Malignant Myoepithelioma of Cranial Dura

Erdoğan, Sibel; Rodríguez, Fausto J.; Scheithauer, Bernd W.; Abell-Aleff, Patrice C.; Rabin, Michael S.

doi:10.1097/pas.0b013e31802c98ae

Cited by 20 publications

(17 citation statements)

References 25 publications

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“…Malignant cases of myoepithelioma have been described in the lacrimal gland, tongue base, nasal cavity, maxillary sinus, nasopharynx, larynx and vulva (2,4). These tumors, however, are increasingly recognized at sites other than the major and minor salivary glands, including breast, skin, lung and soft tissue.…”

Section: Discussionmentioning

confidence: 99%

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A 2‐Year‐Old Girl with Dysmetria and Ataxia

2015

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A 2-year-old girl presented with 1-month history of dysmetria and ataxia. Computer tomography (CT) of the brain showed a large parietal-occipital mass and marked hydrocephalus, for which she required placement of bilateral ventriculoperitoneal shunts. Magnetic resonance imaging (MRI) of the brain also revealed two nodules within the lateral ventricle ( Figure 1A). Spine MRI showed abnormal leptomeningeal enhancement along the cervical spine, with additional enhancement in the thoracic and upper lumbar spine. A metastatic origin was excluded by exhaustive radiologic investigations, including body CT and positron emission tomography. Her disease progressed following completion of 6 months of intravenous and 3 months of oral chemotherapy; MRI showed interval increase in the size of the left temporal lobe ventricular lesions as well as worsening leptomeningeal disease and new parenchymal nodules. The tumor was completely excised. MICROSCOPIC PATHOLOGYLow magnification microscopy (34) of H&E staining of the tumor displayed a variable architecture consisting of solid sheets, islands (310, Figure 1B) and interconnecting cords of cells set in a vascularized fibromyxoid stroma (310, Figure 1C) interspersed with areas of necrosis and calcification. Ductal differentiation was identified in rare foci within <10% of the tumor mass (340 Figure 1D). The cells were polymorphic with round to oval, occasionally spindle-shaped nuclei, conspicuous nucleoli and moderate cytoplasm (320 Figure 1E). Mitotic figures were readily identified (340, Figure 1F) with a high level of immunolabeling with Ki-67/MIB-1 immunostain.The cells demonstrated a divergent immunophenotype with immunoreactivities for S-100 ( Figure 1G), smooth muscle actin ( Figure 1H), CAM 5.2 ( Figure 1I) and EMA ( Figure 1J 340). Retained immunoreactivity with BAF-47/INI1 (340, Figure 1K) and BRG-1 were retained within the tumor cells. The cells were immunonegative for GFAP (340, Figure 1L), pleomorphic adenoma gene 1 (PLAG-1) and germ cell markers including placental alkaline phosphatase (PLAP), Oct 3/4, alpha-feto protein (AFP) and CD30. EWSR1 gene rearrangement was not identified by fluorescent in-situ hybridization. What is your diagnosis? Figure 1.

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Section: Discussionmentioning

confidence: 99%

“…The "multipotent stem cell" is proposed to be the likely cell of origin at these sites (2). The "multipotent stem cell" is proposed to be the likely cell of origin at these sites (2).…”

Section: Discussionmentioning

confidence: 99%

A 2‐Year‐Old Girl with Dysmetria and Ataxia

2015

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“…8,12 They may also have a dural origin or be entirely contained within bone. 17,29,30 Fritchie et al reported a myoepithelioma arising from the parieto-occipital bone with a ESRW-1 mutation 17 and a myoepithelial neoplasm of the clivus is reported as well. 29 Intraorbital myoepitheliomas are thought to arise from the lacrimal gland in most cases, although in one report it was thought to arise from the soft tissues of the lower eye lid.…”

Section: Discussionmentioning

confidence: 99%

“…One patient underwent two subtotal resections of a dural convexity myoepithelioma followed by radiotherapy and remained recurrence free at the end of a 4-month follow-up. 30 Another underwent transphenoidal resection followed by radiation therapy and remained recurrence free at 2 years. 10 Two additional patients underwent subtotal resection and radiation therapy; however, their follow-up was not documented.…”

Section: Discussionmentioning

confidence: 99%

Myoepithelioma of the Orbital Apex and Middle Cranial Fossa: Case Report and Review of the Literature

Hayward

Yoo

Lee

et al. 2014

Neuro-Ophthalmology

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Myoepitheliomas are rare tumours that originate from glandular tissues such as the parotid or salivary glands, and less commonly from soft tissues of the head, neck, and other parts of the body. Intraorbital myoepitheliomas generally arise from the lacrimal gland. Intracranial myoepitheliomas are rare. We report a myoepithelioma of the orbital apex that did not originate from the lacrimal gland. It extended to the middle cranial fossa from the orbital apex and involved the dura and adjacent bone. A diagnostic biopsy via a lateral orbitotomy preceded resection. We review the natural course and histopathology of myoepithelial neoplasms, the surgical nuances of approaching an orbital apex tumour with maximal functional preservation, and the optimal management practices of these rare lesions.

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“…The tumor in this case showed a longer developmental progression with no invasion into the cavernous sinus. In addition, Sibel Erdogan et al [20] described a case of myoepithelial carcinoma arising in the intracranial dura outside of the sellar region. In comparison to these two cases, our case showed a rapid tumor development (the patient survived only four months from initial onset of symptoms to death) and a rare invasion into the cavernous sinus.…”

Section: Discussionmentioning

confidence: 99%

Rapid-developed primary malignant myoepithelioma in the cavernous sinus: a case report

et al. 2013

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BackgroundMalignant myoepithelioma is a relatively rare malignant tumor occurring most frequently in the salivary glands. A few isolated cases have been described in other locations, including soft tissue, bone, lung, bronchus, oral cavity, nasopharynx, larynx, and maxillary sinus. Malignant myoepithelioma, however, is uncommonly involved within the cavernous sinus. To the best of our knowledge, this is the first report of malignant myoepithelioma arising from within the cavernous sinus.Case presentationHerein, we report a case of a 48-year-old woman who presented a 1-month history of diplopia and blepharoptosis as well as radiological evidence of a rapidly developing cavernous sinus tumor. The patient underwent a trans-sphenoidal biopsy and a histological diagnosis indicated a malignant myoepithelioma. After diagnosis, the tumor grew rapidly and her clinical condition deteriorated progressively. Therefore, a pterional craniotomy with partial tumor removal was performed. The patient’s clinical state was worsened, and she died two months after the initial operation. Because the malignant myoepithelioma could not be traced to an organ of origin, other than the cavernous sinus, this case was diagnosed as a primary intracranial malignant myoepithelioma.ConclusionThe purpose of presenting this case report is to raise awareness among clinicians to consider malignant myoepithelioma as a differential diagnosis when a cavernous sinus mass is identified. Furthermore, an ideal management strategy for malignant myoepithelioma is not known and the prognosis seems to be unfavorable; therefore, more cases are needed to enhance our knowledge of the diagnosis, treatment, and prognosis of this rare intracranial lesion.

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Malignant Myoepithelioma of Cranial Dura

Cited by 20 publications

References 25 publications

A 2‐Year‐Old Girl with Dysmetria and Ataxia

A 2‐Year‐Old Girl with Dysmetria and Ataxia

Myoepithelioma of the Orbital Apex and Middle Cranial Fossa: Case Report and Review of the Literature

Rapid-developed primary malignant myoepithelioma in the cavernous sinus: a case report

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