International Journal of Pediatric Otorhinolaryngology Extra 9 (2014) 136–138
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Case Report
Pediatric inverted papilloma of the middle ear: Case report and review of the literature Burak Ulkumen a,1,*, Yunus Kaplan b,2, Ahmet Faruk Kıroglu c,3, Irfan Bayram d,4 a
Department of Otorhinolaryngology, Celal Bayar University, Faculty of Medicine, Manisa, Turkey Department of Otorhinolaryngology, Van Istanbul Hospital, Van, Turkey c Department of Otorhinolaryngology, Yuzuncu Yıl University, Faculty of Medicine, Van, Turkey d Department of Pathology, Yuzuncu Yıl University, Faculty of Medicine, Van, Turkey b
A R T I C L E I N F O
A B S T R A C T
Article history: Received 4 April 2014 Received in revised form 9 June 2014 Accepted 11 June 2014
Inverted papilloma of middle ear is a very rare clinical entity. It has been reported only in the adult population. We report a middle ear inverted papilloma in an 11-year-old male. To the best of our knowledge it is the first pediatric inverted papilloma of middle ear. The patient had a history of chronic suppurative otitis media and the lesion was found during preoperative evaluation for a tympanoplasty procedure. Total excision of the lesion and a tympanoplasty operation was accomplished. We reviewed the middle ear inverted papilloma cases regarding the age distribution, pathophysiology, treatment and prognosis. ß 2014 Elsevier Ireland Ltd. All rights reserved.
Keywords: Inverted papilloma Middle ear Pediatric Schneiderian
1. Introduction Inverted papilloma (IP) which is also known as inverted type of Schneiderian papilloma is a relatively rare lesion of the sinonasal tract [1]. It has a tendency to occur in men between fifth and sixth decades of life [2,3], but there has been few reports concerning pediatric sinonasal involvement [3–8]. Moreover, IP may involve other regions like middle ear [9] and bladder [10,11]. Middle ear involvement is very rare and occurs predominantly secondary to advanced or recurrent sinonasal disease in which invasion of eustachian tube has been determined as the main mechanism [9,12]. Also primary IP of middle ear is extremely uncommon [13] and has not been reported in pediatric population yet. To the best of our knowledge, this is the first case of primary middle ear IP in pediatric population. We present a pediatric middle ear IP
* Corresponding author at: Department of Otorhinolaryngology, Celal Bayar University, Department of Otorhinolaryngology, Uncubozko¨y Yerles¸kesi, Hafsa Sultan Hastanesi, I˙zmir-Manisa Yolu, 5549 Sok., Manisa, Turkey. Tel.: +90 236 2331920/2727; mobile: +90 05322406899. E-mail addresses:
[email protected] (B. Ulkumen),
[email protected] (Y. Kaplan),
[email protected] (A.F. Kıroglu),
[email protected] (I. Bayram). 1 Website: www.burakulkumen.com 2 Van Istanbul Hastanesi, KBB Bo¨lu¨mu¨ Urartu Sok. No: 10, Van, Turkey. 3 ¨ niversitesi Tıp Faku¨ltesi, KBB & BBC AD, Van/Merkez, Turkey. Yu¨zu¨ncu¨ Yıl U 4 ¨ niversitesi Tıp Faku¨ltesi, Patoloji AD, Van/Merkez, Turkey. Yu¨zu¨ncu¨ Yıl U http://dx.doi.org/10.1016/j.pedex.2014.06.003 1871-4048/ß 2014 Elsevier Ireland Ltd. All rights reserved.
without any sign of sinonasal involvement in which the lesion was found during preoperative evaluation for a tympanoplasty procedure. 2. Case report An 11-year-old male was referred to otolaryngology clinic with mild hearing loss and history of recurrent aural discharge from the left ear. At the time of referral, there was no otorrhea. In otomicroscopic examination, there was a 4 mm 3 mm of central perforation of tympanic membrane. The mucosa of cavum tympani was observed as normal but there was a fixed, solid mass over the manubrium mallei extending toward the medial surface and caput mallei. Endoscopic nasal and nasopharyngeal examination did not show any abnormalities. A pure tone audiogram revealed a mild conductive hearing loss. A temporal CT scan was obtained and revealed a hyper dense mass measuring 3.5 mm 6 mm in size attached to the malleus (Fig. 1). A type-1 tympanoplasty with excision of the mass was performed in the left ear. The mass was completely excised without disrupting the ossicular chain. Intraoperatively no evidence of ossicular chain erosion or invasion of the middle ear mucosa was observed. Macroscopic dimensions of the specimen were 8 mm 7 mm 3 mm and it was sent for histopathological examination with hematoxylin and eosin revealing inverted papilloma (Fig. 2). No sign of atypia was determined. After 2 years of follow up, there was no sign of
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Fig. 1. Axial (a), sagittal (b) and coronal (c) view of temporal computed tomography image; revealing a hyperdense mass (arrow) having continuity with the malleus in the left ear.
