPE revealed GOC-like features [Table 1], namely localized hyperplastic plaque-like thickenings of the epithelium, hobnail cells, goblet cells, ciliated and mucous-secreting cells [Figure 2], and multiple cyst compartments with multicystic or multiluminal architecture on the surface layer of a dentigerous cyst (DC) epithelium [Figure 3]. The final diagnosis was established to be GOC-M in a DC. The patient was free of recurrence at the 1 year follow-up.

Figure 1: Preoperative cone-beam computed tomography axial section of anterior maxilla in Case 1: Showing expansion of the buccal cortex (27 mm × 20.5 mm) and the presence of impacted supernumerary tooth

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Figure 2: Postoperative histopathologic findings of glandular odontogenic cyst in Case 1 showing hobnail cells and varying thickness of epithelium (Hematoxylin and eosin, 400×)

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Figure 3: Postoperative histopathologic findings of the glandular odontogenic cyst in Case 1 showing multiple cyst compartments with multicystic or multiluminal architecture (Hematoxylin and eosin, 40×)

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Case 2

A 50-year-old male patient reported to the department of OMFS with the complaint of pain with maxillary anterior teeth since a month with intermittent swelling and no history of associated trauma to the teeth. On examination, intraorally, a solitary, tender, and fluctuant swelling was seen in the maxillary labial vestibule with respect to all maxillary anterior teeth. Vertical pain on percussion was present with noncarious and nonmobile teeth #4, #5, #6, #7, #8, #9, #10, #11. On pulp vitality testing, no response was seen with #6 and #7, remaining showed delayed response. CBCT demonstrated a single radiolucent lesion of 40.6 mm × 21.3 mm size [Figure 4] extending from the mesial aspect of tooth #3 to the distal aspect of tooth #13 mesio-distally also involving the nasal floor and approximating the right maxillary sinus. Following RCT, enucleation of the lesion, extraction of the supernumerary tooth and apicoectomy of the involved teeth was performed under local anesthesia. HPE of the enucleated specimen showed GOC-like features [Table 1] in a DC along with microcyst formation [Figure 5]. Hence, the final diagnosis of GOC-M in a DC was established. The patient was free of recurrence at 1 year follow-up.

Figure 4: Preoperative cone beam computed tomography axial section of anterior maxilla showing the expansion of the buccal cortex in Case 2 (40.3 mm × 21.6 mm)

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Figure 5: Postoperative histopathologic findings of glandular odontogenic cyst in Case 1 showing microcyst formation (Hematoxylin and eosin, 400×)

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Table 1: Comparison of the Kaplan's major and minor histopathological examination criteria and Fowler's parameters to the microscopic features of the glandular odontogenic cyst mimickers case reports described

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Discussion

GOC was originally described as sialo-odontogenic cyst.^[3],[4] But Gardner et al. stated that the cyst wall epithelium contained only glandular elements with no evidence of salivary tissue involvement.^[5] The World Health Organization in 1992 adopted the term glandular odontogenic cyst (GOC).^[6] Approximately, 114 cases of GOC have been reported in the literature and the mandible is the most frequently involved arch (70%).^[7],[8]

It is important to note the radiologic properties and histopathological variations of DCs. In contrast to GOC, the important radiographic feature specific to DC is a radiolucent area associated with an impacted tooth. No specific pathognomonic radiographic features pertaining to GOC and GOC-M have been described. Resorption and displacement of associated teeth, unilocular or multilocular lesion with cortical involvement and perforation has been reported with GOC.^[9],[10] These lesions can show extensive involvement; thus, a CBCT evaluation is important.^[4],[10]

