SOLITAIRE 2 ™

REVASCULARIZATION DEVICE IS THE STANDARD IN

INTERVENTIONAL STROKE CARE

VOLUME 28 - No. 2 - april 2015

CLINICAL STUDIES

ISSN 1971-4009 Online ISSN 2385-1996

• Solitaire FR With the Intention For Thrombectomy as Primary Endovascular Treatment for Acute Ischemic Stroke (SWIFT PRIME). The New England Journal of ™

NRJ - THE NEURORADIOLOGY JOURNAL - VOLUME 28, No. 2 - pages 81- 228, 2015

Medicine, April 2015

• Solitaire™ FR With the Intention For Thrombectomy (SWIFT). The Lancet, 2012 • Solitaire™ FR Thrombectomy for Acute Revascularization (STAR). Stroke AHA Journals, 2013

• The Endovascular Treatment for Small Core and Proximal Occlusion Ischemic Stroke (ESCAPE) Study. The New England Journal of Medicine, February 2015 • Extending the Time for Thrombolysis in Emergency Neurological Deficits – Intra-Arterial (Extend-IA) Study. The New England Journal of Medicine, February 2015 • MR CLEAN: A Multi-center Randomized Clinical trial of Endovascular treatment for Acute Ischemic Stroke in the Netherlands. The New England Journal of Medicine, December 2014

• REVASCAT: Update on a Recently Terminated Randomized Trial of Mechanical Embolectomy With The Solitaire™ Device versus Best Medical Therapy In Acute Stroke. The New England Journal of Medicine, April 2015

The Solitaire™ device has been observed to double the rate of good neurological outcomes (71% vs. 40%) and reduce the rate of mortality by half (9% vs. 20%) compared to standard of care treatment alone. 1, 2, 3, 4, 5, 6 Clinical References: 1. EXTEND-IA Study presented by Dr. Bruce Campbell at the International Stroke Congress 2015 2. ESCAPE Study presented by Dr. Michael Hill at the International Stroke Congress 2015

Official Journal of:

3. Trevo Versus Merci Retrievers for Thrombectomy Revascularisation of Large Vessel Occlusions in Acute Ischaemic Stroke (TREVO 2): a randomised trial. Nogueira et. al., doi:10.1016/S0140-6736(12)61299-9.

AINR - Associazione Italiana di Neuroradiologia

4. Broderick, Joeseph, et. Al. Endovascular Therapy after Intravenous t-PA versus t-PA Alone for Stroke. NEJM. vol. 368 no. 1 0

and: The Neuroradiologists of Alpe-Adria ANRS - Albanian Neuroradiological Society PANRS - Pan Arab NeuroRadiology Society Radiological Society of Saudi Arabia, Division of Neuroradiology Egyptian Society of Neuroradiology ISNR - Indian Society of Neuroradiology Indonesian Society of Neuroradiology Neuroradiology Section of the Radiology Society of Iran Israeli Society of Neuroradiology College of Radiology Malaysia

5. Presented by Dr. Dippel, Erasmus University at World Stroke Congress 2014 6. ADAPT FAST Study: Turk AS, et al. J NeuroIntervent Surg 2014;0:1–5. doi:10.1136/neurintsurg-2014-011125

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Neuroradiology Section - Pakistan Psychiatry Research Center Section of Neuroradiology - Polish Radiological Society The Neuroradiologists of Romania Section of Neuroradiology of Serbia and Montenegro SILAN - Sociedad Ibero Latino Americana de Neurorradiologia Neuroradiology Section of Singapore Radiological Society Slovenian Society of Neuroradiology The Neuroradiological Society of Taiwan TSNR - Turkish Society of Neuroradiology

Case report

Intraosseous Hemangioma of the Middle Turbinate: A Case Report of a Rare Entity and Literature Review

The Neuroradiology Journal 2015, Vol. 28(2) 148–151 ! The Author(s) 2015 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/1971400915576653 neu.sagepub.com

Ryan Goff1, Steven Weindling1, Vivek Gupta1 and Aziza Nassar2

Abstract Intraosseous hemangiomas arising from the nasal turbinate are exceedingly rare, with few reported cases in the literature. We describe a 61-year-old man found to have a nasal cavity mass on sinus computed tomograph (CT) and magnetic resonance imaging (MRI). Although an atypical site of occurrence, distinctive internal honeycomb bony trabeculations demonstrated on CT allowed the correct diagnosis of an intraosseous hemangioma to be prospectively proposed by the interpreting radiologist which had direct clinical and surgical impacts.

Keywords hemangioma, nasal cavity, CT, MRI

Introduction Soft tissue and intraosseous hemangiomas are frequently encountered in the head and neck. The most common sites of head and neck intraosseous hemangiomas include the skull (53%), mandible (10.7%), nasal bones (9%), and the cervical spine (6%).1 Hemangiomas are uncommon within the nasal cavity and paranasal sinuses, making up less than 20% of benign nasal cavity lesions.2 Nasal cavity and paranasal sinus hemangiomas most frequently originate from the mucosa and are rarely intraosseous,3 with few previously reported cases arising from the nasal turbinate.2–6 We describe a case of an intraosseous cavernous hemangioma of the nasal turbinate and propose that this rare diagnosis may be suggested when a nasal cavity mass demonstrates characteristic internal bony trabeculations on computed tomography (CT).

