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Ghadyani F, Behrad S. Myoepithelioma of the upper lip: A rare case report with a literature review of diagnostic approaches. SAGE Open Med Case Rep 2025; 13:2050313X241305185. [PMID: 40104810 PMCID: PMC11915260 DOI: 10.1177/2050313x241305185] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/30/2024] [Accepted: 11/19/2024] [Indexed: 03/20/2025] Open
Abstract
Myoepithelioma is an extremely rare salivary gland neoplasm with diverse histological variants. Understanding myoepithelioma's clinical, radiological, and molecular features is crucial for accurate diagnosis and management. We present a case of a 40-year-old Iranian male with a painless, firm, solitary exophytic mass on the upper lip. Surgical excision was curative, with no recurrence at 36 months. Histopathological analysis confirmed myoepithelioma with spindle and plasmacytoid cell variants. Myoepithelioma's rarity is reflected in epidemiologic studies and its features are mainly recognized by the information in the case reports. Majorly it appears at the site of major salivary glands and occasionally the palate. The occurrence at the site of the upper lip is extremely rare and only appeared in a few case reports. Seeking the history of previous exposures to possible etiologic factors might not be a facilitator. Differential diagnosis includes pleomorphic adenoma, myoepithelial carcinoma, and basal cell adenoma. The main diagnostic approach is histopathological analysis. Following that, the tumor's nature, cell variants, and possible transformation could be evaluated. While molecular studies supplement diagnosis, their routine clinical use is limited and their necessity is debated. The application of artificial intelligence could be helpful when uncertainty arises, or for analyzing microscopic images. Surgical excision with healthy margins is curative, and follow-up is essential.
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Affiliation(s)
- Farid Ghadyani
- Student Research Committee, Dental School, Semnan University of Medical Sciences, Semnan, Iran
- Maxillofacial Surgery & Implantology & Biomaterial Research Foundation, Tehran, Iran
- Universal Scientific Education and Research Network (USERN), Semnan, Iran
| | - Samira Behrad
- Department of Oral and Maxillofacial Pathology, Dental School, Semnan University of Medical Sciences, Semnan, Iran
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Kalra Y, Koppolu S, Babu M, Prashant R, Raj A, Ingale M. A Rare Presentation of Myoepithelioma of the Parotid Gland Manifesting as an Infra-Auricular Swelling. Cureus 2024; 16:e70746. [PMID: 39493007 PMCID: PMC11530963 DOI: 10.7759/cureus.70746] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/24/2024] [Accepted: 10/03/2024] [Indexed: 11/05/2024] Open
Abstract
This report describes an uncommon tumor of the salivary glands, myoepithelioma, that primarily affects major and minor glands, with a notable predilection for the parotid gland. Typically benign, this tumor arises from aberrant myoepithelial cells situated between the basement membrane and acinar cells. Myoepitheliomas are considered a subset of pleomorphic adenomas, distinguished by excessive myoepithelial cell growth. Despite their initial discovery, the precise histopathological and immunohistochemical characteristics of these tumors remain elusive, posing a diagnostic challenge because of their complex nature. We discuss a case of a 42-year-old female who had a 2 x 2 cm lump in the right infra-auricular area. The lump was examined with ultrasonography (USG) and later surgically removed. The initial frozen section analysis indicated an oncocytic lesion, but further histopathological and immunohistochemical evaluations confirmed that it was a myoepithelioma of the parotid gland.
