Intravascular papillary endothelial hyperplasia (IPEH) represents an uncommon reactive endothelial hyperplastic proliferation. A 46-year-old man experienced increased volume in the right maxilla, elevation of the nasal ala, and swelling of the hard palate with a reddish hue for 3 months. Computed tomography revealed an expansive hypodense region and cortical bone destruction associated with an impacted supernumerary tooth and an endodontically treated tooth. Under the differential diagnoses of a radicular cyst, dentigerous cyst, and ameloblastoma, an exploratory aspiration and incisional biopsy were performed. This revealed the formation of blood vessels of various diameters lined by endothelium, forming intravascular papillae positive for CD-34. The definitive diagnosis was IPEH, and the patient was treated by embolization and surgery. Histological analysis confirmed the presence of IPEH associated with an odontogenic cyst. After 12 months of follow-up, no recurrence was observed. Also, we reviewed case reports of IPEH affecting the maxilla and mandible. Fourteen intraosseous cases were reported in the maxilla and mandible, with a preference for males and affecting a wide age range. Complete surgical excision was the treatment of choice, and recurrences were not reported. The pathogenesis of IPEH is controversial and may originate from trauma or inflammatory processes. To the best of our knowledge, this is the first report of an association of IPEH with an odontogenic cyst. The importance of IPEH in the differential diagnosis of intraosseous lesions in the jaws is emphasized, and preoperative semiotic maneuvers are needed to prevent surgical complications.

OBJECTIVE: Odontogenic keratocysts exhibit frequent recurrence, distinctive histopathological traits, a tendency towards aggressive clinical behavior, and a potential linkage to the nevoid basal cell carcinoma syndrome. The aim of this systematic review is to compile insights concerning the control of this condition and assess the effectiveness of various treatment approaches in reducing the likelihood of recurrence.
METHODS: The following systematic review adhered to the PRISMA guidelines. The systematic revision was registered on PROSPERO and  structured around the questions related to the population, intervention, control, outcome and study design (PICOS).
RESULTS: After conducting a search on the PubMed database, we initially identified 944 records. After using end-note software to remove duplicate entries, results totally with 462 distinct records. A thorough review of the titles and abstracts of these articles led to the selection of 50 papers for in-depth examination. Ultimately, following the application of our eligibility criteria, we incorporated 11 articles into our primary outcome analysis.
CONCLUSIONS: Among the studies examined, the most common location for these lesions was found to be in the area of the mandibular ramus and the posterior region of the mandible. In cases where the exact location wasn\'t specified, the mandible emerged as the predominant site. When we considered the characteristics of these lesions in studies that mentioned locularity, most were described as unilocular in two studies, while in two other studies, the prevalence of multilocular lesions was observed. Risk factors associated with keratocyst recurrence include younger patient age, the presence of multilocular lesions, larger lesion size, and a longer anteroposterior dimension. Certain treatment methods have demonstrated a lack of relapses. These include the use of 5-fluorouracil, marsupialization, enucleation with peripheral ostectomy or resection, enucleation and curettage, as well as resection without creating continuity defects. However, it is important to note that further research is essential. Prospective studies and randomized trials are needed to collect more comprehensive evidence regarding the effectiveness of various treatment approaches and follow-up protocols for managing odontogenic keratocysts.
CONCLUSIONS: Odontogenic keratocysts still enter into differential diagnoses with other lesions that affect the jaw bones such as ameloblastama and other tumor forms, furthermore it is not free from recurrence, therefore the therapeutic approach to the lesion aimed at its elimination can influence both the possible recurrence and complications, knowledge of the surgical methods that offer the most predictable and clinically relevant result for the management of follow-up and recurrences.

