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Ashdin Publishing Journal of Case Reports in Medicine Vol. 1 (2012), Article ID 235635, 4 pages doi:10.4303/jcrm/235635 ASHDIN publishing Case Report A Case of a Child with a Lipoma of the Middle Ear and a Concomitant Chondroma of the External Auditory Canal Tomoyasu Tachibana, 1 Kazunori Nishizaki, 2 Masayoshi Fujisawa, 3 Yuya Ogawara, 1 Yuko Matsuyama, 1 Iku Abe, 1 and Michihiro Nakada 4 1 Department of Otolaryngology, Himeji Red Cross Hospital, 12-1 Shimoteno 1-Chome, Himeji City, Hyogo 670-8540, Japan 2 Department of Otolaryngology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, 2-5-1, shikata-cho, kita-ku, Okayama City, Okayama 700-8558, Japan 3 Department of Pathology, Himeji Red Cross Hospital, 12-1 Shimoteno 1-Chome, Himeji City, Hyogo 670-8540, Japan 4 Nakada ENT Clinic, 2-2-20, Shirakuni, Himeji City, Hyogo 670-0808, Japan Address correspondence to Tomoyasu Tachibana, [email protected] Received 20 September 2012; Revised 30 October 2012; Accepted 10 December 2012 Abstract The case of a 2-year-old boy with a right middle ear lipoma and a concomitant chondroma of the left external auditory canal is presented. The patient’s preoperative diagnosis was cholesteatomas of the right middle ear and left external ear canal. A right-sided epitympanotomy was performed. A lump of fatty tissue in the right anterior middle ear cleft that obstructed the Eustachian tube orifice was found. The white mass in the left external auditory canal, which easily separated from the tympanosquamous fissure of the temporal bone, was excised with a Rosen needle. Histopathologic examination revealed a lipoma of the right middle ear and a chondroma of the external ear canal. No cases of concomitant lipoma and chondroma have been previously reported. In the present case, the middle ear lipoma could be classified as a congenital anomaly because of its association with a contralateral side chondroma, which would be a choristoma. Keywords chondroma; choristoma; lipoma 1 Introduction Either lipoma or chondroma could be developed from any- where in the body. Both middle ear lipoma and chondroma of the external auditory canal are rare, and it is difficult to differentiate preoperatively from cholesteatomas or other tumors. In addition, although several hypotheses have been proposed from the etiological perspective, it remains unclear whether the so-called lipomas and chondromas are true tumors. As far as we know, simultaneous occurrence of the two diseases has not been previously reported. The case of a young boy with a lipoma in the middle ear canal and a chondroma of the contralateral external auditory canal is reported. 2 Case report The patient was a 2-year-old boy with no complaints who was evaluated by a local otolaryngologist because of right- sided otitis media with effusion. A right-sided myringotomy was performed twice. A yellowish-white mass was observed in the right middle ear at the time of the treatment for the otitis media. The mass did not change in size, form, or color during the course of treatment. The doctor also discovered a white mass in the left external auditory canal, which was suspected to be a congenital cholesteatoma. The patient was referred to our hospital for further evaluation. Otoscopy revealed an opacity through the anterosuperior quadrant of the right tympanic membrane, and a flat and smooth white mass in front of the short process of the malleus on the superior wall of the left bony external auditory canal (Figure 1). The left tympanic membrane Figure 1: Macroscopic findings of the right (left panel) and left (right panel) ear during the operation. Otoscopy reveals an opacity in the anterosuperior quadrant of the right membrane and a flat, smooth, white mass in front of the short process of the malleus on the superior wall of the left bony external auditory canal.

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Ashdin PublishingJournal of Case Reports in MedicineVol. 1 (2012), Article ID 235635, 4 pagesdoi:10.4303/jcrm/235635

ASHDINpublishing

Case Report

A Case of a Child with a Lipoma of the Middle Ear and a ConcomitantChondroma of the External Auditory Canal

Tomoyasu Tachibana,1 Kazunori Nishizaki,2 Masayoshi Fujisawa,3 Yuya Ogawara,1 Yuko Matsuyama,1 Iku Abe,1

and Michihiro Nakada4

1Department of Otolaryngology, Himeji Red Cross Hospital, 12-1 Shimoteno 1-Chome, Himeji City, Hyogo 670-8540, Japan2Department of Otolaryngology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences,2-5-1, shikata-cho, kita-ku, Okayama City, Okayama 700-8558, Japan3Department of Pathology, Himeji Red Cross Hospital, 12-1 Shimoteno 1-Chome, Himeji City, Hyogo 670-8540, Japan4Nakada ENT Clinic, 2-2-20, Shirakuni, Himeji City, Hyogo 670-0808, Japan

Address correspondence to Tomoyasu Tachibana, [email protected]

Received 20 September 2012; Revised 30 October 2012; Accepted 10 December 2012

Abstract The case of a 2-year-old boy with a right middleear lipoma and a concomitant chondroma of the left externalauditory canal is presented. The patient’s preoperativediagnosis was cholesteatomas of the right middle ear andleft external ear canal. A right-sided epitympanotomy wasperformed. A lump of fatty tissue in the right anteriormiddle ear cleft that obstructed the Eustachian tube orificewas found. The white mass in the left external auditorycanal, which easily separated from the tympanosquamousfissure of the temporal bone, was excised with a Rosenneedle. Histopathologic examination revealed a lipoma ofthe right middle ear and a chondroma of the external earcanal. No cases of concomitant lipoma and chondroma havebeen previously reported. In the present case, the middle earlipoma could be classified as a congenital anomaly becauseof its association with a contralateral side chondroma, whichwould be a choristoma.

