Immunohistochemical evaluation of cyclin D1 and p63 in odontogenic keratocyst and unicystic ameloblastoma.

INTRODUCTION: Odontogenic keratocyst (OKC) and unicystic ameloblastoma (UA) are lesions of odontogenic origin. Both lesions are morphologically cysts. However, they are classified as developmental cysts and epithelial odontogenic tumours, respectively. Cyclin D1 (CCD1) dysregulation is associated with oncogenic activity and malignancies, while tumour protein p63 (p63) alterations are associated with tumourigenesis. AIM: To evaluate and compare the protein expression of CCD1 and p63 in sporadic OKC (OKC-sp), syndromic OKC (OKC-sy), and UA. MATERIAL AND METHODS: 45 cases from the Anatomical Pathology Department, Faculty of Dentistry, University of Chile were analysed and divided into groups: OKC-sp (n=15), OKC-sy (n=15) and UA (n=15), the latter categorised into intraluminal and/or luminal (n=7) and mural (n=8). Immunohistochemical staining for CCD1 and p63 proteins was performed from paraffin-embedded sections. Statistical analysis included the Shapiro-Wilk test, one-way ANOVA with Tukey's multiple comparisons, and Spearman's correlation coefficient (p<0.05). RESULTS: There was an involvement mainly in women in the mandibular area, and a high frequency of jaw expansion, especially in the mural UA. P63 protein expression was higher than CCD1 in all cystic lesions, particularly in mural UA (p<0.001). No correlation was found between CCD1 and p63 expression. CONCLUSION: P63 may serve as a valuable marker for evaluating cell proliferative activity in odontogenic cystic lesions, providing insights into the aggressive behaviour of mural UA.

Calcifying Odontogenic Cyst Presenting Odontogenic Keratocyst-Like Areas: A Rare Case Report.

An 81-year-old male patient presented with a well-demarcated, unilocular radiolucent lesion in the right mandibular body, identified during a routine radiographic examination. Based on the clinical hypothesis of a residual cyst, enucleation with curettage was performed, and the specimen was submitted for histopathological analysis. Microscopically, the cystic lesion was predominantly lined by ameloblastomatous epithelium with numerous ghost cells and dentinoid. Additionally, other cystic cavities lined by stratified squamous epithelium with corrugated parakeratin were observed in the fibrous capsule. Based on these features, a final diagnosis of a calcifying odontogenic cyst with odontogenic keratocyst-like areas was established. No recurrence was observed over a 9-year follow-up period. The association of a calcifying odontogenic cyst with odontogenic keratocyst or odontogenic keratocyst-like areas is very rare. To date, this is the second case report in the literature presenting these findings.

Immunohistochemical Analysis of CD117 in the Mast Cells of Odontogenic Keratocysts.

BACKGROUND: Odontogenic lesions contain mast cells (MCs), particularly those with a cystic appearance. Because of their high recurrence rates and aggressive clinical behaviour, odontogenic keratocysts (OKCs) require special treatment. A particular kind of protein called cluster of differentiation (CD) 117/ receptor tyrosine kinase (c-KIT) is present on the surface of many cells. Most hematopoietic cells lose their expression of KIT during the differentiation process, with the exception of MCs, which continue to express KIT throughout their lifetime. AIM: Using the CD117 immunomarker, this immunohistochemical investigation sought to assess the presence and location of MCs in OKCs and examine the relationship between MC numbers in sporadic, syndromic, and recurrent OKCs. METHODS: The study comprised 30 paraffin-embedded tissue specimens, and a histopathological diagnosis was made from hematoxylin and eosin-stained sections with a thickness of 4-5 µ. Out of 30 specimens, 21 were sporadic, six were recurrent OKCs, and three were syndrome-associated OKCs. CD-117/c-kit rabbit polyclonal primary antibody was used to stain the sections for observing MCs, which were then viewed under a light microscope with a digital camera and a desktop computer with MICAPS software for viewing images. RESULT: To compare the number of MCs among OKCs, a one-way ANOVA test was used. Our study revealed that a statistically significant increase in MCs has been observed in the subepithelial and deep connective tissue of recurrent OKC (p < 0.05). However, a comparison of the mean MC value among three OKC subtypes did not reveal any statistically significant differences. An increased mast count was observed in the deep connective tissue layer of syndromic OKC under multiple comparisons. CONCLUSION: Our study concluded that MCs were present in increased numbers both in the superficial and deep connective tissue of recurrent OKCs, indicative of their aggressive clinical behaviour. Increased mean MC counts observed in some of the sporadic cases may be an indicator of their chances of recurrence in the future.

