Rare Histologic Imitator Central Mucoepidermoid Carcinoma Arising from Glandular Odontogenic Cyst of the Mandible: Case Report with Updated Review of Literature.

Central mucoepidermoid carcinoma is a relatively rare salivary gland tumour of the jawbone. Glandular odontogenic cyst is another unique odontogenic developmental cyst characterised by glandular differentiation. Both entities share several histological characteristics, and a pre-existing Glandular odontogenic cyst can evolve into Central mucoepidermoid carcinoma. Case 1: A 56-year-old male presented with chief complaint of swelling in lower left facial region since 1 year. Histopathology revealed multicystic compartments resembling mucoepidermoid carcinoma, but strong positive expression of Cytokeratin 13 upon immunohistochemistry helped us in rendering the final diagnosis as Glandular odontogenic cyst Case 2: A 34-year-old female presented with a lesion on right side of face. Histologically, the biopsy specimen revealed both typical findings of a Glandular odontogenic cyst component and a recognizable component of Mucoepidermoid carcinoma. The results from cytokeratin profiling demonstrated that, while both Mucoepidermoid carcinoma and Glandular odontogenic cyst expressed Cyokeratins 7, 18, and 19. Cytokeratin 13 was interestingly exclusively expressed in Glandular odontogenic cyst. Present case findings showed that central mucoepidermoid carcinoma and Glandular odontogenic cyst may be part of the same disease spectrum. However, because the expression profile of Cytokeratin13 in mucoepidermoid carcinoma and Glandular odontogenic cyst was so diverse, it can be used to differentiate both.

Glandular odontogenic cyst misdiagnosed and treated as a periapical inflammatory lesion for 6 years in an older adult.

OBJECTIVES: To document the case of a patient who underwent several endodontic treatments due to a glandular odontogenic cyst misdiagnosed as an inflammatory periapical lesion. BACKGROUND: Glandular odontogenic cysts behave more aggressively, while others have an indolent course. There is limited information on this cyst in the gerodontologic literature. MATERIALS AND METHODS: A 76-year-old male patient presented with an asymptomatic expansive lesion in the anterior mandible resistant to several endodontic treatments. Cone-beam computed tomography revealed a multilocular osteolytic lesion measuring 6.0 × 4.0 cm, with cortical bone perforation. RESULTS: Histopathological analysis of a biopsy specimen was consistent with glandular odontogenic cyst. The patient underwent marginal mandibulectomy with preservation of the base of the mandible. CONCLUSION: A strict diagnostic process is important to avoid unwanted consequences, particularly in the geriatric population.

Recurrence of Glandular Odontogenic Cysts: A Systematic Review.

BACKGROUND: The glandular odontogenic cyst (GOC) is a benign developmental cyst of the jaws that is characterized by a high recurrence rate. METHODS: A systematic review is presented of reported cases, case series, and retrospective studies of recurrent cases of glandular odontogenic cysts, to determine the overall and detailed demographic features with documentation of the specific histologic features of the initial presentation of each cyst. Searches of detailed databases were carried out to identify articles published in the English language from 1988 to 2023. The variables were demographics, patient symptoms, cyst location, radiographic features, histopathological findings, type of treatment, and minimum eight months of follow-up. RESULTS: Eighteen cases were identified: with an equal gender presentation of 50% females and 50% males. The average age was 44.7. The mean size was 3.5 cm. The most common location was in the anterior mandible in 50% (n = 9) of cases, followed by the posterior mandible 27.8% (n = 5). Most patients were asymptomatic 55.6% (n = 10). The most common histologic features at first diagnosis were mucous cells in 88.9% (n = 16), variable thickness with 83.3% (n = 15), eosinophilic cuboidal cells 88.9% (n = 16), microcysts 83.3% (n = 15), and clear cells 77.8% (n = 14) cases. CONCLUSION: GOC has an aggressive behavior. Evidence was not conclusive to link any single or combination of histologic features to recurrence, and the strongest correlation for recurrence was the type of treatment. Since this is an uncommon cyst, more cases are needed. Follow-up should continue for at least five years, because recurrences were higher between years 3 and 5.

Glandular Odontogenic Cyst in the Anterior Mandible: A Case Report of a Conservative Approach and a Recurrence Detection.

