Nasal Inverting Papilloma Extending to Bilateral Temporal Bones with Intracranial Extension: A Case Report and Literature Review

Objective: To report a rare case of inverting papilloma involving bilateral temporal bones with intracranial extension in a patient with a distant history of surgically excised sinonasal inverting papilloma.

Study Design: Present study is a case report and review of the literature.

Methods: This study is presented as a retrospective chart review.

Case Description: A 62 -year-old male presented to our tertiary care otology clinic with complaints of bilateral otorrhea and hearing loss. He had a history of sinonasal inverting papilloma 8 years prior at an outside hospital requiring left maxillectomy and free flap reconstruction. On examination, he had papillomatous lesions filling his right external auditory canal and left middle ear. Audiogram showed right profound and left mild to severe sensorineural hearing loss.

CT scan of the temporal bones showed extensive bony erosion of the tegmen, sigmoid, and posterior fossa plate with opacification of bilateral middle ears and mastoids. MRI IAC showed bilateral mastoid enhancement compatible with neoplastic disease with extension of lesion into middle and posterior fossa causing mass effect and extension into sigmoid sinuses with compression of cerebellum and significant temporal dural involvement. Deep biopsies from bilateral middle ears confirmed inverting papilloma with negative biopsy from nose and nasopharynx. After discussion at tumor board and with neurosurgery, options of extensive bilateral surgical intervention, or radiation were discussed with the patient. He was not keen for extensive surgery; therefore, he opted for radiation therapy. He responded very well to radiation; at 46 months of postradiation, the patient is clinically and radiographically free of disease and has bilateral normal tympanic membranes.

Literature Review: Review of the current English literature on inverting papilloma involving the temporal bone is limited due to the rare nature of this disease. Barry et al report only 33 cases of inverting papilloma with temporal bone involvement. Ramey et al were the first to report synchronous bilateral involvement of the temporal bones. Based on currently available literature, our patient is the first extensive bilateral temporal bone inverted papilloma diagnosed 8 years of postsinonasal inverted papilloma. Additionally, this case is the only one with bilateral extension directly from EAC to middle ear to mastoid through sigmoid and into middle cranial fossa dura.

Conclusion: Long-term follow-up of patients with sinonasal inverting papilloma is essential to identify recurrence and malignant transformation. Our case shows that along with sinonasal examination, otological examination remains important during surveillance. Surgeons should keep in mind the rare chances of inverted papilloma recurrence in temporal bones. Although inverted papilloma is a benign lesion, radiation is an option in rare cases, such as ours where the surgical resection of bilateral intracranial lesions along with extensive disease in bilateral temporal bones and sigmoid sinuses carries the potential for serious morbidity.

Publication History

Article published online:
12 February 2021