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Journal of Medical Case Reports Aug 2022Adenomyoepithelioma of the breast is an uncommon subtype of breast neoplasm that occurs in adults over a wide age range but most commonly in middle-aged and older...
BACKGROUND
Adenomyoepithelioma of the breast is an uncommon subtype of breast neoplasm that occurs in adults over a wide age range but most commonly in middle-aged and older adults. It usually presents as a solitary palpable mass or is detected on breast radiographic images. Histologically, it is a biphasic tumor with proliferation of both the epithelial and myoepithelial components of the glands, with variable types of tissue metaplasia.
CASE PRESENTATION
A 64-year-old Saudi woman who underwent regular breast screening (mammogram) presented to our hospital following radiographic detection of a suspicious grouped microcalcification in the upper outer quadrant of her right breast on the mammogram. A wide local excision of the right breast lump was performed. Following histopathological examination of the breast lump, the final diagnosis was breast adenomyoepithelioma with mucoepidermoid/divergent differentiation, with no evidence of malignancy. About two years after the operation, a clinical follow-up conducted outside our hospital showed the development of ductal carcinoma in situ in the same breast.
CONCLUSION
Although the prognosis and the plan of treatment remains the same, our case highlights the complexities in making an accurate diagnosis between the various types of metaplasia within adenomyoepithelioma on one hand and the presence of mucoepidermoid differentiation in adenomyoepithelioma on the other.
Topics: Adenomyoepithelioma; Aged; Breast; Breast Neoplasms; Female; Humans; Metaplasia; Middle Aged; Myoepithelioma
PubMed: 35934703
DOI: 10.1186/s13256-022-03507-3 -
Journal of Cancer Research and... 2022Primary epithelial-myoepithelial carcinoma (EMC) is a rare low-grade malignant neoplasm of the lung that originates from the submucosal bronchial glands. It behaves in...
Primary epithelial-myoepithelial carcinoma (EMC) is a rare low-grade malignant neoplasm of the lung that originates from the submucosal bronchial glands. It behaves in an indolent fashion, although rare cases with high-grade transformation have been reported. Because of the rarity, optimal therapy for this entity has not been clearly defined. Herein, we report a case of primary pulmonary EMC in a 38-year-old Indian man who had a short history of dyspnea and a computed tomographic (CT) scan revealed a 3 cm diameter homogeneous mass in the lower lobe of the right lung. A CT-guided biopsy revealed a mildly atypical and mitotically quiescent tumor with solid and focal acinar arrangement; foci with biphasic arrangement by inner epithelial and outer myoepithelial cells were identified. The neoplasm revealed cytokeratin (CK) 7 positivity in the epithelial cells, while the myoepithelium expressed smooth muscle actin and p63. The tumor had a low (8%) Ki-67 proliferation index. The neuroendocrine markers, thyroid transcription factor 1, CK5/6, p40, and napsin A were negative. Positron emission tomography-CT was negative for any other mass lesion. The mass was excised with negative margins and the patient was on close follow without any evidence of disease for the past 17 months. A custom made, targeted DNA- and RNA-based 5 gene lung cancer next-generation sequencing panel (Epidermal growth factor receptor (EGFR), anaplastic lymphoma kinase (ALK), Ros protocol-oncogene 1 tyrosine kinase (ROS1), B-rapidly accelerated fibrosarcoma family oncogene (BRAF), and mesenchymal epithelial transition molecule (MET)), compatible with the Ion S5 system was performed; however, no mutation was identified. This case depicts awareness about this entity and use of appropriate immunostains, particularly the myoepithelial markers are essential to arrive at a correct diagnosis. Importantly, high-grade transformation, recurrence, and metastases are not very uncommon in EMC, warranting a correct and timely diagnosis for therapeutic decision-making and prognostication of the patient.
