Central Annals of Otolaryngology and Rhinology

Research Article *Corresponding author

Jae-Jun Song, Department of Otorhinolaryngology – Head and Neck Surgery, Korea University College of Clinical Features of Benign Medicine, 148 Gurodong-ro, Guro-gu, Seoul, 152-703, South Korea, Tel: 82-2-2626-3191; Fax: 82-2-868-0475; Tumors of the External Auditory Email: Submitted: 31 March 2017 Accepted: 20 April 2017 Canal According to Pathology Published: 21 April 2017 ISSN: 2379-948X Jeong-Rok Kim, HwibinIm, Sung Won Chae, and Jae-Jun Song* Copyright Department of Otorhinolaryngology-Head and Neck Surgery, Korea University College © 2017 Song et al. of Medicine, South Korea OPEN ACCESS Abstract Keywords Background and Objectives: Benign tumors of the external auditory canal (EAC) • External auditory canal are rare among head and neck tumors. The aim of this study was to analyze the clinical • Benign tumor features of patients who underwent surgery for an EAC mass confirmed as a benign • Surgical excision lesion. • Recurrence • Infection Methods: This retrospective study involved 53 patients with external auditory tumors who received surgical treatment at Korea University, Guro Hospital. Medical records and evaluations over a 10-year period were examined for clinical characteristics and pathologic diagnoses. Results: The most common pathologic diagnoses were nevus (40%), osteoma (13%), and cholesteatoma (13%). Among the five pathologic subgroups based on the origin organ of the tumor, the most prevalent pathologic subgroup was the skin lesion (47%), followed by the epithelial lesion (26%), and the bony lesion (13%). No significant differences were found in recurrence rate, recurrence duration, sex, or affected side between pathologic diagnoses. The overall recurrence rate after excision was 11% and higher in patients with fibro epithelial polyp, intra epidermal pilarepithelioma, and chronic inflammation. Culture of otorrhea identified pathogens in half of patients with recurrence, and the recurrence rate was considerably higher (50%) in cases confirmed to be infected by microorganism. Conclusion: Our results indicated that nevus was the most common pathologic diagnoses. The overall recurrence rate was 11%. Benign external auditory canal lesions that originate from the epithelium and bony canal should be observed closely in the postoperative follow-up period. Furthermore, treatment of infection is thought to be important for preventing recurrence.

ABBREVIATIONS as causative factors in EAC tumor; however, conclusive evidence of this is lacking [2]. Symptoms are rare but can include EAC: External Auditory Canal; MSSA: Methicillin-sensitive conductive hearing loss, otalgia, otorrhea, vertigo, and tinnitus. Staphylococcus aureus In previous studies, osteomas were the most commonly found INTRODUCTION pathologic type in EAC [1,3]. Spielmann et al. reported a 10- year case series of 48 surgically resected EAC masses, including Benign tumors of the external auditory canal (EAC) are malignant tumors, benign tumors, infective lesions, and epithelial uncommon among head and neck tumors. The EAC begins at abnormalities. In their study population, bony lesions were the the opening of the cup-shaped portion (concha) of the ear, and extends downward to the tympanic membrane. The adult EAC [1]. Wiatr et al. also retrospectively investigated 26 benign EAC measures 2-3cm in length. The EAC is covered with keratinizing tumormost frequently cases, and encountered osteoma was diagnosis the most of commonEAC benign pathologic tumors diagnosis [3]. However, there are no concrete data supporting this is thicker and contains a subcutaneous layer, hair follicles, conventional idea, especially in Asian countries. Furthermore, sebaceousstratified squamous glands, and epithelium. wax-secreting The skin ceruminous over the lateralglands. portion These the prevalence and clinical aspects of various pathological benign skin appendages are absent in the medial two thirds of EAC, where tumors arising from the EAC have not yet been investigated with the bony wall is closely adherent to the underlying periosteum

sufficient sample size. The purpose of this study was to describe [1]. Chronic irritation and inflammation have been considered Cite this article: Kim JR, Im H, Chae SW, Song JJ (2017) Clinical Features of Benign Tumors of the External Auditory Canal According to Pathology. Ann Otolaryngol Rhinol 4(3): 1169. Song et al. (2017) Email: Central the correlation of pathologic diagnoses with clinical features in many cases, however, patients with large EAC tumors complained surgically removed benign tumors of the EAC.