recurrence both in magnetic resonance imaging and otomicroscopic examination. 3. Discussion IP is an uncommon lesion of sinonasal tract which predominantly seen in elderly men [1–3]. It typically originates from the Schneiderian membrane which lines the sinonasal tract [2]. Presumably due to this unique origin, middle ear involvement is extremely rare and there are only 32 cases in the English literature. Ramey et al. reviewed 29 cases consisting of 14 primary and 15 secondary IP of middle ear [14]. In the literature, we found 3 additional secondary cases constituting total of 32 cases. Moreover, IP is very rare in pediatric population and the reported cases were predominantly originating from sinonasal tract [2–8] and few from bladder [10,11]. There are no primary or secondary reported cases of middle ear IP in children. To the best of our knowledge, our case is the first pediatric primary IP of the middle ear. Pathophysiology of middle ear IP involvement has been hypothesized by three mechanisms [16–21]. The first is that the lesion can invade the cavum tympani through the eustachian tube directly from a sinonasal origin. It is the leading mechanism for secondary involvement. A violated eustachian tube could only be
Fig. 2. Inverted squamous papilloma. Endophytic growth of the hyperkeratotic and hyperplastic squamous epithelium toward the stroma (hematoxylin–eosin stain; magnification 4).
shown in one case of secondary IP of the middle ear [22]. So, there should be some other mechanisms for secondary involvement. The second mechanism is the presence of a synchronous, metachronous or an isolated lesion originating from an embryological remnant of Schneiderian membrane in the cavum tympani. This may explain the secondary cases with noninvolved eustachian tube and this mechanism could also play a role in primary cases. The third mechanism is chronic inflammation due to suppurative otitis media evoking the emergence of Schneiderian mucosa from which IP originates [23]. In our case, the patient had a history of chronic suppurative otitis media and we postulate that is the mechanism. Briefly, we can classify middle ear involvement as primary or secondary according to the presence of synchronous or metachronous sinonasal disease [1]. According to the English literature, secondary involvement seems to be slightly more common with a number of 18 cases when compared with primary involvement of 14 cases. IP of middle ear typically presents with hearing loss and/or otorrhea [19,24]. In the presented case, there was a mild conductive type of hearing loss with a history of recurrent otorrhea. Treatment of middle ear IP is total surgical excision [26]. Although IP is a benign tumor, it may exhibit aggressive behavior. Thus, we must be quite precise while dealing with the tumor boundaries in order to prevent local recurrence [23]. Computed tomography (CT) is the preferred modality for visualization of the middle ear and it is also the best choice for analyzing the borders of the lesion throughout the middle ear. In our case, a CT was done, revealing a hyperdense lesion over the manubrium mallei with no other involvement of the adjacent structures which was compatible with the microscopic examination. Besides the locally aggressive behavior, IP of middle ear has a greater risk of malignant transformation [1] when compared with the sinonasal counterpart and may necessitate the use of radiotherapy [14]. In the present case, the lesion was relatively small and could be easily excised and there was no sign of malignant transformation in histopathological examination. Long term follow-up after surgery is essential for IP especially in cases with middle ear involvement owing to higher recurrence rate than its sinonasal counterpart [24]. Magnetic resonance imaging (MRI) is more suitable for recurrent cases due to higher sensitivity in distinguishing postoperative changes from recurrent lesions [14]. In our case, the temporal MRI at 1st and 2nd year had shown no evidence of recurrence. Absence of recurrence may be related to the relatively early excision of the lesion during the tympanoplasty.
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In conclusion, IP of middle ear is extremely rare and it has been reported only in adults. It typically occurs secondary to sinonasal disease but some primary cases have been also reported. This is the first case of middle ear IP in pediatric population. The definitive treatment is total surgical excision with disease free borders and could be accomplished in this case without recurrence in 2 years of follow up. References [1] R. Inoue, T. Kanazawa, M. Morita, Y. Iino, S. Yamada, T. Ishida, Inverted papilloma of the middle ear, Otol. Neurotol. 32 (January (1)) (2011) e7–e8. , http:// dx.doi.org/10.1097/MAO.0b013e3181d2f07e. [2] A.P. Limaye, N. Mirani, J. Kwartler, S. Raz, Inverted Schneiderian papilloma of the sinonasal tract in children, Pediatr. Pathol. 9 (5) (1989) 583–590 (Review). [3] C. Ozcan, K. Go¨ru¨r, D. Talas, Recurrent inverted papilloma of a pediatric patient: clinico-radiological considerations, Int. J. Pediatr. Otorhinolaryngol. 69 (June (6)) (2005) 861–864. [4] A.J. D’Angelo Jr., A. Marlowe, F.I. Marlowe, M. McFarland, Inverted papilloma of the nose and paranasal sinuses in children, Ear Nose Throat J. 71 (June (6)) (1992) 264–266. [5] R.J. Stanley, J.A. Kelly, J. Matta I.I., Falkanberg inverted papilloma in a 10-year-old boy, Arch. Otolaryngol. 110 (1984) 813–815. [6] H. Ngo, T.L. Tewfik, Inverted papillomas of the nose and paranasal sinuses in children, J. Otolaryngol. 16 (August (4)) (1987) 244–246 (Review). [7] A.J. D’Angelo Jr., A. Marlowe, F.I. Marlowe, M. McFarland, Inverted papilloma of the nose and paranasal sinuses in children, Ear Nose Throat J. 71 (1992) 264–266. [8] R.D. Eavey, Inverted papilloma of the nose and paranasal sinuses in childhood and adolescence, Laryngoscope 95 (January (1)) (1985) 17–23. [9] M.J. Seshul, T.L. Eby, D.R. Crowe, G.E. Peters, Nasal inverted papilloma with involvement of middle ear and mastoid, Arch. Otolaryngol. Head Neck Surg. 121 (September (9)) (1995) 1045–1048. [10] H. Yagi, M. Igawa, H. Shiina, K. Shigeno, T. Yoneda, Y. Wada, S. Urakami, Inverted papilloma of the urinary bladder in a girl, Urol. Int. 63 (4) (1999) 258–260 (Review).
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