-
Journal of Oral and Maxillofacial... 2023Odontogenic, non-inflammatory maxillofacial cysts and tumours vary greatly in their ability to grow and cause local tissue destruction. Despite their common embryologic...
INTRODUCTION
Odontogenic, non-inflammatory maxillofacial cysts and tumours vary greatly in their ability to grow and cause local tissue destruction. Despite their common embryologic origin, the biologic mechanisms responsible for this diverse array of clinical behaviour are largely unknown. Unfortunately, even with accurate tissue diagnosis and appropriate surgical management, these tumours have relatively high recurrence rates. While this may be related to surgical technique, it may also be due to intrinsic tumour biology. SOX2 is differentially expressed in odontogenic cysts and tumours, which has an impact over patient prognosis. This could be related to their diverse cells of origin or stages of histogenesis. SOX2 is expressed in OKC and ameloblastoma, and in this study, we look forward to find altered levels and intensity of SOX2 in the above-mentioned lesions.
AIM AND OBJECTIVES
To profile the expression of SOX2 in odontogenic keratocyst (OKC) and ameloblastomaTo compare the intensity of these lesions, analyse their intrinsic feature and predict their recurrence.
MATERIAL AND METHODS
Histopathologically diagnosed cases of OKC and ameloblastoma will be selected ( = 40). Paraffin-embedded, formalin-fixed sections of these lesions will be stained for SOX2 marker using a standard immunohistochemical technique. Positive control will be taken as oral squamous cell carcinoma and negative control will be taken as normal oral mucosa.
RESULTS
A comparison between the stained cell types in odontogenic keratocyst and ameloblastoma revealed statistically significant differences. The immunoreactivity scores of SOX2 were analysed in both groups. The results indicated that 45% of OKC cases exhibited strongly positive reactivity, while 65% of ameloblastoma cases were negative. Statistical analysis demonstrated highly significant differences in the frequency of SOX2 expression between the two groups, with a higher frequency of negative expression in ameloblastoma.
CONCLUSION
Stem cell markers have been observed in these lesions, suggesting the acquisition of stem-like properties by tumour cells, which can affect patient prognosis. Specifically, the marker SOX2 shows differential expression in odontogenic cysts and tumours. High expression of SOX2 in OKC indicates the presence of stem cells with significant self-renewal and proliferative properties, potentially signifying neoplastic behaviour. In contrast, weak or absent expression of SOX2 in ameloblastoma suggests different molecular pathways involved in its neoplastic behaviour.
PubMed: 38304494
DOI: 10.4103/jomfp.jomfp_265_23 -
Journal of Toxicologic Pathology Jan 2024Ameloblastoma is a locally aggressive tumor derived from the odontogenic epithelium of the developing tooth germ. It is rarely reported in experimental Sprague-Dawley...
Ameloblastoma is a locally aggressive tumor derived from the odontogenic epithelium of the developing tooth germ. It is rarely reported in experimental Sprague-Dawley (SD) rats. In this 90-day percutaneous repeated-dose toxicity study, mandibular nodules were observed from day 56 to 90. Upon necropsy, a well-demarcated nodule, approximately 1.2×1.0×1.0 cm, was found attached to the mandibular bone, alongside the unerupted left incisor. Histopathologically, the epithelial cells formed islands, nests, or anastomosing strands. The epithelial islands were surrounded by a peripheral layer of tall columnar cells with antibasilar nuclei arranged in a palisading pattern. Several mitotic cells were observed. Some epithelial islands displayed cystic degenerative changes and squamous metaplasia. Necrotic tissue with inflammatory cell infiltration was observed at the tumor margins. Immunohistochemically, the neoplastic epithelial islands and mesenchymal components exhibited positivity for pan-cytokeratin and vimentin, respectively. Based on these features, the case was diagnosed as follicular ameloblastoma in an SD rat.
PubMed: 38283374
DOI: 10.1293/tox.2023-0072 -
Molecular Medicine Reports Mar 2024Keratoameloblastoma (KA) and solid variant of odontogenic keratocyst (SOKC) are rare odontogenic lesions, and their relationship and differences are unclear. The present...