MATERIALS AND METHODS of earfulness. In the case of inflammation, otalgia or otorrhea was frequentlyIn 47 cases,observed. tumors were simply excised via an endaural From 2003 to 2014, a retrospective, single-center study approach. In 4 patients, canaloplasty was performed with of EAC lesions was performed using the medical records of excision; 2 of these also underwent tympanoplasty. In one patients who had received surgical treatment for EAC tumors at Department of Otorhinolaryngology-Head and Neck Surgery, postauricular approach was used because of the large size of Korea University Guro Hospital. A total of 53 patients (26 male case, pathologically confirmed as a branchial cleft anomaly, a and 27 female) were included in the present study. All study differences in age, sex, affected side, recurrence rate, and protocols were approved by the Institutional Review Board of recurrencethe mass (3×3cm). duration Statisticalbetween the analysis pathologic revealed diagnoses no significant (P > 0.5). Korea University Guro Hospital (KUGH15144-001). EAC masses Incidence were surgically excised under and general anesthesia in an In this case series, nevus was the most common pathologic operatingwere identified room. with Medical rigid otoendoscopy, records were reviewedand discovered for patients’ lesions age, sex, affected side, pathologic diagnosis, operation records, diagnosis. There were 21 patients diagnosed with nevus (40%), followed by osteoma (7 patients, 13%), EAC cholesteatoma concurrent infection, and recurrence. Patients were classified into (7 patients, 13%), chronic inflammation (6 patients, 11%), one of five categories according to tumor origin (bony, epithelial, epidermal cyst (5 patients, 9%), and others. Among the 21 skin, inflammatory and other benign lesions). The epithelial nevus patients, there were 19 intradermal (90%), 1 compound lesion means the diseases in the most superficial layer of skin, prevalent pathologic subgroup was the skin lesion (25 patients, as the lesions which arise in the dermis and skin appendages (5%), and 1 junctional (sebaceous) pathologic types. The most excludingstratified squamousepithelium. epithelium.Statistical analyses And skin were lesion performed was defined using the Statistical Package of the Social Sciences (SPSS, version 21). 47%), followed by the epithelial lesion (14 patients, 26%), and Results related to patient age are expressed as means (standard theRecurrence bony lesion (7 patients, 13%) (Table 1). eviations, SDs). During the postoperative follow-up period, recurrent lesions RESULTS AND DISCUSSION

Patient characteristics were identified under otoendoscopic evaluation in 6 patients (6 From 2003 to 2014, a total of 53 patients were diagnosed with of 53, 11%). The mean follow-up period was 18.4 months. The EAC tumor under otoendoscopy; all patients underwent excision. recurrence rate was 28% (2 of 7) in patients with osteoma, 20% (1 of 5) in those with epidermal cyst, and 17% (1 of 6) in those male and 27 female patients, with an average age of 37.0 ± 19.2 with chronic inflammation. All patients who were diagnosed Baseline characteristics are shown in Table (1). There were 26 epithleialwith fibro polyp epithelial patient, polyp the or tumor intra recurredepidermal twice, pilar necessitating epithelioma aexperienced second revision relapse surgery. and underwent re-excision. In one fibro years. The right side was affected in 26 patients, and the left in 27 (Table 1). Patients with small EAC tumors had no symptom in Table 1: Characteristics of patients with benign EAC tumors. Recurrence Incidence Recurrence Infection Pathologic diagnosis N duration (%) (n [%]) (n) (months) Osteoma 7 13 2(29) 45 0 Chronic inflammation 6 11 1(17) 1 2 Epidermal cyst 5 9 1(20) 1 1 Cholesteatoma 7 13 0 1 Fibroepithelial polyp 1 2 1(100) 1 1 Intraepidermalpilarepithelioma 1 2 1(100) 6 0 14 26 3(21) 3 Skin lesion Nevus 21 40 0 1 Fibrofolliculoma 1 2 0 0 Pilomatricoma 1 2 0 0 Apocrine mixed tumor 1 2 0 0 Apocrine metaplasia of eccrine gland 1 2 0 0 25 47 0 1 Branchial cleft anomaly 1 0 0 Total 53 6(11) 16.5 Abbreviations: EAC: External Auditory Canal 6(11%)

