A Comparative immunohistochemical analysis of epithelial-mesenchymal transition biomarkers in odontogenic keratocyst, dentigerous cyst, and radicular cyst.

Background: Odontogenic keratocyst (OKC) is one of the common odontogenic cysts with aggressive clinical behavior and a high recurrence rate. Epithelial-mesenchymal transition (EMT) is a process, in which the epithelial cell loses its epithelial characteristics and acquires mesenchymal features. Since the evidence for the involvement of EMT in the development of OKC is still limited, the present study aimed to investigate the immunohistochemical expression of EMT-related proteins (E-cadherin and N-cadherin) in OKC and compare them to radicular cyst (RC) and dentigerous cyst (DC). Materials and Methods: In this descriptive analytical study, 75 paraffin blocks, including 25 DCs, 25 OKC, and 25 RCs, were selected. Immunohistochemical staining was performed to determine the expression and staining intensity of E-cadherin and N-cadherin proteins. The specimens were examined under an optical microscope, and the data were analyzed using the Kruskal-Wallis test in SPSS statistical software (version 23) with a significance level of 5%. Results: The expression of N-cadherin in OKC was higher than that in other cysts; nonetheless, there was no statistically significant difference (P = 0.331). The staining intensity of N-cadherin was weak in most cases, and this difference was not statistically significant (P = 0.252). E-cadherin expression in OKC was significantly lower than that in radicular and DCs (P = 0.003). In addition, the staining intensity of E-cadherin in OKC was weak and moderate (P = 0.003). Conclusion: In this study, we observed an increase in the expression of N-cadherin in OKC. In addition, the protein expression levels of E-cadherin in OKC were significantly lower compared to DC and RC. Therefore, it appears that the EMT process likely occurs in OKC and may contribute to its local aggressive behavior.

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.

Conservative Approach in Managing Complex Odontogenic Lesions: A Case Report and Literature Review.

Radicular cysts are among the most common odontogenic cystic lesions in the maxillofacial region. This case report details the management of a large radicular cyst in the anterior maxillary region of a 32-year-old female patient and includes a literature review on such cysts. The patient underwent cyst decompression, surgical enucleation, tooth extractions, root canal treatments, periapical curettage, and prosthetic rehabilitation. This study underscores the effectiveness of conservative approaches, such as decompression, in reducing cyst size and highlights the importance of individualized treatment plans for achieving optimal outcomes and preventing recurrence. Collaborative efforts between clinicians and multidisciplinary teams are crucial for managing radicular cysts and ensuring long-term oral health for patients.

Postoperative Maxillary Cyst Following Maxillary Tooth Extraction: A Rare Surgical Predicament.

A postoperative maxillary cyst (POMC) is an epithelium-lined cyst that can develop following surgery or trauma in the maxillary antral region. This condition arises from the entrapment of the sinonasal mucosa in the maxilla, and rarely in the mandible, due to trauma or instrumentation near the maxillary sinus. Literature indicates that POMCs, or surgical ciliated cysts, can appear as delayed complications from five months to 56 years after trauma or surgical procedures in the sinus area. Despite its potential for aggressive local destruction, it often presents incidentally with minimal symptoms. This clinical case report describes the occurrence of such a cyst in a 30-year-old male and discusses the diagnosis and management of this rare pathology.

Calcifying Odontogenic Cyst Associated with Complex Odontoma: Report of a Rare Case.

Calcifying odontogenic cyst, also known as Gorlin cyst is a rare benign cystic lesion primarily found in the jawbones, accounting less than 1% of odontogenic cysts. It can be associated with odontogenic tumors such as odontomas. We report a rare case of COC associated with complex odontoma in a young patient and discuss its clinical features, diagnosis, and treatment options. An 18-year-old female patient presented with a painless radiopaque lesion of the right mandibular bone at Oral Medicine and Oral Surgery department. Radiographs revealed irregular tooth-like structures in the canine-premolar area. The lesion was surgically removed, and histopathology confirmed COC with a complex odontoma. As of the World Health Organization's 2022 definition, COC is a developmental odontogenic cyst characterized by calcified ghost cells. It typically affects individuals during their second and third decades of life, with no gender preference, almost equally in the maxilla and the mandible. The main treatment is total enucleation, with a generally favorable prognosis. Histopathology is essential for diagnosis due to its mimicry of other jaw conditions. Long-term follow-up is needed to prevent recurrences.

Odontogenic Keratocyst of the Maxilla With Fungal Sinusitis: A Rare Case.

