Non-syndromic Odontogenic Keratocysts Research Articles

Does YAP influence cell proliferation and apoptosis in benign epithelial odontogenic lesions?

To analyze the immunohistochemical expression of YAP and its correlation with markers involved in cell proliferation and apoptosis in benign epithelial odontogenic lesions. The sample consisted of 95 cases of odontogenic lesions (25 dentigerous cysts, 30 non-syndromic odontogenic keratocysts, 30 conventional ameloblastomas, and 10 unicystic ameloblastomas) and 10 dental follicles used as normal odontogenic tissue. The histological sections were submitted to immunohistochemistry with YAP, cyclin D1, Ki-67, and Bcl-2 antibodies. Immunoexpression was analyzed qualitatively and quantitatively using an adapted method. The collected data were analyzed descriptively and statistically (p ≤ 0.05). The highest YAP expression was observed in odontogenic keratocysts, followed by unicystic ameloblastomas and conventional ameloblastomas, which exhibited moderate immunoreactivity predominantly in peripheral cells. Furthermore, significant differences in YAP immunoexpression were observed between the groups analyzed, with significant positive correlations between YAP and cyclin D1 in dentigerous cysts and unicystic ameloblastomas and between YAP and Ki-67 in unicystic ameloblastomas (p < 0.05). However, there were no statistically significant correlations between YAP and Bcl-2 immunoexpression in the groups studied. YAP may influence epithelial cell proliferation in odontogenic cysts and tumors, suggesting its possible participation in the progression of the odontogenic lesions studied.

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.

IMMUNOHISTOCHEMICAL ANALYSIS OF CXCL12 AND CXCR4 IN SYNDROMIC AND NONSYNDROMIC ODONTOGENIC KERATOCYSTS

Objectives To evaluate the immunoexpression of chemokine CXCL12 and C-X-C chemokine receptor type 4 (CXCR4) in odontogenic keratocysts associated with Gorlin-Goltz syndrome (SOKCs) and nonsyndromic odontogenic keratocysts (NSOKCs). Study Design The percentages of cytoplasmic (CXCL12 and CXCR4) and nuclear (CXCR4) staining in epithelial and fibrous capsule cells of 22 SOKCs and 22 NSOKCs were determined. The results were analyzed statistically using the nonparametric Mann-Whitney test and Spearman correlation test. Results Higher cytoplasmic expression of CXCL12 was observed in the epithelial lining and fibrous capsule of NSOKCs compared to SOKCs (P < .001). No statistically significant differences in the cytoplasmic expression of CXCR4 were observed between SOKCs and NSOKCs (P > .05). Compared to SOKCs, NSOKCs exhibited higher nuclear expression of CXCR4 in the epithelial lining and lower immunoexpression in the fibrous capsule (P < .05). In the epithelial lining of SOKCs, a positive correlation was observed between cytoplasmic and nuclear expression of CXCR4 (P = .003). In the fibrous capsule of SOKCs and NSOKCs, cytoplasmic and nuclear expressions of CXCR4 were positively correlated (P < .001). Conclusions The results suggest a potential involvement of CXCL12 and CXCR4 in the development of OKCs. The heterogenous expression of these proteins in SOKCs and NSOKCs may reflect differences in their pathogenesis and biological behavior. To evaluate the immunoexpression of chemokine CXCL12 and C-X-C chemokine receptor type 4 (CXCR4) in odontogenic keratocysts associated with Gorlin-Goltz syndrome (SOKCs) and nonsyndromic odontogenic keratocysts (NSOKCs). The percentages of cytoplasmic (CXCL12 and CXCR4) and nuclear (CXCR4) staining in epithelial and fibrous capsule cells of 22 SOKCs and 22 NSOKCs were determined. The results were analyzed statistically using the nonparametric Mann-Whitney test and Spearman correlation test. Higher cytoplasmic expression of CXCL12 was observed in the epithelial lining and fibrous capsule of NSOKCs compared to SOKCs (P < .001). No statistically significant differences in the cytoplasmic expression of CXCR4 were observed between SOKCs and NSOKCs (P > .05). Compared to SOKCs, NSOKCs exhibited higher nuclear expression of CXCR4 in the epithelial lining and lower immunoexpression in the fibrous capsule (P < .05). In the epithelial lining of SOKCs, a positive correlation was observed between cytoplasmic and nuclear expression of CXCR4 (P = .003). In the fibrous capsule of SOKCs and NSOKCs, cytoplasmic and nuclear expressions of CXCR4 were positively correlated (P < .001). The results suggest a potential involvement of CXCL12 and CXCR4 in the development of OKCs. The heterogenous expression of these proteins in SOKCs and NSOKCs may reflect differences in their pathogenesis and biological behavior.