Topics: Adult; Carcinoma; Humans; Lung; Lung Neoplasms; Male; Myoepithelioma; Protein-Tyrosine Kinases; Proto-Oncogene Proteins
PubMed: 35900560
DOI: 10.4103/jcrt.JCRT_559_20 -
Frontiers in Oncology 2022Malignant myoepithelioma of the head and neck (HNMM) is a rare malignancy, and its characteristics and survival rates have not been well-defined. This study aimed to...
Malignant myoepithelioma of the head and neck (HNMM) is a rare malignancy, and its characteristics and survival rates have not been well-defined. This study aimed to define the epidemiology of HNMM and identify the prognostic factors associated with the disease. Data on all patients diagnosed with HNMM between 1991 and 2016 were gathered from the Surveillance Epidemiology and End Results (SEER) database. The demographics, clinicopathological characteristics, treatment, and prognoses of the patients were described. Cox regression analysis was used to identify the prognostic factors, and the prognostic nomograms for overall survival (OS) and disease-specific survival (DSS) were constructed. A total of 333 cases of HNMM were identified. The average age at diagnosis was 60.6 years, and 50.1% of the patients were men. After diagnosis, 46.2% of patients underwent surgery alone, 43.5% of patients underwent surgery and radiotherapy, and 3.6% of patients received only radiotherapy. Survival analysis showed that the 5-year OS and DSS for all HNMM patients were 69.7 and 82.1%, respectively. In the multivariate analysis model, the undifferentiated pathological grade (P <0.05) and M1 in the M category (P <0.01) were independent prognostic factors for poor OS and DSS, whereas the use of surgical resection was an independent favorable prognostic factor for both OS and DSS (P <0.05). The prognostic nomograms for OS and DSS prediction were constructed; the C-index values for OS and DSS prediction were 0.78 (95% CI 0.70-0.86) and 0.79 (95% CI 0.67-0.90), respectively. In conclusion, this SEER data-based study demonstrated that HNMM patients often had a favorable prognosis, and distant metastasis, pathological grade, and the use of surgery contributed to their survival. Furthermore, we developed a prognostic nomogram to predict OS and DSS for HNMM patients to aid physicians in the clinical management of this rare disease.
PubMed: 35847870
DOI: 10.3389/fonc.2022.754967 -
Journal of Medical Cases Jun 2022We report the case of a 53-year-old man with psoriatic arthritis, suffering from a malignant and recidivant myoepithelioma in his right axilla and arm, and undergoing...
We report the case of a 53-year-old man with psoriatic arthritis, suffering from a malignant and recidivant myoepithelioma in his right axilla and arm, and undergoing two surgeries, with the last one being performed a month prior to actual admission. After the last surgery, he was admitted to hospital with fever without a source. After physical examination, laboratory tests, blood cultures and transthoracic and transesophageal echocardiography, he was diagnosed with infectious endocarditis (IE) on a bicuspid aortic valve (BAV) caused by (PA). Antibiogram-guided antibiotic therapy with meropenem and tobramicin was initiated. However, in the presence of repetitive spleen infarctions and a large vegetation, 12 days after admission, a bioprosthesis aortic valve implantation was performed. The postsurgical evolution was favorable and prolonged antibiotic course with meropenem and tobramicin was completed. The pathological anatomy and the native valve cultured confirmed an IE caused by PA. Gram-negative non-HACEK IE cases are infrequent, accounting for 1.8% of the total IE cases. PA is the second most frequent bacillus in this group, causing endocarditis more prevalently when associated with healthcare procedures rather than injectable drug use. No prior case study has identified IE caused by PA related to a BAV in the last years.
