Case Of Odontogenic Keratocyst Research Articles

Analysis of Germline and Somatic Mutation in Patients With Developmental Odontogenic Cysts Using Targeted Gene Panel.

Odontogenic keratocyst (OKC) is a partial manifestation of Gorlin syndrome (GS), resulting from the abnormal activation of the hedgehog signaling pathway. OKC predominantly occurs in young adults and is mostly asymptomatic at the time of initial diagnosis. As OKC is asymptomatic, GS can be challenging to diagnose in certain instances. In this study, we attempted to identify asymptomatic GS from sporadic OKC cases using a previously developed gene panel for GS. Genomic DNA was extracted from patient samples. These DNA samples were analyzed using the AmpliSeq Custom DNA Panel (Illumina), which was specifically designed to target four previously established genes (PTCH1, PTCH2, SMO, and SUFU). Mutations from patients were predicted using tools, such as MutationTaster, CADD, and Polyphen-2. Thirty-one patients with OKC were included: 22 sporadic, 9 syndromic, 14 cases with dentigerous cysts, and 3 patients with orthokeratinized odontogenic cysts. One patient with sporadic OKC carried 50% genetic mutation in the cyst and blood, indicative of GS. PTCH1 mutations were found in one of the 14 patients with dentigerous cysts, 3 of the 17 first-time sporadic cases, and all four recurrent cases. Resected OKC tissues revealed a PTCH1 mutation. We found one patient with GS from those diagnosed with sporadic OKC. Our findings suggest that PTCH1 mutations are associated with postoperative recurrence of OKC, implying that hedgehog-related gene variations may contribute to jaw cyst development and improve the prognosis of OKC.

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 andanalyze 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.

Neuroectodermal influence in odontogenic cyst and tumor: evidence through CD56 immunoexpression.

CD56, associated with neuroectodermal differentiation of the embryonal cells, is often considered a marker of neural lineage. Odontogenic keratocysts (OKCs) are of particular interest because of their characteristic histopathologic features, high recurrence rate, and aggressive behavior. CD56 immunoreactivity in these lesions has been reported with very high frequency. The present study analyzes the immunoexpression of CD56 in ameloblastoma (AM) and OKC to infer neuroectodermal influence in the pathogenesis of odontogenic lesions and its correlation with clinicopathologic parameters. Fifty histopathologically confirmed cases of OKC and AM, 25 from tooth-bearing (TB) and molar-ramus (MR) regions each, and 5 dental follicular tissues as control were collected from the department archives and immunohistochemical analysis with CD56 was carried out. CD56 immunopositivity was seen in 64% AM and 36% OKC cases. The majority of AM cases showed cytoplasmic expression in the peripheral cells of odontogenic islands; similarly, OKC cases showed continuous and uniform cytoplasmic expression in the basal and parabasal cells of the cystic lining. CD56 immunopositivity was found in more AM cases as compared to OKC cases in both the TB and MR regions. The assessment of CD56 immunoexpression in odontogenic cyst and tumor (AM) may aid in understanding the role of neuroectodermal influence in the etiopathogenetic pathways and a possible influence of CD56 on the clinical behavior and aggressiveness of the odontogenic lesions. A correlation of CD56 expression with the clinical outcome of the disease (site, perforation, root resorption, and tooth displacement) can help envisage possible prognostic assessment for these lesions.

Differential Diagnosis of OKC and SBC on Panoramic Radiographs: Leveraging Deep Learning Algorithms.

This study aims to determine whether it can distinguish odontogenic keratocyst (OKC) and simple bone cyst (SBC) based solely on preoperative panoramic radiographs through a deep learning algorithm. (1) Methods: We conducted a retrospective analysis of patient data from January 2018 to December 2022 at Pusan National University Dental Hospital. This study included 63 cases of OKC confirmed by histological examination after surgical excision and 125 cases of SBC that underwent surgical curettage. All panoramic radiographs were obtained utilizing the Proline XC system (Planmeca Co., Helsinki, Finland), which already had diagnostic data on them. The panoramic images were cut into 299 × 299 cropped sizes and divided into 80% training and 20% validation data sets for 5-fold cross-validation. Inception-ResNet-V2 system was adopted to train for OKC and SBC discrimination. (2) Results: The classification network for diagnostic performance evaluation achieved 0.829 accuracy, 0.800 precision, 0.615 recall, and a 0.695 F1 score. (4) Conclusions: The deep learning algorithm demonstrated notable accuracy in distinguishing OKC from SBC, facilitated by CAM visualization. This progress is expected to become an essential resource for clinicians, improving diagnostic and treatment outcomes.

