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Pleomorphic Adenoma of External Auditory Canal

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증례6

J Korean Skull Base Society 9권 1호 : 41~45, 2014

논문 접수일 : 2014년 4월 25일 심사 완료일 : 2014년 5월 20일

주소 : Department of Otolaryngology-Head

& Neck Surgery, College of medicine, The Catholic University of Korea

222 Banpo-daero, Seocho-gu, Seoul 137-701, Korea Tel : 82-2-2258-6213 Fax : 82-2-595-1354 E-mail : khpent@catholic.ac.kr

Kyoung Ho Park, MD, PhD

교신저자

종설1 종설2 종설3 증례1 증례2 증례3 증례4

가톨릭대학교 의과대학 이비인후과학교실 최용석, 정영훈, 맹재환, 박경호 증례5

Department of Otolaryngology-Head & Neck Surgery, College of medicine, The Catholic University of Korea, Seoul, Korea

Yongsug Choi, MD, Young Hoon Jung MD., Jae Hoan Maeng, MD., Kyoung Ho Park, MD, PhD

외이도에 발생한 다형선종

Pleomorphic Adenoma of External Auditory Canal

Pleomorphic adenoma is the most common benign salivary gland tumor, it comprises about 80% of benign salivary gland tumors, and about 65% of all type of salivary gland tumors.

Case reports of this tumor arising from the nasal septum, tongue, turbinate, upper lip, lungs, trachea and lacrimal glands have been found in the literature. But case reports of pleomorphic adenoma from the external auditory canal have been extremely few.2)

We report the case of a pleomorphic adenoma in a 70-year-old man, and do an overall review about this tumor.

Pleomorphic adenoma External auditory canal

Key Words

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▒ Introduction

Pleomorphic adenoma occurs most frequently in 4-5th decade and in female, and extremely rare in infant.1) Of 80%

Pleomorphic adenoma occurs in parotid gland, and it constitutes approximately 40-45% of parotid gland tumor.

But the case of pleomorphic adenoma occurred in external auditory canal is extremely rare.

Glandular structure of the EAC is composed of several glands; ceruminous glands, sebaceous glands, and apocrine glands. But Eccrine sweat glands are not present in the EAC.

The first case was reported by Haugh in 1894, and he called it ceruminoma, and this term includes both benign and malignant tumor. This term has been used long time but it has leaded to confusion and misunderstanding about this tumor. Nowadays, this term is gradually unused.

Glandular orgin tumors of EAC is rare and reported less than 150 cases. Furthermore the pleomorphic adenoma is even rarer, only less than 40 cases were reported and published in English are more fewer.2-7)

In this article, we will present a case of Pleomorphic adenoma occurs in EAC, and do an overall review about this tumor.

▒ Case

A 70 year old male patient, presented as an outdoor patient with chief complaints of left external auditory canal swelling for about 1 year. He had hearing impairment and earfullness on left side, and did not complain about otalgia, otorrhea, tinnitus, dizziness. He took medication for hypertension, and aspirin for right cerebral infarction history in 2004. He was a current smoker and had 15 pack years smoking history.

On physical examination, right tympanic membrane and external auditory canal was normal, left tympanic membrane couldn’t be examined due to severe external auditory canal swelling. In addition to that there was not abnormal finding in nose and pharynx & larynx.

Pure tone audiometry showed 60db mixed type hearing loss on left side. On Speech audiometry, SRT 45dB, SDS 60 checked on left side(Fig. 1).

Temporal bone CT showed an about 1.2×1.2 cm size round

& soft tissue density mass on cartilage segment left external auditory canal, an about 1.7×0.5 cm size soft tissue density ovoid mass like lesion on bony segment of external auditory canal(fig. 2).

Temporal bone MRI T1 GD enhance images showed an about 1.2×1.2 cm size high signal density mass in cartilage segment of left external auditory canal, an about 1.7×0.5 cm

Pure tone audiometry showed 60db mixed type hearing loss on left side.

On Speech audiometry, SRT 45dB, SDS 60 checked on left side.