Histopathological Comparative Analysis between Syndromic and Non-Syndromic Odontogenic Keratocysts: A Retrospective Study

(1) Background: The aim of this study was to compare the histopathological features of syndromic and non-syndromic odontogenic keratocysts (OKs) using conventional and Confocal Laser Scanning Microscopy (CLSM) with their biological behaviour. (2) Methods: Data from the medical records of 113 patients with histological diagnosis of (single and/or multiple) OKs were collected. Globally, 213 OKs (120 syndromic and 93 sporadic) were retrieved, and their histological slides were re-evaluated with conventional H&amp;E staining and with autofluorescence on the same slides using CLSM (Nikon Eclipse E600 microscope). (3) Results: Syndromic OKs showed more satellite cysts than sporadic cases, as well as a basophilic layer in the basement membrane, which was absent in sporadic OKs; both were highlighted with CLSM. The basement membrane in syndromic OKs appeared amorphous and fragile, thus possibly being responsible for the epithelial detachment and collapse, with scalloped features. Furthermore, the basal epithelial layers in such cases also showed increased cellularity and proliferative activity. All these histological features may possibly justify their higher tendency to recur. (4) Conclusions: CLSM is useful advanced technology that could help to quickly and easily discriminate between syndromic and non-syndromic OKs and to more accurately predict their biological behaviour in order to set fitter clinico-radiological follow-ups for individual patients.

Expression of Matrix Metalloproteinases 7 and 9, Desmin, Alpha-Smooth Muscle Actin and Caldesmon, in Odontogenic Keratocyst Associated with NBCCS, Recurrent and Sporadic Keratocysts.

Nevoid basal cell carcinoma syndrome (NBCCS) associated odontogenic keratocysts (OKCs) show more aggressive behavior and it has a higher frequency of relapse than non-syndromic OKCs. Stromal myofibroblasts (MFs), characterized by α-smooth muscle actin (αSMA), desmin and caldesmon expression, and metalloproteinases (MMPs) have an essential role in the remodeling of the extracellular matrix (ECM). The aim of the study is to analyze the immunohistochemical expression of MMP-7, MMP-9, αSMA and other new markers in the study of OKCs MFs such as desmin and caldesmon in NBCCS-associated OKCs compared to recurrent and sporadic keratocysts. Fourty 40 patients (23 M and 17 F) underwent surgery to remove the OKCs. The histological sections in paraffin were incubated with markers antibodies and a semi-quantitative score was used to evaluate the immunoreactivity. Densitometric analysis showed a very significantly increased expression of αSMA, caldesmon, MMP-7 and MMP-9 in NBCCS-OKCs compared to non-syndromic OKCs (p < 0.001). However, desmin showed a not significant increased expression in non-syndromic OKC compared to NBCCS-OKCs specimens in which desmin was slightly or not at all expressed. NBCSS-OKCs showed a greater distribution of MFs compared to the other OKCs subtypes. Further studies will be needed to evaluate whether the different expressions of these markers can be correlated to a different clinical behavior.

Presentation and management of syndromic and non-syndromic patients with multiple odontogenic keratocysts