Case Report A 61-year-old man was referred to our otorhinolaryngology department for intermittent right epistaxis for approximately two to three years. Sinonasal endoscopy revealed a right nasal cavity mucosal ulceration, likely responsible for the epistaxis. On the left, edematous intact nasal mucosa was noted between the middle turbinate and nasal septum. Sinus CT demonstrated a 2.5  3.6  3.4 cm expansile left nasal cavity enhancing mass (Figure 1a) causing secondary remodeling of the left maxillary antrum, medial orbital wall, and rightward deviation of the nasal septum.

Secondary obstruction of the left frontoethmoidal recess resulted in left frontal sinus and anterior ethmoidal air cell opacification by hyperdense secretions that could not be readily differentiated from the adjacent enhancing mass. The mass lesion’s most distinctive CT feature was thin internal honeycomb bony trabeculations best seen on bone algorithm reformations (Figure 1b), and hemangioma was suggested on the basis of this finding. Magnetic resonance imaging (MRI) was subsequently performed for further lesion delineation, assessment of lesion vascularity and operative planning. While T1-weighted images demonstrated a well-defined, homogeneous, isointense mass inseparable from adjacent obstructed secretions (Figure 2a), T2-weighted images allowed the moderately hyperintense mass to be easily differentiated from very hyperintense post-obstructive secretions (Figure 2b). On post-gadolinium T1-weighted images, the avidly enhancing mass contained non-enhancing thin, serpiginous, hypointense foci corresponding with bony trabeculations seen on CT, but was lacking internal ‘‘flowvoids’’ suggestive for hypervascularity that might alter surgical approach or elicit preoperative embolization (Figure 2c). The MRI imaging appearance suggested 1

Department of Radiology, Mayo Clinic, Jacksonville, FL, USA Department of Laboratory Medicine and Pathology at Mayo Clinic; Jacksonville, FL, USA

2

Corresponding author: Ryan Goff, MD, Department of Radiology, Mayo Clinic4500 San Pablo Road, Jacksonville, FL 32224. Email: [email protected]

Goff et al.

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Figure 1. a) Coronal post-contrast CT image shows an enhancing left nasal cavity mass (white arrow) causing smooth bony remodeling of the nasal septum, inferior orbital wall, and left maxillary sinus. The mass obstructs the ipsilateral frontoethmoidal recess with poor differentiation from adjacent post-obstructive secretions (black arrow). b) Sagittal bone window CT image demonstrates an expansile mass in the left nasal cavity with fine internal honeycomb bony trabeculations.

Figure 2. a) Coronal T2 MRI image nicely shows the demarcation of the soft tissue mass (white arrow) and the obstructed secretions (black arrow). Coronal T1 pre- (b) and post-gadolinium (c) images show a T1 isointense, avidly enhancing mass (white arrow) without internal ‘‘flow voids’’ to indicate hypervascularity. Post-contrast T1-weighed images confirm lack of tumor intraorbital or intracranial extension.

a benign pathology, and hemangioma was again proposed as the most likely etiology on the basis of internal bony trabeculations seen at CT, commonly seen with intraosseous hemangiomas elsewhere in the body. The patient underwent uncomplicated, complete endoscopic resection of the mass, which was contiguous with the left middle turbinate and contained internal bony components. The patient did well postoperatively and was free of symptoms at follow-up. Pathology revealed a benign cavernous hemangioma. The specimen demonstrated characteristic large dilated blood-filled venous spaces lined by flattened endothelium with a background fibrotic stroma and interspersed trabecular bone elements (Figure 3). There was no evidence of cytologic atypia or tufting of the vascular endothelial lining to suggest malignancy.

Discussion Hemangiomas, originally classified based on the size of the intralesional vascular channels, include capillary,

strawberry, cavernous, and mixed subtypes. Although this nomenclature is still common among many subspecialties, including pathologists, it is being replaced by a system described in 1982 by Mulliken and Glowacki7 which takes into account the lesion’s histology, biologic behavior, and natural history. Under this system, vascular anomalies are classified as either neoplasms or malformations based on the presence or absence of endothelial proliferation. Vascular malformations are further subcategorized into low-flow and high-flow lesions. Most of the so-called ‘‘hemangiomas’’ seen on adult head and neck imaging studies are low-flow venous malformations,8 and are of little clinical significance unless complicated by hemorrhage, secondary mass effect, or compromise of the organ’s integrity. Hemangiomas, or low-flow vascular malformations, of the paranasal sinuses and nasal cavity may arise from the mucosa or underlying bony structures. When encountered in the nasal cavity, hemangiomas of mucosal origin most commonly arise from the nasal septum (65%), lateral nasal wall (18%) and

