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Asian Journal of Surgery Feb 2022
Topics: Adenomatoid Tumor; Fallopian Tube Neoplasms; Fallopian Tubes; Female; Humans
PubMed: 34955345
DOI: 10.1016/j.asjsur.2021.11.045 -
International Journal of Surgery Case... Jul 2022An adenomatoid odontogenic tumor is a rare medical condition. Large tumor (or several) often appears in the maxillae. In a minority of cases, the tumor(s) appear in the...
INTRODUCTION AND IMPORTANCE
An adenomatoid odontogenic tumor is a rare medical condition. Large tumor (or several) often appears in the maxillae. In a minority of cases, the tumor(s) appear in the mandible.
CASE PRESENTATION
We report on a case of a 24-year-old female diagnosed with a mandibular adenomatoid odontogenic tumor, a giant tumor measuring approximately 22 × 25 × 17 cm. The tumor was located on the side of the mandible, causing facial deformity, malnutrition, and hemorrhaging. We assessed the patient's overall condition, carried out a resection of the tumor and mandible from the right condyle to the left mandibular angle, and reconstructed the mandibular defect with a fibula free flap. After the treatment, the patient was followed up for 1 year, with no recurrence detected over this period.
CLINICAL DISCUSSION
Because adenomatoid odontogenic tumors are benign odontogenic lesions, which are painless and slow-growing, most are surgically removed or treated conservatively. However, the above treatment measures cannot be applied in the case of a giant tumor that causes facial deformity, destroys the entire jawbone, and has complications such as hemorrhaging and malnutrition. After the tumor resection, the defect is still significant. Accordingly, reconstruction using a microsurgical bone flap is an effective method instead.
CONCLUSION
Large adenomatoid odontogenic tumors in the mandible are rare, and treatment cannot follow conventional methods. Accordingly, defect reconstruction after tumor resection is essential.
PubMed: 35714392
DOI: 10.1016/j.ijscr.2022.107295 -
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 -
Andrology Sep 2021Ultrasound (US) is the primary modality for the investigation of scrotal pathology, including both intra- and paratesticular abnormalities. (Review)
Review
BACKGROUND
Ultrasound (US) is the primary modality for the investigation of scrotal pathology, including both intra- and paratesticular abnormalities.
OBJECTIVE
To describe the abnormalities of the paratesticular space.
MATERIALS/METHODS
The paratesticular space contains the epididymis, spermatic cord and the tunica vaginalis cavity and is affected by a variety of inflammatory or tumoral entities. Differential diagnosis based on US criteria is frequently problematic, as the findings are non-specific.
RESULTS
Some general rules apply: (i) unlike testicular lesions, extra-testicular entities are usually benign in the adult, (ii) the first steps to accurate diagnosis include careful localization of the lesion and assessment of its consistency (solid or cystic) and (iii) magnetic resonance imaging can be useful for further tissue characterization of lesions suspected to contain fat, but surgical biopsy will often provide the definite diagnosis. Contrast-enhanced ultrasound (CEUS) has been applied with limited experience indicating a narrow role, primarily for the differential diagnosis of echogenic cystic entities and the delineation of a necrotic abscess from a solid neoplasm.
DISCUSSION
The various abnormalities are discussed and illustrated.
CONCLUSION
This manuscript summarizes the literature on paratesticular lesions and the value of US in diagnosis.
Topics: Adult; Contrast Media; Diagnosis, Differential; Epididymis; Genital Diseases, Male; Humans; Male; Scrotum; Spermatic Cord; Testicular Diseases; Testis; Ultrasonography
PubMed: 33864338
DOI: 10.1111/andr.13021 -
Case Reports in Dentistry 2015Adenomatoid Odontogenic Tumor (AOT) is a well-established benign epithelial lesion of odontogenic origin. Rightfully called "the master of disguise," this lesion has...
