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Head and Neck Pathology Mar 2022The 5 edition of the World Health Organization (WHO) Classification of Head and Neck Tumours (2022) comes out only five years after the previous edition, however it...
The 5 edition of the World Health Organization (WHO) Classification of Head and Neck Tumours (2022) comes out only five years after the previous edition, however it presents important updates that run in parallel with the rapid progression involving the increasingly sophisticated molecular investigation and its interpretation, some of which already have therapy-related impact. This manuscript provides an overview of the leading changes introduced in the classification of Odontogenic and Maxillofacial Bone Tumours that encompasses cysts of the jaws, odontogenic tumours, giant cell lesions and bone cysts, and bone and cartilage tumours. This is the first edition that Essential and Desirable Diagnostic Features were added for each entity, so that the most important clinical, microscopic and/or radiologic features were encapsulated and briefly highlighted. Surgical ciliated cyst was added to the group of odontogenic cysts, adenoid ameloblastoma was a newly recognized benign epithelial odontogenic tumour, and segmental odontomaxillary dysplasia was introduced in the group of fibro-osseous tumours and dysplasia. In addition, rhabdomyosarcoma with TFCP2 rearrangement, was introduced into the group of malignant jawbone tumours. The unique genetic aberrations distinguish it from other types of rhabdomyosarcomas. On the other hand, melanotic neuroectodermal tumour of infancy and osteoid osteoma were deleted from the benign bone and cartilageneous tumours, as was the hematolymphoid tumour of solitary plasmacytoma of bone. We systematically reviewed each entity in this chapter and provided important updated findings for selected topics that can further aid in the diagnostic process for challenging cases, broaden insights on the logic of the present classification, and finally, emphasize the potential that some of the molecular results may have in the near future to set new treatment approaches.
Topics: Bone Neoplasms; DNA-Binding Proteins; Head and Neck Neoplasms; Humans; Odontogenic Cysts; Odontogenic Tumors; Transcription Factors; World Health Organization
PubMed: 35312978
DOI: 10.1007/s12105-021-01404-7 -
The British Journal of Oral &... Jun 2023The odontogenic keratocyst (OKC) is a common cystic lesion in the jaw. Its management, however, is highly debated with no consensus on the best treatment option.... (Review)
Review
The odontogenic keratocyst (OKC) is a common cystic lesion in the jaw. Its management, however, is highly debated with no consensus on the best treatment option. Clinicians base their approach on treatment efficacy and associated morbidity. Management often consists of enucleation with peripheral ostectomy and adjunctive therapy to prevent recurrence. The aim of our systematic review was to evaluate the safety and efficacy of these different modalities. Embase, Medline, and Cochrane were searched according to the PRISMA guidelines for articles that presented non-syndromic patients with histopathologically confirmed OKC treated with 5-fluorouracil (5FU), Carnoy's solution (CS), or modified Carnoy's solution (MCS) as adjunctive therapy after enucleation and peripheral ostectomy. The outcomes of interest were safety (measured as adverse events) and efficacy (expressed as recurrence). Risk of bias was evaluated using the Newcastle-Ottawa scale. Four studies were included and 62 patients were evaluated. The results show that recurrence occurred only in patients treated with MCS. Reported adverse events were mostly limited to paraesthesia that could be permanent (in the CS and MCS treatment groups) or transient (across all adjunctive therapies). With the prohibition of CS, both MCS and 5FU are promising replacement adjunctive therapies. From a safety and efficacy perspective we consider 5FU, which was associated with the lowest recurrence and fewest adverse events, to be the most viable option. More high-evidence prospective studies, such as randomised controlled trials, with a longer follow-up period are necessary to draw definite conclusions.
Topics: Humans; Prospective Studies; Odontogenic Cysts; Acetic Acid; Chloroform; Odontogenic Tumors
PubMed: 37248124
DOI: 10.1016/j.bjoms.2023.04.006 -
Sultan Qaboos University Medical Journal Aug 2023Papilliferous keratoameloblastoma (PKA) is a rare entity, and not much is known about its clinicodemographic features or biological nature. This review aimed to provide... (Review)
Review
Papilliferous keratoameloblastoma (PKA) is a rare entity, and not much is known about its clinicodemographic features or biological nature. This review aimed to provide clarity regarding the characterisation of the demographic, clinical, radiological and histopathological features of PKA. Case reports of PKA were identified through a systematic search across multiple databases. The search yielded a total of 10 cases, half of which were of Indian origin. All the cases invariably occurred in the mandibular posterior region and involved the right side; only one case primarily involved the left side of the mandible. PKA should be considered a variant of the conventional ameloblastoma that is towards the more aggressive end of the spectrum. It tends to occur in older individuals (in their fifth decade or older), with a marked propensity to occur in the right mandibular posterior region. Surgical resection with diligent follow-up is warranted in the treatment of PKA.
