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Corresponding Author:

	Nancy King, M.D.  
	The New York Eye and Ear Infirmary 
	Department of Otolaryngology
	310 E. 14th Street 
	New York, NY 10003
	Email:  HYPERLINK "mailto:nkinghkim@nyee.edu" nking@nyee.edu
	Phone: 212-979-4200
	Fax: 212-979-4315



Additional Authors:

Lisa Mao, M.D., New York Medical Colllege, Valhalla, NY
Codrin Iacob, M.D., New York Eye and Ear Infirmary, New York, NY
Ana H Kim, M.D., New York Eye and Ear Infirmary, New York, NY	


Abstract
Meningiomas are the second most common brain tumor.  Though most are found intracranially,  a few exhibit extracranial extension.  Even fewer meningiomas are found extracranially without an intracranial component.  We present a case of a 50 year old woman diagnosed with meningioma of the middle ear.  Concurrent dural and ophthalmic artery aneurysms were also found on imaging. 



Keywords: Meningioma; Aneurysm; Middle Ear




Introduction

	Meningiomas, constituting nearly 20% of all intracranial tumors, are the second most common primary brain tumor behind gliomas [1]. They arise from the meningocytes of arachnoid granulations. Some meningiomas exhibit extracranial extension, though this happens rarely, with less than 100 cases reported to date [2]. Extracranial meningiomas without an intracranial component, also known as primary extracranial meningiomas, are even more rare comprising < 2% of all meningiomas [3, 4].  Most of these primary extracranial meningiomas occur in the head and neck region, with 26% occurring in the ear and temporal bone [3].  We present a case of a 50-year-old woman diagnosed with meningioma of the middle ear with concurrent dural and ophthalmic artery aneurysms. 

Case Report

A 50-year-old woman presents with progressive hearing loss, aural fullness, and pulsatile tinnitus of three years� duration, all on the left side.  During the last 2 years, she also experienced multiple bouts of otitis media in the left ear, requiring multiple antibiotic treatments. No otalgia, otorrhea, or vertigo was described. Upon physical exam of the left ear, a non-pulsatile, reddish-tan, bulging mass was seen behind a seemingly intact tympanic membrane, posterior to the handle of the malleus. Physical exam of the right ear was normal. Facial nerve was House-Brackmann I/VI (normal function). The patient�s medical history is significant for incidental findings of a 2.5 mm ophthalmic artery aneurysm and distal dural ring aneurysm both also on the left side found on MRA ordered during pulsatile tinnitus work-up (Figure 1).
Audiometry showed maximal conductive hearing loss at all frequencies in the left ear with normal hearing on the right. Speech discrimination was excellent at 96%. Computed tomography (CT) of the temporal bone revealed opacification of the left tympanomastoid cavity without ossicular erosion (Figure 2). The carotid and jugular bulb were intact. Both the tympanic and mastoid portion of the facial nerve had full bony encasement, ruling out the possibility of facial neuroma. Magnetic resonance imaging (MRI) with gadolinium showed enhancing granulation tissue within the left tympanic cavity and adjacent mastoid air cells, with no enhancing mass within the internal auditory canal or cerebellopontine angle where meningioma and vestibular schwannoma most commonly occur (Figure 3).
Intraoperatively, granulation and necrotic tissue was found filling the mastoid air cells.    As the mastoid and facial recess were drilled out, firm vascular tissue was found filling the mastoid cavity and superior aspect of the facial recess. The mass was dissected off the horizontal semicircular canal and a small piece was sent for frozen pathology, which was found to be consistent with meningioma. Once the tympanomeatal flap was elevated, the mass was seen to fill the entire middle ear cavity (Figure 4). As the mass was highly vascular, it could not be safely dissected from the ossicles. In addition, the tympanic portion of the facial nerve could not be readily appreciated due to brisk bleeding. Therefore, only the hypotympanic and mastoid portions wereremoved. 
Formal histopathological assessment showed numerous islands of proliferating meningothelial cells with bland nuclei, indistinct cell borders and foci of whorling. The cells showed epithelial membrane cytoplasmic antigen (EMA) and progesterone receptor nuclear positivity, which is specific to meningiomas in general [5]. The specimen was immunonegative for cytokeratin AE1/AE3, S-100, synaptophysin and chromogranin. Final diagnosis was meningothelial meningioma, World Health Organization (WHO) grade one [6].  (Figure 5)
Post-operatively, the tinnitus had moderately improved. The hearing, as expected, was unchanged. A repeat MRI 6 months later found residual meningioma at the petrous apex. In addition, the ophthalmic artery aneurysm had increased in size to 5 mm. The patient elected to monitor the residual meningioma and was referred to neurosurgery for care of the aneurysm.