ObjectiveThis case series reports the clinical presentation and management of multiple odontogenic keratocysts (OKCs) in patients with Gorlin Goltz syndrome and in non-syndromic patients. Material and methodsEight patients presented with features of multiple odontogenic keratocyst at department of Oral and Maxillofacial Surgery. The diagnosis was confirmed following clinical, radiographic and histopathological examination. Initially the diagnosis of OKC was confirmed on an incisional biopsy. Major and minor criteria were followed for the diagnosis of Gorlin Goltz syndrome. Smaller cysts in all patients were enucleated and for larger cysts marsupialization was planned either alone or followed by enucleation. Patients were followed at 1 month, 3 months, 6 months and yearly interval to check for bone healing and recurrences. ResultsAssociation of Gorlin Goltz syndrome was identified in four patients all of whom were males with age range of 12–37 years. Among four patients that had non-syndromic OKCs, three were female and one was male with age range of 09–50 years; two patients had familial non-syndromic multiple OKCs. In syndromic patients, multiple OKCs, recognized manifestations of Gorlin Goltz syndrome were identified with variable frequency: calcification of falx cerebri and chest deformity (100%); macrocephaly with frontal bossing (100%); hypertelorism (75%); basal cell carcinoma (25%); pectus deformity with flame shaped hands and feet (25%) and syndactyly (50%). Palmar or plantar pits, cleft lip or palate, ovarian fibroma and medulloblastoma was not identified in any patient. In patients with non-syndromic multiple OKCs mandible was more commonly involved than maxilla. ConclusionPatients with multiple OKCs, should be evaluated thoroughly and basal cell carcinomatous lesions should be ruled out. Meticulous follow up is vital as Gorlin Goltz syndrome is associated with malignancies and OKCs may be the first manifestation of this syndrome. Given the fact that OKCs associated with this syndrome have higher rate of recurrence than the isolated OKCs, long term follow up is warranted.

A case of non-syndromic multiple odontogenic keratocyst with isolated dentigerous cyst: A diagnostic conundrum

Odontogenic keratocyst (OKC) is a common developmental odontogenic cyst affecting the maxillofacial region. They can occur in two different forms, either as solitary (non-syndromic OKCs) or as multiple OKCs (syndromic OKCs). Multiple OKCs are usually seen in association with Gorlin–Goltz syndrome or otherwise called nevoid basal cell carcinoma syndrome. However, approximately 5% of patients with OKC have multiple cysts without concomitant syndromic presentation. This article reports a case of multiple OKCs of orthokeratinised variant in a non-syndromic patient along with an isolated dentigerous cyst associated with impacted teeth in one of the quadrants. Simultaneous occurrence of dentigerous cyst along with multiple OKCs may be coincidental. Impacted teeth must be radiographed and evaluated histopathologically to rule out varied lesions and associated complications with them.

Odontogenic Keratocyst: A Clinical Entity With Favorable Response to Active Decompression and Distraction Sugosteogenesis.

The purpose of this study was to determine the usefulness of active decompression and distraction sugosteogenesis (ADDS) for the management of non-syndromic odontogenic keratocysts (OKC). A retrospective case series study was designed and implemented. The study observed the Declaration of Helsinki on medical protocol and ethics and it was approved by the university's Institutional Review Board (IRB). The medical files of all patients who underwent ADDS for OKCs of the jaws at the Department of Oral and Maxillofacial Surgery of a tertiary university-affiliated medical center were reviewed. Data were collected on patient's age, gender, presenting signs and symptoms, lesion location, locularity, pre-ADDS, size of the lesion, post-ADDS, pain, days with bloody discharge and/or proteinaceous fluid inside the system's external unit, days to achieve hermetic seal, size of the lesion 2 weeks after ADDS, percentage of reduction, patient's complaints/complications, and follow-up period. Pre- and post-ADDS panoramic radiographs were reviewed for reduction parameters. Six patients, 5 males and 1 female, with an average age of 45.16 years (range 16-74 years) were studied. ADDS was performed during 4 weeks in all patients. During the therapy, the extraoral unit collected blood during 2.83 days in average. In average, after the third day, the cystic cavity started to drain a proteinaceous fluid for about 9.33 days (range 6-15 days). The average pre-ADDS Standard Lesional Area Index (SLAI) was 18.17 cm2 (range 4.40 cm2-34.58 m2) and, after 2 weeks of ADDS, the average SLAI was 5.47 cm2 (range 0.49 cm2-15.39 cm2). The average percentage of reduction, after 2 weeks, was 73.93% (range 55.49%-97.51%), which yielded an overall good reaction of OKCs to ADDS. No significant reduction of the lesions was observed from week 2 to week 4, when ADDS ceased. All lesions were enucleated after 3 months. After an average of 14 months of follow-up (12 to 17 months), no signs of recurrence have been observed.