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Figure 3. Medium power microscopic image shows cavernous dilated blood vessels filled with red blood cells. There is a background fibrotic stroma with interspersed trabecular bone elements (black arrows). The overlying ciliated columnar respiratory epithelium of the nasal turbinate mucosa is depicted at the far right of the image. (Hematoxylin and eosin stain – 20  magnification).

vestibule (16%).1 The predominant septal location is attributed to the presence of the anteriorly located Kiesselbach’s plexus at the anastomosis of the anterior ethmoidal, sphenopalatine, greater palatine, and septal branch of the superior labial arteries.1 Hemangiomas arising from the nasal cavity mucosa have been well described in the literature.9–12 CT depicts a well-circumscribed enhancing mass with or without adjacent bony remodeling. Phleboliths, often found within cavernous hemangiomas, are rarely seen in mucosal hemangiomas, which are mostly of the capillary subtype. MRI features include a T1 isointense to hypointense mass with high signal intensity on T2 weighted sequences and intense post-contrast enhancement. Internal vascular ‘‘flow voids’’ are unusual, although they have been described in larger lesions. Intraosseous hemangiomas arising from the inferior turbinate8,10 and middle turbinate4 have been described in case reports. Patients typically present with recurrent epistaxis or sinus obstruction. Our patient’s intraosseous cavernous hemangioma of the middle turbinate demonstrated nonspecific MR features similar to previously described nasal mucosa hemangiomas and many other nasal tumors. However, the lesion’s distinctive thin internal honeycomb bony trabeculations demonstrated on CT, characteristic of intraosseous hemangiomas found in other bones, suggested this rare diagnosis. Previous reports in the literature have shown similar CT findings, with their appearance described as ‘‘soap bubble,’’ ‘‘sunburst spiculations,’’ and ‘‘radiating trabeculae.’’ In our patient, MRI demonstrated a circumscribed, T1 isointense, T2 hyperintense avidly enhancing mass. Thin, serpentine internal hypointensities on T1-weighted post-contrast images were consistent with the internal trabecula demonstrated on CT and were felt not to represent intralesional vascular ‘‘flow-voids.’’ MRI

The Neuroradiology Journal 28(2) findings of nasal turbinate intraosseous hemangioma have not been described in prior case reports. However, our findings are in keeping with previously described mucosal and intraosseous sinonasal hemangiomas. Although the diagnosis of an intraosseous hemangioma is primarily based on the characteristic CT appearance, we believe MRI was a complementary imaging modality in our case, affording better delineation of lesion margins in relation to obstructed mucous and assessment of lesion vascularity prior to surgical resection. Additionally, MRI may be useful in detecting intracranial, intraorbital and perineural extension. Surgical excision via sinonasal endoscopy is the treatment of choice for symptomatic nasal cavity and paranasal sinus hemangiomas and is typically curative. Preoperative embolization has been advocated to reduce the risk of intraoperative bleeding in hypervascular lesions.13 However, the lack of internal vascular ‘‘flow voids’’ in our case obviated the need for this intervention. While the differential diagnosis for a soft tissue mass of the nasal cavity is lengthy, it is more limited for masses containing fragments of destroyed bone, intratumoral calcification, or matrix and would include squamous cell carcinoma (SCC), sinonasal undifferentiated carcinoma (SNUC), inverted papilloma, ossifying fibroma, esthesioneuroblastoma, and chondrosarcoma. Entrapped destroyed bony fragments seen with both SCC and SNUC could mimic internal trabeculation, but an aggressive, infiltrative growth pattern helps distinguish these lesions from hemangiomas. Although inverted papillomas and esthesioneuroblastomas can occasionally have intratumoral calcifications or entrapped bony fragments, important differentiating imaging features exist between these tumors and hemangiomas. Inverted papillomas typically arise from the lateral wall of the nasal cavity with maxillary antral extension and may demonstrate a so-called ‘‘cerebriform’’ MRI appearance. Esthesioneuroblastomas are classically centered around the roof of the nasal cavity, frequently extending into the anterior cranial fossa with waisting at the level of the cribriform plate, and may demonstrate cysts at the brain-tumor interface. Facial ossifying fibromas most commonly arise in the mandible but can be seen in the nasal cavity and paranasal sinuses. They are wellcircumscribed expansile masses, which, in contrast to hemangiomas, often demonstrate a peripheral ossified rim, central fibrous matrix and heterogeneous or peripheral enhancement. Lastly, chondrosarcoma characteristically contains ‘‘arc and ring’’ chondroid matrix with a more aggressive imaging appearance. Hemangioma should be considered in the differential diagnosis of a nasal cavity mass with internal calcifications even when arising primarily from a nasal turbinate. While MRI is commonly performed to characterize paranasal sinus masses, the thin internal bony trabeculations demonstrated on bone algorithm CT are key to the radiologic diagnosis of nasal turbinate hemangiomas. Unnecessarily aggressive treatment for a

Goff et al. sinonasal mass may therefore be avoided when a benign appearing expansile nasal turbinate mass containing fine internal honeycomb trabeculations is demonstrated on paranasal sinus CT imaging. Funding This research received no specific grant from any funding agency in the public, commercial or not-for-profit sectors.

Conflict of interest The authors declare no conflict of interest.

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