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Affiliation(s)
- Yash Kalra
- Department of Otolaryngology, Head and Neck Surgery, Dr. D. Y. Patil Medical College, Hospital and Research Centre, Dr. D. Y. Patil Vidyapeeth (Deemed to be University), Pune, IND
| | - Saahiti Koppolu
- Department of Otolaryngology, Head and Neck Surgery, Dr. D. Y. Patil Medical College, Hospital and Research Centre, Dr. D. Y. Patil Vidyapeeth (Deemed to be University), Pune, IND
| | - Manu Babu
- Department of Otolaryngology, Head and Neck Surgery, Dr. D. Y. Patil Medical College, Hospital and Research Centre, Dr. D. Y. Patil Vidyapeeth (Deemed to be University), Pune, IND
| | - Rashmi Prashant
- Department of Otolaryngology, Head and Neck Surgery, Dr. D. Y. Patil Medical College, Hospital and Research Centre, Dr. D. Y. Patil Vidyapeeth (Deemed to be University), Pune, IND
| | - Akshi Raj
- Department of Pathology, Dr. D. Y. Patil Medical College, Hospital and Research Centre, Dr. D. Y. Patil Vidyapeeth (Deemed to be University), Pune, IND
| | - Mayur Ingale
- Department of Otolaryngology, Head and Neck Surgery, Dr. D. Y. Patil Medical College, Hospital and Research Centre, Dr. D. Y. Patil Vidyapeeth (Deemed to be University), Pune, IND
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Viswanathan V, Londhe M, Gurwale S, Buch A. Clear cell myoepithelioma of palate: A rare case report with brief review of literature. J Oral Maxillofac Pathol 2024; 28:493-496. [PMID: 39670128 PMCID: PMC11633919 DOI: 10.4103/jomfp.jomfp_372_23] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/23/2023] [Revised: 08/29/2024] [Accepted: 08/30/2024] [Indexed: 12/14/2024] Open
Abstract
Myoepitheliomas are very rare salivary gland neoplasms seen mainly in the major salivary glands (~40%) followed by minor salivary glands (~21%). Palatal myoepitheliomas are rare, with clear cell types being even rarer. Due to the rarity of this tumor, it lacks specific features that are diagnostic in cytology. Histopathology, along with immunohistochemistry, is needed for its confirmative diagnosis. We present a 28-year-old female with slow-growing, painless palatal swelling diagnosed as clear cell myoepithelioma on histopathology and immunohistochemistry.
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Affiliation(s)
- Vidya Viswanathan
- Department of Pathology, Dr. D.Y. Patil Medical College, Hospital and Research Centre, Dr. D.Y.Patil Vidyapeeth, Pune, Maharashtra, India
| | - Mangesh Londhe
- Department of Pathology, Dr. D.Y. Patil Medical College, Hospital and Research Centre, Dr. D.Y.Patil Vidyapeeth, Pune, Maharashtra, India
| | - Sushama Gurwale
- Department of Pathology, Dr. D.Y. Patil Medical College, Hospital and Research Centre, Dr. D.Y.Patil Vidyapeeth, Pune, Maharashtra, India
| | - Archana Buch
- Department of Pathology, Dr. D.Y. Patil Medical College, Hospital and Research Centre, Dr. D.Y.Patil Vidyapeeth, Pune, Maharashtra, India
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Mughal A, Danish H, Hassan S, Iftikhar H, Awan MO, Awan MS. Myoepithelioma of the Parotid Gland: A Case Report and Literature Review. Indian J Otolaryngol Head Neck Surg 2022; 74:6087-6090. [PMID: 36742563 PMCID: PMC9895647 DOI: 10.1007/s12070-021-02763-x] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/13/2021] [Accepted: 07/04/2021] [Indexed: 02/07/2023] Open
Abstract
Tumors of the salivary glands are rare and have various histo-pathological subtypes. The overall incidence is 2.5-3 per 100,000 patients per year. Myoepithelioma of the parotid gland is very rare with an incidence of only 1-1.5% among all salivary gland tumors. The diagnostic criteria for benign myoepithelioma were laid down on the basis of the work of Barnes and Sciubba. A 30-year-old lady presented to our university with a mass slowly increasing in size. There were no aggravating and/or relieving factors found. On examination a 3 × 3 cm diffuse swelling was found around the right pre auricular area. Fine Needle Aspiration Cytology (FNAC) was performed. The findings were suggestive of a pleomorphic adenoma. The patient underwent a right sided superficial parotidectomy to remove the tumor. The final histopathology report revealed the tumor to be a myoepithelioma. Myoepithelioma is a benign salivary gland neoplasm formed almost entirely of myoepithelial cells arranged in a sheet, island or cord-like fashion. On CT scans, it presents as a well-circumscribed homogenous lesion with lobulated or smooth margins. Myoepitheliomas may also have nodules that enhance under contrast and areas of linear bands that do not. The recommended treatment for a myoepithelioma is complete surgical excision with wide margins. It has a very low recurrence rate compared to the 15-18% recurrence rate for pleomorphic adenomas. Utilization of immunohistochemical staining is of utmost importance in suspected myoepithelioma cases to ensure proper treatment and follow-up. Myoepitheliomas are rare tumors which must be included in the differential if a patient presents with a tumor of characteristics similar to those described above. Immunohistochemistry and electron microscopy are useful tools for the diagnosis of myoepitheliomas.