Decompression: a first-intention treatment for “large” non-syndromic odontogenic keratocysts

Though odontogenic keratocysts (OKCs) are benign lesions, they have a high recurrence rate. Because of their aggressive behavior, they have been classified as tumors by the WHO until 2017. Main differential diagnoses are amelobastoma and dentigerous cyst. Anatomopathological examination can reach a final diagnosis. Several treatments have been proposed: curettage, resection, enucleation (alone or together with peripheral ostectomy) and decompression. Decompression aims to decrease the volume of the lesion of “large” OKCs, in order to prevent surgery-related fractures and to preserve the surrounding important anatomical structures such as the inferior alveolar nerve. It could lead to a complete regression. If not, secondary enucleation can be performed in better conditions: a reduced volume to remove, a thicker epithelium to detach, a lower risk to damage neighboring anatomic structures and a lower recurrence rate. Long-term follow-up however remains necessary. Nowadays, minimally invasive surgery prevails. And since OKC was returned into the odontogenic cysts group in the WHO classification, decompression should be considered as the first intention treatment. The purpose of this paper is to provide an update about OKC features and biological mechanisms, to review the different treatment options and to provide a step-by-step protocol for decompression.

IMMUNOHISTOCHEMICAL ANALYSIS OF AUTOPHAGY-RELATED PROTEINS IN ODONTOGENIC KERATOCYSTS

Objective: To evaluate the immunoexpression of autophagy-related proteins (p62 and Atg7) in odontogenic keratocysts associated with Gorlin syndrome (SOKCs) and nonsyndromic odontogenic keratocysts (NSOKCs).Study design: The percentages of immunopositive cells (nuclear and cytoplasmic/membrane) for p62 and Atg7 in the epithelial component of 25 SOKCs and 25 NSOKCs were assessed. The results were analyzed statistically using the nonparametric Mann-Whitney U test and Spearman correlation test. Results: Expression of p62 and Atg7 was observed in all cases studied. Compared with NSOKCs, SOKCs exhibited higher nuclear expression of p62 (P< .001) and Atg7 (P< .001). No significant difference in the cytoplasmic/membrane expression of p62 was observed between groups (P = .594). On the other hand, SOKCs exhibited higher cytoplasmic/membrane expression of Atg7 compared with NSOKCs (P< .001). In NSOKCs, positive correlations were observed between cytoplasmic/membrane expressions of Atg7 and p62 (P = .004). In SOKCs, positive correlations were found between nuclear and cytoplasmic/membrane expressions of Atg7 and p62 (P< .05).Conclusion: The results suggest a potential role of autophagy in the pathogenesis of odontogenic keratocysts. The more aggressive biological behavior of SOKCs compared with NSOKCs may be related to increased nuclear translocation of p62 and higher cytoplasmic/membrane expression of Atg7. Financial support: CNPq (427931/2016-9). Objective: To evaluate the immunoexpression of autophagy-related proteins (p62 and Atg7) in odontogenic keratocysts associated with Gorlin syndrome (SOKCs) and nonsyndromic odontogenic keratocysts (NSOKCs).Study design: The percentages of immunopositive cells (nuclear and cytoplasmic/membrane) for p62 and Atg7 in the epithelial component of 25 SOKCs and 25 NSOKCs were assessed. The results were analyzed statistically using the nonparametric Mann-Whitney U test and Spearman correlation test. Results: Expression of p62 and Atg7 was observed in all cases studied. Compared with NSOKCs, SOKCs exhibited higher nuclear expression of p62 (P< .001) and Atg7 (P< .001). No significant difference in the cytoplasmic/membrane expression of p62 was observed between groups (P = .594). On the other hand, SOKCs exhibited higher cytoplasmic/membrane expression of Atg7 compared with NSOKCs (P< .001). In NSOKCs, positive correlations were observed between cytoplasmic/membrane expressions of Atg7 and p62 (P = .004). In SOKCs, positive correlations were found between nuclear and cytoplasmic/membrane expressions of Atg7 and p62 (P< .05).Conclusion: The results suggest a potential role of autophagy in the pathogenesis of odontogenic keratocysts. The more aggressive biological behavior of SOKCs compared with NSOKCs may be related to increased nuclear translocation of p62 and higher cytoplasmic/membrane expression of Atg7. Financial support: CNPq (427931/2016-9).

A systematic review on the expression of bcl-2 in the nonsyndromic odontogenic keratocyst: should it be considered a cyst or a tumor?