Odontogenic keratocysts (OKCs) in the maxilla are rare. Although destructive, their clinical presentation can mimic inflammatory conditions like radicular cysts and osteomyelitis. OKCs originate primarily from odontogenic sources. On radiography, they present a unilocular to multilocular radiolucency and may involve the maxillary sinus. Enucleation with chemical cauterization is the best treatment for limiting cyst recurrence. However, superaided infections, such as fungal sinusitis, can adversely affect the condition and require a more comprehensive treatment plan. The present case report describes an OKC of the upper jaw involving the maxillary sinus in a 42-year-old male with a superadded fungal infection. The treatment plan included enucleation with chemical cauterization along with inferior meatal antrostomy. In addition, an antifungal protocol was instituted. A follow-up period of one year was not associated with any complications.

An unusual presentation of calcifying odontogenic cyst with literature review.

The calcifying odontogenic cyst (COC) is an uncommon developmental odontogenic cyst, the oral counterpart of Malherbe's cutaneous calcifying epithelioma (pilomatricoma). This article presents two unique cases of calcifying odontogenic cysts each exhibiting distinctive histopathological features and its literature review. One case with an unexpected finding of cholesterol granuloma (CG), a rare occurrence in non-inflammatory cysts within an unusual location between two maxillary central incisors. One more instance involves the presence of a compound odontome in conjunction with COC. The cases underscore the clinical and histopathological diversity of COC and highlight the importance of radiological and histopathological assessments for accurate diagnosis. The unexpected association of COC with cholesterol granuloma challenges traditional diagnostic expectations. Additionally, the second case suggests that COCs may warrant sub-categorization to better understand their varied presentations and biological behavior. This article contributes to the expanding knowledge of COC, emphasizing the significance of documenting rare cases to enhance comprehension of its nature, pathogenesis, and oral cavity origin.

Calcifying Odontogenic Cyst Presenting Odontogenic Keratocyst-Like Areas: A Rare Case Report.

An 81-year-old male patient presented with a well-demarcated, unilocular radiolucent lesion in the right mandibular body, identified during a routine radiographic examination. Based on the clinical hypothesis of a residual cyst, enucleation with curettage was performed, and the specimen was submitted for histopathological analysis. Microscopically, the cystic lesion was predominantly lined by ameloblastomatous epithelium with numerous ghost cells and dentinoid. Additionally, other cystic cavities lined by stratified squamous epithelium with corrugated parakeratin were observed in the fibrous capsule. Based on these features, a final diagnosis of a calcifying odontogenic cyst with odontogenic keratocyst-like areas was established. No recurrence was observed over a 9-year follow-up period. The association of a calcifying odontogenic cyst with odontogenic keratocyst or odontogenic keratocyst-like areas is very rare. To date, this is the second case report in the literature presenting these findings.

Peripheral calcifying odontogenic cyst in maxillary anterior gingiva: A case report.

BACKGROUND: Calcifying odontogenic cysts (Gorlin cysts) most commonly present centrally and have only rarely been reported in peripheral locations. The purpose of this report is to describe a new case of peripheral calcifying odontogenic cyst (PCOC) occurring in the anterior maxillary gingiva and to review the management and differential diagnosis of such a lesion. METHODS: A 37-year-old female presented with a long-standing submucosal nodule on the gingiva between the maxillary central incisors, with asymptomatic growth over the last three years. Following an initial incisional biopsy, a diagnosis of PCOC was established. To exclude the possibility of a central process, a corresponding small field of view cone beam CT scan was obtained and the patient returned for a 6 mm excisional biopsy to the depth of the periosteum. RESULTS: Results of these additional assessments supported the original diagnosis of PCOC. Following uneventful healing of the second biopsy, no recurrence or other clinical findings were noted at 1-year follow-up. CONCLUSION: While rare, the peripheral variant of calcifying odontogenic cyst, and other peripheral counterparts to recognized central cysts and tumors, should be considered in a differential diagnosis for a benign gingival nodule. Gingival tissue should be submitted for histologic evaluation to ensure a neoplastic process is not present. KEY POINTS: Various lesions may present on the gingiva as a "bump"; these can represent common clinical entities, such as pyogenic granuloma, peripheral ossifying fibroma, peripheral giant cell granuloma, and fibroma, or more rare conditions that may not be adequately considered in the differential diagnosis. A rarely documented case of peripheral calcifying odontogenic cyst (PCOC; Gorlin cyst) on the maxillary anterior gingiva of an adult female is reported here and compared with the few other similar PCOC cases in the literature. A biopsy of gingival lesions is always necessary to establish the correct diagnosis and provide the appropriate treatment. PLAIN LANGUAGE SUMMARY: Several different lesions can appear on the gingiva (gums). Some are quite common, and some are rare. This report documents the occurrence of a new case of calcifying odontogenic cyst (Gorlin cyst), a type of cyst that has been rarely found outside the jawbone, presenting as a "bump" on the gingiva between the maxillary central incisor teeth of an adult female. Because of the patient history, a peripheral calcifying odontogenic cyst (PCOC) was not initially suspected. Following a biopsy, a PCOC diagnosis was given. The possibility of a lesion within the bone was then excluded by an X-ray (cone beam CT) scan examination. A second, more extensive biopsy confirmed the diagnosis and the removal of the lesion. The patient had no complications or recurrence for the following 12 months. This case highlights the need to always biopsy lesions presenting on the gums to obtain a proper diagnosis and provide the correct treatment.