Fig. 1 A

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size low signal density lesion in bony segment of external auditory canal.

Otherwise, a large old infarction was found in right frontal lobe including precentral gyrus, which totally obstructed Rt.

ICA, and it was considered due to CVA history in 2004(fig. 3).

On chest PA film, there was subsegmental atelectasis in right lower lung, so we consulted pulmonology. On chest CT, though there was bronchiolitis in right lower lung field, pulmonary function test was with in normal limit, we

received reply from pulmonology that the lung lesion is not contraindication of operation.

Due to the lesion of brain, we consult neurology. On Neck MRA scan showed right MCA territory large old infarction, right ICA proximal portion total occlusion, left PCA stenosis.

On neurology reply for operability, there is a few risk for infarction, they recommend us to use some medications (asprin, clopidogrel, pravastatin, acetylcarnitine) pre & post operation.

Temporal bone MRI T1 GD enhance images showed an about 1.2 x 1.2 cm size high signal density mass in cartilage segment of left external auditory canal(black arrow), an about 1.7 x 0.5 cm size low signal density lesion in bony segment of external auditory canal.

A large old infarction was found in right frontal lobe including precentral gyrus, which totally obstructed Rt.

ICA, and it was considered due to CVA history in 2004.

(white arrow)

Fig. 3

Temporal bone CT showed an about 1.2×1.2cm size round & soft tissue density mass on cartilage segment left external auditory canal(black arrow), an about 1.7×0.5cm size soft tissue density ovoid mass like lesion on bony segment of external auditory canal(white arrow)

Fig. 2

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There was no abnormal finding in CBC, blood chemistry, urine analysis.

We performed mass excision under general anesthesia on 2012.9.13. We used posterior auricle approach, an about 1.3 cm size encapsulated mass in cartilage segment of external auditory canal was resected clearly.

On bony segment of external auditory canal, there was filled with cerumen, we removed it and irrigated with saline.

The lesion of bony segment of external auditory canal, which was found in CT & MRI scan, seems to this.

The tympanic membrane was intact. Subcutaneous layer approximation with vicryl, skin incision was covered by surgiseal. After external auditory canal packing with furacin gauze packing and compression dressing, the operation was finished(fig. 4).

On POD 1, the compression dressing and packing furacin gauze was removed, and we discharged the patient, and operation wound was healed well without complication.

The pathologic result was ceruminous pleomorphic adenoma(fig. 5,6). We recommended patient to follow up regularly, but he didn’t come our clinic again.

▒ Discussion

Pleomorphic adenoma is also known as mixed tumor and it derives this name because morphologic diversity with both epithelioid and connective tissue components in various proportions.

Pleomorphic adenomas occur most frequently in the major salivary glands but may also arise from the minor salivary glands of the upper respiratory and alimentary tracts.

Pleomorphic adenomas of the minor salivary glands most commonly occur in the palate, the second most common site is the upper lip.9)

The case reports of pleomorphic adenoma from the external auditory canal have been extremely few.

pleomorphic adenoma is a benign tumor that originate from

An about 1.3 cm size encapsulated mass in cartilage segment of external auditory canal was resected clearly

Fig. 4

The mass is composed of various structures. some cystic portions between solid stroma. This figure is completed by joining together several pieces of slide.(H-E stain)

Fig. 5

Fig. 6

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Anatomically the ceruminal glands of external auditory canal are close to cartilage segment and the inner bony portion of the canal & middle ear cavity do not contain ceruminal glands. Therefore pleomorphic adenoma of external auditory canal can occur in cartilage segment, and tympanic membrane nearly always intact.11)

Some authors insist that the ectopic salivary gland tissue or the adjacent parotid gland can be origin of this tumor.

Ceruminous gland adenomas are the most common external auditory canal tumors. The neoplasms have been divided into three major groups based on specific histologic findings: ceruminous adenoma, ceruminous pleomorphic adenoma, and syringocystadenoma papilliferum12)

Other benign tumours of the external auditory canal include papilloma, sebaceous adenoma, exostoses, osteoma, fibroma, chondroma, myoma, and angioma.8)From among these, ceruminous pleomorphic adenoma is a rare variant of ceruminous adenoma.