Calcifying Odontogenic Cyst Associated with Complex Odontoma: Report of a Rare Case.

Calcifying odontogenic cyst, also known as Gorlin cyst is a rare benign cystic lesion primarily found in the jawbones, accounting less than 1% of odontogenic cysts. It can be associated with odontogenic tumors such as odontomas. We report a rare case of COC associated with complex odontoma in a young patient and discuss its clinical features, diagnosis, and treatment options. An 18-year-old female patient presented with a painless radiopaque lesion of the right mandibular bone at Oral Medicine and Oral Surgery department. Radiographs revealed irregular tooth-like structures in the canine-premolar area. The lesion was surgically removed, and histopathology confirmed COC with a complex odontoma. As of the World Health Organization's 2022 definition, COC is a developmental odontogenic cyst characterized by calcified ghost cells. It typically affects individuals during their second and third decades of life, with no gender preference, almost equally in the maxilla and the mandible. The main treatment is total enucleation, with a generally favorable prognosis. Histopathology is essential for diagnosis due to its mimicry of other jaw conditions. Long-term follow-up is needed to prevent recurrences.

Three-dimensional analysis of decompression efficacy and influencing factors in the maxillofacial cystic lesions: a retrospective study.

Objectives: To evaluate the effectiveness of decompression and various parameters that may affect volume change in cystic lesions. Patients and. Methods: This retrospective study included patients who visited the Department of Oral and Maxillofacial Surgery at Ewha Womans University Medical Center between 2012 and 2022 for decompression of cystic lesions of the jaw. To measure volume changes, pre- and post-decompression cone-beam computed tomography was performed and reconstructed in three dimensions using Mimics 25.0 software (Materialise NV). A comparative analysis was performed based on sex, age, initial cyst volume, location, degree of cortical layer expansion, and pathologic diagnosis using the Mann-Whitney U and Kruskal-Wallis tests. Results: In all 20 cases, the duration of decompression was 7.84±3.35 months, and all patients successfully completed the decompression period without any complications. Significant differences were observed in the reduction rate and shrinkage speed based on the degree of cortical layer expansion. However, only the shrinkage speed (not the reduction rate) showed a significant difference with respect to the initial cyst volume. Significant differences were not observed based on sex, age, location, or pathologic diagnosis. Conclusion: Although the present study involved a small number of cases, the effectiveness of decompression was confirmed. In particular, 3D analysis overcame the shortcomings of previous studies of decompression and allowed earlier resection. Further studies with more patients are required to provide a rationale for these results and identify factors that influence decompression.

Can CAPRIN-1 Be Responsible for the Recurrence Potential of Odontogenic Keratocysts?

Objectives: The purpose of this retrospective study is to compare dentigerous cysts and odontogenic keratocysts for cytoplasmic activation/proliferation - associated protein-1 antibodies via immunohistochemical staining to obtain a new perspective about the specific behavioural characteristics of odontogenic keratocysts at the molecular level. Material and Methods: Forty dentigerous cysts (DC) and forty odontogenic keratocysts (OKC) tissue samples were examined using immunohistochemical staining to detect cytoplasmic activation/proliferation - associated protein-1 (CAPRIN-1) antibodies. Nuclear and/or cytoplasmic staining was evaluated as "positive". Cell staining rate (%) and cell staining intensity were determined, and a staining intensity distribution (SID) score was calculated for each sample. Cases were considered "negative" if they showed no staining for CAPRIN-1 antibodies, thus were given a SID score of zero. According to the SID scores, the expression levels were rated as negative, mild, moderate, or high. Results: Of 80 samples, 16 that could adversely affect immunohistochemical evaluation were excluded. Ten negative, 21 positive and three negative, 30 positive CAPRIN-1 expressions were observed in DC and OKC groups, respectively. The difference between the negative and positive cases within groups was significant only in the OKC group (P = 0.000). The SID score range and mean were 0 to 160 and 31.1 (SD 35.7) for DC and 0 to 160 and 57.3 (SD 42.3) for OKC groups. CAPRIN-1 expression was significantly higher in the OKC group (P = 0.043). Conclusions: The molecular basis for increased mitotic activity, high recurrence rates or presence of satellite cysts in odontogenic keratocysts may be attributed to the expression of cytoplasmic activation/proliferation - associated protein-1.

Immunohistochemical analysis of p53 and p63 in selected odontogenic cysts and tumours.

