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Table of Contents
CLINICAL CASES
Year : 2013  |  Volume : 15  |  Issue : 2  |  Page : 41-44

Arteriovenous hemangioma in concha of pinna causing pulsatile tinnitus: Rare case report


1 Departments of ORL, Head and Neck Surgery, Armed Forces Hospital Southern Region, Khamis Mushayt, Saudi Arabia
2 Department of Pathology, Armed Forces Hospital Southern Region, Khamis Mushayt, Saudi Arabia

Date of Web Publication21-Jul-2020

Correspondence Address:
FRCS (Edin), FRCS (Glas) Nadeem W Malik
Consultant ENT Head and Neck Surgeon, Department of ORL, Head and Neck Surgery, Armed Forces Hospital Southern Region
Saudi Arabia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/1319-8491.290348

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  Abstract 


Arteriovenous hemangiomas are benign vascular lesions. A network of arterial and venous structures is characteristic of these cutaneous vascular tumors. Vascular lesions are commonly found in the head and neck region. These lesions may involve the ear, usually extending from external ear canal to involve the tympanic membrane. Lesions limited to the external ear canal without involvement of tympanic membrane are very few and those reported may be counted on fingers. However there is no documented cases of lesions involving and limited only to the concha of the external ear in the English literature. Here we report the first such case of arteriovenous hemangioma involving and limited to the concha of external ear.

Keywords: Arteriovenous hemangioma, arteriovenous malformation, concha of pinna, pulsatile tinnitus


How to cite this article:
Etwadi H, Malik NW, Jamil S. Arteriovenous hemangioma in concha of pinna causing pulsatile tinnitus: Rare case report. Saudi J Otorhinolaryngol Head Neck Surg 2013;15:41-4

How to cite this URL:
Etwadi H, Malik NW, Jamil S. Arteriovenous hemangioma in concha of pinna causing pulsatile tinnitus: Rare case report. Saudi J Otorhinolaryngol Head Neck Surg [serial online] 2013 [cited 2022 Nov 28];15:41-4. Available from: https://www.sjohns.org/text.asp?2013/15/2/41/290348




  Introduction Top


Vascular tumors are common in all ages but they are the commonest tumors in children [1]. In spite of this, vascular tumors continue to present diagnostic and management challenges. One of the reasons for this being the differences among pathologist in diagnosing hemangiomas and vascular malformations. However in 1982 Mulliken and Glowacki [2] proposed a simple classification to reduce the confusion. This was later modified and accepted by the International Society for the Study of Vascular Anomalies [3].

Vascular lesions (hemangiomas and malformations) are widely distributed in the body. They arc benign but may present with variety of complaints due to their size, location and type. When these tumors occur in and or around the ear, they may causc symptoms like deafness, tinnitus and bleeding etc. However occurrence of hemangiomas in the external ear canal is not common. When they occur they usually extend to involve the tympanic membrane.

Hemangioma limited to the external ear canal is very rare and only 7 cases have been described [4]. There is no reported case of hemangioma involving and limited only to the concha of pinna (without extending into the external ear canal). Here we report the first such case in English literature.


  Case Report Top


A 44 years old female patient who presented with left sided tinnitus for three months. The tinnitus was of pulsatile in nature. It was synchronous with her heart beat and increased with physical exertion. There was no associated deafness, otalgia, otorrhoea or vertigo. She gave no history of ear surgery or trauma. Laparoscopic cholecyctectomy was done ten years ago, other than this there was no significant past surgical history. She was a known hypertensive on medical treatment.

