Home Ahead of print Instructions Contacts
About us Current issue Submit article Advertise  
Editorial board Archives Subscribe Login   


 
 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 28  |  Issue : 3  |  Page : 252-254

Recurrent oncocytic papilloma of the middle ear: A case report and literature review


Department of Otolaryngology, Head and Neck Surgery, Nihon University School of Medicine, Tokyo, Japan

Date of Submission17-May-2022
Date of Acceptance10-Aug-2022
Date of Web Publication21-Nov-2022

Correspondence Address:
Dr. Tadayoshi Koda
30-1 Oyaguchi-Kamicho, Itabashi-Ku, Tokyo 173-8610
Japan
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/indianjotol.indianjotol_81_22

Rights and Permissions
  Abstract 


Schneiderian papillomas commonly occur in the nasal sinuses and septal mucosa. This type of papilloma rarely develops in the middle ear, with only 51 previously reported cases. The oncocytic papilloma (OP) subtype is even rarer, with only seven cases. We present a case of primary OP in the middle ear that recurred during long-term postoperative follow-up. The patient was a 31-year-old man who experienced papilloma recurrence after three surgeries. These recurrences appeared as a shadow on computed tomography. No recurrence has been observed since his fourth surgery. Our review showed increased recurrence and malignant transformation rates in inverted papillomas that occur in the middle ear. In contrast, OPs occurring in the middle ear were reported to have a high recurrence rate and no malignant transformation. Close and careful follow-up is necessary for any type of papilloma.

Keywords: Malignant transformation, middle ear, oncocytic papilloma, recurrence, Schneiderian papilloma


How to cite this article:
Koda T, Shigihara S. Recurrent oncocytic papilloma of the middle ear: A case report and literature review. Indian J Otol 2022;28:252-4

How to cite this URL:
Koda T, Shigihara S. Recurrent oncocytic papilloma of the middle ear: A case report and literature review. Indian J Otol [serial online] 2022 [cited 2022 Dec 6];28:252-4. Available from: https://www.indianjotol.org/text.asp?2022/28/3/252/361648




  Introduction Top


Schneiderian papillomas most commonly arise in the nasal sinuses and septal mucosa. They rarely develop in the middle ear, with only 52 reported cases (including the present case).[1],[2],[3],[4],[5],[6] The oncocytic papilloma (OP) subtype is even rarer, with only seven previously reported cases.[2],[3],[4] We present a case of primary OP in the middle ear that recurred during long-term postoperative follow-up. A review of related literature was also performed.


  Case Report Top


A 31-year-old Japanese man presented to a nearby hospital in 2011 with a 2-year history of left-sided hearing loss. Myringotomy was performed on suspicion of otitis media with effusion, during which a neoplastic lesion in the middle ear was noted. In the same year, the patient sought consultation at our hospital.

Otomicroscopy revealed that the patient's left tympanic membrane was cloudy and bulging. Examination of his right tympanic membrane, nasal cavity, pharynx, and larynx was unremarkable. A standard pure-tone audiometry test noted a mild conductive hearing loss of 35 dB in his left ear. Temporal bone computed tomography (CT) revealed a soft-tissue shadow filling the left tympanic cavity. No abnormal findings were observed in the nasal sinuses. A biopsy of the tumor in the tympanic chamber revealed a Schneiderian papilloma. His first operation was performed in 2012. No lesions were observed in the mastoid antrum. The tumor filling the tympanic chamber was resected, and a canal wall-up tympanomastoidectomy was performed. Histopathological examination revealed a Schneiderian papilloma (OP), but there were no malignant findings.