Background: It is a well-recognized fact that abnormal cell proliferation plays a crucial role in the development of odontogenic lesions. p53 is a tumour-suppressor gene which assists in cell cycle regulation and p63 is a homolog of p53 responsible for ectodermal differentiation and maintenance of stratified epithelial progenitor-cell. Analysing the tissue expression of p53 and p63 in odontogenic lesions may provide us with an insight into their potential role in the development of these lesions. Objective: The objective is to study the expression of p53 and p63 in selected odontogenic lesions using immunohistochemistry. Materials and Methods: Formalin-fixed paraffin-embedded tissues of 15 ameloblastomas, 10 adenomatoid odontogenic tumours (AOT), 15 odontogenic keratocysts (OKCs), 10 dentigerous cysts (DCs) along with 10 cases of normal mucosa were retrieved from the departmental archives. These specimens were then subjected to immunohistochemical staining using p53 and p63 oncoproteins. Results: p53 and p63 immune-expression showed mainly intranuclear localization. The mean positivity of p53 in ameloblastoma (59.45%) and OKC (26.38%) was significantly higher than AOT (6.77%) and DC (4%). In contrast, there was no significant difference in the positivity of p63 in between ameloblastoma (77.55%), AOT (69.50%), OKC (76.47%), and DC (50.69%). Conclusion: p53 expression can be correlated with the clinical behaviour of the odontogenic lesions and it can be used as a prognostic marker in odontogenic cysts and tumours. In contrast, p63 expression does not corelate with the biological behaviour of odontogenic lesions.

Podoplanin immunoexpression in odontogenic lesions: a systematic review, meta-analysis, and integrated bioinformatic analysis.

BACKGROUND: Podoplanin (PDPN) is a transmembrane glycoprotein implicated in the pathogenesis of odontogenic lesions (OL). It is localized at the membrane and cytoplasmic level, and its interaction with other proteins could trigger cell proliferation, invasion and migration. The main objective of this systematic review is to explore the immunoexpression pattern of podoplanin in OL. In addition, as secondary objectives, we aimed to compare the immunostaining intensity of PDPN in OL, to analyze its interaction networks by bioinformatic analysis and to highlight its importance as a potential diagnostic marker useful in the pathogenesis of OL. METHODS: The protocol was developed following PRISMA and Cochrane guidelines. The digital search was performed in the databases: PubMed/MEDLINE, ScienceDirect, Scopus, Web of Science and Google Schoolar from August 15, 2010 to June 15, 2023. We included cross-sectional and cohort studies that will analyze the pattern of PDPN immunoexpression in OL. Two investigators independently searched for eligible articles, selected titles and abstracts, analyzed full text, conducted data collection, and performed assessment of study quality and risk of bias. In addition, part of the results were summarized through a random-effects meta-analysis. STRING database was used for protein-protein interaction analysis. RESULTS: Twenty-nine relevant studies were included. The ages of the subjects ranged from 2 to 89 years, with a mean age of 33.41 years. Twenty-two point two percent were female, 21.4% were male, and in 56.4% the gender of the participants was not specified. A total of 1,337 OL samples were analyzed for PDPN immunoexpression pattern. Ninety-four (7.03%) were dental follicles and germs, 715 (53.47%) were odontogenic cysts, and 528 (39.49%) were odontogenic tumors. Meta-analysis indicated that the immunostaining intensity was significantly stronger in odontogenic keratocysts compared to dentigerous cysts (SMD=3.3(CI=1.85-4.82, p=0.000*). Furthermore, bioinformatic analysis revealed that PECAM-1, TNFRF10B, MSN, EZR and RDX interact directly with PDPN and their expression in OL was demonstrated. CONCLUSIONS: The results of the present systematic review support the unique immunoexpression of PDPN as a potential useful diagnostic marker in the pathogenesis of OL.

Large nasopalatine duct cyst in a child patient: a case report.