PubMed: 35837082
DOI: 10.14740/jmc3943 -
Journal of Medicine and Life May 2022This study is a systematic review and meta-analysis to assess the overexpression rate of HER2 in patients with salivary gland tumors. We included peer-reviewed... (Meta-Analysis)
Meta-Analysis Review
This study is a systematic review and meta-analysis to assess the overexpression rate of HER2 in patients with salivary gland tumors. We included peer-reviewed publications from 1995 to 2020, indexed in medical databases, using search terms such as "human epidermal growth factor receptor 2 (HER2)" and "salivary gland tumors", and extracted relevant data. The extracted data were analyzed with RevMan 5.3 software. Intra-and intergroup post hoc analyses of outcome variables were performed using t-tests, and the rates of HER2 positivity among studies were evaluated. 80 studies were included in the analysis. The positive rates of HER2 ranged from 3.3% to 84.0% and 1% to 9% in malignant and benign subtypes, respectively. The highest HER2 overexpression rate among malignant tumors was in salivary ductal carcinomas (SDC), with a 45% positive rate (CI 95%: 21.9-70.3%). Mucoepidermoid carcinoma (MEC) had the highest positive rate of 84% (CI 95%: 74.1-90.0%). Among benign salivary gland tumors, the highest rate was found in myoepithelioma, with a positive rate of 9% (CI 95%: 1.7-33.6%). The highest rate of HER2 overexpression is present in malignant subtypes of salivary gland tumors, more specifically in salivary ductal carcinoma, mucoepidermoid carcinomas, salivary duct carcinoma in situ, and carcinoma ex pleomorphic adenoma.
Topics: Biomarkers, Tumor; Carcinoma, Mucoepidermoid; Humans; Receptor, ErbB-2; Salivary Gland Neoplasms; Salivary Glands
PubMed: 35815077
DOI: 10.25122/jml-2021-0394 -
BMC Musculoskeletal Disorders Jul 2022The purpose of this study was to investigate the feasibility of using a three-dimensional (3D)-printed arthrodesis prosthesis for reconstruction of the proximal humeral...
Efficacy and safety of a 3D-printed arthrodesis prosthesis for reconstruction after resection of the proximal humerus: preliminary outcomes with a minimum 2-year follow-up.
BACKGROUND
The purpose of this study was to investigate the feasibility of using a three-dimensional (3D)-printed arthrodesis prosthesis for reconstruction of the proximal humeral defect after tumor resection.
METHODS
A novel proximal humeral prosthesis was designed to restore bone continuity and shoulder arthrodesis and was fabricated via 3D printing technology. Ten patients with primary malignancies in the proximal humerus underwent intra-articular resection and replacement with this prosthesis from 2017 to 2019. Baseline and operative data, oncological and prosthetic survival, and functional status were summarized.
RESULTS
This cohort consisted of 9 males and 1 female with a mean age of 32.1 ± 16.1 years. Diagnoses included 5 cases of osteosarcoma, 3 cases of chondrosarcoma and 1 each case of undifferentiated pleomorphic sarcoma and malignant myoepithelioma. The mean operative duration, intraoperative hemorrhage and postoperative length of hospitalization were 151.5 ± 61.0 min, 410.0 ± 353.4 ml and 5.3 ± 1.9 d, respectively. The mean follow-up duration was 29.3 ± 6.4 months, with a minimum of 24 months for the surviving patients. Two patients experienced local recurrence, and four patients developed distant metastases. Detachment of the taper occurred in two patients. One was managed conservatively, and the other received amputation due to concurrent tumor recurrence. The mean MSTS-93 and ASES scores and ranges of forwards flexion and abduction were 24.9 ± 3.1, 79.4 ± 8.3, 71.3 ± 19.4°, and 61.3 ± 16.4°, respectively. The functional outcomes were independent of the preservation of the axillary nerve. Histological study of the glenoid component showed evidence of bone ingrowth at the bone-prosthesis porous interface.
CONCLUSION
Application of the 3D-printed arthrodesis prosthesis might be a safe and efficacious method for functional reconstruction in patients who underwent resection of the proximal humerus, especially for those without preservation of the axillary nerve.
