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Journal of Oral Pathology & Medicine :... Jan 2022Hybrid odontogenic lesions combine histopathological characteristics of two or more odontogenic cysts and/or tumours. The aim of this study was to evaluate the available... (Review)
Review
BACKGROUND
Hybrid odontogenic lesions combine histopathological characteristics of two or more odontogenic cysts and/or tumours. The aim of this study was to evaluate the available data on hybrid odontogenic lesions (HOL) and to analyse their epidemiological/clinical features and biological behaviour.
METHODS
An electronic search was done in January 2021 using multiple databases. Eligibility criteria encompassed publications with sufficient clinical and histological information to confirm the tumours' diagnoses.
RESULTS
A total of 147 articles were included in this study, comprising 203 cases. Calcifying odontogenic cyst associated with odontoma (COC/OD) (37/18.2%) was the most common HOL. Females were more affected with a mean age of 24.9 years. Lesions presented as asymptomatic swellings, with a mean evolution time of 8.2 months (0.3-96), and mean tumour size of 4.8 cm (0.3-7). Radiographic aspects frequently showed radiolucent (139/68.4%) and unilocular (52/25.6%) images with well-defined limits (48/23.6%). The lesions mostly affected mandibular pre-molars (69/34%) and mandibular molars (69/34%) regions. Enucleation (89/43.8%) and surgical excision (59/29%) were the most common treatment modalities. The mean follow-up time was 33.8 months (0.5-216 months) and recurrences were observed in four cases (1.9%), all of which were central odontogenic fibroma associated with central giant cell granuloma (COF/CGCG).
CONCLUSION
COC/OD is the most common HOL and recurrence is a rare event, being usually associated with the diagnosis of COF/CGCG.
Topics: Adult; Female; Granuloma, Giant Cell; Humans; Odontogenic Cyst, Calcifying; Odontogenic Cysts; Odontogenic Tumors; Odontoma; Young Adult
PubMed: 34469012
DOI: 10.1111/jop.13238 -
Cureus Dec 2021The glandular odontogenic cyst (GOC) is a rare odontogenic cyst that can develop in the maxillofacial region with aggressive behavior. It tends to develop into enormous...
The glandular odontogenic cyst (GOC) is a rare odontogenic cyst that can develop in the maxillofacial region with aggressive behavior. It tends to develop into enormous proportions with high recurrence rates. The mandibular anterior area is the common site of occurrence of GOC, and it appears as an asymptomatic slow-growing swelling. GOC mimics other odontogenic cysts and tumors both clinically and radiographically, thus posing difficulty in diagnosis. Due to the aggressive potential of GOC, precise diagnosis and prompt treatment are crucial. Both conservative and aggressive surgical therapies have been advocated for GOC with a preference for aggressive therapy due to its high potential for recurrence. In this report, we present a case of GOC of the mandible in an adult female patient, which was successfully treated by segmental resection and primary reconstruction with stainless steel recon plate with uneventful healing during the one-year postoperative follow-up period.
PubMed: 35106238
DOI: 10.7759/cureus.20701 -
Journal of Oral & Maxillofacial Research 2023The glandular odontogenic cyst is now a well-known entity comprising < 0.5% of all odontogenic cysts with a recent review tabulating about 200 cases in the English...
BACKGROUND
The glandular odontogenic cyst is now a well-known entity comprising < 0.5% of all odontogenic cysts with a recent review tabulating about 200 cases in the English literature. Glandular odontogenic cyst shows epithelial features that simulate salivary gland or glandular differentiation. The importance of glandular odontogenic cyst relates to the fact that it has a high recurrence rate and shares overlapping histologic features with central mucoepidermoid carcinoma. The purpose of this paper is to describe the clinical, radiological, and histopathological features of a case of glandular odontogenic cyst with the course of treatment and 9-years follow-up, followed by a review of the literature.
METHODS
A 63-year-old male was referred for further investigation of a mandibular radiolucency observed by his general dental practitioner. The main complaint was a murmuring sensation in the lower jaw right side. Radiological examination revealed a well-defined, unilocular, radiolucent lesion, involving the right mandible with 17 and 68 mm in mediolaterally and anteroposterior dimension, respectively.
RESULTS
A total enucleation of the cystic lesion and surgical extraction of tooth #46, #47 and #48, was performed under local anaesthesia. Histopathologic examination revealed a glandular odontogenic cyst.
CONCLUSIONS
Glandular odontogenic cyst shows no pathognomonic clinico-radiographic characteristics, and therefore in many cases it resembles a wide spectrum of lesions. Diagnosis can be extremely difficult due to histopathological similarities with dentigerous cyst, lateral periodontal cyst and central mucoepidermoid carcinoma. Therefore a careful histopathological examination and a long-term follow-up (preferably seven years) are required to rule out recurrences.
