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Frontiers in Oncology 2024Since 1964, there has been a scarcity of reported cases of primary ameloblastoma (AM) or ameloblastic carcinoma (AMCa) of the skull. The clinical presentation and...
BACKGROUND
Since 1964, there has been a scarcity of reported cases of primary ameloblastoma (AM) or ameloblastic carcinoma (AMCa) of the skull. The clinical presentation and distinctive features of this uncommon condition at specific anatomical sites remain unclear. We report a case of malignant transformation of a primary AM of the skull situated in the frontal-temporal-parietal region and highlight its similarities to other cases reported in the literature.
CLINICAL PRESENTATION
A 53-year-old female patient presented with a 20-day history of headaches and bilateral lower limb weakness for 10 days. Physical examination revealed slow and unsteady gait. An occupying lesion was observed in the right frontal-temporal-parietal region of the skull on the Cranial imaging. A right cranial bone tumor margin expansion resection was performed. The patient's motor functions recovered normally after surgery. Postoperative imaging examinations showed10 tumor resection. Follow-up imaging examinations showed tumor recurrence. The patient underwent resection of the recurrent tumor. Postoperative pathological analysis revealed malignant transformation of the AM.Follow-up imaging examinations showed tumor recurrence again. The patient was admitted for stereotactic radiotherapy. Follow-up imaging examinations demonstrated no evidence of tumor recurrence and subsequent chest CT revealed no signs of metastasis.
CONCLUSION
Primary AM or AMCa of the skull is increasingly being described in the literature, but detailed reports on the malignant transformation of primary AM of the skull are lacking. The pathogenesis of this condition remains unclear. Aggressive treatment and close follow-up may be crucial for preventing disease recurrence and malignant transformation.
PubMed: 38577340
DOI: 10.3389/fonc.2024.1365625 -
Journal of Dentistry (Shiraz, Iran) Mar 2024Periapical cyst and granuloma are inflammatory endodontic lesions. Periapical granuloma usually heals spontaneously after endodontic treatment; however, periapical cyst...
STATEMENT OF THE PROBLEM
Periapical cyst and granuloma are inflammatory endodontic lesions. Periapical granuloma usually heals spontaneously after endodontic treatment; however, periapical cyst mostly needs to be removed via surgical approaches. Although some clinicians believe that microscopic examination of periapical lesions is unnecessary, it is proved that some of them has non-endodontic nature that need critical consideration.
PURPOSE
The purpose of this study was to assess the disagreement between clinico-radiographic and microscopic diagnosis of periapical cysts and granulomas in a major center of oral pathology service in Iran.
MATERIALS AND METHOD
In this retrospective, descriptive cross-sectional study, the archives of the oral and maxillofacial pathology department of Shahid Beheshti University of Medical Sciences served as the source of the material during an 18-year-period for this retrospective, descriptive cross-sectional study. The reports of all patients whose initial clinical diagnosis was a periapical cyst/granuloma were extracted.
RESULTS
In the present study, 474 cases were diagnosed with a periapical cyst/granuloma clinico-radiographically, of which 61 cases (12.86%) received a microscopic diagnosis of a non-endodontic pathology. The most frequent lesion was odontogenic keratocyst (n= 12, 19.67%) followed by infected odontogenic cyst (n= 12, 19.67%). About 21.31% of diagnoses were non-cystic lesions and 4.9% were malignancies. The most odontogenic tumors that were diagnosed as periapical cyst/granuloma in clinico-radiography were the ameloblastoma variants (n= 4, 6.55%).
CONCLUSION
A wide variety of microscopic diagnoses, including aggressive lesions such as ameloblastoma, as well as other malignant lesions was noted in this study. These misdiagnoses can lead to an inappropriate treatment plan. It is important to microscopically examine all lesions removed from the jaw.
PubMed: 38544772
DOI: 10.30476/dentjods.2023.96819.1967 -
BMC Oral Health Mar 2024Ameloblastoma (AM) is the most common benign odontogenic tumor, which is more often detected in the mandible than maxilla, especially the mandibular body and mandibular...