Epidemiology of odontogenic tumours and selected cysts diagnosed at a single New Zealand oral pathology centre- A 15-year retrospective study.

PURPOSE: This research aimed to investigate the relative frequency of odontogenic tumours (OT) and selected odontogenic cysts in a single oral pathology center in New Zealand from 2008 to 2023. METHODS: Histopathological records from the Oral Pathology Centre, University of Otago (2008-2023) were examined to identify OT. Odontogenic keratocyst (OKC) and calcifying odontogenic cyst (COC), previously classified as OT were also included. Patient demographics, clinical details and histopathologic diagnoses were recorded. Data were analyzed using SPSS. RESULTS: Of the 34,225 biopsies over the 15-year period, 1.8% were identified as OTs, COC and OKCs and accounted for 47%, 1.5% and 51.5% respectively. The most prevalent OT types were odontoma (43.7%), ameloblastoma (27%) and cemento-ossifying fibroma (7.5%). Malignant OT, ameloblastic carcinoma, constituted 1.4% of OT. The average age at diagnosis for OKC, COC and OT patients were 48.2 ± 20.9, 33.7 ± 23.3 and 28.9 ± 19.3 years. Overall, male and mandibular site predilections were observed. Recurrence of OKC and ameloblastoma occurred in 15.2% and 13.7% of patients. The time for recurrence for OKC and Ameloblastoma were 61.7 ± 56.5 months and 122 ± 152 months respectively. CONCLUSION: The demographic features and range of OT, COC and OKC in New Zealand align with those of other western countries. The study also confirms need for long term follow up for patient with OKC and ameloblastoma.

Multilocular Radiolucent Pathology in the Body and Ramus of the Mandible: A Case Report.

Odontogenic keratocyst (OKC), a type of epithelial developmental cyst, is frequently found in the jaw region. It has invasive characteristics such as satellite cysts, rapid progression, and tissue expansion. The OKC often favors the mandibular angle and ascending ramus. OKC symptoms include pain, swelling, displacement or malpositioning of adjacent teeth, and erosion or thinning of the limited or no bucco-lingual cortical expansion. There is radiographic evidence of a distinct, often scalloped, radiolucent lesion with a characteristic "soap bubble" or "honeycomb" appearance. This article reports a female patient, aged 40 years, with the main concern of unilateral pain and swelling of the mandibular left side and the provisional diagnosis of ameloblastoma. After histopathological examination, the final diagnosis of the patient was OKC. This article also includes previously published literature on OKC with differential diagnosis and relevant clinical and radiologic findings of the case.

Squamous odontogenic tumour-like proliferation in a maxillary dentigerous cyst - An unusual finding.

Squamous odontogenic tumour-like proliferations (SOTLPs) in the wall of odontogenic cysts are rare occurrences. Due to the histopathological similarity of these proliferations to neoplasms, such as squamous odontogenic tumour, intraosseous well-differentiated squamous cell carcinoma, and acanthomatous ameloblastoma, their correct elucidation is of paramount importance to avoid unnecessary and unwanted treatment. SOTLPs are uncommon in dentigerous cysts and rare in those that occur in the maxilla particularly the anterior region. This paper presents a case of maxillary dentigerous cyst involving 33 and a mesiodens in a 32 year old male which on histopathological examination showed SOTLPs in a dentigerous cyst.

Unravelling the mystery of the central dentinogenic ghost cell tumor- a rare case report and recurrent insights.

Dentinogenic ghost cell tumor (DGCT) is a rare benign neoplasm form of calcifying odontogenic cyst (COC) characterized by ghost cells. Although benign, it presents an aggressive behavior. DGCT accounts for 2% to 14% of all COCs and less than 0.5% of all odontogenic tumors. It is a benign odontogenic tumor despite its local invasion and the likelihood of recurrence. To detect recurrence, central DGCT patients must be monitored long-term. We present the case of a 51-year-old male who reported pain in the right upper back tooth region. On examination, a soft to firm, bright red swelling was present in the buccal vestibule and gingival margin of the maxillary right first and second molar, which extended up to the palate. Histopathological analysis confirmed the diagnosis of a DGCT, which occurred in a previously treated calcifying odontogenic cyst. The case is reported here, along with a review of the literature update of such recurred instances in the past.