Discussion

Meningiomas are slow-growing, benign tumors, accounting for 13-26% of all intracranial neoplasms [7].  They occur most commonly in women between the ages of 45 and 55. Sometimes, meningiomas extend extracranially. However, extracranial extension to the middle ear is uncommon, with fewer than 100 cases reported to date [2].  Presumed routes of extension are along the internal auditory canal (IAC), tegmen, and the jugular foramen [2].  
Our patient had a meningioma in the middle ear with extension into the mastoid cavity, but no intracranial component found on MRI. Her IAC, tegmen, and jugular foramen were all intact. Extracranial meningiomas without an intracranial component, true primary extracranial meningiomas, are extremely rare, accounting for < 2% of all meningiomas [3, 4].  Of these, 26% occur in the middle ear and temporal bone [3].  Primary extracranial meningiomas are believed to originate from embryonal nests of arachnoid cells that detached during embryologic development [8]. Another theory speaks of extracranial meningiomas arising from altered maturation of pluripotential mesenchymal cells [8].  Other possible tumors in the middle ear include glomus tumors, schwannomas, and middle ear adenomas.
Meningiomas are characterized by slow, indolent growth and rare malignant transformation. Approximately 90% of meningiomas are benign (WHO Grade I), 6% are atypical (WHO Grade II), and 2% are malignant (WHO Grade III) [9]. Our patient�s pathology diagnosis of WHO grade I meningioma indicates a likely benign course, though invasive behavior has been noted in the literature, particularly of the osseous labyrinth [10]. More advanced WHO grades exhibit greater rates of malignancy.  The classic treatment for all meningiomas is complete tumor excision, as this offers the greatest chance of tumor-free survival. Gross resection results in 85%, 75%, and 70% overall survival rates at five, ten, and fifteen years respectively [9]. Progression-free survival rates are 90%, 80%, and 67% at five, ten, and fifteen years respectively [9]. However, complete resection is often difficult as meningiomas often infiltrate the bone and clear margins are difficult to obtain. In addition, the risk of injuring important neural and vascular structures makes complete resection even more challenging. For this reason, radiotherapy is often considered as an adjuvant treatment [10]. Radiotherapy is currently standard treatment for atypical, malignant, or recurrent meningiomas based on level 3 evidence [9,11]. Considering that the long term prognosis for most meningiomas is very good with five-year survival rates > 80%, and slow growth rates with almost 60% showing no tumor growth [12], conservative follow-up management with serial imaging is often recommended for patients with asymptomatic meningiomas or meningiomas whose location carries a significant risk of morbidity [9,11,12].
	Interestingly, though the patient�s dural and ophthalmic artery aneurysm occurred on the same side as the meningioma, they are seemingly unrelated. Associations between meningiomas and intracranial aneurysms are rare and limited to case series and case reports in the literature [13,14]. Javalkar et al. found the incidence of meningiomas associated with aneurysms to be 1.1%, which is not higher than the incidence of aneurysms in the general population [13].  A recent 2009 review of over 100 patients with concurrent meningioma and aneurysms concluded that their coexistence seems to be coincidental [14].  
	To our knowledge, this is the first report in the English language literature of a primary middle ear meningioma found in conjunction with aneurysms. 

References
Longstreth WT Jr, Dennis LK, McGuire VM, Drangsolt MT, Koepsell TD. Epidemiology of intracranial meningioma. Cancer. 1993; 72: 639-648.
Chang CY, Cheung SW, Jackler RK. Meningiomas presenting in the temporal bone: the pathways of spread from an intracranial site of origin. Otolaryngol Head Neck Surg. 1998; 119: 658-664.
EJ, Boouffard JP, et al.  Primary extracranial meningiomas: An analysis of 146 cases.  Head and Neck Pathol 2009; 3:116-130. 
Jun HJ, Im, GJ, et al. Temporal Bone Histopathology Case of the Month � An isolated Middle ear meningioma.  Otology & Neurotology 2012;33:c63-64.
Carroll RS, Glowacka D, Dashner K, Black P. Progesterone receptor expression in meningiomas.  Cancer Res 1993;53:1312.
Louis DN, Ohgaki H, Wiestler OD, Cavenee WK. World Health Organization classification of tumors of the central nervous system. 4th ed. Lyon: IARC Press; 2007.
Whittle IR, Smith C, Navoo P, Collie D. Meningiomas.  Lancet 2004; 363: 1535-43.
O�Reilly RC, Kapadia SB, Kamerer DB. Primary extracranial meningioma of the temporal bone. Otolaryngol Head Neck Surg. 1998; 118: 690-694.
Johnson WD, Loredo LN, Slater JD. Surgery and radiotherapy: complementary tools in the management of benign intracranial tumors. Neurosurg Focus 2008; 24:E2. 
Aghi MK, Carter BS, Cosgrove GR, et al. Long-term recurrence rates of atypical meningiomas after gross total resection with or without postoperative adjuvant radiation. Neurosurgery. 2009; 64: 56-60.
Marosi C, Hassler M, Roessler K, Reni M, Sant M, Massa E et al. Meningioma. Crit Rev Oncol Hematol 2008; 67:153-71.
Yano S, Kuratsu J. Indications for surgery in patients with asymptomatic meningiomas based on an extensive experience.  J Neurosurg 2006; 105: 538-43.
Fischer BR, Palkovic S, Holling M, Niederstadt T, Jeibmann A, Wassman H.  Coexistence of cerebral aneurysm and meningioma � pure accident?  Clinical Neurol and Neurosurg 2009; 111: 647-654.
Javalkar V, Guthikonda B, Vannemreddy P, Nanda A.  Association of meningioma and intracranial aneurysm: report of five cases and review of literature. Neurol India 2009; 57(6):772-6. 














Legends for Figures

Figure 1. MRA.  2.5 mm aneurysm (red arrow) of the left internal carotid artery at the origin of the left ophthalmic artery.

Figure 2. CT of the temporal bone. Axial (A)  and coronal (B) views of the middle ear completely opacified without ossicular erosion.  

Figure 3. MRI. Enhancing granulation tissue within the left tympanic cavity and adjacent mastoid (red arrow).  No enhancing IAC/CPA mass.

Figure 4. Transcanal view of the middle ear meningioma (*). C, chorda tympani; TF, tympanomeatal flap; M, mastoid.

Figure 5. Meningothelial meningioma, WHO grade I. Hematoxylin and eosin stain, 40x.  Inset: Progesterone receptor positivity, 40x.  	





	








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