Glandular odontogenic cyst in a young Indian female: A rare case report with review of literature

Vishwas D Kadam; Govind R Changule; Sneha H Choudhary; Lalitkumar P Gade

doi:10.4103/jiaomr.jiaomr_34_20

CASE REPORT

Year : 2020 | Volume : 32 | Issue : 3 | Page : 300-302

Glandular odontogenic cyst in a young Indian female: A rare case report with review of literature

Vishwas D Kadam¹, Govind R Changule², Sneha H Choudhary³, Lalitkumar P Gade⁴
¹ Department of Oral Medicine and Radiology, C.S.M.S.S. Dental College and Hospital, Aurangabad, Maharashtra, India
² Oral and Maxillofacial Surgery, C.S.M.S.S. Dental College and Hospital, Aurangabad, Maharashtra, India
³ Department of Oral Medicine and Radiology, Teerthanker Mahaveer Dental College and Research Centre, Moradabad, Uttar Pradesh, India
⁴ Department of Oral Pathology, S.M.B.T. Dental College and Post-Graduate Institute and Hospital, Sangamner, Maharashtra, India

Date of Submission	03-Mar-2020
Date of Decision	26-Jul-2020
Date of Acceptance	29-Jul-2020
Date of Web Publication	29-Sep-2020

Correspondence Address:
Dr. Sneha H Choudhary
Department of Oral Medicine and Radiology, Teerthanker Mahaveer Dental College and Research Centre, Moradabad, Uttar Pradesh
India

Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/jiaomr.jiaomr_34_20

Abstract

Glandular odontogenic cyst (GOC) is a developmental odontogenic cyst of jaw bones, described in 1987 by Gardner et al. The cyst is rare in occurrence, especially in Indians, unpredictable in nature, potentially aggressive in its behavior and has a very high recurrence rate. Clinically, the age, gender, and site of occurrence of GOC as per the literature are 5^th- 6^th decade of life, slight male predilection and mandibular anterior region. This article presents a case of GOC in a 28-year-old female patient in the posterior mandibular region which is a rare combination of age, gender and site of occurrence for GOC.

Keywords: Developmental cyst, glandular odontogenic cyst, mandible, odontogenic cyst

How to cite this article:
Kadam VD, Changule GR, Choudhary SH, Gade LP. Glandular odontogenic cyst in a young Indian female: A rare case report with review of literature. J Indian Acad Oral Med Radiol 2020;32:300-2

How to cite this URL:
Kadam VD, Changule GR, Choudhary SH, Gade LP. Glandular odontogenic cyst in a young Indian female: A rare case report with review of literature. J Indian Acad Oral Med Radiol [serial online] 2020 [cited 2022 Jul 3];32:300-2. Available from: https://www.jiaomr.in/text.asp?2020/32/3/300/296581

Introduction

Glandular odontogenic cyst (GOC) was first documented as 'sialo-odontogenic cyst' by Padayachee and Van Wyk in 1987 based on the possibility of its salivary gland origin.^[1] Gardner et al. first described its odontogenic origin in 1988 suggesting the name GOC because of the odontogenic nature of the cystic epithelium which contained mucin without any evidence of salivary tissue involvement.^[2] 1992 World Health Organization (WHO) typing of odontogenic tumors, defined GOC as “a cyst arising in the tooth-bearing areas of the jaws characterized by an epithelial lining with cuboidal or columnar cells at the surface as well as the lining crypts or cyst-like spaces within the thickness of the epithelium”.^[3] High recurrence and aggressive growth potential are the two major attributes of GOC.^[4] It occurs in the 5^th-7^th decade of life,^[5] and has slight male predilection. The common site is mandibular and maxillary anterior region presenting as an asymptomatic, slow-growing swelling.^[6] Radiologically, these cysts appear as unilocular or multilocular radiolucency with a well-defined border and with scalloped and sclerotic margins.^[7] Here we present an asymptomatic case of GOC in the posterior mandible.

Case Report

A 28-year-old female patient reported with a complaint of decayed teeth in the lower right and left back region of jaw. Extraoral examination revealed no abnormalities [Figure 1]. Intraoral examination revealed root stumps of 36,46 and deep caries with 47. Since the root stumps were too superficial they were extracted without radiographic investigation. The patient was advised an IOPA radiograph of 36 and 46 regions at 2 months follow-up visit to plan for implant prosthesis. The radiograph revealed a well defined, corticated, oval-shaped radiolucency measuring approximately 2.5 cm × 3.0 cm in the edentulous space of 46. Also, there was presence of an artifact as a solitary semicircular black spot overlying the central part of the base of the lesion (processing error) [Figure 2]. An orthopantomogram (OPG) revealed a well defined, corticated, ovoid radiolucency of size approximately 2.5 cm × 3.0 cm in 46 region above the mandibular canal with no lesion in other regions [Figure 3]. Because of the inability of the patient to remove earrings and nose ring, radioopacities of the same are visible on the radiograph. Based on these findings, a diagnosis of residual cyst with 46 region was given. Thereafter, aspiration and enucleation with curettage was performed and the cyst was completely enucleated under local anesthesia. The aspirate yielded brownish-red fluid and the cystic lining was submitted for histopathological evaluation. The H and E stained slides showed non keratinized stratified squamous epithelium of variable thickness with a flat interface connective tissue wall and lack of basal palisading, eosinophilic material in the cystic lumen, pseudoglandular structures with intraepithelial clefts and eosinophilic material [Figure 4]a, intraepithelial duct-like structures [Figure 4]b. Also, papillary proliferation of the lining epithelium exhibiting variations in thickness along the cystic lining with the presence of whorls and clear or vacuolated cells [Figure 4]c. The histopathological picture was suggestive of GOC and the final diagnosis of GOC was confirmed. The patient was kept on regular follow-up for 1 year without any signs of recurrence.