Management regulations for odontogenic keratocyst: a case report and review of the literature

BackgroundReconstruction of the entire dentition with odontogenic keratocyst is a very challenging quandary. Most cases of odontogenic keratocyst are often reported to be benign, resulting in severe occlusal discrepancies with the maxillary and mandibular dentition. Dental radiographs occasionally reveal an uncommon, locally aggressive developing cyst termed as odontogenic keratocyst, which is typically located in the posterior jaw. When this cyst occurs in the anterior region, it is often misdiagnosed with other periapical lesions due to its lack of response to pulp vitality tests.Case presentationThis clinical case scenario demarcates the endodontic management of a patient diagnosed with odontogenic keratocyst. A 37-year-old Indian male patient reported to the department with throbbing pain in the lower left posterior tooth requiring endodontic therapy. This patient also presented with odontogenic keratocyst in the anterior region of the jaw, for which he had undergone surgical rehabilitation. This case report highlights the clinical protocol for the endodontic therapy in patient diagnosed with ododntogenic keratocyst. Masticatory impairment was not visible after the follow-up period and the treatment outcome was successful.ConclusionThis case report details the presentation, characteristic radiographic findings, and endodontic management of a patient with an extremely rare condition of odontogenic keratocyst. The management involves multidisciplinary approach for the rehabilitation.

A Case of Odontogenic Keratocyst in the Posterior Ramus and Body of the Mandible

The dental lamina gives rise to the odontogenic keratocyst (OKC), a common odontogenic cyst that affects the maxillofacial region. The OKC is distinct from other jaw cysts and is more likely to return when combined with aggressive clinical behaviour. The recurrence rate in OKC is 25%–30%. In 2005, the World Health Organization (WHO) group categorized odontogenic keratocyst (OKC) as a tumour and proposed the abbreviation KCOT to differentiate the condition from the ortho keratinizing variant. The WHO reclassified KCOT as OKC in 2017 based on data demonstrating non-neoplastic clinical behaviour. The 19-year-old male in this case study has OKC in the ramus and body of his jaw, near the left mandibular molar tooth. This case study aims to demonstrate the need for dentists to do in-depth investigations into each circumstance and offer patients better treatment options with ongoing patient monitoring and follow-up. Enucleation, marsupialization, and other surgical techniques are possible; however, in this case, our objective was to preserve the patient's mandible and facial features.

Immunohistochemical expression of SOX2 in OKC and ameloblastoma: A comparative study.

Odontogenic, non-inflammatory maxillofacial cysts and tumours vary greatly in their ability to grow and cause local tissue destruction. Despite their common embryologic origin, the biologic mechanisms responsible for this diverse array of clinical behaviour are largely unknown. Unfortunately, even with accurate tissue diagnosis and appropriate surgical management, these tumours have relatively high recurrence rates. While this may be related to surgical technique, it may also be due to intrinsic tumour biology. SOX2 is differentially expressed in odontogenic cysts and tumours, which has an impact over patient prognosis. This could be related to their diverse cells of origin or stages of histogenesis. SOX2 is expressed in OKC and ameloblastoma, and in this study, we look forward to find altered levels and intensity of SOX2 in the above-mentioned lesions. To profile the expression of SOX2 in odontogenic keratocyst (OKC) and ameloblastomaTo compare the intensity of these lesions, analyse their intrinsic feature and predict their recurrence. Histopathologically diagnosed cases of OKC and ameloblastoma will be selected (n = 40). Paraffin-embedded, formalin-fixed sections of these lesions will be stained for SOX2 marker using a standard immunohistochemical technique. Positive control will be taken as oral squamous cell carcinoma and negative control will be taken as normal oral mucosa. A comparison between the stained cell types in odontogenic keratocyst and ameloblastoma revealed statistically significant differences. The immunoreactivity scores of SOX2 were analysed in both groups. The results indicated that 45% of OKC cases exhibited strongly positive reactivity, while 65% of ameloblastoma cases were negative. Statistical analysis demonstrated highly significant differences in the frequency of SOX2 expression between the two groups, with a higher frequency of negative expression in ameloblastoma. Stem cell markers have been observed in these lesions, suggesting the acquisition of stem-like properties by tumour cells, which can affect patient prognosis. Specifically, the marker SOX2 shows differential expression in odontogenic cysts and tumours. High expression of SOX2 in OKC indicates the presence of stem cells with significant self-renewal and proliferative properties, potentially signifying neoplastic behaviour. In contrast, weak or absent expression of SOX2 in ameloblastoma suggests different molecular pathways involved in its neoplastic behaviour.