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Affiliation(s)
- Ainulakbar Mughal
- Department of Undergraduate Medical Education, Aga Khan University Hospital, Karachi, Pakistan
| | - Hassan Danish
- Department of Undergraduate Medical Education, Aga Khan University Hospital, Karachi, Pakistan
| | - Saba Hassan
- Department of Undergraduate Medical Education, Aga Khan University Hospital, Karachi, Pakistan
| | - Haissan Iftikhar
- Department of Undergraduate Medical Education, Aga Khan University Hospital, Karachi, Pakistan
| | - Muhammad Ozair Awan
- Department of Undergraduate Medical Education, Aga Khan University Hospital, Karachi, Pakistan
| | - M. Sohail Awan
- Department of Undergraduate Medical Education, Aga Khan University Hospital, Karachi, Pakistan
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5
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Gökçe E, Beyhan M. Advanced magnetic resonance imaging findings in salivary gland tumors. World J Radiol 2022; 14:256-271. [PMID: 36160835 PMCID: PMC9453317 DOI: 10.4329/wjr.v14.i8.256] [Citation(s) in RCA: 7] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/12/2022] [Revised: 03/22/2022] [Accepted: 08/07/2022] [Indexed: 02/08/2023] Open
Abstract
Salivary gland tumors (SGTs) make up a small portion (approximately 5%) of all head and neck tumors. Most of them are located in the parotid glands, while they are less frequently located in the submandibular glands, minor salivary glands or sublingual gland. The incidence of malignant or benign tumors (BTs) in the salivary glands varies according to the salivary gland from which they originate. While most of those detected in the parotid gland tend to be benign, the incidence of malignancy increases in other glands. The use of magnetic resonance imaging (MRI) in the diagnosis of SGTs is increasing every day. While conventional sequences provide sufficient data on the presence, localization, extent and number of the tumor, they are insufficient for tumor specification. With the widespread use of advanced techniques such as diffusion-weighted imaging, semi-quantitative and quantitative perfusion MRI, studies and data have been published on the differentiation of malignant or BTs and the specificity of their subtypes. With diffusion MRI, differentiation can be made by utilizing the cellularity and microstructural properties of tumors. For example, SGTs such as high cellular Warthin’s tumor (WT) or lymphoma on diffusion MRI have been reported to have significantly lower apparent diffusion values than other tumors. Contrast agent uptake and wash-out levels of tumors can be detected with semi-quantitative perfusion MRI. For example, it is reported that almost all of the pleomorphic adenomas show an increasing enhancement time intensity curve and do not wash-out. On quantitative perfusion MRI studies using perfusion parameters such as Ktrans, Kep, and Ve, it is reported that WTs can show higher Kep and lower Ve values than other tumors. In this study, the contribution of advanced MRI to the diagnosis and differential diagnosis of SGTs will be reviewed.
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Affiliation(s)
- Erkan Gökçe
- Department of Radiology, Faculty of Medicine, Tokat Gaziosmanpasa University, Tokat 60100, Turkey
| | - Murat Beyhan
- Department of Radiology, Faculty of Medicine, Tokat Gaziosmanpasa University, Tokat 60100, Turkey
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Saripalli RRK, Alluri LSC, Jakkula A, Yadavilli SS. Extracapsular Dissection for Small Benign Tumors of Parotid Gland: A Case Report on Contemporary Technique. Cureus 2021; 13:e17007. [PMID: 34540408 PMCID: PMC8423323 DOI: 10.7759/cureus.17007] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 08/08/2021] [Indexed: 11/30/2022] Open
Abstract
Salivary gland tumor looms as painless enlarging mass which may embrace in both major or minor glands. Pleomorphic adenoma (PA) accord about 40-70% of all salivary gland tumors, where Warthin tumor, basal cell adenoma (BCA), adenoid cystic carcinoma (ACC), and sebaceous tumors have a strong predilection for major salivary gland. However, polymorphous low-grade adenocarcinoma (PLGA) has a marked predilection for the minor salivary gland. We present a case of PA in a 26-year-old male patient that has been successfully managed by extracapsular dissection (ECD) without any post-operative complications.