Introduction and importance: Nasopalatine duct cyst (NPDC) is one of the most common non-odontogenic cysts of the oral cavity, which it can arise at any age but is seen rarely in children. These cysts are usually asymptomatic, and are detected by routine radiographs. Case presentation: This case report presents the management of a large nasopalatine duct cyst in an 11-year-old pediatric patient. The patient presented with symptoms such severe mobility in central incisors and swelling in the anterior part of the palate. Diagnostic imaging confirmed the presence of a large cyst in the nasopalatine region. Surgical intervention was performed to excise the cyst, and the central incisors were splinted with a rigid splint for 4 weeks. Clinical discussion: The case of a large nasopalatine duct cyst in a child patient poses several important clinical considerations. Nasopalatine duct cysts are relatively rare, especially in pediatric patients, making this case particularly noteworthy. The presentation of a large cyst in a child raises questions about the etiology, diagnosis, and treatment options for such cases. Conclusions: This case report highlights the importance of considering nasopalatine duct cysts as a differential diagnosis in pediatric patients presenting with maxillary swelling and associated symptoms. Early recognition and appropriate management are essential to prevent potential complications and ensure optimal outcomes for the patient. The nasopalatine duct cyst that occurs at children may be aggressive and led to severe loss in the supporting alveolar bone with teeth mobility.

Dentigerous Cysts in Children: Clinical, Radiological, and Healing Aspects.

Background and Objectives: Dentigerous cysts are one of the most frequent pathologies associated with unerupted or impacted teeth. Such cysts show a male predilection and a preference for the mandibular region. Also, they commonly occur in the second and third decades of life, with only 9% occurring in the first decade. The aim of this work is to apply and study the therapeutic algorithms developed for dentigerous cysts and their outcomes, from the early diagnostic stage to the complete healing phase of pediatric patients diagnosed with this medical condition. Materials and Methods: The study included 19 pediatric patients diagnosed with dentigerous cysts who underwent the enucleation and extraction or conservative attitude of the associated tooth. The bony healing was also followed-up 9 months after the surgery. Results: A higher incidence in the posterior area of the mandible and maxilla was observed, as well as a higher incidence in boys. The 9 months postoperative radiographic assessment showed that the bony defects were completely healed. Conclusions: A thorough understanding of the nature of the lesion backed by a good clinical history and by state-of-the-art radiographic and radiologic examinations can go a long way in helping the surgeon to choose the correct therapeutic approach and to ameliorate the medical condition in the best long-term interest of the young patient. The considered dentigerous cyst cases demonstrated that an early diagnosis and treatment of this pathology is followed by a responsive treatment.

Descompressão de cavidades ósseas associadas a cistos dentígeros / Decompression of bone cavities associated with dentigerous cysts / Descompresión de cavidades óseas asociadas a quistes dentígeros

Objetivo: O Cisto Dentígero (CDT) é o tipo mais comum dos cistos odontogênicos de desenvolvimento e o segundo mais frequente entre todos que ocorrem nos maxilares. E uma lesão benigna, radiolúcida, geralmente unilocular, prevalente no sexo masculino, nas três primeiras décadas de vida e com uma maior ocorrência na mandíbula. Possui crescimento lento e assintomático e sua etiopatogenia ainda é considerada desconhecida. Este trabalho tem como objetivo relatar dois casos clínicos de cistos dentígeros, o primeiro em um paciente de 50 anos e o segundo, em uma paciente de 11 anos, ambos localizados em mandíbula e envolvendo molares. Relato de caso: O método utilizado para o tratamento dessa lesão foi a descompressão, no qual foi criada uma comunicação entre o cisto e a cavidade oral, colocando um dispositivo para o condicionamento de sua drenagem e posterior enucleação da lesão e do dente envolvido. Este tratamento, por ser menos invasivo, promove uma maior preservação das estruturas saudáveis adjacentes e consequentemente, um pós-operatório mais favorável. Conclusão: Portanto, é de total importância que o profissional esteja apto para realizar este tipo de procedimento e tenha conhecimento para realizar a técnica, para que assim, o risco de complicações trans e pós-operatórias seja diminuído... (AU)

Objective: The Dentigerous Cyst (DTC) is the most common type of developmental odontogenic cyst and the second most frequent among all that occur in the jaws. It is a benign, radiolucent, usually unilocular lesion, prevalent in males, in the first three decades of life and with a greater occurrence in the mandible. It has slow and asymptomatic growth and its etiopathogenesis is still considered unknown. This paper aims to report two clinical cases of dentigerous cysts, the first in a 50-year-old patient and the second in an 11-year-old patient, both located in the mandible and involving molars. Case report: The method used to treat this lesion was decompression, in which a communication was created between the cyst and the oral cavity, placing a device to condition its drainage and subsequent enucleation ofthe lesion and the tooth involved. This treatment, being less invasive, promotes greater preservation of adjacent healthy stmctures and, consequently, a more favorable postoperative peñod. Conclusion: Therefore, it is of utmost importance that the professional is able to perform this type of procedure and has the knowledge to perform the technique, so that the risk of trans and postoperative complications is reduced... (AU)

