ARTICLE IN PRESS+Model

Braz J Otorhinolaryngol. 2015;xxx(xx):xxx---xxx

www.bjorl.org

Brazilian Journal of

OTORHINOLARYNGOLOGY

CASE REPORT

Maxillary sinus hemangioma: usefulness ofembolization according to classification�

Hemangioma de seio maxilar: utilidade da embolizacão de acordo com aclassificacão

Hisashi Hasegawaa, Hiroumi Matsuzakia,∗, Tohru Furusakaa, Takeshi Oshimaa,Shinobu Masudab, Toshiyuki Unnoc, Osamu Abec

a Department of Otorhinolaryngology, Head and Neck Surgery, Nihon University School of Medicine, Japanb Department of Pathology, Nihon University School of Medicine, Japanc Department of Radiology, Nihon University School of Medicine, Japan

Received 19 June 2015; accepted 12 September 2015

C

A

C

AbsscsawimSat

Introduction

Hemangioma in the nose and nasal sinuses is rare,1 particu-larly in the maxillary sinus.2 Differentiation of hemangiomafrom malignant tumors is important, because both may beaccompanied by bone destruction. With regard to gianthemangioma, this type has a rich blood supply, and surgi-cal treatment requires particular care due to the high riskof massive bleeding and obstructed field of view. Althoughmaxillary sinus hemangioma has been sporadically reported,few reports have described cases of this type diagnosedaccording to the 1996 classification of the International Soci-ety for the Study of Vascular Anomalies (ISSVA) and treatedwith arterial embolization.

Here, we report three cases of massive sinonasal hea-mangioma that were resected after arterial embolization.

� Please cite this article as: Hasegawa H, Matsuzaki H, FurusakaT, Oshima T, Masuda S, Unno T, et al. Maxillary sinus hemangioma:

usefulness of embolization according to classification. Braz J Otorhi-nolaryngol. 2015. http://dx.doi.org/10.1016/j.bjorl.2015.09.002

∗ Corresponding author.E-mail: matuzaki.hiroumi@nihon-u.ac.jp (H. Matsuzaki).

ttwt

http://dx.doi.org/10.1016/j.bjorl.2015.09.0021808-8694/© 2015 Associacão Brasileira de Otorrinolaringologia e Cirureserved.

ase report

n overview of the three cases is shown in Table 1.

ase 1

43-year-old man presented with recurrent right nasalleeding and nasal obstruction for six months. Mucosalwelling was observed from the maxillary sinus to the nasaleptum in the right inferior and middle nasal meatuses. Sinusomputed tomography (CT) revealed a soft tissue densityhadow, with a contrast-enhanced interior in a scatterednd gradually increasing pattern (Fig. 1). No calcificationas observed. Gadolinium contrast magnetic resonance

maging (MRI) showed that the inside of the mass wasarkedly contrast-enhanced, suggesting a vascular tumor.

phenopalatine arteriography showed tumor staining with collection of thin, meandering arteries extending fromhe posterior superior alveolar artery and descending pala-

ine artery. The tumor, which was contrast-enhanced duringhe arterial phase, was a slow-flow vascular lesion, fromhich the contrast medium was partially washed out in

he late phase, and was diagnosed as venous malformation

rgia Cérvico-Facial. Published by Elsevier Editora Ltda. All rights

BJORL-275; No. of Pages 4

ARTICLE IN PRESS+Model

2 Hasegawa H et al.

Table 1 An overview of the three cases.

Case 1 2 3

Age gender 41 y.o. male 43 y.o. female 74 y.o. malePreoperated biopsy Hematoma Necrotic polypoid Nasal mucosaOrigin Lateral wall Upper wall UnknownArtery Posterior superior alveolar

descending palatineInfraorbital dorsal nasal Sphenopalatine deep temporal

Embolic device Micro coil and gelatin sponge Micro coil and gelatin sponge Micro coil and gelatin spongeTechnique ESS and Watsuji---Denker

operationESS and Watsuji---Denker operation ESS and Watsuji---Denker

operationISSVA classification Low flow venous malformation Low flow venous malformation Low flow venous malformation

Bleeding (g) 60 30 100ernou

aebW

3

Fi

Fi(a

Diagnosis Sinusoidal hemangioma Cav

ccording to the ISSVA classification. Both arteries were

mbolized. Three days later, the patient underwent com-ined surgery with an endoscopic procedure and theatsuji---Denker operation. Intraoperative blood loss was

hIh

igure 1 Enhanced CT in Case 1. Axial (A) and coronal (B) imagess seen during the arterial phase.

igure 2 Pathological findings in Case 1 (A) and Case 3 (B). (A) Tntermingling fibrous capsule and aggregation of vessel structures. ThB) The submitted materials contained thick fibrous capsule and vere consistent with cavernous hemangioma.

s hemangioma Cavernous hemangioma

0 mL. Sinusoidal hemangioma, a variant of cavernous

emangioma, was diagnosed by histopathology (Fig. 2A).n 24 months of follow-up conducted every three months,owever, we have yet to recognize recurrence.