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Among the pathologic subgroups, the recurrence rate was darker pigmentation, whereas more elevated compound nevi have a lighter brown to black color (Figure 1A). Intradermal nevi are elevated lesions without pigmentation (Figure 1B). Lim et al. thehighest skin in lesion the bony group, lesion no grouppatients (29%) experienced followed recurrenceby the epithelial after reported that the intradermal type was the most common among lesion group (21%) and the inflammatory lesion group (17%). In and it was the longest in patients with osteoma (45 months). Noexcision. patients The experienced average duration malignant of recurrence transformation was 16.5 during months, the EAC nevi; the same tendency was shown in this study [8]. all cases, or if symptoms are evident [9]. After incomplete removal, follow-up period. neviSome may have reappear insisted as that recurrent acquired lesionsEAC nevi (pseudomelanoma), must be removed in Infection clinically and pathologically similar to malignant melanoma in situ regardless of the initial pathology [10]. Early and complete We performed bacterial culture of otorrhea preoperatively. resection should thus be recommended for EAC nevi. In this study, no nevus patients experienced relapse. Nevus is one of methicillin-sensitivePathogens were confirmed Staphylococcus in 6 patients aureus (11%); (MSSA). among Half of these,these 3 (50%) had Pseudomonas aeruginosa, and 3 (50%) had Most Caucasian adults have about 20 nevi but is less common in Blackthe most individuals frequently or other acquired persons skin with neoplasms highly pigmented in Caucasians. skin patients (3 of 6) had re-excision. Three of six recurrent cases also [11]. Because this review involves only South Korean patients of hadDISCUSSION confirmed pathogens (Table 2). Asian ethnicity, further study on interracial differences of EAC

Benign tumors originating from the EAC are uncommon and the EAC is a solitary, slow-growing, and pedunculated mass in usually asymptomatic; thus, most of them are found incidentally. thenevi unilateral is recommended. bony canal The [2,12] clinical (Figure presentation 1C). It is known of osteoma to arise in But they can sometimes cause aural symptoms such as ear fullness, conductive hearing impairment, otitis externa, otalgia, persistent or repetitive exposure of the ears to cold water [13]. and tinnitus. Surgical removal is the standard treatment. It has Recurrencefrom tympanomastoid had occurred and in tympanosquamous 2 of 7 osteoma patients suture 45 lines months after also been suggested that ear symptoms assumed to be due to EAC tumor could be alleviated by tumor excision [4]. higher than overall rate for EAC tumors, and the duration of after the first excision. The recurrence rate of osteoma is much In this study, occurrence of EAC tumor was not associated recurrence is also longer than average (16.5 months). Long-term follow-up is therefore recommended for bony lesions in EAC. [5]. In previous studies, bony lesions (eg, osteoma, exostoses) werewith sexfound or age;to be this the finding most common is similar pathologic to those oftypes other of reportsbenign (Figure 1D) and recurred twice, so the patient underwent surgical In this study, one case was diagnosed as a fibro epithelial polyp EAC tumors [1,3]. However, nevus was the most common form of excision two times. Fibroepithelial polyp is a benign lesion of EAC tumor in this study. mesodermal origin that is most commonly found in the skin and Nevus, one of the most common benign skin tumors, is a sharply-circumscribed overgrowth of cells associated with genitourinary tract, but very rarely in the EAC. Their etiology is melanocytic migration and proliferation [6]. It usually has a wasthought assumed to be toan beunderlying a secondary, chronic reactive inflammatory change in process, the skin but to uniform surface and coloration pattern, round or oval shape, and remains largely unknown [14,15]. In EAC, fibro epithelial polyp in childhood and puberty and peak in the fourth decade of life polyp.an initial It presentsinflammatory a solitary, change pendunculated or an osteoma growth [4,6]. patternFigure 2,and B [7].relatively (In this regular case series,border the(Figure average 1A,B). age Most was cases 39.6 are years acquired in the and C show the histopathologic characteristics of fibro epithelial nevus group.) Histologically, nevi are divided into three subtypes polyphas benign c squamous epithelial linings with moderate acanthosis nevus in the epidermis, intradermal nevus in the dermis (Figure polypand mild or other papillomatosis ear disorders, [16]. or In mechanical our study, stimulation the fibroepthelial of the according to the location of clusters of melanocytic cells; junctional ase had no clinical findings suggesting history of aural