Objetivo: El Quiste Dentígero (CDT) es el tipo más común de quiste odontogénico del desarrollo y el segundo más frecuente entre todos 10s que se presentan en 10s maxilares. Es una lesión benigna, radiolúcida, generalmente unilocular, prevalente en el sexo masculino, en Ias tres pñmeras décadas de Ia vida y con mayor incidencia en Ia mandíbula. Tiene un crecimiento lento y asintomático y su etiopatogenia aún se considera desconocida. Este trabajo tiene como objetivo reportar dos casos clínicos de quistes dentígeros, el primero en un paciente de 50 años y el segundo en un paciente de 11 años, ambos localizados en Ia mandíbula y afectando molares. Caso clínico: El método utilizado para el tratamiento de esta lesión fue Ia descompresión, en Ia que se creaba una comunicación entre el quiste y Ia cavidad bucal, colocando un dispositivo para condicionar su drenaje y posterior enucleación de Ia lesión y del diente afectado. Este tratamiento, al ser menos invasivo, favorece una mayor conservación de Ias estmcturas sanas adyacentes y, en consecuencia, un postoperatorio más favorable. Conclusión: Por Io tanto, es de suma importancia que el profesional sea capaz de realizar este tipo de procednmento y tenga 10s conocnmentos para realizar Ia técnica, de manera que se reduzca el riesgo de complicaciones trans y postoperatorias... (AU)

Automatic classification and segmentation of multiclass jaw lesions in cone-beam CT using deep learning.

OBJECTIVES: To develop and validate a modified deep learning (DL) model based on nnU-Net for classifying and segmenting five-class jaw lesions using cone-beam CT (CBCT). METHODS: A total of 368 CBCT scans (37 168 slices) were used to train a multi-class segmentation model. The data underwent manual annotation by two oral and maxillofacial surgeons (OMSs) to serve as ground truth. Sensitivity, specificity, precision, F1-score, and accuracy were used to evaluate the classification ability of the model and doctors, with or without artificial intelligence assistance. The dice similarity coefficient (DSC), average symmetric surface distance (ASSD), and segmentation time were used to evaluate the segmentation effect of the model. RESULTS: The model achieved the dual task of classifying and segmenting jaw lesions in CBCT. For classification, the sensitivity, specificity, precision, and accuracy of the model were 0.871, 0.974, 0.874, and 0.891, respectively, surpassing oral and maxillofacial radiologists (OMFRs) and OMSs, approaching the specialist. With the model's assistance, the classification performance of OMFRs and OMSs improved, particularly for odontogenic keratocyst (OKC) and ameloblastoma (AM), with F1-score improvements ranging from 6.2% to 12.7%. For segmentation, the DSC was 87.2% and the ASSD was 1.359 mm. The model's average segmentation time was 40 ± 9.9 s, contrasting with 25 ± 7.2 min for OMSs. CONCLUSIONS: The proposed DL model accurately and efficiently classified and segmented five classes of jaw lesions using CBCT. In addition, it could assist doctors in improving classification accuracy and segmentation efficiency, particularly in distinguishing confusing lesions (eg, AM and OKC).

Report of intraosseous intravascular papillary endothelial hyperplasia associated with an odontogenic cyst in the maxilla and literature review.

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.

Decompression Associated with Irrigation with Elixir Sanativo® Before Enucleation of Odontogenic Keratocysts: Report of Two Cases

Odontogenic Keratocyst (OKC) is an aggressive cystic lesion of the jaws and one of the most common odontogenic cysts, usually affecting the posterior region of the mandible. Recurrences are frequently recorded and may be directly related to the treatment modality adopted. Two patients presenting OKC were treated by combining decompression and intralesional irrigation with the drug Elixir Sanativo® for 8 months before enucleation surgery. In both patients, the association between decompression and irrigation with Elixir Sanativo® represented a safe and effective method to reduce the preoperative lesion dimensions.

El queratoquiste odontogénico (QO) es una lesión quística agresiva de la mandíbula y es uno de los quistes odontogénicos más frecuentes, que suele afectar a la región posterior de la mandíbula. Las recidivas se registran con frecuencia y pueden estar directamente relacionadas con la modalidad de tratamiento adoptada. Presentación del caso: Dos pacientes que presentaban OKC fueron tratados combinando descompresión e irrigación intralesional con el fármaco Elixir Sanativo® durante 8 meses antes de la cirugía de enucleación. En ambos pacientes, la asociación entre descompresión e irrigación con Elixir Sanativo® representó un método seguro y eficaz para reducir las dimensiones de la lesión preoperatoria.