The purpose of this study is to find out if the biological behavior and the capacity of the odontogenic keratocyst (OKC) in maintaining pathologic cells alive are more similar to the dentigerous cyst or to the ameloblastoma by assessing bcl-2. We searched MEDLINE, Web of Science, and Scopus for immunohistochemical studies reporting OKCs, dentigerous cysts, and ameloblastomas. Risk difference between the lesions expressing bcl-2 was the effect measure and a P value < 0.05 was considered to provide evidence to the effect estimates. OKCs have an estimated difference of 91% in the probability to express the bcl-2 over dentigerous cysts, but there is no difference in the expression of bcl-2 between OKCs and ameloblastomas. The present study demonstrated a great risk difference in the expression of bcl-2 between OKCs and dentigerous cysts and no risk difference between OKCs and ameloblastomas. OKC's growth may indirectly be attributed to the anti-apoptotic effect of bcl-2 in the cystic epithelium and not only to the increase of its intraluminal pressure. Therefore, the classification of this lesion into keratocystic odontogenic tumor should be carefully reconsidered.

COMPARISON OF CELLULAR PROLIFERATION AND MELANOCYTES IN NON-SYNDROMIC AND SYNDROMIC ODONTOGENIC KERATOCYSTS

Objective To compare cell proliferation and the presence of melanocytes in non-syndromic and syndromic odontogenic keratocyst (OK). Study Design We studied 20 cases of sporadic OK and 10 cases of OK associated with NBCC. To evaluate cell proliferation and the presence of melanocytes, immunohistochemistry was performed with antibodies against Ki-67 and Melan-A, respectively. Results The OK associated with NBCC presented 23.4% (range 8.6%-36.8%) and the sporadic OK 21.8% (range 7.6%-40.4%) of proliferating cells. This difference was not significant. Cells positive for Melan-A occurred exclusively in 4 of 9 cases of syndromic keratocyst. Conclusions No differences were found in the proliferation between OK associated with NBCC and sporadic. Only in the OK associated with the NBCC, the presence of melanocytes was detected.

Immunoexpression of DNA base excision repair and nucleotide excision repair proteins in ameloblastomas, syndromic and non-syndromic odontogenic keratocysts and dentigerous cysts

ObjectiveTo evaluate the immunoexpression of DNA base excision repair (BER) [apurinic/apyrimidinic endonuclease 1 (APE-1), X-ray repair cross complementing 1 (XRCC-1)] and nucleotide excision repair (NER) [xeroderma pigmentosum complementation group (XPF)] proteins in benign epithelial odontogenic lesions with different biological behaviors. DesignThirty solid ameloblastomas, 30 non-syndromic odontogenic keratocysts (NSOKCs), 29 syndromic odontogenic keratocysts (SKOCs), 30 dentigerous cysts (DCs) and 20 dental follicles (DFs) were evaluated quantitatively for APE-1, XRCC-1 and XPF through immunohistochemistry. ResultsNuclear expression of APE-1 was significantly higher in NSOKCs, SOKCs, and ameloblastomas in comparison to DCs (p < 0.001). Nuclear expression of XRCC-1 was higher in NSOKCs and SOKCs than in DCs (p < 0.05). At the nuclear level, XPF expression was higher in NSOKCs and SOKCs than in DCs and ameloblastomas (p < 0.05). A statistically significant higher expression of APE-1 (nuclear), XRCC-1 (nuclear), and XPF (nuclear and cytoplasmic) was found in all odontogenic lesion samples as compared to DFs (p < 0.05). For all lesions, there was a positive correlation between nuclear expression of APE-1 and XRCC-1 or XPF (p < 0.05). ConclusionsOur results suggest a potential involvement of APE-1, XRCC-1 and XPF proteins in the pathogenesis of benign epithelial odontogenic lesions, especially in those with more aggressive biological behavior, such as ameloblastomas, NSOKCs, and SOKCs. We also showed that the expression of APE-1 was positively correlated with the nuclear expression of XRCC-1 and XPF, which may suggest an interaction between the BER and NER pathways in all odontogenic lesions studied herein.