. A strong, spreading, and gradually increasing contrast effect

he submitted material consisted of abundant coagulation withe vessels are thin and delicate, with an anastomosing structure.ssel structures. Morphological features of the vessel structure

ARTICLE IN PRESS+Model

Maxillary sinus hemangioma: usefulness of embolization according to classification 3

Figure 3 Gadolinium-enhanced MRI of Case 2. Axial (A) and coronal (B) images. A heterogeneously enhanced vascular tumor was

tebrtpabsaviuaeo(m

D

Iootnsoroat

iifs1

suspected.

Case 2

A 41-year-old woman presented with recurrent left nasalbleeding and nasal obstruction for a month. Her left com-mon nasal meatus was filled with edematous mucosa. SinusCT revealed a soft tissue density shadow occupying theleft maxillary sinus, ethmoid sinus, frontal sinus, and nasalcavity, with a bone defect on the medial wall of theleft maxillary sinus. In addition, CT showed a soft tis-sue density shadow including blood retention that filledthe left maxillary sinus. The medial wall of the maxillarysinus showed destruction and was increasingly contrast-enhanced. A 30 mm heterogeneously enhanced vasculartumor was suspected, based on gadolinium-enhanced MRI(Fig. 3). Facial arteriography showed tumor staining fromthe infraorbital artery on the upper wall of the left maxillarysinus, and the artery was embolized. In addition, the dor-sal nasal artery was embolized. The tumor was a slow-flowvascular lesion, and venous malformation was diagnosedaccording to the ISSVA classification. Four days later, thepatient underwent combined surgery with an endoscopicprocedure and the Watsuji---Denker operation, and the entirelesion was extracted. Intraoperative blood loss was 30 mL.The postoperative pathological diagnosis was cavernoushemangioma. In 18 months of follow-up conducted everythree months, however, we have yet to recognize recur-rence.

Case 3

A 74-year-old man presented with right nasal bleedingand nasal obstruction. Two years before, the patient hadexperienced right nasal bleeding lasting a few hours.Myelodysplastic syndrome was diagnosed. An edematouspolyp occupying the right common nasal meatus wasobserved. Sinus CT showed a soft tissue density shadowoccupying the right maxillary sinus, ethmoid sinus, andfrontal sinus to the left nasal cavity, with bone destruc-

tion on the medial wall of the right maxillary sinus. ContrastCT showed a lobulated tumorous lesion that was graduallycontrast-enhanced from the maxillary sinus to the commonnasal meatus, as well as microvascular development in the

tfrt

umor. Gadolinium-enhanced MRI showed a heterogeneouslynhanced lesion, raising suspicion of a vascular tumor. Tissueiopsy showed only nasal mucosa. External carotid arteriog-aphy showed tumor staining in the area corresponding tohe entire right maxillary sinus. Sphenopalatine arteriogra-hy showed that the infraorbital artery, descending palatinertery, and a number of small medial/lateral posterior nasalranches were involved as feeders. The main trunk of thephenopalatine artery and branches of the deep temporalrtery were embolized (Fig. 4). The tumor was a slow-flowascular lesion diagnosed as venous malformation accord-ng to the ISSVA classification. Four days later, the patientnderwent combined surgery with an endoscopic procedurend the Watsuji---Denker operation, and the entire lesion wasxtracted. Intraoperative blood loss was 100 mL. The post-perative pathological diagnosis was cavernous hemangiomaFig. 2 B). In 18 months of follow-up conducted every threeonths, however, we have yet to recognize recurrence.

iscussion

n a report of 281 cases of hemangioma, 55 percent occurredn the face and extracranial head, and 5 percent occurredn the neck,3 with these lesions only rarely occurring inhe nose or nasal sinuses.1 Of those which do occur in theose and nasal sinuses, about 80 percent occur on the nasaleptum, in Kiesselbach’s area in particular, and 15 percentn the lateral wall of the nasal cavity. Hemangioma occur-ing in the sinus cavity is even rarer. Cavernous hemangiomaccurs far less frequently than capillary hemangioma, usu-lly developing on turbinate bone and rarely on the bonyissue of the maxillary sinus wall.4