Appearance and recurrence of odontogenic keratocysts.

The purpose of this study was to evaluate the appearance, histopathological features, and recurrence of odontogenic keratocysts (OKCs) from a large single institute registry over a 36-year period. A total of 226 cases of OKC were identified in 174 patients over a 36-year period in a single institute in Southwestern Finland. Histological specimens were re-evaluated. The patient's age, sex, location, recurrence, and histopathological features of the OKC were the study variables. OKCs occurred more frequently in men, the mean age was 46 years, and the most frequent site was the lower jaw. Recurrence rate was 21%. Histopathologically, inflammation was present in 95% and satellite cysts in 10% of cases. In patients diagnosed with satellite cysts, OKC recurred in 50% of cases, while the corresponding figure for patients without satellite cysts was 17%. Compared with the literature, patients were older and inflamed cysts were found more frequently. Satellite cysts occurred only in association with chronic inflammation. Based on the results, regular radiographic evaluation is important among patients aged 10-29 years to detect OKCs and to treat them before enlargement, infection, and inflammation. Satellite cysts should be reported and may be a sign of increased risk of OKC recurrence.

Cone-beam CT imaging features of common cystic lesions associated with the impacted mandibular third molar

Objective: To analyze the imaging features of cone-beam CT (CBCT) of ameloblastoma (AB), odontogenic keratocyst (OKC) and dentigerous cysts (DC) associated with the mandibular impacted third molars,so as to provide useful information for differential diagnosis of these lesions. Methods: The patients who were with complete clinical data, pathological diagnosis and CBCT images from AB, OKC and DC around the mandibular impacted third molars were collected in Peking University Hospital of Stomatology from August 2016 to December 2021. A total of 109 patients (14 were diagnosed as AB, 23 were diagnosed as OKC and the others were diagnosed as dental cysts) were collected, including 73 males and 36 females. The age ranged from 11 to 70. The analyzed imaging features included location and internal density of the lesions, bone expansion, root resorption of adjacent teeth and types of the impacted teeth. The Chi square test was used to compare the gender of different lesions, and the Fisher's exact test was used to compare imaging features of lesions. When P<0.05, there was a significant difference among the three. Logistic regression analysis was performed to determine the imaging features that significantly contribute to correct imaging diagnosis. Corresponding P-values were calculated for all factors from multivariate models. Results: In the 23 cases of OKC, no special location was observed for the center of the lesion, heterogeneous high-density were seen in 21.7% of the cases, 56.5% of the cases had no significant bone expansion and the impacted teeth were not specially oriented. Among the 14 AB, 7 cases (7/14) were mainly located in the ramus of the mandible, and all cases (14/14) had buccal/lingual expansion of the jaw, 8 cases (8/14) presented root resorption of the adjacent teeth, and mesial impacted mandibular third molar were seen in 6 cases (6/14). Among the 72 DC, 88.9% (64/72) of the cases were mainly limited to the crown of the impacted third molar, 72.2% (52/72) of the cases had no obvious bone expansion, inverted impacted teeth were shown in 56.9% (41/72) of the cases. There was a significant difference among the three groups (χ2=7.30, P=0.026) in gender. AB and odontogenic cyst were more common in men than in women, while the incidence of OKC was roughly equal between men and women.There were significant differences in the location (P<0.001), internal density (P=0.001) of the lesions, bone expansion (P<0.001) and types of the impacted teeth (P<0.001), while no statistical difference was found for root resorption of adjacent teeth (P=0.153). Logistics regression analysis showed that the location of the lesion, internal density, bone expansion, root resorption of adjacent teeth and the types of impacted teeth had significant effects on the accurate diagnosis of the three kinds of lesions. Conclusions: Location, internal density, bone expansion and types of the impacted teeth played an important role in the correct imaging diagnosis. Further analysis indicates that when the classification of impacted teeth and the location of lesions are considered synchronously, DC can be differentiated from AB and OKC.