On examination there was a 0.5 cm X 0.5 cm mass in the concha of the left pinna. It did not extend into the external ear canal and was covered by normal appearing skin [Figure 1]a. On palpation it was compressible with faint pulsations. Digital pressure over the left lateral neck (carotid artery) reduced the linnitus. Otoscopic examination showed normal external ear canal and tympanic membrane. Hearing assessment was within normal limits. Rest of ear. nose, throat, head and neck examination was also normal.
Figure 1: 1a The mass visible in concha. 1b:Vascular lesion exposed during surgery. 1c: Vascular mass after excision. 1d: MRI showing the vascular lesion with enhancement (White arrow)

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Computerized Tomography with contrast was done. Unfortunately the mass was not visualized well but it did not show intracranial extension or connection of the mass. Magnetic Resonance Imaging (MR1) with contrast was done. The conchal mass was visualized as a vascular abnormality with a clear feeding vessel “t ..‘'-., “ [Figure 1]b. It confirmed that there was no intracranial extension or connection of the vascular mass.

Considering the complaint of pulsatile tinnitus and MRI findings, diagnosis of a vascular lesion was made. It was decided to surgicaily excise the vascular mass. Incision was made at periphery of concha (for cosmetic reasons). Sub-perichondrial dissection was done. The vascular mass identified and dissected free from the tissues [Figure 1]c & [Figure 1]d. The feeding vessel was identified, diathermized and divided. The mass was removed enblock and heniostasis was confirmed. The skin perichondrial flap was returned to original position, incision closcd with 5/0 synthetic non-absorbable suture and pressure dressing applied. The mass was sent for histopathological examination. It was reported as arteriovenous hemangioma [Figure 2]a.
Figure 2: 2a: Low power magnification with vessels and nerves visible. 2b:High power magnification with thick walled vessels (labeled A) and thin walled vessels (labeled V). 2c: High power magnification with nerve labeled N. 2d: Nerve stained brown with S-100 immunohistochemistry labeled N

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On follow-up the pulsatile tinnitus had resolved completely.


  Discussion Top


Vascular lcsionsarc distributed widely in the human body. Although most common in infants, they may occur in all ages and both sexes. They have been described as the oldest documented rumor [1]. Still controversy regards histological diagnosis continues. Hemangioma of one histopathologist may be arteriovenous malformation for other and vice versa. In 1982 Mulliken and Glovvacki [2] proposed a simple classification separating vascular lesions into hemangiomas and malformations. Both may be present at birth, hemangiomas showing rapid neonatal growth but malformations grow slowly with the grow th of whole body. Hypercellularity is a characteristic histological feature of hemangiomas during the proliferative phase while the malformations show normal cellularity.

This classification was later modified and accepted by the International Society for the Study of Vascular Anomalies. They consider hemangioma as true tumor arising due to cellular hyperplasia and vascular malformations arising due to errors of vascular development [5].

There is also WHO classification of superficial vascular tumors which is not simple [6].

The presence of many classifications of benign vascular lesions makes things confusing. However two broad philosophies may be considered: one based primarily on anatomic and histopathologic features and the other based on clinicopathologic features: patient history, physical examination, endothelial characteristics, flow dynamics, and disease natural history. These approaches are useful but not without limitation [7].

On the other hand Weiss and Goldblum [8] describe hemangiomas as benign nonreactive vascular lesions. This description is simple and based on histological findings. The pathologists in our hospital followed this philosophy w hen reporting superficial vascular lesions. We acknowledge that some of reported hemangiomas may represent malformations when considered under other present classifications. We believe that by following this description many confusions and controversies may settle.

Hemangiomas occur frequently in the head and neck region but only rarely involve the external ear. When they occur in the external ear canal they usually involve the tympanic membrane. So far only 19 such cases have been reported and out of these only 7 were limited to the external car canal w ithout involvement of tympanic membrane [9]. But hemangiomas involving and limited to concha of pinna have not been described in the English literature. Here we describe the first such case of arteriovenous hemangioma inv olving the concha of pinna with no extension into the external ear canal. On literature search a case of cavernous hemangioma involving concha of pinna was found but in the Spanish literature [10].