BACKGROUND: The glandular odontogenic cyst (GOC) is a benign developmental cyst of the jaws that is characterized by a high recurrence rate.
METHODS: A systematic review is presented of reported cases, case series, and retrospective studies of recurrent cases of glandular odontogenic cysts, to determine the overall and detailed demographic features with documentation of the specific histologic features of the initial presentation of each cyst. Searches of detailed databases were carried out to identify articles published in the English language from 1988 to 2023. The variables were demographics, patient symptoms, cyst location, radiographic features, histopathological findings, type of treatment, and minimum eight months of follow-up.
RESULTS: Eighteen cases were identified: with an equal gender presentation of 50% females and 50% males. The average age was 44.7. The mean size was 3.5 cm. The most common location was in the anterior mandible in 50% (n = 9) of cases, followed by the posterior mandible 27.8% (n = 5). Most patients were asymptomatic 55.6% (n = 10). The most common histologic features at first diagnosis were mucous cells in 88.9% (n = 16), variable thickness with 83.3% (n = 15), eosinophilic cuboidal cells 88.9% (n = 16), microcysts 83.3% (n = 15), and clear cells 77.8% (n = 14) cases.
CONCLUSIONS: GOC has an aggressive behavior. Evidence was not conclusive to link any single or combination of histologic features to recurrence, and the strongest correlation for recurrence was the type of treatment. Since this is an uncommon cyst, more cases are needed. Follow-up should continue for at least five years, because recurrences were higher between years 3 and 5.

BACKGROUND: Reconstruction 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.
METHODS: This 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.
CONCLUSIONS: This 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.

Glandular odontogenic cyst (GOC) is a rare developmental odontogenic cyst from the cell rests of Serres. GOC is locally aggressive with a tendency toward recurrence. The most common site of occurrence is the anterior mandible with an asymptomatic presentation. Radiographically, it presents as unilocular or multilocular radiolucency. It bears histopathological resemblance to low-grade mucoepidermoid carcinoma. We report two cases of GOC occurring in a 16-year-old and a 33-year-old male patient with a review of the clinical presentation, histopathological features, and diagnostic aspects of GOC reported so far in literature.

BACKGROUND: The recent impact of artificial intelligence in diagnostic services has been enormous. Machine learning tools offer an innovative alternative to diagnose cysts and tumors radiographically that pose certain challenges due to the near similar presentation, anatomical variations, and superimposition. It is crucial that the performance of these models is evaluated for their clinical applicability in diagnosing cysts and tumors.
METHODS: A comprehensive literature search was carried out on eminent databases for published studies between January 2015 and December 2022. Studies utilizing machine learning models in the diagnosis of odontogenic cysts or tumors using Orthopantomograms (OPG) or Cone Beam Computed Tomographic images (CBCT) were included. QUADAS-2 tool was used for the assessment of the risk of bias and applicability concerns. Meta-analysis was performed for studies reporting sufficient performance metrics, separately for OPG and CBCT.
RESULTS: 16 studies were included for qualitative synthesis including a total of 10,872 odontogenic cysts and tumors. The sensitivity and specificity of machine learning in diagnosing cysts and tumors through OPG were 0.83 (95% CI 0.81-0.85) and 0.82 (95% CI 0.81-0.83) respectively. Studies utilizing CBCT noted a sensitivity of 0.88 (95% CI 0.87-0.88) and specificity of 0.88 (95% CI 0.87-0.89). Highest classification accuracy was 100%, noted for Support Vector Machine classifier.
CONCLUSIONS: The results from the present review favoured machine learning models to be used as a clinical adjunct in the radiographic diagnosis of odontogenic cysts and tumors, provided they undergo robust training with a huge dataset. However, the arduous process, investment, and certain ethical concerns associated with the total dependence on technology must be taken into account. Standardized reporting of outcomes for diagnostic studies utilizing machine learning methods is recommended to ensure homogeneity in assessment criteria, facilitate comparison between different studies, and promote transparency in research findings.

Odontogenic cysts are bony lesions in the jaws that can reach large sizes. Decompression, a technique that helps in their surgical treatment, aims to reduce their size. We aimed to conduct a systematic review of the main types of device used for the decompression of odontogenic cysts and to analyse the indications, types, advantages, and disadvantages of the devices used. We searched PubMed, Science Direct, LILACS, EMBASE, and Web of Science until February 2023, with no time restriction. We considered studies with a minimum of 10 patients published only in English, those that reported cases and case series, randomised clinical trials of the decompression of odontogenic cysts, and the types of devices used during the decompression period. All reported odontogenic cysts had to have been confirmed by biopsy in their respective publications. We found 713 articles in the selected databases. After removing duplicates, 499 remained. After reading the titles and abstracts, we excluded 461 articles so 38 remained. Nine studies were selected for the review, totalling 244 patients. A total of 206 lesions were identified and confirmed by anatomopathological examination: 123 keratocysts, 40 dentigerous cysts, 34 radicular cysts, one cyst of epithelial origin but without specification, and eight unicystic ameloblastomas. Although we did not find out which device is best for the decompression of odontogenic cysts, our findings show that those that are most effective should be as comfortable as possible and should remain in place. They should have stability in the oral cavity and be easy for the patient to clean.