In vitro assessment of PD-L1+ microvesicles in the cyst fluid of non-syndromic odontogenic keratocysts

Odontogenic keratocysts (OKCs) are jaw cystic lesions which are characterized by local invasion and high recurrence rate. The majority of OKCs are exposed to microorganisms and occur along with focal inflammatory infiltrates. Cyst fluids are biological fluids that contain a large content of cytokines and immune globulins. Inhibitory receptor such as programmed death receptor 1 (PD-1) and its ligand programmed death-ligand 1 (PD-L1), which can induce a coinhibitory signal in activated T cells, plays a vital role in the differentiation, exhaustion and apoptosis of T cells. Cell derived microvesicles, carrying a cargo of functional proteins, nucleic acids and lipids, are important communication tools in the development of diseases. However, the expression of PD-L1 in OKCs tissues and whether PD-L1 could be carried by microvesicles are unexplored. Presently, we have isolated cyst fluid microvesicles and identified cell derived PD-L1+ cyst fluid microvesicles. PD-L1 was located in the membrane of the cyst fluid microvesicles. The main cellular origins of PD-L1+ cyst fluid microvesicles were dendritic cells followed by lymphocytes. Elevated PD-L1+ cyst fluid microvesicles were detected in the OKCs compared with dentigerous cysts. Isolated cyst fluid microvesicles could bind to the membrane of activated CD8 T cells and inhibit proliferation of stimulated peripheral blood CD8 T cells. In conclusion, the present study suggests that elevated PD-L1+ cyst fluid microvesicles might be related with the cyst development of OKCs.

What is the Prevalence of Undiagnosed Nevoid Basal Cell Carcinoma Syndrome in Children With an Odontogenic Keratocyst?

Odontogenic keratocysts (OKCs) can occur in isolation or as part of nevoid basal cell carcinoma syndrome (NBCCS). Patients with NBCCS are younger at OKC diagnosis than those with nonsyndromic OKC (NS-OKC). The purpose of this study was to determine the prevalence of undiagnosed NBCCS in children who present with an OKC and to assess differences in demographic and presenting features between children with NBCCS and those with NS-OKC. This study is a retrospective case series of children with an OKC presenting to Boston Children's Hospital (Boston, MA) from 2007 through 2018. To be included, patients had to be no older than 18years and have a newly diagnosed OKC. Patients were excluded if they had previous or recurrent OKC or NBCCS diagnosis. Records were reviewed for age at presentation, gender, number and location of OKCs, treatment, recurrence, family history, and clinical features consistent with NBCCS. Descriptive data were summarized. The sample included 50 patients (27 boys) diagnosed with an OKC at a mean age of 11.7years (range, 2 to 18yr); 36% (n=18) with NBCCS and 64% (n=32) with NS-OKC. NBCCS diagnosis was made in 8 of 18 patients (44%) because of a family history at presentation, and in 10 patients (56%) the diagnosis was made by genetic testing or documentation of diagnostic criteria. Eight of 18 patients (44%) with undiagnosed NBCCS presented with a single OKC. Patients with NBCCS were significantly younger at presentation (NBCCS, 9.3yr; NS-OKC, 13.0yr), had more cysts at time of diagnosis (NBCCS, 1.7; NS-OKC, 1.0; P<.05), had more maxillary cysts (NBCCS, 13; NS-OKC, 11), and had a higher recurrence rate (P<.05). Given the prevalence of undiagnosed NBCCS in children with OKC, clinicians should have a low threshold for referral for complete examination or genetic testing in children with even a single OKC.

Expression of p53-family members in odontogenic cysts

Expression of p53-family members in odontogenic cysts

Conservative Treatment of Primary and Nonsyndromic Odontogenic Keratocyst: An Overview of the Practice

Odontogenic Keratocyst (OKC) is a locally aggressive odontogenic cyst with a high recurrence rate after treatment. Several conservative surgeries have been suggested in the treatment of Keratocysts but working with an overview of the practice may render better outcomes.

Surgical treatments for nonsyndromic odontogenic keratocysts.

Data sourcesPubMed/MEDLINE, Science Direct, Web of Science, Scopus and the Cochrane Library were searched until October 2017.Study selectionClinical trials, controlled trials, retrospective studies and case series containing at least ten cases with a minimum follow-up of ten years of patients treated with marsupialisation or decompression with or without enucleation, and enucleation alone evaluating the recurrence rates.Data extraction and synthesisTwo reviewers independently assessed for eligibility and extracted data. The level of agreement between reviewers was calculated. The quality of the included studies was assessed using the Methodological Index of Nonrandomised Studies (MINORS) scoring system and the Newcastle Ottawa Quality Assessment Scale (NOS).Data were combined in meta-analysis using a fixed effect model of the calculated odds ratio with 95% CI for each comparison.ResultsTwenty-nine studies were included in the review. Of those 25 were characterised as retrospective studies, one cohort and three case series. Of the 1321 cases 99 were marsupialisation; 45 received marsupialisation followed by enucleation; 27 were treated by decompression; 101 received decompression followed by enucleation and 1049 cases were treated by enucleation alone.The recurrence rate for decompression followed by enucleation was 11.9% (95% CI 5.6 to 18.2%), marsupialisation followed by enucleation 17% (95% CI 6.6 to 28.9%), enucleation 20.8% (95% CI 18.3 to 23.2% ), decompression alone 18.5% (95% CI 8.2 to 28.9%) and marsupialisation 18.2% (95% CI 10.6 to 25.8%).ConclusionsThe authors concluded that there is a significant superiority for OKC treatment when decompression is used before enucleation OR 0.48 (95% CI 0.22 to 1.08).