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 26‑year‑old 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 Ki‑67 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.
Topics: Male; Humans; Adult; Ameloblastoma; Odontogenic Cysts; Mutation; Biopsy; Bone and Bones
PubMed: 38275130
DOI: 10.3892/mmr.2024.13168 -
International Journal of Clinical... Nov 2023Unicystic ameloblastoma is a rare, benign, locally invasive odontogenic neoplasm of young age that shows clinical, radiographic, or gross features of an odontogenic cyst...
BACKGROUND
Unicystic ameloblastoma is a rare, benign, locally invasive odontogenic neoplasm of young age that shows clinical, radiographic, or gross features of an odontogenic cyst but histologically shows typical ameloblastomatous epithelium lining part of the cyst cavity, with or without luminal and/or mural tumor growth.
AIM
To report a case of an asymptomatic unicystic ameloblastoma in a 12-year-old child, along with its management and follow-up.
CASE DESCRIPTION
A 12-year-old boy presented with swelling with respect to the left body of the mandible. The orthopantomogram (OPG) and computed tomography scan revealed a large unilocular radiolucency in the left mandible associated with the primary second mandibular molar. Complete enucleation of the cyst and extraction of the associated primary teeth and underlying permanent teeth were done under general anesthesia. Carnoy's solution was applied in the bone cavity for 3 minutes with cotton applicators. Postoperative healing was uneventful. Prosthetic rehabilitation was done during the follow-up period.
CONCLUSION
Unicystic ameloblastoma is rarely seen in younger children, so a pediatric dentist must be cautious while diagnosing an intraoral swelling. Timely intervention and conservative surgical treatment, along with a proper follow-up, improved the treatment outcome and prevented potential complications in the future.
CLINICAL SIGNIFICANCE
This report highlights the salient features of unicystic ameloblastoma to be able to accurately diagnose and manage the lesion.
HOW TO CITE THIS ARTICLE
Peter J, Emmatty TB, Jose B, Unicystic Ameloblastoma Associated with Primary Mandibular Second Molar: A Case Report. Int J Clin Pediatr Dent 2023;16(S-3):S335-S338.
PubMed: 38268624
DOI: 10.5005/jp-journals-10005-2701 -
Clinical Case Reports Jan 2024We present a case of recurring ameloblastoma in soft tissue, for which we have estimated the growth rate of the lesion. This information could help clinicians to...
CLINICAL KEY MESSAGE
We present a case of recurring ameloblastoma in soft tissue, for which we have estimated the growth rate of the lesion. This information could help clinicians to establish follow-up protocols for the early diagnosis of recurrent ameloblastomas.
ABSTRACT
In the present paper, we present a case of recurring ameloblastoma in soft tissue, for which we have estimated the growth rate of the lesion. The area of the whole resected specimen was measured using the ImageJ guide for complex object area. After dividing the area of the recurrent tumor by the number of years during the follow-up, we found that the lesion growth rate was 5.3 cm per year. Although further studies are still necessary in the literature to assess the growth rate of ameloblastoma, the present report shows a different methodology to estimate it. This information could help clinicians to establish follow-up protocols for the early diagnosis of recurrent ameloblastomas.
PubMed: 38259867
DOI: 10.1002/ccr3.8444 -
Medicina (Kaunas, Lithuania) Dec 2023Ameloblastoma is a benign epithelial tumor that has aggressive, destructive and unlimited growth potential, having the capacity for recurrence and malignant...
Ameloblastoma is a benign epithelial tumor that has aggressive, destructive and unlimited growth potential, having the capacity for recurrence and malignant transformation. Regarding the symptoms and clinical signs, the presentation of ameloblastoma is poor. In children and young people, ameloblastoma can be difficult to diagnose, because it mimics other benign lesions. Its diagnosis requires a combination of imaging data, histopathological analysis and molecular tests. The methods of treatment consist of radical surgery (segmental resection) and conservative treatments (enucleation with bone curettage). The particularity of the presented case is represented by the late request for medical consultation, a direct consequence of the measures implemented to prevent and control the spread of COVID-19.