PURPOSE: The glandular odontogenic cyst (GOC) is considered a rare developmental cyst, with an odontogenic origin and both epithelial and glandular characteristics, with less than 200 reported cases in the literature. METHODS: In the present case, a 29-year-old man was referred for evaluation of an asymptomatic slow-growing swelling in the anterior region of the mandible, with one-year history. The patient's medical history did not reveal any systemic alteration. The extraoral examination did not show enlargement of the facial contour and the intraoral examination showed vestibular and lingual swelling. Panoramic radiography and CT scan revealed a well-defined unilocular radiolucent lesion involving the inferior incisors and canines bilaterally. RESULTS: Histopathological analysis revealed multiple cysts lined by stratified epithelium with varying thickness and characteristics, in addition to duct-like structures filled with PAS-positive amorphous material, suggestive of GOC. Conservative treatment was performed through surgical curettage, peripheral ostectomy of the surgical site and apicectomy of the teeth involved in the lesion. There was one recurrence, which was detected in postoperative follow-up, leading to a new surgical approach. CONCLUSIONS: Fifteen months after the second procedure, no signs of recurrence were identified, and bone neoformation within the surgical site occurred, supporting that a conservative approach for the treatment of GOC is viable.

Pediatric Oral Pathology: Odontogenic Cysts.

Cysts encountered in the head and neck typically arise from epithelium that would normally be programmed to form teeth or tooth-supporting structures (odontogenic epithelium). These cysts come with a confusing array of similar-sounding names and histopathologic features that are sometimes shared between conditions. Here we describe and contrast the relatively-common lesions: hyperplastic dental follicle, dentigerous cyst, radicular cyst, buccal bifurcation cyst, odontogenic keratocyst, glandular odontogenic cyst, and the less-common gingival cyst of the new-born and thyroglossal duct cyst. The goal of this review is to help clarify and simplify these lesions for the general pathologist, pediatric pathologist, and surgeon.

Frequency and Demographic Profile of Odontogenic Cysts in Riyadh, Saudi Arabia: Retrospective Multicenter Study.

Odontogenic cysts (OCs) are etiologically diverse conditions with a shared origin in the jaws. Unfortunately, there is a scarcity of published data regarding OC frequency, treatment, and follow-up information in Saudi Arabia, especially from tertiary centers. OBJECTIVES: The study aims to assess the relative frequency, clinicopathological features, treatment, and follow-up of OCs in three tertiary medical centers. METHODS AND MATERIAL: OCs were identified from King Abdulaziz Medical City, King Fahad Medical City, and Prince Sultan Military Medical City from January 2010 to December 2021. RESULTS: Three hundred and seventy-two cases of OCs were identified from the archive of three medical centers in Riyadh; one hundred and forty-nine (40%) cases were retrieved from Prince Sultan Military Medical City. The mean age of all OCs was 32 years (range 2-90), with 225 (60.4%) cases occurring in males. There was an almost equal distribution of OCs between the maxilla (47.0%) and the mandible (53.0%). The apical radicular cyst (ARC) accounted for half of the cases, followed by dentigerous cyst (DC) (29.3%) and odontogenic keratocyst (OKC) (14.2%). Enucleation was the most common treatment modality (52.8%), followed by excision (35.0%). Thirteen cases showed recurrence: one ARC, four DCs, and eight OKCs. CONCLUSION: This is the first large multicenter study of OCs in Riyadh, Saudi Arabia. All three centers showed that ARC was the most common, followed by DC and OKC.

Diagnostic Reliability of CRTC1/3::MAML2 Gene Fusion Transcripts in Discriminating Histologically Similar Intraosseous Mucoepidermoid Carcinoma from Glandular Odontogenic Cyst: A Systematic Review and Meta-analysis.

