Atypical presentation of a glandular odontogenic cyst in the anterior maxilla: A case report

INTRODUCTION

Granular odontogenic cysts (GOCs) are a unique subtype of odontogenic cysts that are sometimes misdiagnosed because of their histological similarities to other cystic lesions. Due to its locally aggressive character, it most frequently affects the anterior part of the maxilla or mandible and has a high recurrence rate.^[1]

CASE REPORT

For 6 months, a 26-year-old woman had a painless, gradually growing bulge in her front maxilla. A firm, non-fluctuant enlargement that stretched from 23 to 13 cm posterior to the central incisor was discovered during the clinical examination. There were no indications of subsequent infection or ulcers on the mucosa above [Figure 1].

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Figure 1:

Clinical site with swelling - under case presentation.

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Pathological findings

A cystic lining made of non-keratinized stratified squamous epithelium was revealed by histopathological analysis. The epithelial layer contained granular eosinophilic cells and localized nodular thickenings. There were sporadic cholesterol clefts and scattered inflammatory infiltrates in the stroma. There was no indication of cancer, and mitoses were uncommon. One distinctive characteristic that supported the diagnosis of GOC was the existence of granular cells inside the lining epithelium [Figure 2].

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Figure 2:

Photomicrograph showing cystic lesion showing variable thickness of the epithelium lining with metaplastic mucous cells. Red arrow indicate intraepithelial microcysts/duct-like spaces and black arrow shows epithelial lining of the Cyst. (Hematoxylin and Eosin stain x20, scale bar = 500 µm).

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Surgical management

Under general anesthesia, the patient had curettage and enucleation. To reveal the lesion, a full-thickness mucoperiosteal flap was raised. To reduce the chance of recurrence, the cyst was completely removed from the surrounding bone using sharp dissection. A round bur and extensive saline irrigation were used to perform the peripheral ostectomy. Hemostatic material was used to pack the surgical site, and absorbable sutures were used to accomplish primary closure. Following surgery, the patient was prescribed analgesics and antibiotics along with instructions on how to keep their mouth clean [Figures 3 and 4].

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Figure 3:

Excised area without cyst - under surgical management.

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Figure 4:

Cyst sample - after mentioning enucleation in surgical management.

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Prognosis and follow-up

There was no sign of a recurrence throughout the patient’s 12-month follow-up. No functional impairment was observed, and the healing process went smoothly. Because of the possibility of recurrence, long-term surveillance was recommended [Figure 5].

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Figure 5:

Postoperative clinical image -under follow-up and prognosis.

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DISCUSSION

Due to its distinct histological characteristics, GOCs are rare odontogenic cysts that were initially identified as a separate entity.^[2] Granular epithelial cells set it apart from other odontogenic lesions, despite its resemblance to glandular odontogenic cysts and lateral periodontal cysts.^[3] Granular cells are believed to be present because the epithelial lining is either degenerating or has changed metabolically.

Although the exact cause of GOC is unknown, it is thought to result from the dental lamina or its leftovers.^[4] To prevent recurrence, it frequently necessitates substantial surgical intervention due to its slow-growing yet locally aggressive character. According to a review of the research, GOC primarily affects adults between the ages of three and five, with a slight preference for men.^[5]

Our example, however, demonstrates an event in a younger female patient, highlighting the importance of taking GOC into account when making a differential diagnosis for anterior maxillary lesions.^[6]

Histologically, GOC shows an epithelial lining with granular eosinophilic cells, thicker plaques, and occasionally microcystic spaces. Although it is frequently mistaken for central mucoepidermoid carcinoma, its benign character is supported by the lack of mitotic figures and cellular atypia. Although they have been explored to help with differentiation, immunohistochemical markers such as CK19 and p63 still have a limited relevance in routine diagnosis.^[7]

To reduce recurrence, thorough enucleation combined with peripheral ostectomy is the recommended course of treatment. Although there is disagreement on the requirement of adjuvant therapies, certain findings support them, such as cryotherapy or Carnoy’s solution. Because recurrence rates range from 10% to 30%, long-term monitoring is required. Research has shown that inadequate removal raises the chance of recurrence, especially when a multilocular presentation is present.^[8]

All things considered, this case highlights how crucial thorough histological analysis and careful surgical intervention are to obtaining the best results. In order to improve therapeutic approaches, more research is necessary to fully comprehend the pathophysiology and biological behavior of GOC.^[9]

CONCLUSION

GOC is an uncommon but serious condition that needs to be diagnosed carefully and treated surgically to avoid recurrence. It is important to distinguish it from other odontogenic cysts by histopathological analysis. For best results, enucleation combined with peripheral ostectomy is still the recommended course of treatment.