It is a well-differnetiated benign tumor, characterized by myxomatous, pseudochondromatous, hyaline stroma with epithelial or myoepithelia cell formation.

Any age can be affected, no gender predilection exists, but there are too few cases to perform a valid statistical analysis.8)

Clinically, pleomorphic adenomas are characterized as painless, slow-growing masses, and the recommended treatment is surgical excision.13)

Pleomorphic adenomas have been classified as a benign tumor, but they have the capacity to grow to large proportions and may undergo malignant transformation.

The most important factor which determines this tendency is the length of the time for which the tumour has been present.14)

The risk of malignant transformation is 1.5% within the first 5 years of diagnosis but increases to 10% if observed for more than 15 years.15)

References

1. Thangaswamy V, Sivakumar A, Sivakumar A, Sivaraj, Pugazhendi SK, Thambiah L :Pleomorphic adenoma in an adolescent.. J Pharm Bioallied Sci. 2012 Aug;4(Suppl 2):S435-6. doi: 10.4103/0975- 7406.100325.

2. Heraguchi H, Hentona H, Tanaka H, et al. Pleomorphic adenoma of the external auditory canal: a case report and review of literature.J Laryngol Otol 1996;110:52-6.

3. Tsukahara K, Suzuki M, Tokashiki R, et al. Pleomorphic adenoma of the external auditory canal complicated by hearing loss secondary to chronic otitis media. Auris Nasus Larynx 2006;33:183-6.

4. Koyuncu M, Karagoz F, Kiliacarslan H. Pleomorphic adenoma of the external auditory canal. Eur Arch Otorhinolaryngol 2005;262:969-71.

5. Nardone M, Ferrara G, Nappi O, et al. Pleomorphic adenoma in unusual anatomic sites: case reports and review of literature. Acta Otorhinolaryngol Ital 2002;22:158-63.

6. Gerber C, Zimmer G, Linder T, et al. Primary pleomorphic adenoma ofthe external auditory canal diagnosed by fine needle aspiration cytology. A case report. Acta Cytol 1999;43:489-91.

7. Baldus SE, Streppel M, Stennert E, et al. Pleomorphic adenoma (mixed tumor) of the external auditory canal. Differential diagnosis of the tumors of the ceruminal glands. Pathologe 1999;20:125-9.

8. Chadha S, Pannu KK, Gill KS : Pleomorphic adenoma of external auditory canal.. Indian J Otolaryngol Head Neck Surg. 2011 Jul;63(Suppl 1):61-3. doi: 10.1007/s12070-011-0198-6. Epub 2011 Apr 17.

9. Jansisyanont P, Blanchaert Jr RH, Ord RA: Intraoral minor salivary gland neoplasm: a single institution experience of 80 cases. Int J Oral Maxillofac Surg 2002; 31:257-261.

10. Becelli R, Carboni A, Cassoni A, et al: Pleomorphic adenoma of the lachrymal gland: presentation of a clinical case of relapse. J Craniofac Surg 2002; 13:49-52.

11. Collins RJ, Yu HC (1989) Pleomorphic adenoma of the external auditory canal: an immunohistochemical and ultrastructural study.

Cancer 64:870-875

12. Thompson LD, Nelson BL, Barnes EL (2004) Ceruminous adenomas:

a clinicopathologic study of 41 cases with a review of the literature.

Am J Surg Pathol 28(3):308-318

13. Wright D (1997) Diseases of the external ear: Scott Brown’s otolaryngology, 6th ed, vol 3, Oxford, Butterworth-Heinemann 14. Shaheen OH (1997) Benign salivary gland tumours: Scott Brown’s

Otolaryngology, 6th ed, vol 5, Oxford, Butterworth-Heinemann 15. Seifert G: Histopathology of malignant salivary gland tumours. Eur J

Cancer Part B Oral Oncol 1992; 28B:49-56

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