PubMed: 37521326
DOI: 10.5037/jomr.2023.14204 -
Orvosi Hetilap Dec 2020Összefoglaló. A Gorlin-Goltz-szindróma - más néven naevoid basalsejtes carcinoma szindróma - egy ritka, viszont számos orvosi társszakmát érintő, rendkívül... (Review)
Review
Összefoglaló. A Gorlin-Goltz-szindróma - más néven naevoid basalsejtes carcinoma szindróma - egy ritka, viszont számos orvosi társszakmát érintő, rendkívül változatos megjelenésű és genetikailag is heterogén betegség. Bár a tudományos kutatások egyik kedvenc területe, az aránylag alacsony betegszám, valamint a genotípus és a fenotípus közötti, igen komplex összefüggések miatt a kórképről meglévő ismereteink még nem teljesek. A témában megjelent nemzetközi és magyar nyelvű publikációk jelentős része esetközlésekre és a szindróma általános ismertetésére szorítkozik. A közlemény célja, hogy áttekintést adjon a szindróma genetikai vonatkozásairól. A nemzetközi és a magyar nyelvű szakirodalom áttanulmányozását végeztük. A naevoid basalsejtes carcinoma szindróma genetikai hátterének, az egyelőre azonosítatlan örökletes tényezőknek pontos megismerése még várat magára. A genetikai vizsgálatok a szindróma pontosabb megértéséhez, könnyebb diagnosztizálásához, a pozitív családtervezéshez és a személyre szabott terápiákhoz is hozzájárulhatnak. Orv Hetil. 2020; 161(49): 2072-2077. Summary. Gorlin-Goltz syndrome, or nevoid basal cell carcinoma syndrome, is a rare disease that requires multidisciplinary approach in patient management. The disease is genetically heterogenous and has an extremely variable expressivity. Although the syndrome is in the focus of scientific research, our knowledge of it is still limited due to the relatively low number of recognised patients and the complexity of genotype-phenotype correlation. Several papers in this field have been published in the international and also in the Hungarian literature but most of these reports are single cases or small case series of families and outline general information about the disease. Authors aimed to review the literature of the syndrome and to report the genetic background and its role in the diagnosis and treatment. A review of the English and Hungarian literature was performed. The full genetic background of the syndrome is not yet discovered. Increasing the awareness of the syndrome, collecting and thoroughly analysing the medical records and performing genetic tests on the patients may lead to the better understanding of the disease; they may also help early diagnosis and treatment, positive family planning and may establish personalized medicine. Orv Hetil. 2020; 161(49): 2072-2077.
Topics: Basal Cell Nevus Syndrome; Carcinoma, Basal Cell; Humans; Hungary; Interdisciplinary Research; Odontogenic Cysts; Rare Diseases
PubMed: 33279882
DOI: 10.1556/650.2020.31933 -
International Dental Journal Feb 2023Odontogenic lesions evolve as a result of altered dental development. This study aimed to evaluate the prevalence and the coinfection of Epstein-Barr virus (EBV) and...
OBJECTIVES
Odontogenic lesions evolve as a result of altered dental development. This study aimed to evaluate the prevalence and the coinfection of Epstein-Barr virus (EBV) and Kaposi sarcoma-associated herpesvirus (KSHV) in radicular cysts, dentigerous cysts, odontogenic keratocysts, and ameloblastomas.
METHODS
Polymerase chain reaction (PCR) was used to analyse 66 cases of odontogenic lesions for the presence of EBV-DNA and KSHV-DNA. These lesions were 15 radicular cysts, 16 dentigerous cysts, 18 odontogenic keratocysts, and 17 ameloblastomas.
RESULTS
EBV-DNA was detected in 24 (36.4%) of the studied samples as follows: 6 samples (40.0%) of radicular cysts, 4 (25.0%) of dentigerous cysts, 10 (55.6 %) of odontogenic keratocysts, and 4 (23.5%) of ameloblastomas (P = .168). KSHV-DNA was found in 16 (24.2%) of the studied samples as follows: 1 sample (6.7%) of radicular cysts, 6 (37.5%) of dentigerous cysts, 8 (44.4 %) of odontogenic keratocysts, and 1 (5.9%) of ameloblastomas (P = .001). Additionally, EBV and KSHV were positively correlated in all studied samples (P = .002).
CONCLUSIONS
Both EBV and KSHV are found in odontogenic cysts and ameloblastomas. KSHV and EBV are more prevalent in odontogenic keratocysts than in other studied odontogenic lesions. Further, there is a high prevalence of EBV and KSHV coinfection in odontogenic cysts and ameloblastomas.