Topics: Humans; Aged; Ameloblastoma; Mandible; Thorax
PubMed: 37655071
DOI: 10.18295/squmj.5.2023.021 -
Sultan Qaboos University Medical Journal Aug 2022This article aimed to collectively present the demographic, clinical, radiographic and histopathological features as well as the treatment performed along with its... (Review)
Review
This article aimed to collectively present the demographic, clinical, radiographic and histopathological features as well as the treatment performed along with its outcome for all the cases of adenoid ameloblastoma with dentinoid (AAD) reported in scientific literature till date. Ameloblastoma and adenomatoid odontogenic tumours are the most common odontogenic neoplasms. However, AAD, a hybrid variant of the two lesions, is found to be extremely rare. The lesion comprises of characteristic histopathological features of ameloblastoma and adenomatoid odontogenic tumour and shares certain clinical characteristics with either of the entities. AAD may be considered to be present at the more aggressive end of spectrum of benign odontogenic neoplasms. Owing to the frequent tendency of the lesions to be underdiagnosed, careful histopathological screening of submitted biopsies is warranted. With the increase in number of reported cases in the recent years, it is likely to be included as a separate entity in the upcoming World Health Organization classification.
Topics: Adenoids; Ameloblastoma; Biopsy; Humans; Odontogenic Tumors
PubMed: 36072074
DOI: 10.18295/squmj.9.2021.127 -
Head and Neck Pathology Sep 2022Clear cell Odontogenic Carcinoma (CCOC) is an uncommon malignant odontogenic tumor (MOT). It is the fifth most common MOT. A systematic review is presented of reported... (Review)
Review
Clear cell Odontogenic Carcinoma (CCOC) is an uncommon malignant odontogenic tumor (MOT). It is the fifth most common MOT. A systematic review is presented of reported cases, case series and retrospective studies of CCOC, to determine trends in presentation, diagnostic features, treatment, and patient outcome. Searches of detailed databases were carried out to identify papers reporting CCOC. The variables were demographics, patient symptoms, tumor location, histopathological findings, immunohistochemical studies, treatment, follow-up, and recurrence. 117 cases were identified; CCOC was most frequently seen in mature females 65% (n = 76). The total average age was 55.4 with a range from 17 to 89 years, for females 56.4 and males 53.6 years. The mean size was 3.41 cm. The most common location was in the mandibular body 36.2% (n = 42), followed by the anterior mandible 23.3% (n = 27). The most common clinical presentation was a swelling 80.4% (n = 74), and the main symptom was pain 41.3% (n = 31), followed by painless lesion 24% (n = 18). The most common Immunohistochemistry positive expression was CK19, EMA, and CEA, and for special staining periodic acid Shiff (PAS); 97% of cases were treated surgically. The average follow-up was 30.3 months, and recurrence was reported in 52.4% of the cases. Conclusion: CCOC shows a strong predilection for the body and anterior mandible, and females are more frequently affected. CCOCs can be painful and the principle clinical sign is swelling, CCOCs can metastasize, and the prognosis is fair.
Topics: Adolescent; Adult; Aged; Aged, 80 and over; Carcinoma; Female; Humans; Male; Mandible; Middle Aged; Mouth Neoplasms; Odontogenic Tumors; Retrospective Studies; Young Adult
PubMed: 34618301
DOI: 10.1007/s12105-021-01383-9 -
Genes Aug 2023Stem cells have been associated with self-renewing and plasticity and have been investigated in various odontogenic lesions in association with their pathogenesis and... (Review)
Review
BACKGROUND
Stem cells have been associated with self-renewing and plasticity and have been investigated in various odontogenic lesions in association with their pathogenesis and biological behavior. We aim to provide a systematic review of stem cell markers' expression in odontogenic tumors and cysts.
METHODS
The literature was searched through the MEDLINE/PubMed, EMBASE via OVID, Web of Science, and CINHAL via EBSCO databases for original studies evaluating stem cell markers' expression in different odontogenic tumors/cysts, or an odontogenic disease group and a control group. The studies' risk of bias (RoB) was assessed via a Joanna Briggs Institute Critical Appraisal Tool. Meta-analysis was conducted for markers evaluated in the same pair of odontogenic tumors/cysts in at least two studies.