BACKGROUND
Ameloblastoma (AM) is the most common benign odontogenic tumor, which is more often detected in the mandible than maxilla, especially the mandibular body and mandibular angle. Pediatric AM is a rare disease, especially in patients aged 10 and younger. Compared with the mainstream osteotomy and reconstructive surgery for adult ameloblastoma, there is more room for discussion in the treatment of pediatric ameloblastoma. The postoperative functional and psychological influence can not be ignored. Especially for children in the period of growth and development, an osteotomy is often challenging to be accepted by their parents. We report two patients with ameloblastoma under 10 years old who are treated with curettage and fenestration, which is a beneficial method for children with ameloblastoma.
CASE PRESENTATION
We present two cases of classic ameloblastoma in children. We describe in detail the patients' characteristics, treatment processes, and follow-up result. The bone formation and reconstruction in the lesion area after fenestration decompression and curettage are recorded at every clinic review. The surgical details and principles of curettage and decompression are also described and discussed. The two patients have good bone shape recovery and no recurrence.
CONCLUSIONS
Children are in the growth and development period and possess an extremely strong ability of bone formation and reconstruction. Based on the principles of minimally invasive and functional preservation, we believe that curettage combined with decompression can be the first choice for treating AM in children, especially for mandibular lesions.
Topics: Child; Humans; Ameloblastoma; Curettage; Decompression; Mandible; Mandibular Neoplasms
PubMed: 38519948
DOI: 10.1186/s12903-024-04126-8 -
The American Journal of Case Reports Mar 2024BACKGROUND Adamantinoma is a rare low-grade malignant bone tumor, usually found in the tibial diaphysis and metaphysis, with histological similarities to mandibular...
BACKGROUND Adamantinoma is a rare low-grade malignant bone tumor, usually found in the tibial diaphysis and metaphysis, with histological similarities to mandibular ameloblastoma. The most effective treatment of recurrent adamantinoma is not yet clear. This report is of a 22-year-old woman with recurrent tibial adamantinoma treated with the tyrosine kinase inhibitor pazopanib. CASE REPORT We report the case of a 22-year-old woman who was referred to our center for a suspicious bone lesion in the right tibia. Bone biopsy findings were consistent with an adamantinoma. En bloc resection was completed successfully, with no postoperative complications. Five years later, a positive emission tomography scan revealed mildly increased tracer uptake near the area of the previous lesion and in the right inguinal lymph node. Biopsies of the lesion and inguinal lymph node confirmed recurrence of the adamantinoma. Due to abdominal and pelvic metastasis, the patient underwent surgical debulking, along with an appendectomy, right salpingo-oophorectomy, intraoperative radiation therapy, and hyperthermic intraperitoneal chemotherapy. Subsequently, the patient was placed on pazopanib for 4 months; however, her tumor continued to worsen after 4 months of chemotherapy. Currently, the patient is receiving gemcitabine and docetaxel as second-line medical therapy. CONCLUSIONS This report showed that pazopanib as standalone treatment does not appear to have promising role on patient outcomes. To the best of our knowledge, this is the second report of pazopanib in the treatment of adamantinoma.
Topics: Female; Humans; Young Adult; Adamantinoma; Ameloblastoma; Bone Neoplasms; Indazoles; Pyrimidines; Sulfonamides; Tibia
PubMed: 38486378
DOI: 10.12659/AJCR.941248 -
International Journal of Oral Science Feb 2024Ameloblastoma is a benign tumor characterized by locally invasive phenotypes, leading to facial bone destruction and a high recurrence rate. However, the mechanisms...
Ameloblastoma is a benign tumor characterized by locally invasive phenotypes, leading to facial bone destruction and a high recurrence rate. However, the mechanisms governing tumor initiation and recurrence are poorly understood. Here, we uncovered cellular landscapes and mechanisms that underlie tumor recurrence in ameloblastoma at single-cell resolution. Our results revealed that ameloblastoma exhibits five tumor subpopulations varying with respect to immune response (IR), bone remodeling (BR), tooth development (TD), epithelial development (ED), and cell cycle (CC) signatures. Of note, we found that CC ameloblastoma cells were endowed with stemness and contributed to tumor recurrence, which was dominated by the EZH2-mediated program. Targeting EZH2 effectively eliminated CC ameloblastoma cells and inhibited tumor growth in ameloblastoma patient-derived organoids. These data described the tumor subpopulation and clarified the identity, function, and regulatory mechanism of CC ameloblastoma cells, providing a potential therapeutic target for ameloblastoma.