Our patient presented with a short 3 months history of pulsatile tinnitus and swelling in the concha of pinna. Whether it appeared in this period or increased in size from previously smaller swelling could not be ascertained. There w as no history of trauma or infection which may have possibly caused this to happen. Pulsatile tinnitus may be caused by variety of conditions and may need extensive investigations. But here it was clearly caused by this vascular lesion. CT and MRI were done only to delineate the vascular lesion. A clear feeding vessel was visualized on the MRI. Surgical excision was done which resolved the pulsatile tinnitus completely. The microscopic examination of the lesion showed arteries, veins, capillaries and nerves [Figure 2]b, [Figure 2]c, [Figure 2]d. Our pathologist following Weiss and GoldblunTs [8] description of hemangioma reported it as arteriovenous hemangioma. But some may disagree and consider this lesion as arteriovenous malformation [11]. In this case the management would have been same whether it was hemangioma or malformation. But in some cases the correct histological diagnosis may affect the management. The confusion which is prevalent regards hemangioma and malformation needs resolution for uniformity of documentation.


  Conclusion Top


We have described the first case in English literature of hemangioma involving the concha of pinna. It presented with pulsatile tinnitus which resolved completely after enbloc excision. CT and MRI were done to determine the extent of the lesion before excision. We also highlighted the confusion and controversy that exists regards vascular lesions being histologically labeled as hemangioma or malformation.



 
  References Top

1.
Blei F. Vascular anomalies: from bedside to bench and back again. Curr Probl Pediatr Adolesc Health Care.2002; 32:72-93.  Back to cited text no. 1
    
2.
Mulliken JB, Glowacki J. Hemangiomas and vascular malformations in infants and children: a classification based on endothelial characteristics. Plast Reconstr Surg. 1982; 69: 412—422.  Back to cited text no. 2
    
3.
Enjolras O, Mulliken JB. Vascular tumors and vascular malformations (new issues). Adv Dermatol. 1997; 13:375- 423.  Back to cited text no. 3
    
4.
Martines F, Bentivegna D, Maira E, Marasa S, Ferrara S. Cavernous haemangioma of the external auditory canal: clinical case and review of the literature. Acta Otorhinolaryngol Ital. 2012 February; 32(1): 54-57.  Back to cited text no. 4
    
5.
Enjolras O, WassefM, Chapot R. Introduction: ISSVA classification. In: Color atlas of vascular tumors and vascular malformations. Cambridge, UK: Cambridge University Press, 2007.  Back to cited text no. 5
    
6.
Sangueza OP, Kasper RC, LeBoit P ct al. Vascular tumors. In: LeBoit PE, Burg G, Weedon D, Sarasin A, eds. WHO classification of tumors, pathology, and genetics: skin tumors. Lyon, France: [ARC Press, 2006:233-246.  Back to cited text no. 6
    
7.
Kransdorf MJ, Murphey MD, Fanburg-Smith JC. Classification of Benign Vascular Lesions: History, Current Nomenclature, and Suggestions for Imagers. Am J Roentgenol. July 2011 vol. 197 no. 1 8-11.  Back to cited text no. 7
    
8.
Weiss SW, Goldblum JR. Enzingers & Weiss’s soft tissue pathology, 5th ed. Philadelphia. PA: Elsevier, 2008:633- 679.  Back to cited text no. 8
    
9.
Martines F, Bentivegna D, Maira E, Marasa S.Ferrara S. Cavernous haemangioma of the external auditory canal: clinical case and review of the literature. Acta Otorhinolaryngol Ital. 2012 February; 32( 1): 54-57.  Back to cited text no. 9
    
10.
Garcia-Purrinos FJ, Lemberg P, Calvo J. Subcutaneous sinusoid cavernous hemangioma of the outer ear. Review of literature [Article in Spanish]. Acta Otorrinolaringol Esp. 1998 Oct;49(7):576-8.  Back to cited text no. 10
    
11.
Adegboyega PA, Suimin Qiu S. Hemangioma Versus Vascular Malformation. Presence of Nerve Bundle Is a Diagnostic Clue for Vascular Malformation. 772-775 Arch Pathol Lab Med. Vol 129, June 2005.  Back to cited text no. 11
    


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  [Figure 1], [Figure 2]



 

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