Surgical Approach of Maxillary Cysts in Pediatric Patients: Report of Two Clinically Similar Cases with Different Diagnosis

Extensive odontogenic cysts in children may represent surgical challenges, as they may have common clinical characteristics and different approaches. The main objective of this study is to compare two cases of pediatric odontogenic cysts in maxilla with similar surgical treatment and different histopathological diagnosis. The case series collected included two children, both 12 years old, with encapsulated osteolytic lesions in the region of the maxilla and zygoma body, with clinical and imaging characteristics that suggested odontogenic cysts. The histopathological diagnosis was dentigerous cyst and radicular cyst. In this way, we address the clinical-surgical diagnostic and therapeutic process adopted, analyzing clinical data, such as signs and symptoms, as well as pre- and postoperative tomography scans. Outpatient visits at regular intervals were planned. Both patients achieved significant regression of initial signs and symptoms and returned to their daily activities. It is noticeable that a good stratification of surgical need and planned action in diagnosis and surgery offer benefits with a favorable prognosis for pediatric odontogenic cysts of the jaw.

Los quistes odontogénicos extensos en niños pueden representar desafíos quirúrgicos, ya que pueden tener características clínicas comunes y diferentes abordajes. El objetivo principal de este estudio fue comparar dos casos de quistes odontogénicos en mandíbulas de niños con tratamiento quirúrgico similar y diagnóstico histopatológico diferente. La serie de casos recolectada incluyó dos niños, ambos de 12 años, con lesiones osteolíticas encapsuladas en la región mandibular y cuerpo cigomático, con características clínicas e imagenológicas que sugerían quistes odontógenos. El diagnóstico histopatológico fue quiste dentígero y quiste radicular. De esta manera abordamos el proceso diagnóstico y terapéutico clínico-quirúrgico adoptado, analizando datos clínicos, como signos y síntomas, así como tomografías pre y postoperatorias. Se planificaron visitas ambulatorias a intervalos regulares. Ambos pacientes lograron una regresión significativa de los signos y síntomas iniciales y regresaron a sus actividades diarias. Se destaca que una buena estratificación de la necesidad quirúrgica y una acción planificada en diagnóstico y cirugía ofrecen beneficios con un pronóstico favorable para los quistes odontogénicos de la mandíbula en pediatría.

Nasopalatine Duct Cyst Treated by Transnasal Endoscopic Marsupialization: A Case Report.

Introduction: Nasopalatine duct cyst (NDC) is the most prevalent non-odontogenic cyst emerging from the epithelial remnants in the maxillary incisive canal. A sublabial or transpalatal approach is performed to enucleate NDC completely. More recently, transnasal endoscopic marsupialization has been used gradually. Case Presentation: A 24-year-old male patient with a large nasopalatine duct cyst with a diameter of 51 mm was managed by transnasal endoscopic marsupialization under general anesthesia. The presentation involves painless swelling around the left side of the anterior maxilla and bulging of the hard palate. No postoperative complications were observed after a 3-month follow-up. Transnasal endoscopic marsupialization is a minimally invasive surgery for large NDC. Clinical discussion: Approximately 1% of the population has a nasopalatine duct cyst. Surgical treatment was carried out under general anesthesia; the cyst was dissected and removed using a typically transnasal endoscopic marsupialization technique. Conclusion: The cause of the NDC is unclear. Simple surgical resection and clinical and radiological control are recommended to ensure the case is resolved correctly.

CLIC4 Function in the Epithelial-Mesenchymal Transition of Epithelial Odontogenic Lesions.

