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.

New perspectives in the differential diagnosis of jaw lesions: Machine learning and inflammatory biomarkers.

This study aimed to assess the diagnostic performance of a machine learning approach that utilized radiomic features extracted from Cone Beam Computer Tomography (CBCT) images and inflammatory biomarkers for distinguishing between Dentigerous Cysts (DCs), Odontogenic Keratocysts (OKCs), and Unicystic Ameloblastomas (UAs). This retrospective study involves 103 patients who underwent jaw lesion surgery in the Maxillofacial Surgery Unit of Federico II University Of Naples between January 2018 and January 2023. Nonparametric Wilcoxon-Mann-Whitney and Kruskal Wallis tests were used for continuous variables. Linear and non-logistic regression models (LRM and NLRM) were employed, along with machine learning techniques such as decision tree (DT), k-nearest neighbor (KNN), and support vector machine (SVM), to predict the outcomes. When individual inflammatory biomarkers were considered alone, their ability to differentiate between OKCs, UAs, and DCs was below 50 % accuracy. However, a linear regression model combining four inflammatory biomarkers achieved an accuracy of 95 % and an AUC of 0.96. The accuracy of single radiomics predictors was lower than that of inflammatory biomarkers, with an AUC of 0.83. The Fine Tree model, utilizing NLR, SII, and one radiomic feature, achieved an accuracy of 94.3 % (AUC = 0.95) on the training and testing sets, and a validation set accuracy of 100 %. The Fine Tree model demonstrated the capability to discriminate between OKCs, UAs, and DCs. However, the LRM utilizing four inflammatory biomarkers proved to be the most effective algorithm for distinguishing between OKCs, UAs, and DCs.

Decompression-first or direct enucleation: The choice of treatment for medium-sized odontogenic jaw cysts.

BACKGROUND: The treatment of medium-sized odontogenic jaw cysts is inconsistent at present. Two main treatments, namely decompression and enucleation, are used overlappingly. This retrospective analysis aims to provide useful references for technique selection for future management of medium-sized odontogenic jaw cysts. METHODS: Odontogenic cysts with lesion sizes ranging 2-4 cm were included. The clinical and radiological data of the patients were reviewed. Decompression-first and direct enucleation treatments were the two main surgical techniques. The preoperative parameters and postoperative outcomes were compared between the two groups. RESULTS: Out of 69 patients included, 40 (58 %) were in the decompression group and 29 (42 %) in direct enucleation group. The logistic regression analysis of preoperative parameters demonstrated that the maximum lesion size and the chief surgeon's preference could affect the selection of surgical techniques for medium-sized cysts (P < 0.05). Receiver operating characteristic curve analyses demonstrated that a lesion size >2.5 cm was the best cutoff value for predicting a decompression selection. Most postoperative outcomes differed significantly between the two groups (P < 0.05). Lower-risk anesthesia, shorter hospitalization, tooth function protection, and fewer neurosensory impairments were decompression-favoring outcomes. However, more follow-up visits, more postoperative X-rays, and longer postoperative care were outcomes against decompression. The recurrence rate was low and did not differ significantly between the two groups (P > 0.05). CONCLUSIONS: There is no apparent preference for treating medium-sized jaw cysts. The maximum lesion size is a moderate-impact factor for treatment selection. A tendency to prefer decompression-first with larger lesion size was found in medium-sized jaw cysts. The advantages of teeth preservation and low neurosensory impairment of decompression were verified in the medium-size jaw cysts. The burden of postoperative care should be considered when selecting decompression.

Pediatric Odontogenic Cysts and Tumors.

Pediatric odontogenic cysts and tumors are rare and often associated with developing or impacted teeth. Odontogenic cysts are broadly categorized as inflammatory or developmental while odontogenic tumors are classified histologically as epithelial, mesenchymal, or mixed tumors. This article will discuss the presentation, diagnosis, and treatment of odontogenic cysts and tumors in the pediatric population.

Optimal time to definitive enucleation of large cysts following marsupialization: A single center, retrospective study.

OBJECTIVES: Optimal time to enucleation following marsupialization of large odontogenic mandibular cysts is undefined. We aim to assess volume reduction throughout follow-up, to indicate optimal time to enucleation. Secondary objectives include the identification of factors influencing cyst reduction. STUDY DESIGN: We retrospectively enrolled 15 patients with mandibular cysts of different histological types treated with marsupialization at our center between 2018 and 2022. Cyst volume was assessed with cone-beam computed tomography (CBCT) and a semi-automatic segmentation algorithm, at baseline and between 6 and 8 months post marsupialization. RESULTS: The overall mean cyst volume reduction percent (VR%) was 57.7 % or 0.2 % per day. VR% at 8 months was significantly higher than those assessed at 6 and 7 months (67.1% vs 47.1 %, p = 0.003). Time to CBCT was the only independent variable influencing cyst VR%. CONCLUSION: Our study proves that the optimal time to enucleation for mandibular cyst is 8 months, independent of histological cyst type, patient age, baseline cyst volume and the number of pre-operative residual bone walls.