In 2013, a second surgery was performed on suspicion of tumor recurrence. A small tumor was confirmed in the anterior and inferior tympanic cavities and was successfully resected. The histopathological examination result was similar to that of the first surgery. CT scan performed 11 months after the second surgery revealed a small soft-tissue shadow parallel to the lower surface of the tympanic membrane [Figure 1]a. Standard postoperative monitoring was performed.
Figure 1: Computed tomography findings (a) 11 months after the second surgery and (b) 7 years after the second surgery. A soft-tissue shadow is observed in the mastoid antrum

Click here to view


Seven years after the second surgery, CT revealed a soft-tissue shadow in the mastoid antrum. This finding provided the impression that the shadow in the tympanic cavity had slightly increased [Figure 1]b. Magnetic resonance imaging (MRI) also revealed a contrasting effect of the shadow in the tympanic cavity. The possibility of recurrence was considered [Figure 2]. Tumor resection was performed in 2019. The papilloma located just above the round window in the inferior tympanic cavity was successfully resected. Partial adhesion to the tympanic membrane was observed; thus, resection of this area was performed. No tumor was found in the mastoid antrum. Effusion, possibly due to ventilation impairment, was observed. Although the tumor bled easily, we were able to preserve the posterior wall of the ear canal. Histopathological examination showed findings consistent with tumor recurrence. No signs of malignancy were noted [Figure 3].
Figure 2: Magnetic resonance imaging reveals an enhanced tumor mass (arrow)

Click here to view
Figure 3: The tumor exhibits inverted and exophytic growth patterns, with interspersed multilayered eosinophilic columnar epithelium and cystic structures (hematoxylin-eosin staining ×200)

Click here to view


CT scan performed 5 months postoperatively revealed soft-tissue opacities in the left tympanic cavity and mastoid antrum. Confirmation surgery was performed in 2020. Only granulated tissue was observed in the tympanic membrane and mastoid antrum. Histopathological examination revealed no signs of recurrence. To date, no recurrence has been reported.


  Discussion Top


Schneiderian papilloma of the middle ear is rare and was first reported by Stone et al. in 1987.[1] To the best of our knowledge, only 52 cases of Schneiderian papillomas of the middle ear, including this present case, have been reported.[1],[2],[3],[4],[5],[6]

Schneiderian papillomas are classified into three types. Inverted papillomas (IPs) are the most frequent, followed by exophytic papillomas (EPs) and OPs.[7] Nasal sinus IPs have an increased risk of repeated local recurrence and malignant transformation. According to reports, the local recurrence rate of IPs is 37%,[8] and their malignant transformation rate is 5%–15%.[7] Middle ear IPs also have a high rate of recurrence and malignant transformation.[1],[2],[3],[4],[5],[6] The recurrence rate is 70% and the malignant transformation rate is 51%.

However, the clinical features of middle ear OPs remain unclear. The local recurrence rate of nasal sinus OPs has been reported to be 39%,[9] whereas their malignant transformation rate is 4%–17%.[7] There are currently only seven reported cases of middle ear OPs,[2],[3],[4] including the present case. [Table 1] summarizes the reported cases of middle ear OPs. Middle ear OPs have a local recurrence rate of 71% (five/seven cases) and a malignant transformation rate of 0%. Compared to nasal sinus OPs, middle ear OPs have a higher local recurrence rate. However, no malignant transformations have been observed. Histopathologically, OPs present with a multilayered eosinophilic columnar epithelium, with a cystic structure containing mucous components with IP-and EP-like growth patterns.[10] There have been reports of cases presenting with both IP and OP.[2] From these facts, OP seems to have the same characteristics as IP, but there was no malignant transformation.
Table 1: Clinical findings of oncocytic papillomas of the middle ear (literature review and present case)

Click here to view


Furthermore, 54% of middle ear IPs also develop in the nasal sinus, and 70% of them had a malignant transformation.[1],[2],[3],[4],[5],[6] In the middle ear OPs, there were no combined cases. It may be one of the reasons why there is no malignant transformation. The local recurrence rate remains the highest in middle ear OPs. Thus, immediate resection is necessary in these cases to avoid recurrence.