BACKGROUND: Odontogenic lesions constitute a heterogeneous group of lesions. CLIC4 protein regulates different cellular processes, including epithelial-mesenchymal transition and fibroblast-myofibroblast transdifferentiation. This study analyzed CLIC4, E-cadherin, Vimentin, and α-SMA immunoexpression in epithelial odontogenic lesions that exhibit different biological behavior. METHODS: It analyzed the immunoexpression of CLIC4, E-cadherin, and Vimentin in the epithelial cells, as well as CLIC4 and α-SMA in the mesenchymal cells, of ameloblastoma (AM) (n = 16), odontogenic keratocyst (OKC) (n = 20), and adenomatoid odontogenic tumor (AOT) (n = 8). Immunoexpressions were categorized as score 0 (0% positive cells), 1 (< 25%), 2 (≥ 25% - < 50%), 3 (≥ 50% - < 75%), or 4 (≥ 75%). RESULTS: Cytoplasmic CLIC4 immunoexpression was higher in AM and AOT (p < 0.001) epithelial cells. Nuclear-cytoplasmic CLIC4 was higher in OKC's epithelial lining (p < 0.001). Membrane (p = 0.012) and membrane-cytoplasmic (p < 0.001) E-cadherin immunoexpression were higher in OKC, while cytoplasmic E-cadherin expression was higher in AM and AOT (p < 0.001). Vimentin immunoexpression was higher in AM and AOT (p < 0.001). Stromal CLIC4 was higher in AM and OKC (p = 0.008). Similarly, α-SMA immunoexpression was higher in AM and OKC (p = 0.037). Correlations in these proteins' immunoexpression were observed in AM and OKC (p < 0.05). CONCLUSIONS: CLIC4 seems to regulate the epithelial-mesenchymal transition, modifying E-cadherin and Vimentin expression. In mesenchymal cells, CLIC4 may play a role in fibroblast-myofibroblast transdifferentiation. CLIC4 may be associated with epithelial odontogenic lesions with aggressive biological behavior.

Comparing the characteristics of recurrent versus primary odontogenic keratocysts - A single center retrospective cohort study.

The high recurrence rate of odontogenic keratocysts (OKC) entails a large number of follow-up interventions after primary surgery. This study aimed to compare recurrent with primary OKC in regard to recurrence rate, treatment modality, radiographic and clinical findings. A single center retrospective cohort study with surgically treated OKC between 2012 and 2021 was conducted. The primary predictor was recurrence type of the lesion: primary (P-OKC), first recurrence (R1-OKC) and second recurrence (R2-OKC). The primary outcome variables were recurrence and time to recurrence. 68 surgeries were identified. Recurrence was not significantly associated with recurrence type (p = 0.906) but with the method of surgery (p < 0.001). Marsupialized R1-OKC recurred earlier than marsupialized P-OKC. Diameter was significantly associated with recurrence type (p = 0.002). R1-OKC had a smaller median diameter than P-OKC and R2-OKC were smaller than R1-and P-OKC. P-OKC were associated with teeth in 79% of surgeries, R1-OKC in 61% and R2-OKC in 27% (p = 0.007). Postoperative numbness was present after 40% of surgeries and decreased to 15% at follow-up, independently of recurrence type. Time to recurrence may be shorter for recurrent marsupialized OKC. Recurrent OKC are smaller and less often associated with teeth. Postoperative numbness after OKC surgery has a good prognosis.

L-cysteine contributes to destructive activities of odontogenic cysts/tumor.

BACKGROUND: Odontogenic cysts/tumor can cause severe bone destruction, which affects maxillofacial function and aesthetics. Meanwhile, metabolic reprogramming is an important hallmark of diseases. Changes in metabolic flow affect all aspects of disease, especially bone-related diseases. At present, the researches on pathogenesis of odontogenic cysts/tumor are mainly focused on the level of gene regulation, but the effects of metabolic alterations on odontogenic cysts/tumor have still underexplored. MATERIALS AND METHODS: Imaging analysis was used to evaluate the lesion size of different odontogenic lesions. Tartrate resistant acid phosphatase (TRAP) and immunohistochemistry (IHC) assays were utilized to detect the differences in bone destruction activity in odontogenic cysts and tumors. Furthermore, metabolomics and weighted gene co-expression network analysis (WGCNA) were conducted for the metabolomic features and key metabolite screening, respectively. The effect of ferroptosis inhibition on bone destruction was confirmed by IHC, immunofluorescence, and malondialdehyde colorimetric assay. RESULTS: The bone destruction activity of ameloblastoma (AM) was the strongest and the weakest in odontogenic cysts (OC). High-throughput targeted metabolomics was used to map the metabolomic profiles of OC, odontogenic keratocyst (OKC) and AM. WGCNA and differential analysis identified L-cysteine in OKC and AM. Cystathionine γ-lyase (CTH) was further screened by Kyoto Encyclopedia of Genes and Genomes (KEGG) analysis. The functions of L-cysteine were further validated. Finally, we confirmed that CTH affected destructive activities by regulating the sensitivity of epithelial cells to ferroptosis. CONCLUSION: High-throughput targeted metabolomics performed on diseased tissue confirmed the unique alteration of metabolic profiles in OKC and AM. CTH and its metabolite L-cysteine are the key factors regulating destructive activities.