Discovery of metabolite biomarkers for odontogenic keratocysts.

INTRODUCTION: Odontogenic keratocysts (OKCs) are locally aggressive and have a high rate of recurrence, but the pathogenesis of OKCs is not fully understood. We aimed to investigate the serum metabolomic profile of OKCs and discover potential biomarkers. METHODS: Metabolomic analysis was performed on 42 serum samples from 22 OKC patients and 20 healthy controls (HCs) using gas chromatography‒mass spectrometry to identify dysregulated metabolites in the OKC samples. LASSO regression and receiver operating characteristic (ROC) curve analyses were used to select and validate metabolic biomarkers and develop diagnostic models. RESULTS: A total of 73 metabolites were identified in the serum samples, and 24 metabolites were dysregulated in the OKC samples, of which 4 were upregulated. Finally, a diagnostic panel of 10 metabolites was constructed that accurately diagnosed OKCs (sensitivity of 100%, specificity of 100%, area under the curve of 1.00). CONCLUSION: This study is the first to investigate the metabolic characteristics and potential metabolic biomarkers in the serum of OKC patients using GC‒MS. Our study provides further evidence to explore the pathogenesis of OKC.

Transformation of an odontogenic keratocyst into a solid variant of odontogenic keratocyst/keratoameloblastoma during long­term follow­up: A case report.

Keratoameloblastoma (KA) and solid variant of odontogenic keratocyst (SOKC) are rare odontogenic lesions, and their relationship and differences are unclear. The present study described a case that started as an odontogenic keratocyst (OKC) and transformed to SOKC/KA upon recurrence. Briefly, a 26­year­old man presented with swelling in the right cheek and was referred to the Department of Oral and Maxillofacial surgery, Hiroshima University Hospital (Hiroshima, Japan). At the initial visit, unicystic bone permeation was observed extending from the right canine to the molar, maxillary sinus and nasal cavity. After the biopsy, the patient underwent excisional surgery and was diagnosed with OKC. Thereafter, the lesion recurred six times over a period of 13 years and showed different histopathological features from those of the primary lesion, all consisting of numerous cysts with keratinization, which were diagnosed as SOKC/KA. The Ki­67 positivity rate was ~10%, which was higher than that of the primary lesion, but there was no atypia. Genetic analysis of the recurrent lesion revealed mutations in adenomatous polyposis coli and Kirsten rat sarcoma viral oncogene homolog. This case originated from OKC, and the morphological features of OKC and KA were mixed upon recurrence, supporting the commonality and association between the two. However, multiple mutations different from those of OKC and ameloblastoma were detected, suggesting an association of SOKC/KA with increased proliferative activity and a high recurrence rate.

Pediatric Oral Pathology: Odontogenic Cysts.

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

Focal Abscess: Description of a clinical case.

Purpose The purpose of this article is to present a case report of a periapical lesion found in a 24-year-old female who presented to a university dental hygiene clinic with a chief complaint of vestibular pain around tooth #22. Radiographically, the lesion appeared radiolucent and unilocular with well-defined borders and significant bone resorption. The area of radiolucency increased in size over a period of five weeks indicating an aggressive pathosis. Given the clinical and radiographic presentations, the differential diagnosis included an odontogenic keratocystic tumor (odontogenic keratocyst), central giant cell granuloma, periapical cyst, and squamous cell carcinoma. Biopsy revealed the diagnosis of a focal abscess. The patient's presenting signs and symptoms are reviewed. Management and contributing factors are discussed.

A retrospective study of the malignant change of odontogenic keratocyst.

The odontogenic keratocyst (OKC) is a common cystic lesion of the maxilla and mandible. Squamous cell carcinoma (SCC) arising from OKC or dysplasia occurring in OKC is rare. This study aimed to explore the incidence and clinical features of the dysplasia and malignant transformation of OKC. In this study, 544 patients diagnosed with OKC were collected. Among them, 3 patients were diagnosed as SCC arising from OKC, and 12 patients were diagnosed as OKC with dysplasia. The incidence was calculated. Clinical features were analyzed by chi-square test. In addition, a representative case reconstructing mandible with vascularized fibula flap under general anesthesia was reported. And cases reported before were reviewed. The incidence of the dysplasia and malignant transformation of OKC, which are highly associated with the clinical features of swelling and chronic inflammation, is about 2.76%. But the relevance between the dysplasia and malignant transformation and age, gender together with pain is not statistically high. All in all, the clinical features of swelling and chronic inflammation can be considered as characteristics of the dysplasia and malignant transformation of OKC. Although the pain isn't statistically relevant, it may be a dangerous clew. Also, combined with earlier literatures, the dysplasia and malignant transformation of OKC shows unique features of radiographs and histopathology.