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Affiliation(s)
| | | | - Ananthnag Jakkula
- Department of Oral and Maxillofacial Surgery, GSL Dental College & Hospital, Rajahmundry, IND
| | - Sai Sarat Yadavilli
- Department of Oral and Maxillofacial Surgery, St. Joseph Dental College, Eluru, IND
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Mireles MG, Julián A, Salgado-Chavarría F, González GM. Benign myoepithelioma of the soft palate: an unusual clinical entity. BMJ Case Rep 2021; 14:e240384. [PMID: 34187791 PMCID: PMC10577746 DOI: 10.1136/bcr-2020-240384] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 05/20/2021] [Indexed: 11/06/2022] Open
Abstract
Myoepitheliomas are rare benign tumours that affect the exocrine glands and are sporadically located in the salivary glands. The most common location of myoepithelioma in the oral cavity is the parotid gland and it is seldom encountered in the palate. The diagnosis of this entity is challenging since its clinical presentation may resemble those of more common neoplasms, rendering a complex histopathological diagnosis. The aim of the present case report is to describe an unusual case of myoepithelioma of the soft palate in a male patient, which developed as an asymptomatic, slowly growing mass. The tumour was assessed with histopathological examination and the diagnosis was verified via immunohistochemistry. Finally, the treatment included surgical resection of the tumour and no signs of recurrence were noted 2.5 years after the surgical procedure. Early diagnosis and treatment plays an important role in the prognosis of this pathological entity.
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Affiliation(s)
| | - Angelica Julián
- Cirugia Maxilofacial, Hospital Regional de Alta Especialidad de Ixtapaluca, Estado de México, México
| | - Fabiola Salgado-Chavarría
- Especialidad de Cirugía Oral y Maxilofacial, Universidad Nacional Autónoma de México, Ciudad de México, México
| | - Guillermo Manuel González
- Anatomía Patológica, Hospital Regional De Alta Especialidad de Ixtapaluca, Estado de México, Estado de México, México
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Guo X, Watanabe J, Nakatani H, Fukushima K, Yamada S. Mucinous myoepithelioma: A report of a new variant. SAGE Open Med Case Rep 2020; 8:2050313X20940567. [PMID: 33101682 PMCID: PMC7550940 DOI: 10.1177/2050313x20940567] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/18/2020] [Accepted: 06/15/2020] [Indexed: 11/16/2022] Open
Abstract
Myoepitheliomas account for approximately 1.5% of all salivary gland tumors and arise most frequently from the parotid gland. Recently, a new myoepithelioma variant, called mucinous myoepithelioma, has attracted widespread attention. These tumors are recognized as a unique subtype of myoepithelioma, characterized by the presence of abundant mucin. We herein report the findings of an 86-year-old Japanese woman who presented with a hard mass of the right parotid gland behind her right ear which was gradually increasing in size. The patient had undergone a fine-needle aspiration biopsy 4 years earlier, and a cytological evaluation of a biopsy specimen had shown features of pleomorphic adenoma. A resection was thus performed and the tissue was found to be an encapsulated, soft and solid mass, and the cut surface was observed to be a capsulated and well-defined tumor lesion with myxoid-looking foci of gray-white coloration. Microscopic examination revealed that this lesion was composed of a proliferation of bland-looking epithelial and myoepithelial cells, arranged in a solid or reticular growth fashion in an abundant myxomatous or hyalinized stroma. These neoplastic epithelial cells had centrally located small nuclei with fine chromatin and abundant clear to eosinophilic cytoplasm, often containing mucin in a uniform pattern. Immunohistochemical staining demonstrated the tumor cells to be positive for AE1/AE3, S-100 and mucicarmine. Our findings suggest this case to be one myoepithelioma variant of mucinous myoepithelioma, and more experience related to this myoepithelioma variant is necessary to better understand its biological behavior and make an accurate diagnosis for a proper treatment.