Case of Odontogenic Keratocyst in the Posterior Ramus and Body of the Mandible Mimicking Ameloblastoma

The dental lamina gives rise to the odontogenic keratocyst (OKC), a common odontogenic cyst that affects the maxillofacial region.The OKC is distinct from other jaw cysts and is more likely to return when combined with aggressive clinical behavior. The recurrence ratein OKC is 25%–30%. In 2005, the World Health Organization (WHO) group categorized odontogenic keratocyst (OKC) as a tumor andproposed the abbreviation KCOT to differentiate the condition from the ortho keratinizing variant. The WHO reclassified KCOT as OKC in2017 based on data demonstrating non-neoplastic clinical behavior. The 30-year-old male in this case study has OKC in the ramus and body ofhis jaw, near the right mandibular molar tooth. When the patient reported to us, he complained of pain and swelling, 48 was clinically absent,and there was an enlargement of the buccal and lingual cortical plates. Palpation revealed tenderness in the region distal to 48. CBCT revealeda sizable radiolucent lesion, making it difficult to determine whether it was an odontogenic keratocyst or ameloblastoma in the body and lowerpart of the ramus on the right side. Histopathology findings corroborated OKC's diagnosis. As a precaution, the affected teeth were extracted,marsupialization was done, and an ongoing follow-up was done for an additional 1.5 years. There hasn't been a recurrence of OKC, and itshowed good healing. This case study aims to demonstrate the need for dentists to do in-depth investigations into each circumstance and offerpatients better treatment options with ongoing patient monitoring and follow-up. Enucleation, marsupialization, and other surgical techniquesare possible; however, in this case, our objective was to preserve the patient's mandible and facial features.

Appraisal of an Innovative Surgical Approach in Treating Odontogenic Keratocyst

Objective: To estimate the recurrence of odontogenic keratocyst (OKCs) and increment of bone height upon an innovative treatment protocol of the OKC, i.e., enucleation along with the adjuvant therapy of Carnoy’s solution, followed by marsupialization. Methods: Twenty cases of OKC treated at the Department of Oral and Maxillofacial Surgery from 2020-2021 were studied retrospectively. Clinical, radiological features and histologic features were reviewed. The patients diagnosed with OKCs were planned for enucleation with the use of Carnoy's solution and followed by marsupialization. Recurrence of the same and bone formation was analyzed concerning sites of involvement, based upon the gender and age group of patients, after undergoing the proposed treatment. Results: Mean age of the patients was 35.15±13.02, ranging from 11 to 56 years. Most (85%) patients were symptomatic, and the remaining (15%) were asymptomatic and diagnosed on a routine check-up. Males (65%) were mostly affected than females (35%). Mandible (75%) was the most frequent site of occurrence. Most lesions were diagnosed histologically as OKC on incisional biopsy. All patients were followed for six months, an increment of 11.11±1.68mm bone height was seen, and none reported recurrence. Conclusions: The steady growth of bone without any cases of recurrence was reported. Hence, clinicians can consider enucleation along with adjuvant therapy using Carnoy's solution followed by marsupialization as the treatment of choice.

The Role of Adjunctive Chemical Solutions in the Treatment of Odontogenic Keratocysts: A Scoping Review.