Beneficios de la descompresión y enucleación en lesiones quísticas de gran tamaño en la mandíbula / Benefits of decompression and enuclation in large cystic lesions in the jaw / Benefícios da descompressão e enuclação nas grandes lesões císticas na mandíbula

Las lesiones quísticas ocurren en los maxilares, por la presencia de células remanentes del neuroectodermo embrionario. La descompresión es una técnica conservadora para disminuir la presión intraquística mediante drenaje constante, permitiendo el crecimiento de nuevo hueso centrípeto de las paredes óseas del quiste. Objetivo: determinar los beneficios de la descompresión y enucleación en lesiones quísticas mandibulares, tomando como base la metodología de un caso clínico. Descripción del caso: se diagnosticó una lesión quística mandibular en paciente masculino de 27 años, que acudió a consulta mostrando secreción purulenta en mucosa trígono retromolar de UD 37, inicialmente asintomática. Se utilizaron como materiales la tomográfica computarizada de haz cónico, artefacto de drenaje autocurado, hemiarcada izquierda elaborada con Metil Metacrilato y aparato a base de cilindro. Como resultados se reveló imagen hipodensa de bordes definidos localizada en el límite posterior de cuerpo mandibular, borde anterior y parte de la rama ascendente mandibular del lado izquierdo; extendida en sentido cefálico caudal desde la cresta alveolar y borde anterior de la rama hasta la cortical superior del conducto mandibular. Conclusión: Se confirmó diagnóstico de quiste periapical, quiste residual y ameloblastoma. Se realizó biopsia incisional de la lesión para estudio histopatológico y la descompresión con dispositivo personalizado a enucleación conminada con solución de Carnoy, resultando el tratamiento conservador efectivo complementado por la enucleación de una membrana quística más gruesa y menos friable.

Cystic lesions occur in the jaws due to the presence of remnant cells of the embryonic neuroectoderm. Decompression is a conservative technique to decrease intracystic pressure by constant drainage, allowing the growth of new centripetal bone from the bony walls of the cyst. Objective: to determine the benefits of decompression and enucleation in mandibular cystic lesions, based on the methodology of a clinical case. Case description: a cystic mandibular lesion was diagnosed in a 27 year old male patient, who came for consultation showing purulent secretion in the trigone retromolar mucosa of UD 37, initially asymptomatic. The materials used were cone beam computed tomography, self-curing drainage device, left hemiarch made with Methyl Methacrylate and cylinder based apparatus. The results revealed a hypodense image with defined borders located in the posterior limit of the mandibular body, anterior border and part of the ascending mandibular branch on the left side; extended in a caudal cephalic direction from the alveolar crest and anterior border of the branch to the superior cortical of the mandibular duct. Conclusion: Diagnosis of periapical cyst, residual cyst and ameloblastoma was confirmed. An incisional biopsy of the lesion was performed for histopathological study and decompression with a customized device to enucleation with Carnoy's solution, resulting in effective conservative treatment complemented by enucleation of a thicker and less friable cystic membrane.

As lesões císticas ocorrem nos maxilares, devido à presença de células remanescentes da neuroectoderme embrionária. A descompressão é uma técnica conservadora que visa reduzir a pressão intracística por meio de drenagem constante, permitindo o crescimento de novo osso centrípeto a partir das paredes ósseas do cisto. Objetivo: determinar os benefícios da descompressão e da enucleação em lesões císticas mandibulares, com base na metodologia de um caso clínico. Descrição do caso: foi diagnosticada uma lesão cística mandibular em um paciente do sexo masculino, 27 anos, que se apresentou para consulta apresentando secreção purulenta na mucosa do trígono retromolar do UD 37, inicialmente assintomática. Os materiais utilizados foram tomografia computadorizada de feixe cônico, dispositivo de drenagem autopolimerizável, hemiarco esquerdo confeccionado com metacrilato de metila e aparelho de base cilíndrica. Os resultados revelaram uma imagem hipodensa com limites definidos localizada no limite posterior do corpo mandibular, bordo anterior e parte do ramo mandibular ascendente do lado esquerdo; estendendo-se em direção cefálica caudal desde a crista alveolar e bordo anterior do ramo até ao córtex superior do ducto mandibular. Conclusão: Foi confirmado o diagnóstico de quisto periapical, quisto residual e ameloblastoma. Foi efectuada uma biopsia incisional da lesão para estudo histopatológico e descompressão com um dispositivo adaptado à enucleação cominutiva da solução de Carnoy, resultando num tratamento conservador eficaz complementado pela enucleação de uma membrana quística mais espessa e menos friável.