Keywords chondroma; choristoma; lipoma

1 Introduction

Either lipoma or chondroma could be developed from any-where in the body. Both middle ear lipoma and chondromaof the external auditory canal are rare, and it is difficult todifferentiate preoperatively from cholesteatomas or othertumors. In addition, although several hypotheses havebeen proposed from the etiological perspective, it remainsunclear whether the so-called lipomas and chondromas aretrue tumors. As far as we know, simultaneous occurrence ofthe two diseases has not been previously reported. The caseof a young boy with a lipoma in the middle ear canal anda chondroma of the contralateral external auditory canal isreported.

2 Case report

The patient was a 2-year-old boy with no complaints whowas evaluated by a local otolaryngologist because of right-sided otitis media with effusion. A right-sided myringotomywas performed twice. A yellowish-white mass was observedin the right middle ear at the time of the treatment for theotitis media. The mass did not change in size, form, or colorduring the course of treatment. The doctor also discovereda white mass in the left external auditory canal, which wassuspected to be a congenital cholesteatoma. The patient wasreferred to our hospital for further evaluation.

Otoscopy revealed an opacity through the anterosuperiorquadrant of the right tympanic membrane, and a flat andsmooth white mass in front of the short process of themalleus on the superior wall of the left bony externalauditory canal (Figure 1). The left tympanic membrane

Figure 1: Macroscopic findings of the right (left panel)and left (right panel) ear during the operation. Otoscopyreveals an opacity in the anterosuperior quadrant of the rightmembrane and a flat, smooth, white mass in front of theshort process of the malleus on the superior wall of the leftbony external auditory canal.

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Figure 2: Computed tomography of the right temporalbone. A soft tissue density fills the epitympanum andmesotympanum in the right middle ear.

Figure 3: Computed tomography of the left temporal bone.A minute nodular mass on the anterior wall of the left bonyexternal auditory canal is seen.

was normal. Auditory brainstem response (ABR) testingdemonstrated a 50-dB conductive hearing loss in the rightear, with no hearing loss in the left ear.

On temporal bone computed tomography (CT) scan, asoft tissue density filled the epitympanum and mesotympa-num in the right middle ear (Figure 2), and a minute nodularmass on the anterior wall of the left bony external auditorycanal (Figure 3) was identified. The preoperative diagnosiswas cholesteatomas in the right middle ear and left externalear canal. A right-sided epitympanotomy was performedthrough a transmeatal approach. A lump of fatty tissuethat filled the right anterior middle ear cleft, producingobstruction of the Eustachian tube orifice, was found(Figure 4). The mass (5.5 mm × 4.5 mm) was dissectedcompletely; it was yellow and round, surrounded withepithelium (Figure 5). The left-sided surgery was performedthrough the ear canal. The white mass of the external earcanal, which separated easily from the tympanosquamousfissure of the temporal bone, was excised with a Rosenneedle. It was 1.4 mm in diameter. Histopathologicexamination of the removed masses revealed a lipoma

Figure 4: Macroscopic findings of the right ear before (left)and after (right) removing the mass. A lump of fatty tissueis seen to fill the right anterior middle ear cleft, obstructingthe Eustachian tube orifice intermittently.

Figure 5: The mass extirpated from the right ear. The massis yellow and round, surrounded by epithelium. It measures5.5 mm×4.5 mm.

in the right middle ear and a chondroma in the left externalear canal (Figure 6). There were no signs of chronic orsevere inflammation in the epithelium around the lipoma.

3 Discussion

A patient who had a lipoma in the middle ear associatedwith a chondroma in the contralateral external ear canal wasdescribed. Both middle ear lipoma and chondroma of theexternal auditory canal are rare. Further, to the best of ourknowledge, there have been no previous reports concerning

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Table 1: Past reports of lipoma of the middle ear.Author Publication year Age Sex Location Conductive hearing

lossOtitis media witheffusion

Complications

Kasbekar et al. 1984 33 M Epitympanum (++) (+)

Stegehuis et al. 1985 64 F Epitympanum (+) (−)

Selesnick et al. 1990 4 F Epitympanum (++) (+) Meatal atresia

Suetake et al. 1991 7 M Mesotympanum (++) (−) Middle ear anomaly

Abdullah et al. 1993 5 M Mesotympanum (++) (+)

Edmonds et al. 1997 7 F Mesotympanum (+) (+) Craniofacial abnormalities

Ito et al. 2002 5 M Mesotympanum (−) (+)

Kobayashi et al. 2002 12 M Mesotympanum (−) (−) Congenital cholesteatoma

Gierek et al. 2002 8 F Unidentified Unidentified Unidentified UnidentifiedPresent case 2012 2 M Mesotympanum (+) (+) Chondroma

Figure 6: Histopathologic examination of the removedmasses shows a lipoma (A, left panel) and a chondroma (B,right panel). (H & E stain × 100).

lipoma co-existing with chondroma. In addition, althoughseveral hypotheses have been proposed from the etiologicalperspective, it remains unclear whether the so-called lipo-mas and chondromas are true tumors.