BACKGROUND: Nevoid basal cell carcinoma syndrome (NBCCS, Gorlin syndrome) is a rare autosomal dominantly inherited disorder that is characterized by multisystem disorder such as basal cell carcinomas, keratocystic odontogenic tumors and skeletal abnormalities. Bilateral and/or unilateral ovarian fibromas have been reported in individuals diagnosed with NBCCS.
METHODS: A 22-year-old female, presented with low back pain, and was found to have bilateral giant adnexal masses on pelvic ultrasonography, which had been suspected to be malignant ovarian tumors. Positron emission tomography/computed tomography showed multiple intracranial calcification and skeletal abnormalities. The left adnexa and right ovarian tumor were resected with laparotomy, and pathology revealed bilateral ovarian fibromas with marked calcification. We recommended the patient to receive genetic testing and dermatological examination. No skin lesion was detected. Germline testing identified pathogenic heterozygous mutation in PTCH1 (Patched1).
CONCLUSIONS: The possibility of NBCCS needs to be considered in patients with ovarian fibromas diagnosed in an early age. Skin lesions are not necessary for the diagnosis of NBCCS. Ovarian fibromas are managed with surgical excision with an attempt at preserving ovarian function. Follow-up regime and counseling on options for future fertility should be offered to patients.

Lowe syndrome (LS) is a rare disease (1:500,000) with X-linked recessive inheritance involving the kidneys, eyes, and nervous system. A Mexican 25-year-old male patient presented for diagnosis of multiple radiolucent lesions observed on routine radiographic examination. General aspects revealed cognitive delay, eye alterations, and kidney involvement, which support the diagnosis of LS. Radiolucent well-delimited lesions were observed in both mandibular angle and symphysis. Under general anesthesia, incisional biopsy and decompression were performed. Histological aspects led to diagnosing odontogenic keratocyst (OKC) for all lesions. The lesions in the right and left mandibular angles were decompressed, and the symphyseal lesion was enucleated. A 2-month follow-up shows the bone healing process. There are few reports detailing oral findings in LS. Here, we reported the first case of multiple OKC in a patient with LS. In addition, we performed a literature review on odontogenic lesions in patients affected by LS.

BACKGROUND: CTNNB1 gene encodes beta catenin, a transcriptional activator of Wnt pathway involved in the pathogenesis of odontogenic lesions. Though located intramembranously, its translocation into cytoplasm and nucleus could trigger cell proliferation, inhibition of apoptosis, invasion and migration of the tumour cell.
METHODS: Five electronic databases including MEDLINE by PubMed, Google scholar, Scopus, Trip, Cochrane library and EMBASE until 1 January 2023 without period restriction were thoroughly searched. Those articles that identified CTNNB1 mutation and beta catenin in odontogenic lesions were included for review. Risk of bias was analysed for each study using QUADAS 2 tool and Review Manager 5.3 was used to output its result.
RESULTS: Thirty four published articles were included for data synthesis. A total of 1092 cases of odontogenic lesions were assessed for both CTNNB1 mutation and beta catenin expression. CTNNB1 mutation was observed in ameloblastoma, calcifying odontogenic cyst, calcifying cystic odontogenic tumour and all malignant odontogenic tumours. The beta catenin expression (nuclear and cytoplasmic) was maximum in odontogenic keratocyst and calcifying odontogenic cyst. The expression was variable in ameloblastomas, membranous in odontomas, calcifying cystic odontogenic tumour and nuclear in all malignant tumours.
CONCLUSIONS: High recurrence of odontogenic keratocyst and aggressiveness of solid ameloblastoma and malignant odontogenic tumours could be associated with the nuclear translocation of beta catenin. Disparity between CTNNB1 mutation and beta catenin expression within odontogenic lesions suggests alternate routes of beta catenin activation. The review results support the unique localisation of beta catenin as a helpful diagnostic factor in the pathogenesis of odontogenic lesions.