Histopathologically, the superficial layer of the GOC resembles the lateral periodontal cyst, botryoid odontogenic cyst, radicular cyst, DC with metaplasia, surgical ciliated cysts, and low-grade central mucoepidermoid carcinoma.^[11],[12] These lesions are termed GOC-M as it is sometimes difficult to distinguish whether the cyst is a true GOC or a distinct pathology with GOC-like features.^[13] Only 0.012% of all jaw cysts fulfill the microscopic criteria to be termed as GOC.^[7] Histologic features of DC are connective tissue wall with a thin layer of the stratified squamous epithelium lining the lumen. As stated by Marx and Stern in 2003, DC lining retains some primordial odontogenic cells with potential for transformation to unicystic ameloblastoma or muco-epidermoid carcinoma.^[14] Initial reports by Gorlin and Shear have stated another feature mimicking the GOC is its potential to differentiate into mucous secreting and ciliated epithelium.^[15],[16] Kaplan and Fowler have described specific criteria for diagnosis based on histopathological features to distinguish GOC from GOC-M.^[12],[17] Kaplan divided the histopathological features into diagnostic major and minor criteria based on the frequency of each feature in reported cases from the literature [enlisted in [Table 1]].^[17] Compliance with all major criteria was required for a diagnosis while minor criteria played a supportive role. Fowler in a retrospective analysis of 46 cases of GOC, stated certain mandatory criteria to differentiate a true GOC from a GOC-M [enlisted in [Table 1]].^[12] He concluded that the presence of 7 or more microscopic parameters was highly predictive of a diagnosis of GOC and the presence of 5 or less microscopic parameters was highly predictive of a diagnosis of a GOC-M.

The cases mentioned in the case report are seen in the anterior maxillary region in association with supernumerary tooth (mesiodens) impacted between two central incisors. In Case 1 it occurred in a young female, and in Case 2 in a middle-aged male with the only symptom of nontender swelling. The aspirate obtained in Case 1 was straw-colored. Incisional biopsy was not performed, but incisional biopsies taken only from a single site itself has its own disadvantages as it can give an incorrect diagnosis of GOC, leading to overtreatment like enucleation followed by peripheral ostectomy or marginal resection to avoid recurrence. On the contrary, these features may not be evident in the representative incisional biopsy tissue. Overlooking these features altogether in HPE may result in misdiagnosis and under-treatment like mere marsupialization or enucleation, increasing the patient's susceptibility to recurrence. Incisional biopsy compulsorily should be taken from a minimum from two sites of the lesion before the final treatment. The treatment performed in above-mentioned cases was enucleation with apicoectomy and RCT of the involved teeth. Excisional biopsy report stated GOC-like changes in the DC. Hence it can be concluded to be a GOC-M instead of a true GOC.

The widely accepted treatment of GOC is enucleation with adjunctive treatment due to its high recurrence rate.^[18] Majority of the cases reported in the literature have been treated with marsupialization, enucleation and curettage.^[12] Carnoy's solution and cryotherapy, in addition, have been used with satisfactory outcomes.^[17] However, marginal or segmental resection has been suggested because of the potential for recurrence and the aggressive nature of GOC.^[19] Cell kinetics in the GOC epithelium has been postulated by some authors for the propensity of these cysts to recur.^[20] A post-operative follow-up period is crucial in these cases because the recurrence potential is high.^[17]

The rate of recurrence of GOC ranges from 21% to 55% as described in literature, and 50% as mentioned by Fowler in 2011 with the longest follow-up period of 20 years with two cases recurring thrice.^[12],[18] Shear in 2003 stated that such data are not a reliable indicator of the incidence for any lesion as it varies from institution to institution.^[21] Although the diagnosis given was of a GOC-M and not true GOC, substantial follow-up is mandatory to record the rate of recurrence in such lesions.

Conclusion

In the cases described above, GOC-M were the examples of DC with GOC like changes of the impacted supernumerary teeth in the anterior maxilla. Histopathologic study of the excised specimen confirmed the diagnosis of GOC-M as per Kaplan and Fowler criteria.^[12],[17] GOC-M can be placed at a transitional stage between odontogenic cysts and GOC. Meticulous examination of histopathologic samples of the specimen serves as the gold standard for diagnosis. Incisional biopsy at least from two areas should be made mandatory followed by treatment. Extensive and close postoperative follow-up and monitoring minimum for 5 years are vital due to the high potential for recurrence.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the legal guardian has given his consent for images and other clinical information to be reported in the journal. The guardian understands that names and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Acknowledgment

The authors would like to acknowledge the Department of Oral Pathology, MGM Dental College and Hospital, Navi Mumbai for their expertise in the histopathological diagnosis.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

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