2A) and compound nevus in both areas. Flat junctional nevi have EAC. Tanaka et al. reported the first known case of independent Table 2: Characteristics of patients with recurrence of benign EAC tumor. Duration Infection Age Case Pathologic diagnosis Classification Sex Direction (months) (pathogen) (years) 1 Osteoma Bony lesion 46 - 13 M R 2 Osteoma Bony lesion 44 - 34 M R + 3 Chronic inflammation 1 24 F L (Pseudomonas aeruginosa) Inflammation + 4 Epidermal cyst Epithelial lesion 1 63 M R (Pseudomonas aeruginosa) + 5 Fibroepithelial polyp Epithelial lesion 1 63 F L (MSSA) Intraepidermal 6 Epithelial lesion 6 - 51 M R pilarepithelioma Abbreviations: MSSA: Methicillin

Ann Otolaryngol Rhinol 4(3): 1169 (2017) 3/5 Song et al. (2017) Email: Central cyst presents as a solitary, rounded mass that has an epidermal- fibroepithelial polyp of the EAC [5]. In epithelial lesions, epidermal like wall (Figure 1E). A chronic inflammatory mass in the EAC has tumoran erythematous was thought nodular to be the appearance main cause (Figureof relapse. 1F). Furthermore, The overall recurrence rate was 11% (6 of 53); incomplete resection of the recurrence, and the recurrence rate was considerably higher culture of otorrhea identified pathogens in half of patients with suggests that infection may increase the risk of recurrence of EAC tumor.(50%) inFurther cases confirmedresearch should to be infectedclarify the by causal microorganism. relationship This of tumor recurrence and infection in a larger sample size. surgically were included in this case series; patients who had conservative,This study medicalhas some management limitations. First, or were only lost patients to follow-up treated were excluded, which could represent a selection bias. Second, the sample size is relatively small. Although the sample size of this analysis is larger than that of any other previous studies of the strong correlation between pathologic diagnoses and benign EAC benign tumors [1,3], it is still insufficient to clarify environmentalclinical characteristics. differences. Third, In this the study future, was a larger,performed multicenter only in studySouth shouldKorea andbe performed thus does notto get reflect a more interracial, meaningful, regional, powerful and result. CONCLUSION In conclusion, our results indicated that nevus was the most common pathologic type, and the overall recurrence rate