Adenomatoid Odontogenic Tumor (AOT) is a well-established benign epithelial lesion of odontogenic origin. Rightfully called "the master of disguise," this lesion has been known for its varied clinical and histoarchitectural patterns. Not only does AOT predominantly present radiologically as a unilocular cystic lesion enclosing the unerupted tooth (which is commonly mistaken as a dentigerous cyst) but the lesion also presents rarely with a cystic component histopathologically. We present one such unusual case of cystic AOT associated with an impacted canine, mimicking a dentigerous cyst. The present case aims to highlight the difference between cystic AOT and dentigerous cyst radiographically. The exact histogenesis of AOT and its variants still remains obscure. An attempt has been made to hypothesize the new school of thought regarding the origin of AOT.
PubMed: 26579317
DOI: 10.1155/2015/503059 -
Modern Pathology : An Official Journal... Jun 2019Adenomatoid odontogenic tumor is a benign encapsulated epithelial odontogenic tumor that shows an indolent clinical behavior. We have reported in a few adenomatoid...
Adenomatoid odontogenic tumor is a benign encapsulated epithelial odontogenic tumor that shows an indolent clinical behavior. We have reported in a few adenomatoid odontogenic tumors mutations in KRAS, which is a proto-oncogene frequently mutated in cancer such as lung, pancreas, and colorectal adenocarcinomas. We aimed to assess KRAS mutations in the hotspot codons 12, 13, and 61 in a large cohort of adenomatoid odontogenic tumors and to test the association of these mutations with clinical (age, site, tumor size, follicular/extrafollicular subtypes) and histopathological parameters. Thirty eight central cases were studied. KRAS codon 12 mutations were assessed by TaqMan allele-specific qPCR (p.G12V/R) and/or Sanger sequencing, and codon 13 and 61 mutations were screened by Sanger. Histological tumor capsule thickness was evaluated by morphometric analysis. Additionally, the phosphorylated form of the MAPK downstream effector ERK1/2 was investigated. Statistical analysis was carried out to test the association of KRAS mutations with clinicopathological parameters. KRAS c.35 G >T mutation, leading to p.G12V, was detected in 15 cases. A novel mutation in adenomatoid odontogenic tumor, c.34 G >C, leading to p.G12R, was detected in 12 cases and the other 11 were wild-type. Codon 12 mutations were not associated with the clinicopathological parameters tested. RAS mutations are known to activate the MAPK pathway, and we show that adenomatoid odontogenic tumors express phosphorylated ERK1/2. In conclusion, a high proportion of adenomatoid odontogenic tumors (27/38, 71%) have KRAS codon 12 mutations, which occur independently of the clinicopathological features evaluated. Collectively, these findings indicate that KRAS mutations and MAPK pathway activation are the common features of this tumor and some cancer types. Although it is unclear why different codon 12 alleles occur in different disease contexts and the complex interactions between tumor genotype and phenotype need clarification, on the basis of our results the presence of KRAS p.G12V/R favors the adenomatoid odontogenic tumor diagnosis in challenging oral neoplasm cases.
Topics: Adolescent; Adult; Ameloblastoma; Child; Female; Humans; MAP Kinase Signaling System; Male; Middle Aged; Mutation; Proto-Oncogene Mas; Proto-Oncogene Proteins p21(ras); Young Adult
PubMed: 30643167
DOI: 10.1038/s41379-018-0194-4 -
Modern Pathology : An Official Journal... Feb 2020Localized pleural mesothelioma is a rare solitary circumscribed pleural tumor that is microscopically similar to diffuse malignant pleural mesothelioma. However, the...