Sinusoidal hemangioma,5,6 diagnosed on histopathologyn Case 1, is a markedly rare histological type (Fig. 4) whichs relatively unknown among pathologists. Because only aew cases of sinusoidal hemangioma have been reportedince the initial description by Calonje and Fletcher in991, a clear recognition of the clinicopathological charac-

eristics of this type will avoid diagnostic pitfalls. Patientollow-up has revealed no tendency toward either localecurrence or metastasis5; however, Ciurea et al. reportedhree reoperations for recurrence.7 In our cases, the

ARTICLE IN PRESS+Model

4 Hasegawa H et al.

F A) ans ery (

pivrlh

amnvtsgitami

otmiFbptesievFnofl

C

TfAdTta

C

T

R

report. Dentomaxillofac Radiol. 2012;41:340---1.10. Song CE, Cho JH, Kim SY, Kim SW, Kim BG, Kang JM. Endoscopic

resection of haemangiomas in the sinonasal cavity. J Laryngol

igure 4 External carotid arteriography of Case 3. Frontal (ponding to the entire right maxillary sinus. Sphenopalatine art

reoperative CT showed bone destruction. Given the rar-ty of sinusoidal hemangioma and the possibility that otherascular neoplastic lesions might also be present, a highisk of local recurrence and metastasis lesions with simi-ar CT and MRI findings, such as hemangiopericytoma andemangiosarcoma in these cases, cannot be denied.

Hemangioma is commonly diagnosed radiographically,ccording to the classification adopted at the 1996 ISSVAeeting. In the ISSVA classification, conventionally diag-

osed hemangiomas are divided into vascular tumors andascular malformations.8 Vascular malformations, conven-ionally classified as cavernous hemangiomas, do not regresspontaneously and sometimes require treatment when theyradually increase in size. They may be further subdividednto slow- and fast-flow lesions; fast-flow lesions can bereated with semipermanent and reliable embolization, suchs via micro coils. Knowing the flow speed helps to deter-ine the embolization method and is a useful reference for

ntravascular treatment.We considered that our surgical method was effective

n the basis of two criteria: the low rate of periopera-ive hemorrhage and the reported successful postoperativeanagement. We were able to limit intraoperative bleed-

ng to 60 mL, 30 mL, and 100 mL for Cases 1---3, respectively.urther, postoperative bleeding was sufficiently controlledy the placement of gauze tampons. In a previous report, aatient who did not undergo arterial embolization requiredwo emergency room visits for the treatment of postop-rative bleeding.9 Another study reported that massiveinonasal hemangioma was able to be resected endoscop-cally, albeit with effort, including preoperative arterialmbolization and an increase in surgical access to the tumoria the construction of a control hole at the canine fossa.10

or our three patients, in contrast, we chose the combi-ation of an endoscopic procedure and the Watsuji---Denkerperation with preoperative arterial embolization, whichacilitated our investigation and care of the postoperative

esion.

d profile (B) views, showing tumor staining in the area corre-arrow).

onclusion

hese three cases were diagnosed as slow-flow venous mal-ormations, in accordance with the ISSVA classification.ngiography and arterial embolization both aided accurateiagnosis of the primary site and the control of bleeding.hese very rare cases are amenable to relatively rou-ine treatment following careful assessment and a plannedpproach.

onflicts of interest

he authors declare no conflicts of interest.

eferences

1. Fu YS, Perzin KH. Non-epithelial tumors of the nasal cavity,paranasal sinuses, and nasopharynx: a clinicopathologic study.3. Cartilaginous tumors (chondroma, chondrosarcoma). Cancer.1974;34:453---63.

2. Batsakis JG, Rice DH. The pathology of head and neck tumors:vasoformative tumors, part 9A. Head Neck Surg. 1981;3:231---9.

3. Macomber W, Wang M. The hemangioma. GP. 1953;8:41---9.4. Hellquist HB. Pathology of the nose and paranasal sinuses. But-

terworths Editions; 1990.5. Calonje E, Fletcher CD. Sinusoidal hemangioma. A distinctive

benign vascular neoplasm within the group of cavernous heman-giomas. Am J Surg Pathol. 1991;15:1130---5.

6. Jammal H, Barakat F, Hadi U. Maxillary sinus cavernous heman-gioma: a rare entity. Acta Otolaryngol. 2004;124:331---3.

7. Ciurea M, Ciurea R, Popa D, Parvanescu H, Marinescu D, VrabeteM. Sinusoidal hemangioma of the arm: case report and reviewof literature. Rom J Morphol Embryol. 2011;52:915---8.

8. Enjolras O, Wassef M, Chapot R. Color atlas of vascular tumorsand vascular malformations. Cambridge University Press; 2007.

9. Vargas MC, Castillo M. Sinonasal cavernous haemangioma: a case

Otol. 2009;123:868---72.