Figure 1: Patient's extraoral profile picture with no signs of swelling or asymmetry

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Figure 2: IOPA radiograph showing single, well defined, corticated, oval shaped radiolucency in 46 region

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Figure 3: OPG showing single, well defined, corticated, oval shaped radiolucency in 46 region

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Figure 4: Pictomicrograph showing (a) non keratinized stratified squamous lining epithelium exhibiting variations in thickness along the cystic lining, pseudoglandular structures along with intraepithelial clefts and eosinophilic material (black arrow); (b) Intraepithelial cleft and duct like structures (black arrow); (c) presence of clear or vacuolated cells (black arrow)

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Discussion

Although rare, GOC is a well-known clinical entity and is important to recognize its aggressive behavior and tendency to recur. GOC accounts for only about 0.012-1.3% of all jaw bone cysts.^[8] According to literature, less than 200 cases of GOC have been reported all over the world so far and only 26 cases have been reported in Indians since 1987^[5],[9],[10] and the present case adds one more to the Indian literature.

In the present case, the patient was a 28-year-old female, and the cyst was present in the posterior region of the mandible that is quite uncommon. Out of the 26 cases of GOC reported in Indians, the posterior mandibular region was involved in only 7 cases.^[5],[9] The common clinical presentation of GOC is an asymptomatic swelling,^[6] but in our case it was an incidental radiographic finding. An observation in the literature review of GOC cases reported in India showed that 7 out of 19 cases were located in edentulous or partially edentulous areas, as in the present case. This could be indicative of the asymptomatic initial growth of the lesion which might cause visible swelling and cortical expansion at a later stage.^[10] A preoperative aspiration and fluid inspection is advisable since the presence of watery, clear, low viscous fluid can be a helpful clinical indication of GOC.^[11] The brownish-red aspirate in the present case is because of the presence of blood.

A diagnosis based on clinical and radiological examination is difficult as the findings are not pathognomonic because of the similarities with a radicular cyst or residual cyst, hence histopathological evaluation becomes mandatory.

Histologically, GOC shows a non keratinized squamous epithelial lining of varying thickness interfaced with the connective tissue wall without basal palisading. The hallmark finding is the presence of intraepithelial glandular, microcystic, or duct like structures. Also cuboidal eosinophilic cells or “hobnail” cells and mucous (goblet) cells with intraepithelial mucous pools are seen.^{[2],[3],[8],[9]} Histologically GOC should be distinguished from the lateral periodontal cyst (LPC), botryoid odontogenic cyst (BOC), radicular cyst with mucous metaplasia, and central mucoepidermoid carcinoma (CMEC) as it exhibits considerable overlapping features.^[11] The presence of ciliated epithelium and duct-like spaces with mucous cells differentiated GOC from both LPC and BOC. Epithelial whorls, superficial cuboidal cells, ciliated cells, duct-like structures, or intraepithelial microcyst are not indicative for CMEC. Certain studies showed that GOC exhibited decreased p-53 positivity, increased Ki-67 index and positive immunostaining with MASPIN, Ki-67, and CKs 18 and 19 that may be helpful to distinguish GOC from CMEC.^[12] These markers could be used as auxiliary aids in the confirmatory diagnosis.^[9] Special stains like mucicarmine, alcian blue, and periodic acid-schiff can be used,^[12] but in the present case no special stain was used.

Literature suggests that GOC is a relatively aggressive lesion having higher tendency for perforation or erosion of the cortical plates. GOC has a higher recurrence rate of 21-25%^[8] and the following are reported as reasons for recurrence:^[12],[13]

The size and its multilocular nature. 14.4% of the small lesions recur in contrast to 85.6% of the larger lesions.
Cell kinetics in the lining epithelium with an increased Ki-67 index, increased Bc12 and decreased P53 positivity.
Tendency of the epithelial separation from connective tissue.
Thin cystic wall and the presence of microcysts.
The conservative treatment.

The treatment is still controversial and several treatment modalities have been used for GOCs and the prognosis remains unclear. The enucleation with or without curettage and peripheral ostectomy are used generally for small unilocular lesions.^[9] However, multicystic lesions treated by curettage or enucleation alone showed an increased rate of recurrence. Therefore, larger lesions should be treated more aggressively.^[9],[10] The aggressive nature and the high recurrence rate necessitate long-term follow-up and our patient has been kept under regular follow-up till date without any signs of recurrence.

Conclusion

GOC is one of the rare odontogenic cysts. Since GOC presents with many similarities as other odontogenic cysts, a careful clinical and radiological evaluation along with a precise histopathological examination must always be carried out. The unpredictable nature, the aggressive behavior and high recurrence rate make GOC one of the high alert cysts to deal with.

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 initial s 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

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