Localized pleural mesothelioma is a rare solitary circumscribed pleural tumor that is microscopically similar to diffuse malignant pleural mesothelioma. However, the molecular characteristics and nosologic relationship with its diffuse counterpart remain unknown. In a consecutive cohort of 1110 patients with pleural mesotheliomas diagnosed in 2005-2018, we identified six (0.5%) patients diagnosed with localized pleural mesotheliomas. We gathered clinical history, evaluated the histopathology, and in select cases performed karyotypic analysis and targeted next-generation sequencing. The cohort included three women and three men (median age 63; range 28-76), often presenting incidentally during radiologic evaluation for unrelated conditions. Neoadjuvant chemotherapy was administered in two patients. All tumors (median size 5.0 cm; range 2.7-13.5 cm) demonstrated gross circumscription (with microscopic invasion into lung, soft tissue, and/or rib in four cases), mesothelioma histology (four biphasic and two epithelioid types), and mesothelial immunophenotype. Of four patients with at least 6-month follow-up, three were alive (up to 8.9 years). Genomic characterization identified several subgroups: (1) BAP1 mutations with deletions of CDKN2A and NF2 in two tumors; (2) TRAF7 mutations in two tumors, including one harboring trisomies of chromosomes 3, 5, 7, and X; and (3) genomic near-haploidization, characterized by extensive loss of heterozygosity sparing chromosomes 5 and 7. Localized pleural mesotheliomas appear genetically heterogeneous and include BAP1-mutated, TRAF7-mutated, and near-haploid subgroups. While the BAP1-mutated subgroup is similar to diffuse malignant pleural mesotheliomas, the TRAF7-mutated subgroup overlaps genetically with adenomatoid tumors and well-differentiated papillary mesotheliomas, in which recurrent TRAF7 mutations have been described. Genomic near-haploidization, identified recently in a subset of diffuse malignant pleural mesotheliomas, suggests a novel mechanism in the pathogenesis of both localized pleural mesothelioma and diffuse malignant pleural mesothelioma. Our findings describe distinctive genetic features of localized pleural mesothelioma, with both similarities to and differences from diffuse malignant pleural mesothelioma.
Topics: Adult; Aged; Biomarkers, Tumor; Cyclin-Dependent Kinase Inhibitor p16; Female; Gene Deletion; Genetic Predisposition to Disease; Humans; Male; Middle Aged; Mutation; Neurofibromin 2; Phenotype; Pleural Neoplasms; Solitary Fibrous Tumor, Pleural; Tumor Necrosis Factor Receptor-Associated Peptides and Proteins; Tumor Suppressor Proteins; Ubiquitin Thiolesterase
PubMed: 31371807
DOI: 10.1038/s41379-019-0330-9 -
Journal of Conservative Dentistry : JCD 2022The objective of the study is to describe the clinical and radiographic features of nonmalignant nonendodontic periapical lesions (NMNPLs) mimicking lesions of... (Review)
Review
The objective of the study is to describe the clinical and radiographic features of nonmalignant nonendodontic periapical lesions (NMNPLs) mimicking lesions of endodontic cause. Five electronic databases, PubMed, Web of Science, Scopus, Embase, and ProQuest, were searched (till July 2021) for case reports, case series, and cross-sectional studies, in English language, reporting NMNPLs, which were clinically and/or radiographically simulating periapical pathosis of endodontic origin. Data extraction was done followed by quality assessment of the included articles using the Joanna Briggs Institute tool for case reports and case series. Seventy-three articles comprising 176 cases were included. Sixty-one articles were case reports, nine articles were case series, and three articles were retrospective studies. Male:female ratio was 1.5:1, with a higher prevalence of lesions occurring in the fourth and second decades of life. The majority of the lesions were located in the anterior maxilla. Radiographically, most of the lesions were well defined, radiolucent, and unilocular. Histologically, 29 different types of NMNPLs were reported, with the most common ones being odontogenic keratocyst (25.56%), dentigerous cyst (17.61%), ameloblastoma (11.36%), nasopalatine duct cyst (10.79%), and adenomatoid odontogenic tumor (5.68%). As all the included studies were observational, the quality of available evidence is considered low. Various features such as loss of tooth vitality, history of trauma, and presence of periapical radiolucency may lead to misdiagnosis of NMNPLs and must be considered during diagnosis of the lesion. Additional imaging modalities and histopathology can aid in right diagnosis.
PubMed: 35836562
DOI: 10.4103/jcd.jcd_13_22 -
Magnetic Resonance in Medical Sciences... Apr 2024Adenomatoid tumor is a rare benign genital tract neoplasm of mesothelial origin. Uterine adenomatoid tumors occur in the outer myometrium and may mimic leiomyomas....