As far as we could determine, only 9 cases of lipomaof the middle ear have been reported previously [1,2,3,5,6,7,12,14,15]. The details, including the case reported here,are summarized in Table 1. Patients’ ages ranged from 2to 64 years, with an average of 14.7 years. The sex distri-bution was approximately equal (6 males, 4 females). Allbut one developed in the epitympanum or mesotympanum.Conductive hearing loss was reported in 7 of 9 cases. Otitismedia with effusion was reported in 6 of 9 cases. In all caseswith secretory otitis media, the lipoma was located aroundthe Eustachian tube. In the case presented, it appears thatthe lipoma obstructed the Eustachian tube orifice, resultingin secretory otitis media with conductive hearing loss. Fourof eight previously reported cases had congenital disease,including meatal atresia, middle ear anomaly, craniofacialabnormalities, and congenital cholesteatoma.

In a case report and review by Ito et al. [5], the lipomacould be congenital, because their case was associated witha middle ear anomaly and a subcutaneous lipoma at the timeof birth. On the other hand, Selesnick et al. [12] reported alipoma patient with meatal atresia, but they suggested that

this disease could be acquired; the lipoma may have devel-oped due to repeated or long-term inflammation in the mid-dle ear. The present patient was 2 years of age, which istoo young to consider that chronic inflammation was themajor cause of this disease. In addition, otitis media in thispatient was not long-term, and there were no signs of severeinflammation in the epithelium around the lipoma. There-fore, the lipoma in the present case was most likely causedby congenital factors.

Chondroma of the external auditory canal is also rare.Nelms Jr. and Paparella [10] divided tumors found in theexternal auditory canal into three groups (epithelial anlage,interstitial anlage, and melanoma), and they classifiedchondroma as interstitial anlage. Recently, Yokogawa etal. [17] reviewed 26 cases of chondroma, and Tanigawaet al. [16] analyzed 48 previously reported cases [4,7,8,9,16] of this disease. According to their investigations,the patients’ ages ranged from 2 to 70 years. There wasan approximately equal distribution between males andfemales. Most of the patients were asymptomatic, as inthe present case, but some others complained of otalgia,an abnormal sensation around the ear, otorrhea, facialnerve paresis, or conductive hearing loss. Chondroma wasmost frequently observed in the medial portion of theanterior wall of the bony external auditory canal just infront of the short process of the malleus. The etiologicalmechanism of chondroma is unknown, but many authorshave strongly suggested a congenital origin. Kobayashi etal. [7] hypothesized that chondroma developed from straygerm tissue at the tympanosquamous fissure. In the casereport and review by Yokogawa et al. [17], one patienthad an inner ear malformation of the Mondini type. Theysuggested a relationship between chondroma and Mondini-type malformation, because the formative period of theexternal auditory canal is compatible with the embryonicperiod of 7 weeks when the malformation of Mondini typeis caused. Quercetani et al. [11] reported that chondroma ofthe external auditory canal could be a hamartoma. Lee [8]

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recently proposed that cartilaginous choristoma rather thanchondroma may be more appropriate terminology for thelesion. All of these authors suggested that chondroma in theear would be congenital.

Simoni et al. [13] reported that choristomatous polyps ofthe aural and pharyngeal regions in the same patient wouldbe thought to be due to errors in development of the secondand first branchial arches. In our case, it would be reasonableto consider that two different types of choristoma developedin a single patient, although we have no idea why the twopossible anomalies of the ear developed in the same patient;whether they were accidental or there were common con-genital factors is unknown.

4 Summary

(1) A middle ear lipoma with a chondroma of the externalauditory canal is rare.

(2) The case of a young male patient with a lipoma inthe middle ear canal associated with a chondroma of theexternal auditory canal was described.

(3) The lipoma in the present patient was probably nota true tumor but a congenital anomaly called choristoma,because the chondroma, which is strongly suspected to be acongenital anomaly, was associated with a lipoma.

References

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[3] T. Gierek, L. Klimczak-Golab, A. Slaska-Kaspera, andK. Majzel, An extremely rare case of primary lipoma ofthe middle ear in an 8-year old girl, Otolaryngol Pol, 56 (2002),489–491.

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[11] R. Quercetani, R. Gelli, N. Pimpinelli, and U. M. Reali, Bilateralchondroma of the auricle, J Dermatol Surg Oncol, 14 (1988),436–438.

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[13] P. Simoni, B. J. Wiatrak, and D. R. Kelly, Choristomatous polypsof the aural and pharyngeal regions: first simultaneous case, IntJ Pediatr Otorhinolaryngol, 67 (2003), 195–199.

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