Figure 2 A. Nevus (intradermal), view of melanocyts with brown pigmentation (arrow), (x100)Microscopic findings of each pathologic type. B. Fibroepithelial polyp, solitary, pendunculated growth pattern,

arrow), mild papillomatosis (black arrow) (x40) benign squamous epithelial lining with moderate acanthosis (white with vessels (black arrow), (x200) C. Fibroepithelial polyp, view of fibrotic backgrounds (white arrow)

affected side, recurrence rate, and recurrence duration between pathologicwas 11%. Therediagnoses. were Benign no significant EAC lesions differences that originate in age, from sex, the epithelium and bony canal should be observed closely in the

important for preventing recurrence. postoperative period. Treatment of infection is also thought to be REFERENCES 1. Spielmann PM, McKean S, White RD, Hussain SS. Surgical management of external auditory canal lesions. J Laryngol Otol. 2013; 127: 246-251. 2. Carbone PN, Nelson BL. External auditory osteoma. Head Neck Pathol. 2012; 6: 244-246. 3. Wiatr M, Skladzien J. Benign tumors of the external and middle ear from data collected at the Otolaryngologyst Department of the Jagiellonian University in Cracow between1992-2001. Przegl Lek. Figure 1 2007; 64: 1004-1005. A. Nevus (compound type), B. Nevus (intradermal type), C. Osteoma, 4. Otoendoscopic findings of each pathologic type. presenting as an aural polyp. J Laryngol Otol. 1993; 107: 935-936. Toma AG, Fisher EW. Osteoma of the external auditory meatus D. Fibroepithelial polyp, E. Epidermal cyst, F. Chronic inflammation Ann Otolaryngol Rhinol 4(3): 1169 (2017) 4/5 Song et al. (2017) Email: Central

5. 11. Wolff K, Johnson RA, Saavedra AP. Fitzpatrick’s Color Atlas and external auditory canal: a case report and a literature review. Case Synopsis of Clinical Dermatology 7th ed. United States: Mac Graw Hill; Tanaka N, Matsunobu T, Shiotani A. Fibroepithelial polyp of the 2013. 12. Ebelhar AJ, Gadre AK. Osteoma of the external auditory canal. Ear 6. RepStegmaier Otolaryngol. Oc, Becker 2013: Sw 818197.. Jr. Incidence of melanocytic nevi in young adults. J Invest Dermatol. 1960; 34: 125-129. 7. 13. NoseCumming Throat CW, J. 2012; Fredrickson 91: 96-100. JM, Harker LA, Krause CJ, Schuller DE, editors. Otolaryngology-Head and Neck Surgery. 6th ed. St Louis: Pariser RJ. Benign neoplasms of the skin. Med Clin North Am. 1998; Mosby Year Book; 2015; 2200-2210. 82: 1285-1307. histological characteristics of melanocytic nevus in external auditory 14. Schuster D, Sweeney AD, Eisenberg R, Wanna GB. A case of 8. canalsLim HJ, andKim auricles.YT, Choo Eur OS, ArchPark Otorhinolaryngol.K, Park HY, Choung 2013; YH. Clinical 270: 3035- and 3042. sensorineural hearing loss involving a fibroepithelial polyp of the 9. Alves RV, Brandã 15. middle ear. Am J Otolaryngol. 2015; 36: 475-478. EA, et al. Intradermal melanocytic nevus of the external auditory of the tonsil: case report of a rare, benign tonsillar lesion. J Laryngol Farboud A, Trinidade A, Harris M, Pfleiderer A. Fibroepithelial polyp canal. Braz J Otorhinolaryngol.o FH, Aquino 2005;JE, Carvalho 71: 104-106. MR, Giancoli SM, Younes Otol. 2010; 124: 111-112. 10. Sexton M, Sexton CW. Recurrent pigmented melanocytic nevus. A 16. Elder DE, Elenitsas R, Rosenbach M, Murphy G, Rubin A, Xu X, et al. benign lesion, not to be mistaken for malignant melanoma. Arch Lever s Histopatholoy of the skin. 11th ed. Philadelphia: Wolters Pathol Lab Med. 1991; 115: 122-126. Kluwer. 2014; 1203-1250.

Cite this article Kim JR, Im H, Chae SW, Song JJ (2017) Clinical Features of Benign Tumors of the External Auditory Canal According to Pathology. Ann Otolaryngol Rhinol 4(3): 1169.

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