BACKGROUND: Intraosseous mucoepidermoid carcinoma (IMEC) and Glandular odontogenic cyst (GOC) are those two pathological entities causing diagnostic dilemma due to the histopathological similarity. An accurate distinction between the two entities is difficult as both presents with a common radiological and histological similarities. The aim of our systematic review was to establish the diagnostic reliability of CRTC1/3::MAML2 gene fusion for the distinction between IMEC and GOC. METHODS: A complete electronic literature search was made in MEDLINE by PubMed, Google Scholar, and EMBASE databases. Articles with keywords using molecular genetic findings of CRTC1/3::MAML2 gene fusion transcripts, IMEC and GOC were assessed and included for the systematic review. RESULTS: Twelve subgroups having both qualitative and quantitative analysis revealed CRTC1/3::MAML2 sensitivity of 100% and specificity of 70.59% in differentiating GOC and IMEC. Fixed-effects model confirmed translocation-negative cases to have a decreased risk of association with IMEC (combined odds ratio 8.770, 95% confidence interval - 2.45 to 31.45, p < 0.002). CONCLUSIONS: The current evidence supports that in all cases with positive gene fusion transcript of the CRTC1/3::MAML2 was specific for IMEC and was significantly differentiating it from GOC. Whereas cases of IMEC with negative gene fusion transcript pose diagnostic difficulty in differentiating from a GOC which is negative for CRTC1/3::MAML2 expression.

Odontogenic Keratocyst with Diverse Variations: A Rare Case Report.

Rationale: An odontogenic keratocyst (OKC) is a developmental odontogenic cyst lined by squamous epithelium having intrinsic growth potential. Hence, metaplastic changes such as the formation of mucous cells, ciliated cells, and hyaline bodies with ortho/para keratinisation have been known to create unusual histopathological variations. Patient Concerns: A 34-year-old male patient reported with swelling on the lower right side of the face and numbness on the overlying skin. Diagnosis: Based upon the histopathological findings, a final diagnosis of glandular odontogenic cyst with OKC was confirmed presenting mixed features of basal layer palisading squamous epithelium with goblet cells and satellite cysts appeared to be entrapped in the connective tissue wall. Treatment: Surgical enucleation of the cyst was done. Outcomes: No recurrence was reported in 1 year of follow-up. Take-away Lessons: Diverse variations appear within odontogenic cysts and tumours. The high recurrence rate and aggressive nature of the cyst, divulges appropriate treatment and long-term follow-up.

Surgeons Approach to Glandular Odontogenic Cyst of Mandible Mimicking Mucoepidermoid Carcinoma.

This article reports a case of glandular odontogenic cyst in a 70-year-old male and discusses the clinical, radiological and histopathological features. The present case shows treatment of GOC which effected the body of mandible with infiltration into submandibular gland.

Glandular Odontogenic Cyst Associated with Odontoma: A Rare Case Report.

Background: Glandular odontogenic cyst (GOC) is an extremely rare developmental jawbone cyst, tending to recurrence owing to its aggressive behavior. There has been no reported case of presence of GOC simultaneous with odontoma. We presented a case of GOC associated with odontoma with special reference to the diagnostic imaging and the histopathological features. Case Presentation: A 42-year-old Chinese man presented with swelling and pain in the anterior mandible for the past 3 months. Panoramic scan showed a large multiocular well-circumscribed radiolucency accompanied by toothlike morphological abnormality. Histological findings confirmed a GOC associated with odontoma. Conclusion: This case demonstrates GOC with multiple clinical spectrum, and its association with odontoma might enhance the existing knowledge of this rare jawbone cyst.

Glandular odontogenic cysts: A collaborative investigation of 22 cases and proteins related to invasiveness.

BACKGROUND: A glandular odontogenic cyst (GOC) has an intriguing, aggressive behaviour whose mechanisms have not yet been clarified. OBJECTIVE: To conduct a collaborative cross-sectional study on the clinical, demographic, microscopic and immunohistochemical characteristics of GOCs, emphasizing the histopathological characteristics and expression of proteins related to invasiveness. METHODS: Twenty-two cases of GOC from three oral and maxillofacial pathology services in Brazil were selected from 1988 to 2018. Clinical and demographic data were collected. Histopathological features were evaluated in detail. Sixteen cases of GOC were also submitted to immunohistochemistry to detect MT1-MMP, TKS4, TKS5 and cortactin, the key regulators of invadopodia formation. RESULTS: Glandular odontogenic cysts were primarily seen in men over 40 years of age, in the posterior mandible and the anterior maxilla as a unilocular, radiolucent lesion. All cases presented hobnail cells, clear cells and variable thickness of the lining epithelium, 3 of the 10 key histopathological parameters to be evaluated in GOCs. Immunohistochemistry revealed a greater expression of the studied proteins in the GOCs than in the controls (p < 0.0001). CONCLUSION: Overexpression of proteins that regulate cell invasiveness was identified, and the present study's findings suggest that invadopodia activity is a possible mechanism used by GOCs to promote local invasion, which could partly explain its intriguing biological behaviour.