RESULTS
29 studies reported the expression of stem cell markers, e.g., SOX2, OCT4, NANOG, CD44, ALDH1, BMI1, and CD105, in various odontogenic lesions, through immunohistochemistry/immunofluorescence, polymerase chain reaction, flow cytometry, microarrays, and RNA-sequencing. Low, moderate, and high RoBs were observed in seven, nine, and thirteen studies, respectively. Meta-analysis revealed a remarkable discriminative ability of SOX2 for ameloblastic carcinomas or odontogenic keratocysts over ameloblastomas.
CONCLUSION
Stem cells might be linked to the pathogenesis and clinical behavior of odontogenic pathologies and represent a potential target for future individualized therapies.
PubMed: 37761874
DOI: 10.3390/genes14091735 -
Medicina Oral, Patologia Oral Y Cirugia... May 2020The primordial odontogenic tumor (POT) is a recently described benign entity with histopathological and immunohistochemical features suggesting its origin during early...
BACKGROUND
The primordial odontogenic tumor (POT) is a recently described benign entity with histopathological and immunohistochemical features suggesting its origin during early odontogenesis.
AIM
To integrate the available data published on POT into a comprehensive analysis to better define its clinicopathological and molecular features.
MATERIAL AND METHODS
An electronic systematic review was performed up to September 2019 in multiple databases.
RESULTS
A total of 13 publications were included, representing 16 reported cases and 3 molecular studies. The mean age of the affected patients was 11.6 years (range 2-19), with a slight predominance in males (56.25%). The posterior mandible was the main location (87.5%), with only two cases affecting the posterior maxilla. All cases appeared as a radiolucent lesion in close relationship to an unerupted tooth. Recurrences have not been reported to date. Microscopically, POT comprises fibromyxoid tissue with variable cellularity surrounded by a cuboidal to columnar odontogenic epithelium but without unequivocal dental hard tissue formation. A delicate fibrous capsule surrounds (at least partially) the tumor. The epithelial component shows immunohistochemical positivity for amelogenin, CK19, and CK14, and variable expression of Glut-1, Galectin-3 and Caveolin-1, Vimentin, p-53, PITX2, Bcl-2, Bax and Survivin; the mesenchymal tissue is positive for Vimentin, CD90, p-53, PITX2, Bcl-2, Bax, and Survivin, and the subepithelial region exhibits the strong expression of Syndecan-1 and CD34. The Ki-67 index is low (<5%). The negative or weak expression of dentinogenesis-associated genes could explain the inhibition of dentin and subsequent enamel formation in this neoplasm.
CONCLUSION
POT is an entity with a well-defined clinicopathological, immunohistochemical and molecular profile that must be properly diagnosed and differentiated from other odontogenic lesions and treated consequently.
Topics: Adolescent; Adult; Child; Child, Preschool; Epithelium; Humans; Male; Mandible; Neoplasm Recurrence, Local; Odontogenesis; Odontogenic Tumors; Young Adult
PubMed: 32040459
DOI: 10.4317/medoral.23432 -
Annals of Maxillofacial Surgery 2023Odontogenic cysts have the potential to transform into neoplasms. However, the characteristics of those which transformed to neoplastic tissues have not been well... (Review)
Review
BACKGROUND
Odontogenic cysts have the potential to transform into neoplasms. However, the characteristics of those which transformed to neoplastic tissues have not been well described and the exact causes of that phenomenon are not yet clear.
OBJECTIVES
This study aims to describe characteristics of odontogenic cysts that transformed into neoplasms and to look for their potential etiologies.
DATA SOURCES
English-written studies indexed in PubMed, Science Direct, and Proquest were assessed using keywords verified by Medical Subject Headings: 'Odontogenic Cyst' and 'Neoplastic Cell Transformation'.
STUDY ELIGIBILITY CRITERIA
Preferred Reporting Items for Systematic Review and Meta-analysis (PRISMA) guidelines were used as guidance.
PARTICIPANTS
Following steps in PRISMA guidelines, 19 articles were fully reviewed (three case series and 16 case reports) with 27 subjects of 16 males (59%) and 11 females (41%) from 15 to 86 years old.