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.

Calcifying odontogenic cysts: A novel outlook on classification, diagnosis and management.

BACKGROUND: The Calcifying Odontogenic Cysts (COC) displays a wide range of clinical and histopathological variations as well as diverse biological behaviors. This diversity has led to confusion and disagreement regarding the terminology and classification of this lesion. The previous classification attempts to categorize COC into two concepts. The first concept, termed "monistic," suggests that all COCs are neoplastic despite the majority being cystic in structure and seemingly non-neoplastic. The second concept, known as "dualistic," posits that COC comprises two distinct entities: a cyst and a neoplasm. This research discusses various previous classifications of COC found in the literature and proposes a new, straightforward universal classification based solely on histopathology, aiming to facilitate understanding for surgeons. MATERIAL AND METHODS: Fifteen cases of COC have been collected with clinicopathological parameters including detailed information regarding patient demographics, symptoms, anatomical site, radiological characteristics, duration of evolution, recurrence, and types of histopathology according to the proposed classification. RESULT: A total of fifteen cases of COC were analyzed. According to the histological analysis of the proposed classification Type 1: 5 (33.3), Type II: 4 (26.6), Type III: 3(20), and Type IV:3(20) and recurrence in 3 (20 %) of cases. CONCLUSION: It simplifies the complexities arising from variations in the cystic linings of type IV of COC, which can be overlooked and have caused recurrence in the current research. Therefore, the key requirement for arriving at a validated and practical conclusion lies in the accurate histological classification of calcifying odontogenic cysts and their impact on treatment.

Calcifying Epithelial Odontogenic Cysts of the Anterior Maxilla: Report of Two Cases.

This article is a discussion of two cases of young adults with lesions in similar locations in the anterior maxilla, i.e., the canine-to-canine region, similar history, and comparable radiology. Both cases were histologically diagnosed as calcifying odontogenic cysts. Case 1 was a male aged 28 years with diffuse, firm left malar area facial swelling with pain in associated teeth for a month. Intraorally, he had a gingivo-vestibular swelling also extending palatally in the anterior left maxillary region extending from the distal surface of the left maxillary central incisor to the mesial surface of the left maxillary canine. The overlying mucosa was normal in appearance. The radiograph showed a large unilocular radiolucency in the affected region. The lesion was excised followed by curettage and primary closure. Case 2 was a female aged 25 years with a lumpy mass and pain in associated teeth since one year in the left canine-premolar region with an external swelling in the left ala of the nose region that extended superiorly to the zygomatic arch. The color of the skin as well as the intraoral mucosa was normal, and an orthopantomogram (OPG)revealed a unilocular radiolucency in the left maxillary canine-premolar region with resorption of premolar roots. Treatment included surgical enucleation and bone curettage. Both cases have been in follow-up for about a year and have shown non-incidental healing.

CITED1 expression in odontogenic cysts.

BACKGROUND: Originating from odontogenic tissue, Odontogenic cysts are pathological cavities lined with epithelial cells and surrounded by fibrous connective tissue. This study investigated expression of CITED1 protein in different types of odontogenic cysts. MATERIAL AND METHOD: 40 keratocysts, 40 radicular cysts, and 40 dentigerous cysts were excised and processed for routine paraffin wax embedding protocol. Macroscopic and panoramic radiographies images were used for diagnosis. Demographical properties and dental parameters were recorded. Cystic tissues were stained with hematoxylin-eosin dye and CITED1 antibody. Semi-quantitative analysis was performed for immune staining. The protein-protein interaction network, hub gene detection and KEGG analysis were conducted using Cytoscape software. RESULT: Odontogenic keratocysts was imaged with 6-8 layered epithelial cells and fibrous cyst walls with inflammatory cells. Radicular cysts had stratified squamous epithelium with varying thickness, ciliated cells, and Rushton hyaline bodies. Dentigerous cysts presented hyperplastic non-keratinized epithelium, fibrous tissue, rete ridges, and inflammatory cells. CITED1 immunoexpression was highest in odontogenic keratocysts, followed by radicular cysts, and lowest in dentigerous cysts. Nuclear and cytoplasmic CITED1 expression was significantly elevated in odontogenic keratocysts compared to radicular and dentigerous cysts. The top five targets of CITED1 were identified, primarily showing enrichment in hormone and cancer related pathways. CONCLUSIONS: Positive CITED1 expression in all three types of odontogenic cysts suggest a potential role for CITED1 in the pathogenesis of odontogenic cysts, particularly in keratocysts. Further investigations are needed to elucidate the exact mechanisms underlying the differential expression of CITED1 and its implications for the development and progression of odontogenic cysts.