Topics: Humans; Ameloblastoma; Coinfection; Dentigerous Cyst; DNA; Epstein-Barr Virus Infections; Herpesvirus 4, Human; Herpesvirus 8, Human; Odontogenic Cysts; Prevalence; Radicular Cyst; Sarcoma, Kaposi
PubMed: 35907672
DOI: 10.1016/j.identj.2022.06.028 -
F1000Research 2022Various stemness markers (SOX2, OCT4, and NANOG) have been studied in odontogenic cysts and tumors. However, studies on SALL4 having similar properties of stemness has...
BACKGROUND
Various stemness markers (SOX2, OCT4, and NANOG) have been studied in odontogenic cysts and tumors. However, studies on SALL4 having similar properties of stemness has not been documented. Additionally, insight into fascin as a migratory molecule is less explored. In this study, the expression of SALL4 and fascin were evaluated in ameloblastoma, adenomatoid odontogenic tumor (AOT), odontogenic keratocyst (OKC), dentigerous cyst (DC), radicular cyst (RC), and calcifying odontogenic cyst (COC).
METHODS
Semi-quantitative analysis of fascin and SALL4 immuno-positive cells was done in a total of 40 cases of ameloblastoma (11 plexiform, 12 follicular, 12 unicystic, and 5 desmoplastic) variants, 6 cases of AOT, 15 each of OKC, DC, RC and 5 of COC. Chi-square test was applied to evaluate the association between SALL4 and fascin expression in odontogenic cysts and tumors.
RESULTS
Fascin immunopositivity was observed in peripheral ameloblast-like cells, and weak or absent in stellate reticulum-like cells. A moderate to weak immune-reactivity to SALL4 was observed in the cytoplasm of ameloblastoma, epithelial cells of dentigerous and radicular cysts, having a marked inflammatory infiltrate, which is an interesting observation. COC and AOT had negative to weak expressions. No recurrence has been reported.
CONCLUSIONS
Expression of fascin in ameloblastomas elucidate their role in motility and localized invasion. Its expression in less aggressive lesions like DC, COC, AOT will incite to explore the other functional properties of fascin. SALL4 expression in the cytoplasm of odontogenic cysts and tumors may represent inactive or mutant forms which requires further validation.
Topics: Humans; Transcription Factors; Microfilament Proteins; Odontogenic Cysts; Carrier Proteins; Immunohistochemistry; Ameloblastoma; Odontogenic Tumors; Biomarkers, Tumor
PubMed: 38895097
DOI: 10.12688/f1000research.126091.3 -
BMP-2 and Noggin Immunoexpression in Ameloblastomas, Odontogenic Keratocysts, and Dentigerous Cysts.Applied Immunohistochemistry &... Jan 2023BMP-2 and Noggin are expressed in several tissues and participate in cell differentiation and proliferation during odontogenesis and tumor development. We evaluated the...
BMP-2 and Noggin are expressed in several tissues and participate in cell differentiation and proliferation during odontogenesis and tumor development. We evaluated the immunohistochemical expression of these proteins in ameloblastomas (AMs), odontogenic keratocysts (OKCs), and dentigerous cysts (DCs). The expression in AM (n.20), OKC (n.20), and DC (n.20) was evaluated by the percentage of positive cells and expression intensity, resulting in a total immunostaining score. Analysis of BMP-2 and Noggin revealed positivity in all cases. The Mann-Whitney test showed a statistically significant difference for Noggin between AM and DC and between OKC/DC. The mean DC scores were always higher than those of the other groups, regardless of the assessment method. Individual analysis of each lesion showed a positive and significant correlation between the percentage of cells positive for BMP-2 and Noggin in DC. We demonstrated the presence of BMP-2 and Noggin in AMs/OKCs/DCs. Marked expression of BMP-2 was observed in OKCs and AMs. There was also a positive correlation between BMP-2 and Noggin in DCs, suggesting a greater role of these markers in the bone formation and remodeling process since DCs are characterized by phases of bone quiescence and healing.
Topics: Humans; Dentigerous Cyst
PubMed: 36315234
DOI: 10.1097/PAI.0000000000001084 -
Brazilian Dental Journal 2021The Inhibitor of Growth (ING) gene family is a group of tumor suppressor genes that play important roles in cell cycle control, senescence, DNA repair, cell...
UNLABELLED
The Inhibitor of Growth (ING) gene family is a group of tumor suppressor genes that play important roles in cell cycle control, senescence, DNA repair, cell proliferation, and apoptosis. However, inactivation and downregulation of these proteins have been related in some neoplasms. The present study aimed to evaluate the immunohistochemical profiles of ING3 and ING4 proteins in a series of benign epithelial odontogenic lesions.