Topics: Child; Female; Humans; Adolescent; Ameloblastoma; Mandible; Aggression; COVID-19; Conservative Treatment
PubMed: 38256328
DOI: 10.3390/medicina60010066 -
International Journal of Molecular... Jan 2024Craniopharyngiomas present unique challenges in surgical management due to their proximity to critical neurovascular structures. This systematic review investigates... (Review)
Review
Craniopharyngiomas present unique challenges in surgical management due to their proximity to critical neurovascular structures. This systematic review investigates genetic and immunological markers as potential targets for therapy in craniopharyngiomas, assessing their involvement in tumorigenesis, and their influence on prognosis and treatment strategies. The systematic review adhered to PRISMA guidelines, with a thorough literature search conducted on PubMed, Ovid MED-LINE, and Ovid EMBASE. Employing MeSH terms and Boolean operators, the search focused on craniopharyngiomas, targeted or molecular therapy, and clinical outcomes or adverse events. Inclusion criteria encompassed English language studies, clinical trials (randomized or non-randomized), and investigations into adamantinomatous or papillary craniopharyngiomas. Targeted therapies, either standalone or combined with chemotherapy and/or radiotherapy, were examined if they included clinical outcomes or adverse event analysis. Primary outcomes assessed disease response through follow-up MRI scans, categorizing responses as follows: complete response (CR), near-complete response (NCR), partial response, and stable or progressive disease based on lesion regression percentages. Secondary outcomes included treatment type and duration, as well as adverse events. A total of 891 papers were initially identified, of which 26 studies spanning from 2000 to 2023 were finally included in the review. Two tables highlighted adamantinomatous and papillary craniopharyngiomas, encompassing 7 and 19 studies, respectively. For adamantinomatous craniopharyngiomas, Interferon-2α was the predominant targeted therapy (29%), whereas dabrafenib took precedence (70%) for papillary craniopharyngiomas. Treatment durations varied, ranging from 1.7 to 28 months. Positive responses, including CR or NCR, were observed in both types of craniopharyngiomas (29% CR for adamantinomatous; 32% CR for papillary). Adverse events, such as constitutional symptoms and skin changes, were reported, emphasizing the need for vigilant monitoring and personalized management to enhance treatment tolerability. Overall, the data highlighted a diverse landscape of targeted therapies with encouraging responses and manageable adverse events, underscoring the importance of ongoing research and individualized patient care in the exploration of treatment options for craniopharyngiomas. In the realm of targeted therapies for craniopharyngiomas, tocilizumab and dabrafenib emerged as prominent choices for adamantinomatous and papillary cases, respectively. While adverse events were common, their manageable nature underscored the importance of vigilant monitoring and personalized management. Acknowledging limitations, future research should prioritize larger, well-designed clinical trials and standardized treatment protocols to enhance our understanding of the impact of targeted therapies on craniopharyngioma patients.
Topics: Humans; Ameloblastoma; Craniopharyngioma; Imidazoles; Oximes; Pituitary Neoplasms
PubMed: 38255797
DOI: 10.3390/ijms25020723 -
Maxillofacial Plastic and... Jan 2024One-stage jaw reconstruction with fibular flap and prosthetic rehabilitation restores bony and dental continuity simultaneously. It was also called as "jaw-in-a-day...
BACKGROUND
One-stage jaw reconstruction with fibular flap and prosthetic rehabilitation restores bony and dental continuity simultaneously. It was also called as "jaw-in-a-day (JIAD)" technique. However, bone volume and height of fibular flap may be insufficient for dental implant insertion. The provision of a considerable amount of bone makes an iliac flap the ideal choice in these cases. We present the first case report to document the use of one-stage jaw reconstruction and prosthetic rehabilitation with the iliac flap.