In this case report, the tumor recurred after the patient's second surgery. This highlights the importance of regular follow-up imaging examinations to screen for the development of a new soft-tissue shadow in the mastoid antrum. Most recurrences were identified within 1 year of the initial diagnosis.[2] In our case report, recurrence occurred 1 year after the initial diagnosis. Considering the high recurrence rate, repeat surgery should be considered in the presence of suspicious postoperative findings.

Our patient experienced papilloma recurrence twice, with the tumor confined in the tympanic cavity. We were able to preserve the posterior wall of the ear canal during surgery. It is logical that the posterior wall of the ear canal should be removed because complete resection is essential to prevent a recurrence. If the tumor is localized in the tympanic chamber and the tumor base can be confirmed, canal wall-up surgery is also possible. No recurrence was observed after this final surgery. To monitor for possible recurrence, the patient will be continuously followed up with CT and MRI.

Informed consent

Written informed consent was obtained from the patient for the use of images and for the publication of this case report.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initials will not be published and due efforts will be made to conceal his identity, but anonymity cannot be guaranteed.

Acknowledgment

We would like to thank Editage (www.editage.com) for English language editing.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Stone DM, Berktold RE, Ranganathan C, Wiet RJ. Inverted papilloma of the middle ear and mastoid. Otolaryngol Head Neck Surg 1987;97:416-8.  Back to cited text no. 1
    
2.
Wenig BM. Schneiderian-type mucosal papillomas of the middle ear and mastoid. Ann Otol Rhinol Laryngol 1996;105:226-33.  Back to cited text no. 2
    
3.
de Filippis C, Marioni G, Tregnaghi A, Marino F, Gaio E, Staffieri A. Primary inverted papilloma of the middle ear and mastoid. Otol Neurotol 2002;23:555-9.  Back to cited text no. 3
    
4.
Fu ZM, Zhao LP, Guo YY, Guan GF. A rare instance of primary oncocytic schneiderian papilloma of middle ear and eustachian tube with a combined trans oto and nasal approach resection. J Craniofac Surg 2020;31:504-6.  Back to cited text no. 4
    
5.
Blioskas S, Konstantinidis I, Antoniadis I, Kypriotou A, Hytiroglou P, Markou K. Primary inverted papilloma of the middle ear. Ear Nose Throat J 2021. p. 1-4.  Back to cited text no. 5
    
6.
Thompson LD. Middle ear and temporal bone Papilloma: A clinicopathologic study and comprehensive literature review of 57 cases. Head Neck Pathol 2021;15:1212-20.  Back to cited text no. 6
    
7.
Bishop JA. OSPs and ESPs and ISPs, oh my! An update on sinonasal (Schneiderian) papillomas. Head Neck Pathol 2017;11:269-77.  Back to cited text no. 7
    
8.
Busquets JM, Hwang PH. Endoscopic resection of sinonasal inverted papilloma: A meta-analysis. Otolaryngol Head Neck Surg 2006;134:476-82.  Back to cited text no. 8
    
9.
Lilja M, Viitasalo S, Hytönen M, Haapaniemi A, Hagström J, Mäkitie A. Sinonasal oncocytic papilloma-a series of 20 cases with special emphasis on recurrences. Laryngoscope Investig Otolaryngol 2019;4:567-72.  Back to cited text no. 9
    
10.
Hyams VJ. Papillomas of the nasal cavity and paranasal sinuses. A clinicopathological study of 315 cases. Ann Otol Rhinol Laryngol 1971;80:192-206.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]
 
 
    Tables

  [Table 1]



 

Top
 
 
  Search
 
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Access Statistics
    Email Alert *
    Add to My List *
* Registration required (free)  

 
  In this article
Abstract
Introduction
Case Report
Discussion
References
Article Figures
Article Tables

 Article Access Statistics
    Viewed236    
    Printed6    
    Emailed0    
    PDF Downloaded19    
    Comments [Add]    

Recommend this journal