Topics: Adolescent; Adult; Arthrodesis; Artificial Limbs; Bone Neoplasms; Female; Follow-Up Studies; Humans; Humerus; Male; Middle Aged; Printing, Three-Dimensional; Shoulder; Young Adult
PubMed: 35787280
DOI: 10.1186/s12891-022-05581-6 -
Genes, Chromosomes & Cancer Jan 2023The RREB1::MRTFB (former RREB1::MKL2) fusion characterizes ectomesenchymal chondromyxoid tumors (EMCMT) of the tongue. Only five molecularly confirmed extra-glossal...
RREB1::MRTFB fusion-positive extra-glossal mesenchymal neoplasms: A series of five cases expanding their anatomic distribution and highlighting significant morphological and phenotypic diversity.
The RREB1::MRTFB (former RREB1::MKL2) fusion characterizes ectomesenchymal chondromyxoid tumors (EMCMT) of the tongue. Only five molecularly confirmed extra-glossal EMCMT cases have been reported recently; all occurring at head and neck or mediastinal sites. We herein describe five new cases including the first two extracranial/extrathoracic cases. The tumors occurred in three male and two female patients with an age ranging from 18 to 61 years (median, 28). Three tumors were located in the head and neck (jaw, parapharyngeal space, and nasopharyngeal wall) and two in the soft tissue (inguinal and presacral). The tumor size ranged from 3.3 to 20 cm (median, 7). Treatment was surgical without adjuvant treatment in all cases. Two cases were disease-free at 5 and 17 months; other cases were lost to follow-up. Histologically, the soft tissue cases shared a predominant fibromyxoid appearance, but with variable cytoarchitectural pattern (cellular perineurioma-like whorls and storiform pattern in one case and large polygonal granular cells embedded within a chondromyxoid stroma in the other). Two tumors (inguinal and parapharyngeal) showed spindled to ovoid and round cells with a moderately to highly cellular nondescript pattern. One sinonasal tumor closely mimicked nasal chondromesenchymal hamartoma (NCMH). Mitotic activity was low (0-5 mitoses/10 hpfs). Immunohistochemical findings were heterogeneous with variable expression of S100 (2/5), EMA (2/3), CD34 (1/4), desmin (1/4), and GFAP (1/3). Targeted RNA sequencing revealed the same RREB1::MRTFB fusion in all cases, with exon 8 of RREB1 being fused to exon 11 of MRTFB. This study expands the topographic spectrum of RREB1::MRTFB fusion-positive mesenchymal neoplasms, highlighting a significant morphological and phenotypic diversity. Overall, RREB1::MRTFB-rearranged neoplasms seem to fall into two subcategories: tumors with lobulated, chondroid, or myxochondroid epithelioid morphology (Cases 2 and 3) and those with more undifferentiated hypercellular spindle cell phenotype (Cases 1, 4, and 5). Involvement of extracranial/extrathoracic sites and the NCMH-like pattern are novel. The biology of these likely indolent or benign tumors remains to be verified in the future.
Topics: Male; Female; Humans; Biomarkers, Tumor; Tongue Neoplasms; Gene Fusion; Myoepithelioma; Phenotype; Soft Tissue Neoplasms; DNA-Binding Proteins; Transcription Factors
PubMed: 35763541
DOI: 10.1002/gcc.23082 -
Journal of Cardiothoracic Surgery May 2022Endotracheal tumors are rare in the respiratory system. Myoepitheliomas are benign tumors, which are rarely reported in the respiratory system. Herein, we report a rare... (Review)
Review
BACKGROUND
Endotracheal tumors are rare in the respiratory system. Myoepitheliomas are benign tumors, which are rarely reported in the respiratory system. Herein, we report a rare case of endotracheal myoepithelioma, which was resected by rigid bronchoscopy.