CASE PRESENTATION
We modified the conventional JIAD workflow to make it suitable for iliac flap. Two cases were presented who both underwent segmental mandibulectomy for ameloblastoma. Virtual surgical planning was performed in all cases. The iliac crest was positioned upward to provide cortical bone for achieving primary stability of dental implants. Similar to the "all-on-4" procedure, the iliac bone was placed 12 to 15 mm below the occlusal plane to create adequate space for the implant-retained prosthesis. Immediate implant-based dental rehabilitation was performed at same stage. The surgery was successful in all cases without any short-term complications. In the first postoperative week, patients were given a liquid diet through a nasal feeding tube. The liquid diet is advised until 1 month after the surgery. Thereafter, a soft diet is recommended. Patients were advised to resume routine mastication and normal diet 3 months after the surgery. Peri-implantitis occurred in one patient, and additional gingival graft was required. Postoperative function and esthetics were satisfactory at the last follow-up visit.
CONCLUSIONS
One-stage jaw reconstruction and prosthetic rehabilitation with the iliac flap are safe and useful for restoring postoperative function and esthetics. It should be used in more cases with a longer follow-up in further studies.
PubMed: 38231325
DOI: 10.1186/s40902-024-00413-0 -
Materials Today. Bio Feb 2024Stromal cells are key components of the tumour microenvironment (TME) and their incorporation into 3D engineered tumour-stroma models is essential for tumour mimicry. By...
Stromal cells are key components of the tumour microenvironment (TME) and their incorporation into 3D engineered tumour-stroma models is essential for tumour mimicry. By engineering tumouroids with distinct tumour and stromal compartments, it has been possible to identify how gene expression of tumour cells is altered and influenced by the presence of different stromal cells. Ameloblastoma is a benign epithelial tumour of the jawbone. In engineered, multi-compartment tumouroids spatial transcriptomics revealed an upregulation of oncogenes in the ameloblastoma transcriptome where osteoblasts were present in the stromal compartment (bone stroma). Where a gingival fibroblast stroma was engineered, the ameloblastoma tumour transcriptome revealed increased matrix remodelling genes. This study provides evidence to show the stromal-specific effect on tumour behaviour and illustrates the importance of engineering biologically relevant stroma for engineered tumour models. Our novel results show that an engineered fibroblast stroma causes the upregulation of matrix remodelling genes in ameloblastoma which directly correlates to measured invasion in the model. In contrast the presence of a bone stroma increases the expression of oncogenes by ameloblastoma cells.
PubMed: 38226014
DOI: 10.1016/j.mtbio.2023.100923 -
BMC Oral Health Jan 2024Ameloblastoma, a common benign tumor found in the jaw bone, necessitates accurate localization and segmentation for effective diagnosis and treatment. However, the...
BACKGROUND
Ameloblastoma, a common benign tumor found in the jaw bone, necessitates accurate localization and segmentation for effective diagnosis and treatment. However, the traditional manual segmentation method is plagued with inefficiencies and drawbacks. Hence, the implementation of an AI-based automatic segmentation approach is crucial to enhance clinical diagnosis and treatment procedures.
METHODS
We collected CT images from 79 patients diagnosed with ameloblastoma and employed a deep learning neural network model for training and testing purposes. Specifically, we utilized the Mask R-CNN neural network structure and implemented image preprocessing and enhancement techniques. During the testing phase, cross-validation methods were employed for evaluation, and the experimental results were verified using an external validation set. Finally, we obtained an additional dataset comprising 200 CT images of ameloblastoma from a different dental center to evaluate the model's generalization performance.
RESULTS
During extensive testing and evaluation, our model successfully demonstrated the capability to automatically segment ameloblastoma. The DICE index achieved an impressive value of 0.874. Moreover, when the IoU threshold ranged from 0.5 to 0.95, the model's AP was 0.741. For a specific IoU threshold of 0.5, the model achieved an AP of 0.914, and for another IoU threshold of 0.75, the AP was 0.826. Our validation using external data confirms the model's strong generalization performance.
CONCLUSION
In this study, we successfully applied a neural network model based on deep learning that effectively performs automatic segmentation of ameloblastoma. The proposed method offers notable advantages in terms of efficiency, accuracy, and speed, rendering it a promising tool for clinical diagnosis and treatment.
Topics: Humans; Ameloblastoma; Deep Learning; Research Design; Tomography, X-Ray Computed
PubMed: 38195496
DOI: 10.1186/s12903-023-03587-7