Odontogenic keratocysts (OKC) are aggressive cysts with a high recurrence potential. Treating them with surgical enucleation procedures alone is associated with high recurrence rates; therefore, additional or supportive treatment approaches, such as peripheral osteotomy, cryotherapy, and chemical solutions, are warranted.The objective of the present review was to evaluate the existing literature on the efficacy of chemical approaches, such as Carnoy's solution (CS), in preventing recurrence after the enucleation of OKC.An electronic search was conducted on PubMed, Scopus, and Google Scholar databasesto find articles published from January 2010 to December 2022 by using the Medical Subject Headings (MeSH) terms "Odontogenic Keratocyst" "Carnoy's Solution," "Treatment," and "Enucleation." Articles published in the English language were selected for the study. The PICOS criteria (population: patients with non-syndromic OKC with histopathological diagnosis and a minimum follow-up of six months; intervention and comparison: enucleation followed by adjunctive chemical therapy and standard procedure; outcome: recurrence rates; study design: retrospective and prospective studies, randomized controlled trials, and case series involving at least 10 cases of OKC) were employed. Studies involving syndromic (nevoid basal cell carcinoma) cases were excluded from the search.Seventeen studies fulfilled the inclusion criteria and the majority of them were retrospective studies, with a few case series. OKC was found more frequently in the mandible, with a recurrence rate of 11%, when treated with CS following enucleation after four years of follow-up. Modified Carnoy's solution (MC) was used in two studies. The mean follow-up period was 44 months. Based on our findings, adjuvant therapy using a chemical approach following enucleation is a more effective and beneficial modality for the treatment of OKC.

Divulgence of Distribution of Odontogenic Keratocyst Based on Demographic and Radiographic Features: A Retrospective Institutional Study

Introduction: Odontogenic keratocysts (OKC) is a developmental, slow-growing cyst that illustrates aggressive behaviour, a high recurrence rate, extensive radiologic diversity and variation within age groups and gender. The objective of this study is to evaluate the distribution and radiological traits of patients diagnosed with OKC. Materials and Methods: A cross-sectional retrospective assessment of 40 diagnosed cases of OKC archived at the Government College of Dentistry in Indore was conducted. Demographic information and radiological characteristics of the cases were investigated, sorted, tabulated, and placed through descriptive valuation before being expressed as frequencies and percentages. Results: This study investigated 40 diagnosed OKC cases ranging within the age group of 13 to 63 years (57.5% male and 42.5% female). OKC is more often encountered in the 2nd–3rd decade of life. Depending on the anatomical area, OKC often originates in the posterior mandible, followed by the anterior mandible and maxilla. Concerning radiological characteristics, a unilocular variant was more prevalent (65.0%) than a multilocular. About 97.5% of OKC cases had scalloped and well-corticated borders. Also, the mandibular left third molar was frequently involved (69.2%). Only 15% of OKC cases coexisted with root resorption, 25% with tooth displacement, and 40% with inferior alveolar canal displacement. Conclusion: Our study showed significant disparities in the distribution of OKC among various age groups, genders, and radiological features. Our variables involved in this study were comparable to previous research. Several radiological aspects will aid in treatment strategies, effectiveness, and limiting the recurrence rate. However, a substantial sample size is required to validate these findings.

An Insight Into the Effect of Odontogenic Keratocysts on Surrounding Structures: Cone-Beam Computed Tomography-Based Analysis of Cases.

Introduction An odontogenic keratocyst (OKC) is a benign intraosseous lesion with potential to demonstrate aggressive and invasive behavior. The aim of this retrospective study was to analyze the imaging features of the OKCusing cone-beam computed tomography (CBCT) and to evaluate the association between the internal structure of the lesion and the effect of the lesion on surrounding structures. Methods Overall, 32 CBCT scans of histopathologically diagnosed cases of OKC were analyzed retrospectively. The following variables were analyzed: anatomic location of the lesions (mandible body (right/left), ramus (right/left), mandible body+ramus (right/left), maxilla (right/left), and both jaws), the internal structure of the lesion (unilocular/multilocular), and the effect ofthe lesion on the surrounding anatomical structures (involvement of the inferior alveolar nerve canal (IANC), displacement of the IANC, cortical expansion, displacement of the tooth, resorption of the root, associated impacted tooth, associated missing tooth). We also looked for the association between the internal structure and the effect of the lesion on anatomic structures. Results Out of 32 cases, 29 (90.6%) cases involved the mandible alone. Statistically significant association (p-value 0.005) was present between the internal structure and mean age of presentation as well as between the internal structure and impacted tooth (p-value 0.027). The association between the internal structure and other variables was statistically not significant. Conclusions The radiographic features of OKCs can be variable, and these lesions have a considerable effect on the tooth, IANC, and cortical bone. Significant association was found between the internal structure, age, and impacted tooth. Since OKCs have a high recurrence rate, CBCT is advised for evaluating the extent and location of any cortical perforations.