Cystic Lesions of the Jaws: The Top 10 Differential Diagnoses to Ponder.

BACKGROUND: Cystic lesions of the gnathic bones present challenges in differential diagnosis. This category includes a smorgasbord of odontogenic and non-odontogenic entities that may be reactive or neoplastic in nature. While most cystic jaw lesions are benign, variability in biologic behavior makes distinction between these entities absolutely crucial. METHODS: Review. RESULTS: Two clinical cases are presented in parallel and are followed by an illustrated discussion of the ten most likely differential diagnoses that should be considered when confronted with a cystic jaw lesion. Strong emphasis is placed on the histologic differences between these entities, empowering readers to diagnose them with confidence. Perhaps even more importantly, the more common diagnostic pitfalls in gnathic pathology are discussed, recognizing that a definitive diagnosis cannot be rendered in every situation. The histologic diagnoses for the two clinical cases are finally revealed. CONCLUSION: Cystic lesions of the maxilla and mandible may be odontogenic or non-odontogenic. The most common cystic lesions are the reactive periapical cyst, and the dentigerous cyst (which is developmental in nature). It is important to note that cystic neoplasms also occur in the jaws, and that the presence of inflammation may obscure the diagnostic histologic features of lesions like odontogenic keratocyst and unicystic ameloblastoma. Ancillary testing is of limited diagnostic value in most scenarios. However, both clinical and radiographic information (such as the location, size, duration, associated symptoms, and morphology of the lesion in its natural habitat) are significantly useful.

Histopathological and histomorphometric analysis of glandular odontogenic cyst-A diagnostic aid.

OBJECTIVES: Compare recognized microscopic parameters, including variations in width, plaque-like thickenings, intra-epithelial microcysts, clefts, mucous, hob-nail, ciliated and clear cells, between glandular odontogenic cyst (GOC) and GOC-like cysts, investigate the extent of cyst circumference exhibiting these features, and inflammation. MATERIALS AND METHODS: Archival records of cysts with histological features of GOC evaluated between 2000 and2020 were retrieved. Slides were revised, and the expression of features throughout the cyst wall was analyzed. Cysts with at least 5 features were classified as GOC, cysts with 3-4 features as GOC-like. RESULTS: The study included 74 cysts, 47 males M, 25 females (2 unknown gender), aged 19-81 years, 62 (83.8%) GOC, 12 (16.2%) GOC-like. Mandible was involved in 44 (59.5%), maxilla in 30 (40.5%), 18 (25%) were associated with unerupted teeth. Cyst classified as GOC had significantly higher rates of all parameters investigated, (except ciliated and clear cells), than GOC-like cysts (p ≤ 0.05). 26 (40.6%) cases showed GOC features in >50% of cyst circumference, 21 (32.8%) involved 25-50%, 17 (26.6%) <25%. More than 50% circumference involvement was highly and independently predictive for a diagnosis of GOC, <25% was highly and independently predictive for GOC-like (p = 0.003). Hobnail cells (p = 0.008) and plaque-like thickenings (p = 0.038) were significantly more frequent in inflamed cysts. CONCLUSION: Besides the number and type of histological features, GOC can be characterized by their distribution within the cyst circumference (focal Vs diffuse), and it may serve as a new diagnostic aid. It is suggested that GOC and GOC-like may represent a single spectrum.

Different presentations of the Buccal Bifurcation Cyst: A case series.

The buccal bifurcation cyst (BBC) is an uncommon odontogenic inflammatory cyst affecting the vestibular aspects of the first or second mandibular molar of pediatric patients. Its etiopathogenesis is not fully understood, but it is hypothesized that food and detritus impacting buccal periodontal pockets in titled tooth would be responsible for inflammation of the pericoronal tissues, leading to proliferation of epithelial rests and subsequent cystic formation. The true prevalence of the BBC is not known, but it is estimated to be less than 1% of all the inflammatory cysts. Most cases are unilateral but bilateral cases may account for up to 30% of all BBCs, which can generate confusion to unfamiliar clinicians. Maxillary cases are extremely uncommon, and to our knowledge, there are no cases published in the English literature. In this case series, we present five BBC cases; two unilateral, two bilateral, and one affecting the maxilla. We included clinical, imaging, and histopathological information to highlight the different presentations that this cyst might have, with the final aim to aid clinicians in its diagnosis and ultimately, its treatment.

The extra-osseous odontogenic keratocyst: An anachronism?