Central mucoepidermoid carcinoma arising directly from a glandular odontogenic cyst of the mandible: a case report.

BACKGROUND: Central mucoepidermoid carcinoma (MEC) is a rare salivary gland tumor that affects the jawbone. Glandular odontogenic cyst (GOC) is also a rare odontogenic developmental cyst with glandular differentiation. GOC shares some histological features with central MEC, and a pre-existing GOC can develop into central MEC. Here, we present a rare case of central MEC developed directly from a pre-existing GOC of the mandible. CASE PRESENTATION: A 67-year-old Japanese man presented with a cystic lesion in the right third molar region. Histologically, the biopsy specimen demonstrated both typical findings of a GOC component lined with non-keratinized squamous epithelium and a recognizable component of central MEC consisting of polycystic nests with mucous cells, intermediate cells, and epidermoid cells in the cyst wall. The results from the immunohistochemistry for cytokeratin (CK) profiling demonstrated that, while both central MEC and GOC expressed CKs 7, 14, 18, and 19, CK13 was interestingly exclusively expressed in GOC. Fluorescence in-situ hybridization (FISH) revealed the rearrangement of the Mastermind like (MAML)-2 gene in both the MEC and GOC components. CONCLUSIONS: Our case suggests that central MEC and GOC may be in the same spectrum of diseases caused by the rearrangement of the MAML-2 gene. However, given that the expression profile of CK13 was completely different between central MEC and GOC, they can be considered as separate tumors. Overall, we demonstrated a rare case in which central MEC may have originated directly from the GOC.

Glandular Odontogenic Cyst: A Diagnostic and Management Dilemma.

The glandular odontogenic cyst (GOC) is a rare odontogenic cyst that can develop in the maxillofacial region with aggressive behavior. It tends to develop into enormous proportions with high recurrence rates. The mandibular anterior area is the common site of occurrence of GOC, and it appears as an asymptomatic slow-growing swelling. GOC mimics other odontogenic cysts and tumors both clinically and radiographically, thus posing difficulty in diagnosis. Due to the aggressive potential of GOC, precise diagnosis and prompt treatment are crucial. Both conservative and aggressive surgical therapies have been advocated for GOC with a preference for aggressive therapy due to its high potential for recurrence. In this report, we present a case of GOC of the mandible in an adult female patient, which was successfully treated by segmental resection and primary reconstruction with stainless steel recon plate with uneventful healing during the one-year postoperative follow-up period.

Glandular Odontogenic Cyst of Anterior Maxilla - A Rare Case Report.

Rationale: Glandular odontogenic cyst (GOC) is a rare type of cyst of the jaws, which shares histological features with that of lateral periodontal cyst, botryoid cyst, radicular cyst with mucous metaplasia, or low-grade mucoepidermoid carcinoma, making it difficult to come to a definitive diagnosis. Not many cases of GOC have been reported in the literature. Patient Concerns: A 40-year-old male patient presented with pain in the upper right front tooth region. Mild extraoral swelling, obliterating the right nasolabial fold, was present. Tenderness on percussion was present in involved teeth. Diagnosis: On histopathological examination, GOC was confirmed featuring pseudostratified columnar cells with cilia, goblet cells, and mucous cells. Treatment: Complete enucleation of the cyst was done. Outcomes: No recurrence was noted on 1-year follow-up. Take-away Lessons: Due to high recurrence rate and aggressive nature of the cyst, it is important to plan proper management and long-term follow-up.

Florid Cemento-Osseous Dysplasia with a Concurrent Glandular Odontogenic Cyst.

The presence of non-odontogenic cysts associated with benign fibro-osseous lesions of the jaws has been well documented. However, literature is scant when describing benign fibro-osseous lesions with associated odontogenic cysts. This case report highlights the presence of a concurrent developmental odontogenic cyst, glandular odontogenic cyst with extensive squamous metaplasia, in a patient with florid cemento-osseous dysplasia (COD). The postulated pathogenesis of these synchronous lesions is discussed along with a review of current literature. Surgical treatment is discouraged for florid COD, however, radiological follow-up is recommended, especially in lesions with associated cysts.