Clinicopathological Features and Expression of Ki-67 in Odontogenic Keratocyst, Dentigerous Cyst and Radicular Cyst

Background: Biological behaviour of Odontogenic Keratocyst (OKC) is aggressiveness than others Odontogenic Cysts (OCs) like Dentigerous Cyst (DCs) and Periapical Cyst/Residual Cysts (RCs). The aim of the study was to determine clinicopathological features and expression of Ki-67 in Odontogenic Cysts of the oral cavity.&#x0D; Methodology: This cross-sectional study was conducted at de’Montmorency College of Dentistry (DCD from Feb, 2020 to Feb, 2022 after approval from Institutional Review Board (IRB) of DCD. A total 78 cases of OCs were collected from hospitals which are affiliated with the DCD. Routine lab process for Hematoxylin &amp; Eosin and Immunohistochemistry was performed. Data entry and statistical analysis was carried out in SPSS 21. A Chi- square test was applied to observe the association between cyst and Ki-67. P value &lt; 0.05 was taken as statistically significant. &#x0D; Results: Among total 78 cases the mean age was 25.08 ±14.5 years with an age range of 6-70 years. Most OCs (64.1%) were reported in males than females (35.89%). Most OCs were reported in mandible (56.4%. Ki-67 expression in OKC was high 7.7%, low in 76.9% and negative in 15.4%). Most of the DCs and RCs expressed low expression of Ki-67 (84.6% and 76.9% respectively). &#x0D; Conclusion: Most of the odontogenic cysts expressed low expression of Ki-67 while few cases of OKC and DC expressed high expression.&#x0D; Keywords: Dentigerous Cyst, Odontogenic Keratocyst, Immunohistochemistry, Ki-67, Odontogenic Cysts, Periapical Cyst, Radicular Cyst.

Expression of PTCH gene in ameloblastoma and odontogenic keratocyst: A comparative study.

This is a cross-sectional comparative study, aimed to quantify the expression of patched (PTCH) gene in ameloblastoma, odontogenic keratocyst (OKC) and also the comparison of both the expressions. Genomic deoxyribonucleic acid (DNA) was extracted and quantified, and the expression of the PTCH gene was done in 17 cases of ameloblastoma and 17 cases of OKC by quantitative real-time polymerase chain reaction (RT-qPCR). It was observed that there was an overexpression of the PTCH gene in both ameloblastoma and OKC with a good mean cycle threshold (CT) value of 32.71 ± 2.432 and 34.69 ± 1.875, respectively. When comparing the PTCH expression between the two, ameloblastoma showed higher expression than the OKC and the difference is statistically significant with P value of 0.025. Our findings suggest that there is overexpression of PTCH in ameloblastoma and OKC, but it is highly expressed in ameloblastoma when compared to OKC. Overexpression of PTCH may constitute the activation of the Sonic Hedgehog pathway and may suggest the mechanism for the development of ameloblastoma and OKC. Hence it can be used as a valuable marker for early diagnosis and in the identification of therapeutic targets.

Comparative Molecular Genetics of Odontogenic Keratocysts in Sporadic and Syndromic Patients