Alpha-Tocopherol Levels in Patients With Dentigerous Cysts: A Cross-Sectional Study.

Background Dentigerous cysts (DC) form due to fluid accumulation between the crown of the tooth and the reduced enamel epithelium. Due to the diverse clinical characteristics, such as ambiguity concerning their biological origins and the significance of timely diagnosis and detection of these lesions, researchers are presently motivated to undertake further investigations. The aim of the present study was to assess the amount of serum alpha-tocopherol in patients with DC and compare it with that of normal, healthy individuals. Methods A total sample size of n=34 was included in the current study. Group A, designated as the control group, comprised 17 randomly selected healthy subjects, while Group B, the DC diagnostic group, consisted of 17 patients. Blood samples were collected, and the concentration of vitamin E or alpha-tocopherol was evaluated and expressed in mg/mL. Results Compared to the mean vitamin E level in healthy controls (12.08 ± 1.92 mg/mL), patients with DC showed a statistically significant (p<0.0001) reduction in mean vitamin E levels (5.29 ± 1.01 mg/mL). Conclusion Patients with DC have lower levels of vitamin E than healthy individuals. The reduced concentration of vitamin E can have a role in the extension of cystic volume and thus have an impact on the aggressiveness of pathologic lesions. The therapeutic benefits of vitamin E supplementation in reducing the aggressiveness of DC should be evaluated in future research.

Characterization of stromal calcifications in odontogenic keratocyst: a multicentric study.

Odontogenic keratocysts (OKCs) are locally aggressive cysts that exhibit typical histopathological features and have a propensity for recurrence. Though histological variations are observed in OKCs, hard tissue formation and metaplastic changes are rare, and the underlying pathogenesis is not well understood. This study aimed to characterize stromal calcifications and analyze their association with odontogenic components in non-syndromic and syndrome-associated cases of OKCs. We analyzed 153 cases of OKCs from healthcare institutes in India and Japan. The epithelial and stromal features were evaluated, and the relationship of calcifications with odontogenic rests was determined. Immunohistochemistry for cytokeratin-19 and special stains including Masson Trichrome and Van Gieson, were used for identification of odontogenic rests and calcifications respectively. Stromal calcifications were observed in 29.41% OKCs. The calcification patterns included irregular dystrophic, dentinoid with linear or calcospherite-type mineralization, and psammoma calcifications. Psammoma and dentinoid calcifications were found in the proximity of cytokeratin-19-positive odontogenic rests or satellite cysts, whereas majority cases with dystrophic calcifications did not exhibit co-localization with stromal odontogenic components. Distinct patterns of calcifications were observed in OKCs. Calcifications found in proximity of the odontogenic rests were possibly indicative of an inductive or host-mediated response.

Spread asymmetry to differentiate nasopalatine duct cysts from radicular cysts arising in the anterior maxilla on computed tomographic images.

OBJECTIVE: The aim of this study was to clarify numerical values for differentiating nasopalatine duct cysts (NPDCs) from radicular cysts (RCs) arising in the anterior maxilla on computed tomography (CT) or cone-beam CT (CBCT) images. METHODS: CT or CBCT images of histologically proven NPDCs (n = 30) and RCs (n = 33) beyond the midline of the maxilla were investigated to determine two asymmetry indices on axial images of the maximum lesion area. The lateral asymmetry index was calculated based on two distances from each of the lateral ends of the lesion to the midsagittal plane. The index was defined as the difference between the two distances divided by their sum. The labio-palatal asymmetry index was determined by the distance between the labial and palatal ends of the lesion and the coronal plane passing through the central incisor root apex. The performance of these indices was assessed by receiver operating characteristic (ROC) analysis. The cutoff values for differentiating NPDCs from RCs were determined with the Youden procedure on the ROC curve. RESULTS: The area under the ROC curve was 0.97 for the lateral asymmetry index and 0.88 for the labio-palatal asymmetry index. The cutoff values for differentiation were 0.36 and 0.68 for the lateral and labio-palatal asymmetry indices, respectively. CONCLUSION: The lateral asymmetry index appeared to be an effective reference for differentiating NPDCs from RCs on CT or CBCT images. When the index was less than the cutoff value, a diagnosis of NPDC was strongly suggested.