Topics: Humans; Ameloblastoma; Neoplasm Recurrence, Local; Phenotype; Cell Transformation, Neoplastic; Gene Expression Profiling
PubMed: 38424060
DOI: 10.1038/s41368-024-00281-4 -
Annals of Maxillofacial Surgery 2023Ameloblastic carcinoma is defined as an ameloblastoma in which there is histological evidence of malignancy in primary tumour or recurrent tumour regardless if it has...
RATIONALE
Ameloblastic carcinoma is defined as an ameloblastoma in which there is histological evidence of malignancy in primary tumour or recurrent tumour regardless if it has metastasised or not. It is aggressive in nature.
PATIENT CONCERNS
The patient presented with a painful swelling associated with restricted mouth opening.
DIAGNOSIS
This is the clinical profile of a patient who has presented with a recurrent ameloblastic carcinoma.
TREATMENT
Histological features of the excised specimen resemble ameloblastic carcinoma constituting cytological atypia with tall columnar cells.
OUTCOME
Excision of tumour was done. Reconstruction was done using pectoralis major myocutaneous flap.
TAKE-AWAY LESSONS
Ameloblastic carcinoma is an aggressive tumour and constitutes destruction and distant metastatic spread. Hence, aggressive resection is the choice of treatment along with a long-term follow-up for better quality of life.
PubMed: 38405565
DOI: 10.4103/ams.ams_66_23 -
International Journal of Molecular... Feb 2024Ameloblastoma is a rare tumor but represents the most common odontogenic neoplasm. It is localized in the jaws and, although it is a benign, slow-growing tumor, it has...
Ameloblastoma is a rare tumor but represents the most common odontogenic neoplasm. It is localized in the jaws and, although it is a benign, slow-growing tumor, it has an aggressive local behavior and high recurrence rate. Therefore, alternative treatment options or complementary to surgery have been evaluated, with the most promising one among them being a targeted therapy with the murine sarcoma viral oncogene homologue B (), as in ameloblastoma the activating mutation V600E in BRAF is common. Studies in other tumors have shown that the synchronous inhibition of BRAF and human murine double minute 2 homologue (MDM2 or HDM2) protein is more effective than BRAF monotherapy, particularly in the presence of wild type p53 (WTp53). To investigate the MDM2 protein expression and gene amplification in ameloblastoma, in association with BRAF and p53 expression. Forty-four cases of ameloblastoma fixed in 10% buffered formalin and embedded in paraffin were examined for MDM2 overexpression and BRAF and p53 expression by immunohistochemistry, and for ploidy with fluorescence in situ hybridization. Sixteen of forty-four (36.36%) cases of ameloblastoma showed MDM2 overexpression. Seven of sixteen MDM2-positive ameloblastomas (43.75%) were BRAF positive and fifteen of sixteen MDM2-positive ameloblastomas (93.75%) were p53 negative. All MDM2 overexpressing tumors did not show copy number alterations for MDM2. Overexpression of MDM2 in ameloblastomas is not associated with MDM2 amplification, but most probably with MAPK activation and WTp53 expression. Further verification of those findings could form the basis for the use of MDM2 expression as a marker of MAPK activation in ameloblastomas and the trial of dual BRAF/MDM2 inhibition in the management of MDM2-overexpressing/BRAFV600E-positive/WTp53 ameloblastomas.
Topics: Animals; Humans; Mice; Ameloblastoma; In Situ Hybridization, Fluorescence; Mutation; Proto-Oncogene Proteins B-raf; Proto-Oncogene Proteins c-mdm2; Tumor Suppressor Protein p53
PubMed: 38396916
DOI: 10.3390/ijms25042238 -
Indian Journal of Pathology &... Apr 2024Adenoid ameloblastoma with dentinoid (AAD) is a hybrid odontogenic tumor comprising histopathological presentation of ameloblastoma (AM) and adenomatoid odontogenic...