RESULTS
Cystic origins were eight dentigerous cysts, four odontogenic keratocysts, two residual cysts, one radicular cyst, one calcifying odontogenic cyst, one follicular cyst, one glandular odontogenic cyst, and nine unspecified odontogenic cysts that transformed to ameloblastoma (3 cases) and carcinoma (24 cases).
LIMITATIONS
Neoplastic transformations of odontogenic cysts arose from epithelial remnants of inadequate odontogenic cyst removal and chronic inflammation due to infection. However, the exact causes of their transformations remain unclear.
CONCLUSIONS
Therefore, careful removal of odontogenic cysts and regular postoperative follow-ups are key to prevent recurrence and neoplastic transformation. Future studies are needed to investigate potential causes of neoplastic transformation of odontogenic cysts.
PubMed: 37711539
DOI: 10.4103/ams.ams_226_22 -
Frontiers in Oral Health 2021To perform a comprehensive and systematic critical appraisal of the genetic alterations reported to be present in adenomatoid odontogenic tumor (AOT) compared to...
To perform a comprehensive and systematic critical appraisal of the genetic alterations reported to be present in adenomatoid odontogenic tumor (AOT) compared to ameloblastoma (AM), to aid in the understanding in their development and different behavior. An electronic search was conducted in PubMed, Scopus, and Web of Science during March 2021. Eligibility criteria included publications on humans which included genetic analysis of AOT or AM. A total of 43 articles reporting 59 AOTs and 680 AMs were included. Different genomic techniques were used, including whole-exome sequencing, direct sequencing, targeted next-generation sequencing panels and TaqMan allele-specific qPCR. Somatic mutations affecting were identified in 75.9% of all AOTs, mainly G12V; whereas a 71% of the AMs harbored mutations, mainly V600E. The available genetic data reports that AOTs and AM harbor somatic mutations in well-known oncogenes, being KRAS G12V/R and BRAFV600E mutations the most common, respectively. The relatively high frequency of ameloblastoma compared to other odontogenic tumors, such as AOT, has facilitated the performance of different sequencing techniques, allowing the discovery of different mutational signatures. On the contrary, the low frequency of AOTs is an important limitation for this. The number of studies that have a assessed the genetic landscape of AOT is still very limited, not providing enough evidence to draw a conclusion regarding the relationship between the genomic alterations and its clinical behavior. Thus, the presence of other mutational signatures with clinical impact, co-occurring with background mutations or in wild-type cases, cannot be ruled out. Since BRAF and RAS are in the same MAPK pathway, it is interesting that ameloblastomas, frequently associated with BRAFV600E mutation have aggressive clinical behavior, but in contrast, AOTs, frequently associated with RAS mutations have indolent behavior. Functional studies might be required to solve this question.
PubMed: 35048068
DOI: 10.3389/froh.2021.767474 -
Journal of Dental Research, Dental... 2021Ki-67 is a marker of cell proliferation, used as an important diagnostic marker in the pathologic differentiation of various lesions. It is also relevant for developing... (Review)
Review
Ki-67 is a marker of cell proliferation, used as an important diagnostic marker in the pathologic differentiation of various lesions. It is also relevant for developing targeted molecular therapies. We carried out a systematic review to assess the Ki-67 labeling index (LI) in odontogenic cysts and tumors. Databases were searched, including PubMed (MEDLINE), Scopus, CINHAL, PsycoInfo, the Cochrane Library, and Proquest. The meta-analysis was carried out based on the data of 608 lesions. When a 5% cut-off point was set, ki-67 LI of all benign odontogenic tumors dropped below this point. All the malignant tumors demonstrated an LI of over 15.3%; a significantly higher Ki-67 LI in malignant odontogenic lesions (17.59±2.80) was observed. Among benign tumors, the largest and the smallest Ki-67 LIs were seen in ameloblastoma (4.39±0.47) and adenomatoid odontogenic tumor (0.91±1.71). The mean values of Ki-67 LI in tumors and cysts were 4.23 (0.38) and 1.04 (0.07), respectively. Among odontogenic cysts, the highest Ki-67 LI was found in odontogenic keratocyst (OKC) (3.58±0.51), and the lowest in the radicular cyst (1.29±0.62%). Ki-67 LIs in all odontogenic cysts were <3%, except for OKC. This controversial lesion seems to have a profile more similar to a tumor, and a treatment plan similar to tumors might be suggested. We found that odontogenic lesions have diverse proliferative activities that help differentiate between various lesions and suggest therapeutic plans.
PubMed: 33927844
DOI: 10.34172/joddd.2021.012