INTRODUCTION: The aim of this study was to review the literature on extra-osseous odontogenic keratocysts, and to present the best possible approach to these lesions. MATERIAL AND METHODS: A comprehensive literature search was performed using the strategy: (keratocyst) and (soft tissue)). A total of 25 articles were included, 20 reported on extra-osseous odontogenic keratocysts in the soft tissues of the mouth and 17 presented as gingiva cysts. DISCUSSION: Almost all articles were published in the last 20 years and 16 even in the last 10 years. Extra-osseus odontogenic keratocysts are usually found in the cheek or present as gingiva cysts of adults. Muscle abutment of the cheek lesions is common but infiltration of muscles has also been reported. CONCLUSION: In almost none of the reported cases was a preoperative diagnosis available. Based on the clinical presentation and available laboratory tests it is suggested to always do an aspiration biopsy to ascertain the diagnosis preoperatively. This is particularly relevant for those who appear in the soft tissues of the mouth.

Characterization and Classification of Keratinized Odontogenic Cysts in 29 Dogs.

This retrospective case series presents a unique group of odontogenic cysts that are lined by heavily keratinized epithelium and contain laminated keratin. Keratinized odontogenic cyst (KOC) is proposed as appropriate terminology for the described lesions. The series evaluates cysts from 29 dogs, including clinical presentation, diagnostic imaging, and histopathology. All 29 lesions occurred in tooth bearing regions of the jaws; 21 were maxillary and 8 were mandibular. These keratinized odontogenic cysts were unilocular or multilocular, and some demonstrated considerable expansion resulting in bone destruction. In 13 of 29 cases, there was evidence of tooth displacement associated with the expansion of the KOC. The KOCs did not have a distinctive radiographic appearance. 48% of the cysts had a soft tissue defect through which the keratin contents could be visualized. Cyst contents ranged from hard mineralized keratin to fluid consistency with soft flecks of keratin. The pathoetiology of KOCs is unknown; however, the biological behavior is benign and thought to be slowly progressive despite potential for locally destructive growth. Recurrence is uncommon when cyst enucleation and debridement are aggressive or when solid cysts are excised en bloc.

Surgical enucleation of radicular cyst in endodontically treated primary mandibular molar.

Radicular cysts are most common odontogenic cysts seen in the orofacial region, but not commonly detected in paediatric cases. This case report describes the accidental detection of radicular cyst and its surgical management in an 8-year-old boy in lower left back region of the jaw. The article also gives an insight about radicular cyst from its aetiology to the various other treatment modalities.

Unicystic Ameloblastoma of Mandible: A Case Report.

Ameloblastomas of jaws are benign odontogenic tumors of epithelial origin with four clinical variants: solid multicystic type, unicystic type, desmoplastic type, and extraosseous type. The incidence rate of ameloblastoma is 0.92 per million person-years. Unicystic ameloblastoma refers to those cystic lesions that show clinical and radiologic characteristics of an odontogenic cyst but shows a typical ameloblastomatous epithelium lining part of the cyst cavity, with or without luminal and/or mural tumor proliferation on histological examination. Here is a unique case of unicystic ameloblastoma involving the mandible in a 70-year-old patient. The case was managed by segmental mandibulectomy and flap repair. Unicystic ameloblastoma accounts for only 13% of all known cases in scientific literature. Considering the rarity of the lesion, the purpose of presenting this report on a clinical case is to emphasize the importance of radiological evaluation and histopathological examination for the diagnosis of ameloblastoma. Keywords: ameloblastoma; odontogenic cysts; odontogenic tumors; segmental mandibulectomy.

Imaging of odontogenic keratocysts: a pictorial review.

INTRODUCTION: The aim of the present article was to review and depict the main radiological features of odontogenic keratocysts (OKCs), thus helping the differential diagnoses from other odontogenic cysts and neoplasms. EVIDENCE ACQUISITION: A review of articles published between January 2000 and October 2020 using Medline and the MeSH Term "odontogenic keratocyst" in combination with the following terms "imaging," "radiology," "panoramic radiograph," and "computed tomography," was performed. EVIDENCE SYNTHESIS: Radiographically, OKCs are well-defined unilocular or multilocular radiolucencies bounded by corticated margins. Most lesions are unilocular; instead, multilocular OKCs represent about the 30% of cases, mainly involving the posterior mandible. When, particularly in large lesions, OKCs display a multilocular presentation with adjacent satellite cysts (daughter cysts) a "soap-bubble appearance" can be recognized. DISCUSSION: Panoramic radiograph and CT still play an important role in the diagnosis and treatment planning of OKCs. Unfortunately, it may not be easy to differentiate OKCs from other odontogenic lesions, especially when they are small and unilocular. CONCLUSIONS: Histopathological findings are still necessary to obtain a definitive diagnosis.

Peripheral odontogenic keratocysts in buccal soft tissues: two cases report.

Peripheral odontogenic keratocysts are rarely observed, and cases of odontogenic keratocysts of buccal soft tissues are even rarer. This study was performed to present two rare cases of odontogenic keratocysts in buccal soft tissues and review related literature.