PURPOSE
Adenomatoid tumor is a rare benign genital tract neoplasm of mesothelial origin. Uterine adenomatoid tumors occur in the outer myometrium and may mimic leiomyomas. Because hormonal treatment is not applicable to adenomatoid tumors and laparoscopic enucleation is not easy as myomectomy, it is important to differentiate adenomatoid tumors from leiomyomas for the adequate treatment. The purpose of this study is to evaluate the MRI findings of adenomatoid tumor for the differentiation from leiomyoma.
METHODS
MRI findings of surgically proven 10 uterine adenomatoid tumors in 9 women were retrospectively evaluated with correlation to histopathological findings.
RESULTS
All 10 tumors appeared as solid myometrial masses and showed heterogeneous signal intensity with admixture of partially ill-defined slight high-intensity areas containing abundant tubular tumor cells and well-defined myoma-like low-intensity areas reflecting smooth muscle hypertrophy on T2WI including 4 lesions with peripheral ring-like high intensity. High-intensity areas on T2WI tended to show high intensity on diffusion-weighted imaging (DWI) with relatively high apparent diffusion coefficient (ADC), suggesting T2 shine-through effect due to abundant tubules. Intra-tumoral hemorrhage revealed on MRI was rare. Early intense contrast-enhanced areas on dynamic contrast-enhanced study were observed dominantly within the high-intensity areas but rarely within the low-intensity areas on T2WI.
CONCLUSION
The outer myometrial mass with the admixture of well-defined low- and ill-defined high-intensity areas on T2WI may be suggestive of adenomatoid tumor. Peripheral ring-like high intensity on T2WI and DWI may also be suggestive. Dynamic contrast-enhanced MR study may be helpful for the differentiation from leiomyoma.
Topics: Female; Humans; Adenomatoid Tumor; Uterine Neoplasms; Retrospective Studies; Magnetic Resonance Imaging; Leiomyoma; Diffusion Magnetic Resonance Imaging
PubMed: 36697028
DOI: 10.2463/mrms.mp.2022-0067 -
Frontiers in Endocrinology 2021Adenomatoid tumor (AT) is an uncommon benign neoplasm of mesothelial origin, usually occurring in the female and male genital tracts. Extragenital localization such as... (Review)
Review
Adenomatoid tumor (AT) is an uncommon benign neoplasm of mesothelial origin, usually occurring in the female and male genital tracts. Extragenital localization such as the adrenal gland is extremely rare. Until now, only 39 cases of adrenal AT have been reported in the English literature. Here we report two novel cases of adrenal AT that occurred in male patients aged 30 and 31 years. The tumors were discovered incidentally by computed tomography (CT). Macroscopically, the tumors were unilateral and solid, and the greatest dimension of the tumors was 3.5 and 8.0 cm, respectively. Histologically, the tumors consisted of angiomatoid, cystic, and solid patterns and infiltrated the adrenal cortical or medullary tissue. The tumor cells had low nuclear/cytoplasmic ratio, with no pathological mitosis or nuclear pleomorphism. Thread-like bridging strands and signet-ring-like cells could be seen. Immunohistochemically, the tumor cells were positive for epithelial markers (AE1/AE3, CK7) and mesothelial markers (D2-40, calretinin, and WT-1). The Ki-67 index was approximately 1 and 2%, respectively. The differential diagnosis of adrenal AT includes a variety of benign and malignant tumors. The patients had neither local recurrence nor distant metastasis at 21 and 8 months after removal of the tumor. In the literature review, we comprehensively summarized the clinical, morphological, immunohistochemical, and prognostic features of adrenal AT. Adrenal ATs are morphologically and immunophenotypically identical to those that occur in the genital tracts. Combining the histology with immunohistochemical profiles is very supportive in reaching the diagnosis of this benign tumor, helping to avoid misdiagnosis and overtreatment.
Topics: Adenomatoid Tumor; Adrenal Gland Neoplasms; Adult; Humans; Male; Tomography, X-Ray Computed
PubMed: 34248850
DOI: 10.3389/fendo.2021.692553