BACKGROUND
Adenoid ameloblastoma with dentinoid (AAD) is a hybrid odontogenic tumor comprising histopathological presentation of ameloblastoma (AM) and adenomatoid odontogenic tumor (AOT) along with extracellular dentinoid material.
CASE PRESENTATION
A 35-year-old female reported an asymptomatic swelling in the left mandibular posterior region. Histopathological examination revealed composite features of AM with AOT along with dentinoid material, which stained positively with Van Gieson and trichrome stains.
CONCLUSION
The present case report serves to add further to the modicum of literature reports pertaining to AAD, which may gain recognition as a distinct entity in future World Health Organization (WHO) classification of odontogenic tumors.
Topics: Humans; Female; Adult; Ameloblastoma; Odontogenic Tumors; Mandibular Neoplasms; Mandible
PubMed: 38391318
DOI: 10.4103/ijpm.ijpm_186_22 -
Journal of Veterinary Diagnostic... Feb 2024Orofacial masses or swellings are a common presenting complaint in lagomorphs. Similar gross appearances of the masses can complicate clinical interpretation, and...
Orofacial masses or swellings are a common presenting complaint in lagomorphs. Similar gross appearances of the masses can complicate clinical interpretation, and histologic review often provides the final diagnosis. Underlying causes vary from infectious to neoplastic. Although inflammatory changes are most commonly reported, various neoplasms occur, although the prevalence of specific tumor types is relatively unknown. We reviewed retrospectively 120 cases (87.5% biopsy, 12.5% autopsy) of neoplastic and non-neoplastic orofacial masses received from January 2000-February 2023 at 2 institutions: University of Guelph, Canada (Animal Health Laboratory and Department of Pathobiology), and Finn Pathologists, United Kingdom. All final diagnoses were achieved through histologic assessment. We included masses or mass-like swellings from the oral cavity, including the mandible and maxilla, and surrounding skin and soft tissues of the oral cavity and jaw. Submissions included pet and commercial (meat and fur) rabbits. Neoplastic lesions were most common (60%), including trichoblastomas, papillomas, melanocytic neoplasms, sarcomas, round-cell tumors, carcinomas (including squamous cell carcinoma), lipomas, odontogenic neoplasms, polyps, osteoma, neuroma, peripheral keratinizing ameloblastoma, and apocrine adenoma. Inflammatory diagnoses (30%) included abscesses, osteomyelitis, dermatitis, and sialadenitis. Other diagnoses (7%) included cysts, as well as hyperplastic skin and proliferative bone lesions. Three cases had no definitive diagnosis. The importance of histologic assessment in diagnosing orofacial "masses" in rabbits is highlighted, given that the most common diagnostic category overall was neoplasia.
PubMed: 38389410
DOI: 10.1177/10406387241234326 -
Malawi Medical Journal : the Journal of... Dec 2023Odontogenic keratocyst is a benign intraosseous lesion of odontogenic origin which is characterized by its aggressive nature. It is usually present in the mandibular...
Odontogenic keratocyst is a benign intraosseous lesion of odontogenic origin which is characterized by its aggressive nature. It is usually present in the mandibular posterior area, although it can also be found in the maxilla, particularly in the canine region. We discuss a unique example of OKC in the maxillary sinus involving the 27&28 region. Due to comparable clinical signs, this lesion is more prone to be mistaken for other lesions of the maxillary sinus, such as sinusitis or polyps. On the other side, this benign disease has the potential to develop into Ameloblastoma or squamous cell carcinoma. A favorable prognosis thus depends on early identification, precise diagnosis, appropriate treatment, and follow-ups.
Topics: Humans; Animals; Dogs; Maxillary Sinus; Odontogenic Cysts; Carcinoma, Squamous Cell
PubMed: 38362567
DOI: 10.4314/mmj.v35i4.6