METHODS
The sample comprised of 20 odontogenic keratocysts (OKC), 20 ameloblastomas (AM), and 15 adenomatoid odontogenic tumors (AOT) specimens. Nuclear and cytoplasmic immunolabeling of ING3 and ING4 were semi-quantitatively evaluated in epithelial cells of the odontogenic lesions, according to the percentage of immunolabelled cells in each case. Descriptive and statistics analysis were computed, and the p-value was set at 0.05.
RESULTS
No statistically significant differences were found in cytoplasmic and nuclear ING3 immunolabeling among the studied lesions. In contrast, AOTs presented higher cytoplasmic and nuclear ING4 labeling compared to AMs (cytoplasmic p-value = 0.01; nuclear p-value < 0.001) and OKCs (nuclear p-value = 0.007).
CONCLUSION
ING3 and ING4 protein downregulation may play an important role in the initiation and progression of more aggressive odontogenic lesions, such as AMs and OKCs.
Topics: Ameloblastoma; Cell Cycle Proteins; Cell Proliferation; Homeodomain Proteins; Humans; Odontogenic Cysts; Odontogenic Tumors; Tumor Suppressor Proteins
PubMed: 34787253
DOI: 10.1590/0103-6440202104279 -
Journal of Endodontics Mar 2022Radiolucent lesions with gingival swelling found in the premolar and intercanine region can elicit a different clinical diagnosis than one confirmed by histologic...
INTRODUCTION
Radiolucent lesions with gingival swelling found in the premolar and intercanine region can elicit a different clinical diagnosis than one confirmed by histologic findings. The purpose of the study is to identify and present the frequency of the unexpected microscopic diagnosis of odontogenic keratocyst (OKC) in a location preoperatively favoring a lateral periodontal cyst (LPC) with similar clinical and radiographic appearance.
METHODS
A retrospective analysis of biopsies received from 2011 and 2019 was performed, and the number of LPC and OKC cases was assessed. The alignment of clinical and radiographic diagnosis to histologic findings and anatomic location was analyzed, and the number of OKC cases preoperatively misdiagnosed as LPCs was identified.
RESULTS
A total of 79,257 biopsies were received. Of those, 184 were diagnosed as LPCs and 742 as OKCs. For all preoperatively diagnosed LPCs, the clinical and histologic diagnosis aligned; however, 182 of 742 OKCs were submitted with a clinical misdiagnosis of LPCs. The location of these lesions with the unanticipated diagnosis overlapped with those for LPCs, specifically the maxillary and mandibular anterior and premolar regions.
CONCLUSIONS
Radiolucent lesions with gingival swelling in the premolar and intercanine region are frequently clinically and radiographically misdiagnosed. A biopsy should be considered in all cases to establish the correct pathologic diagnosis and treatment course.
Topics: Bicuspid; Diagnostic Errors; Humans; Odontogenic Cysts; Periodontal Cyst; Retrospective Studies
PubMed: 34922990
DOI: 10.1016/j.joen.2021.11.010 -
Heliyon Jun 2023Developmental cysts are pathological epithelial-lined cavities arising in various organs as a result of systemic or hereditary diseases. Molecular mechanisms involved in... (Review)
Review
Developmental cysts are pathological epithelial-lined cavities arising in various organs as a result of systemic or hereditary diseases. Molecular mechanisms involved in the formation of developmental odontogenic cysts (OCs) are not fully understood yet; the cystogenesis of renal cysts originating from the autosomal dominant polycystic kidney disease (ADPKD) has been, however, explored in much greater detail. This narrative review aimed i) to summarize molecular and cellular processes involved in the formation and growth of developmental OCs, especially dentigerous cysts (DCs) and odontogenic keratocysts (OKCs), ii) to find if there are any similarities in their cystogenesis to ADPKD cysts, and, based on that, iii) to suggest potential factors, candidate molecules, and mechanisms that could be involved in the DC formation, thus proposing further research directions. Here we suggest a possible association of developmental OCs with primary cilia disruption and with hypoxia, which have been previously linked with cyst formation in ADPKD patients. This is illustrated on the imagery of tissues from an ADPKD patient (renal cyst) and from developmental OCs, supporting the similarities in cell proliferation, apoptosis, and primary cilia distribution in DC/OKC/ADPKD tissues. Based on all that, we propose a novel hypothesis of OCs formation suggesting a crucial role of mutations associated with the signaling pathways of primary cilia (in particular, Sonic Hedgehog). These can lead to excessive proliferation and formation of cell agglomerates, which is followed by hypoxia-driven apoptosis in the centers of such agglomerates (controlled by molecules such as Hypoxia-inducible factor-1 alpha), leading to cavity formation and, finally, the OCs development. Based on this, we propose future perspectives in the investigation of OC pathogenesis.
PubMed: 37389068
DOI: 10.1016/j.heliyon.2023.e17130