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Affiliation(s)
- Xin Guo
- Department of Pathology and Laboratory
Medicine, Kanazawa Medical University, Kahoku, Japan
- Department of Pathology, Kanazawa
Medical University Hospital, Kahoku, Japan
| | - Jiro Watanabe
- Laboratory of Pathology, NHO Fukuyama
Medical Center, Fukuyama, Japan
| | - Hiroaki Nakatani
- Department of Otolaryngology, NHO
Fukuyama Medical Center, Fukuyama, Japan
| | - Kei Fukushima
- Department of Otolaryngology, NHO
Fukuyama Medical Center, Fukuyama, Japan
| | - Sohsuke Yamada
- Department of Pathology and Laboratory
Medicine, Kanazawa Medical University, Kahoku, Japan
- Department of Pathology, Kanazawa
Medical University Hospital, Kahoku, Japan
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Myoepithelial Tumors of Salivary Gland: A Clinicopathologic and Immunohistochemical Study of 15 Patients with MIB-1 Correlation. Head Neck Pathol 2020; 15:479-490. [PMID: 32959208 PMCID: PMC8134606 DOI: 10.1007/s12105-020-01225-0] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/10/2020] [Accepted: 09/10/2020] [Indexed: 02/07/2023]
Abstract
Myoepithelial neoplasms are rare tumors of the salivary glands with predominant myoepithelial differentiation and a broad histologic spectrum. Their histological features, immunohistochemical profile and biological behavior are not well characterized and pose a diagnostic challenge. A total of 15 myoepithelial tumors, diagnosed during 2012 and 2019 were subcategorized and correlated with MIB-1 labeling index (LI) and various histological parameters. Immunohistochemical stains for MIB-1 and other antibodies were performed. Statistical analysis was done by chi-square test, Fisher's exact test and Kaplan Meier curve. Nine patients were male and six were female with the median age of 44 years (range 21-83 years). Of the 15 patients, 6 cases were classified as myoepithelioma (ME) and 9 cases as myoepithelial carcinoma (MECA). Parotid gland was the most common site (46.7%) followed by the palate. MEs showed well circumscribed tumor borders whereas MECAs exhibited focal capsular to extensive invasion into adjacent tissues. Epithelioid cell morphology was most common followed by mixed cell morphology. MIB-1 LI was significantly associated with invasive tumor borders, necrosis and high mitosis. Increased frequency of recurrence was noted with high MIB-1 LI, though it was not statistically significant. MIB-1 LI was high in nearly all MECAs with focal capsular to extensive invasion while low in MEs. Myoepithelial tumor with multinodular growth pattern and focal capsular invasion may have an indolent behavior if mitotic activity and MIB-1 LI is low. Early diagnosis and treatment of MECAs significantly improves the patient's survival and prognosis.
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Malignant Myoepithelioma of the Parotid Gland in a Rat. J Comp Pathol 2020; 176:162-164. [PMID: 32359631 DOI: 10.1016/j.jcpa.2020.03.004] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/13/2020] [Revised: 03/05/2020] [Accepted: 03/09/2020] [Indexed: 11/23/2022]
Abstract
Myoepitheliomas of the salivary glands have been described in laboratory mice, but not in rats. A 20-week-old Wistar (Han) female rat developed a white to grey firm mass at the left side of the neck. Histologically, the mass was unencapsulated and infiltrated the adjacent tissue. The tumour parenchyma was cell rich without acinar or tubular architecture. The tumour showed a palisading basal cell pattern adjacent to blood vessels. There were areas of necrosis filled with cellular debris. The tumour cells showed strong immunohistochemical labelling for pan-cytokeratin types I and II (AE1/AE3), pan-cytokeratin (cytokeratins 1, 5, 6 and 8), cytokeratin 5, cytokeratin 14, vimentin and podoplanin, and only very slight positivity for cytokeratin 8 in small areas. There was no expression of smooth muscle actin. Based on the histological appearance and the immunohistochemistry, the tumour was diagnosed as a malignant myoepithelioma of the parotid gland originating from the parotid duct.
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Koyama R, Minagawa N, Maeda Y, Shinohara T, Hamada T. Metachronous Pancreatic and Thyroid Metastases from Primary Soft-Tissue Myoepithelioma in the Clavicular Region: A Case Report of a Long-Term Survivor. AMERICAN JOURNAL OF CASE REPORTS 2020; 21:e920702. [PMID: 31983728 PMCID: PMC6998795 DOI: 10.12659/ajcr.920702] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/13/2022]
Abstract
Patient: Male, 69-year-old Final Diagnosis: Metachronous pancreatic and thyroid metastases from primary soft tissue myoepithelioma Symptoms: None Medication:— Clinical Procedure: Surgery Specialty: Surgery
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Affiliation(s)
- Ryota Koyama
- Department of Gastrointestinal Surgery, Hokkaido Cancer Center, Sapporo, Hokkaido, Japan
| | - Nozomi Minagawa
- Department of Gastrointestinal Surgery, Hokkaido Cancer Center, Sapporo, Hokkaido, Japan
| | - Yoshiaki Maeda
- Department of Gastrointestinal Surgery, Hokkaido Cancer Center, Sapporo, Hokkaido, Japan
| | - Toshiki Shinohara
- Department of Gastrointestinal Surgery, Hokkaido Cancer Center, Sapporo, Hokkaido, Japan
| | - Tomonori Hamada
- Department of Gastrointestinal Surgery, Hokkaido Cancer Center, Sapporo, Hokkaido, Japan
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PALOMEQUE VERA JM, López de Huelva E, Fernández Ruiz E, Platero Sánchez-Escribano M, Oliva Dominguez M. Mioepitelioma benigno de parótida con diagnóstico inicial de malignidad. Descripción de un caso. REVISTA ORL 2018. [DOI: 10.14201/orl.18070] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/11/2022] Open
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