CASE PRESENTATION
A 36-year-old man, presenting with chest pain, dyspnea, stridor, and hemoptysis, was referred to our center with radiological features of near-total tracheal obstruction due to mass. Fiberoptic bronchoscopy with argon plasma coagulation and rigid bronchoscopy with grasper forceps was utilized to resect the mass. Pathological evaluation of the mass demonstrated myoepithelioma. The patient was discharged in good condition. Now, after 6 months, the patient is symptom-free with no evidence of tumor recurrence or re-growth.
CONCLUSIONS
Despite being extremely rare, myoepithelioma should be considered a possible differential diagnosis for endotracheal tumors. Fiberoptic and rigid bronchoscopy management is an effective method for the resection of endotracheal tumors.
Topics: Adult; Airway Obstruction; Bronchoscopy; Humans; Male; Myoepithelioma; Neoplasm Recurrence, Local; Trachea
PubMed: 35606819
DOI: 10.1186/s13019-022-01880-0 -
BMJ Case Reports May 2022A young pregnant woman presented to the emergency department with acute lower abdominal pain. The ultrasound examination showed not only a viable fetus with a...
A young pregnant woman presented to the emergency department with acute lower abdominal pain. The ultrasound examination showed not only a viable fetus with a gestational age of 24 weeks but also a heteroechoic mass of about 7×7 cm involving parts of the face. The patient was found to be in active labour with rapid progression, and she gave birth to a dead female neonate. The autopsy showed a large malignant mixed tumour of the left parotid gland and revealed infection with cytomegalovirus. To our knowledge, this is the first reported case of a malignant mixed tumour of salivary glands in fetuses.
Topics: Carcinoma; Cytomegalovirus Infections; Female; Humans; Infant; Infant, Newborn; Mixed Tumor, Malignant; Myoepithelioma; Parotid Gland; Parotid Neoplasms; Pregnancy; Salivary Glands
PubMed: 35568413
DOI: 10.1136/bcr-2021-248616 -
The American Journal of Case Reports Apr 2022BACKGROUND Breast adenomyoepithelioma is a rare benign breast tumor characterized by a biphasic proliferation of epithelial and myoepithelial cells with variable...
BACKGROUND Breast adenomyoepithelioma is a rare benign breast tumor characterized by a biphasic proliferation of epithelial and myoepithelial cells with variable clinical and diagnostic features. Establishing the diagnosis, determining optimal therapy, and predicting outcome are problematic because of the rarity of this entity. There have been only 2 large series of adenomyoepitheliomas of the breast, reported by Tavassoli and Rosen, which included 27 and 18 patients, respectively. In this report, we present 3 cases of breast adenomyoepithelioma. CASE REPORT Herein, we report 3 cases of breast adenomyoepithelioma. The first case is of a 64-year-old woman who was found to have right breast microcalcification on a screening mammogram. The second case is of a 74-year-old woman who had a right breast mass. These 2 patients were managed by wide local excision. Postoperative microscopic examination revealed adenomyoepithelioma. The third case is of a 49-year-old woman with bilateral saline breast implants who presented with a left breast mass. A core needle biopsy was done and revealed adenomyoepithelioma associated with usual ductal hyperplasia and ductal carcinoma in situ. CONCLUSIONS Breast adenomyoepithelioma is a rare condition that can pose diagnostic challenges due to variable imaging presentations, necessitating percutaneous core biopsy for initial diagnosis. Correct diagnosis is usually possible only on excisional biopsy and confirmed by demonstrating the biphasic nature of the tumor by IHC. Clinical suspicion coupled with utilizing both radiological and histopathological facilities can aid in the accurate diagnosis and management. For the most part, they are considered to be benign, but they can locally recur.
Topics: Adenomyoepithelioma; Aged; Breast; Breast Neoplasms; Carcinoma, Intraductal, Noninfiltrating; Female; Humans; Middle Aged; Myoepithelioma; Neoplasm Recurrence, Local
PubMed: 35400730
DOI: 10.12659/AJCR.936070