Conservative surgical treatments for nonsyndromic odontogenic keratocysts: a systematic review and meta-analysis.

This manuscript presents a systematic review of the clinicopathologic features and outcomes of conservative surgical treatments for nonsyndromic odontogenic keratocysts (OKCs) and assesses the recurrence rates through a meta-analysis, in order to indicate the best conservative approach. PRISMA guidelines for systematic reviews were followed, and the protocol was registered (PROSPERO/Nr.: CRD42017060964). An electronic search was conducted using the PubMed/MEDLINE, Science Direct, Web of Science, Scopus, and The Cochrane Library databases, and relevant articles were selected based on specific inclusion criteria. The PICOS criteria (Population: nonsyndromic patients of any age with OKC, with histopathological diagnosis and minimum follow-up of 12months; Intervention and Comparison: marsupialization or decompression with or without enucleation, and enucleation alone; Outcome: recurrence rates; Study design: clinical trials, controlled trials, retrospective studies, and case series containing at least 10 cases of OKC) were employed. A pooled odds ratio (OR) was computed through the Mantel-Haenszel test (M-H) with 95% confidence intervals (CI). One thousand nine hundred OKCs were analyzed; the age of the patients varied from 6 to 90years (mean of 38.6years); a male to female ratio of 1.57:1 was observed; 74.5% of the lesions occurred in the mandible; 75.7% of OKCs were unilocular; the association with impacted tooth was reported for 344 OKCs; and the mean follow-up was 60.1months. One thousand three hundred thirty-one OKCs were treated by conservative surgical treatments, and 261 cases (19.8%) presented recurrence. Nonetheless, minor total recurrence rates were observed after decompression followed by enucleation (11.9%) and marsupialization followed by enucleation (17.8%). In contrast, enucleation alone showed a total recurrence rate of 20.8%. The results suggest a significant superiority of success for OKC treatments that use decompression followed by enucleation, instead of an initial enucleation (M-H, OR = 0.48; 95% CI = 0.22 to 1.08; P = 0.0163). No consensus exists concerning the best management for OKCs. More aggressive treatments (ostectomy, resection, or use of adjunctive therapies like Carnoy's solution and liquid nitrogen) can have many disadvantages and risks. Therefore, it is necessary to identify the conservative approach for OKCs that results in a lower recurrence rate.

The anecdote of viral etiopathogenia in ameloblastoma and odontogenic keratocyst: Why don’t we let it go?

BackgroundAmeloblastoma (AM) and odontogenic Keratocyst (OKC) are destructive odontogenic lesions of the gnathion. Although their exact pathogeneses are not yet totally understood, the viral etiopathogenesis in AM and KCOT has been proposed. True to syndromic keratocystic odontogenic tumor (sKCOT) and non-syndromic OKC is the high recurrence rate. ObjectivesGiven that shared pathways trailed by AM and by sKCOT/OKC have been suggested, this study, however, contrasts the expression of AM and OKC for viral antibodies. MethodA total of archival 80 paraffin blocks of cases of parakeratinized odontogenic keratocyst (non-syndromic KCOTs) and of ameloblastomas (n=40 for each) were included in this study to be sectioned and stained for two immunohistochemical markers: anti-human papillomavirus and Epstein-Barr virus-encoded latent membrane protein. ResultsAll the submitted cases of AM and parakeratinized OKC were negative for both markers: anti-HPV and anti-LMP-1. ConclusionsAlthough results could have been biased, given the same ethnic group and territory examined in this study, all cases were negative for both markers. Therefore, the viral contribution to the etiopathogenesis in AM and OKC could not be established in this study.