Odontogenic keratocysts (OKCs) are common cysts of odontogenic origin that usually occur as a single nonsyndromic cyst in isolation (sporadic) or as syndromic multiple cysts as a manifestation of naevoid basal cell carcinoma syndrome. Alterations involving the PTCH gene are the most commonly identified factor associated with up to 85% and 84% of naevoid basal cell carcinoma syndrome and sporadic cases, respectively. Other Hedgehog pathway and non-Hedgehog pathway-associated genes have been implicated in the pathogenesis of OKCs. This pilot study used the Affymetrix OncoScan molecular assay to perform a comparative genomic analysis between 4 sporadic and 3 syndromic cases of OKC to identify molecular drivers that may be common and/or distinct in these 2 groups. The majority of alterations detected in both groups were copy number neutral loss of heterozygosity. Despite distinct molecular signatures observed in both groups, copy number neutral loss of heterozygosity alterations involving chromosome 9q affecting not only PTCH but also the NOTCH1 gene were detected in all syndromic and 3 sporadic cases. Loss of heterozygosity alterations involving 16p11.2 affecting genes not previously described in OKCs were also detected in all syndromic and 3 sporadic cases. Furthermore, alterations on 22q11.23 and 10q22.1 were also detected in both groups. Of note, alterations on 1p13.3, 2q22.1, and 6p21.33 detected in sporadic cases were absent in all syndromic cases. This study demonstrates that a more common group of genes may be affected in both groups of OKCs, whereas other alterations may be useful in distinguishing sporadic from syndromic cysts. These findings should be validated in larger OKC cohorts to improve molecular diagnosis and subsequent patient management.

DIAGNOSIS OF ODONTOGENIC KERATOCYST IN ANTERIOR REGION OF THE MAXILLA: CLINICAL CASE REPORT

Odontogenic keratocyst (OKC) is an aggressive behavioral cyst, which has a predilection for men and usually affects the posterior mandibular region (49%). The aim of this study is to report a case of OKC in the anterior maxillary region. Physical examination showed a slight increase in volume in the anterior maxillary region. Panoramic radiography showed a radiolucent image at the apices of units 21 and 22, treated endodontically, suggestive of an inflammatory cyst. Computed tomography showed the presence of a hypodense, well-defined area with spacing and disruption of the buccal cortex and growth in the anteroposterior maxillary direction. An incisional biopsy was performed, resulting in an OKC diagnosis. The treatment instituted was enucleation and curettage under general anesthesia and exodontia of teeth 21, 22, and 23 associated with the lesion. The anatomopathologic was confirmatory for the previously obtained report and the patient is being followed up with no signs of recurrence so far.

Odontogenic Keratocyst of the maxilla: Case report

Odontogenic Keratocyst (OKC) is a pathology that presents high proliferative activity and frequent relapses after treatment. The aim of this study was to report a case of OKC with uncommon location in a 22-year-old patient presenting swelling and drainage of purulent secretion in the region of the third right upper molar. In examinations, a radiolucent density unilocular image involving impacted element 18 was observed. After excisional biopsy accompanied by severe bone curettage, the removed specimen was sent for histopathological analysis, where final diagnosis of odontogenic keratocystic was obtained. The patient has been in clinical and radiographic follow-up for more than 6 months and, to date, there are no signs of relapse.

Glucose transporter 1 expression in ameloblastoma and odontogenic keratocyst - A comparative immunohistochemical study.

Introduction:Facilitative glucose transporters (GLUTs), which mediate glucose transport across the cell membrane, differ in their tissue distribution and affinity for glucose. GLUT1 is ubiquitously present and help in the basal uptake of glucose into the cells. Its expression is known to be elevated in conditions that induce hypoxia and by growth factors. GLUT1 is known to be increased in many malignant tumors to meet the metabolic requirements, but its role in odontogenic tumors is not known.Objective:The objective of this study is to evaluate and compare the immunohistochemical expression of GLUT1 in ameloblastoma (AM) and odontogenic keratocyst (OKC).Materials and Methodology:Thirty cases each of AM and OKCs were immunohistochemically stained using anti-GLUT1 antibody according to the standard protocol. Qualitative assessment of GLUT1 expression was done under the categories of distribution, intensity and localization of staining. Quantitative assessment was done using Image J software. The results were tabulated and statistically analyzed.Results:GLUT1 positivity was observed in 25 (83.3%) cases of OKC and 26 (86.7%) of AM cases. The majority of cells in the suprabasal layer of OKC showed positivity, whereas the equal distribution of staining was observed in the central and peripheral cells of AM.Conclusion:GLUT1 expression in these tumors is suggestive of an increased glucose uptake and probably increased utilization of energy, which may be correlated with their aggressive behavior.