multifocal inverting papilloma of the sinonasal cavity and temporal bone

Imaging demonstrated a left sided nasal mass and a separate non-contiguous soft tissue massfilling the left middle ear without involvement of the eustachian tube.. Review of the literatur

Multifocal inverting papilloma of the sinonasal cavity and temporal

bone

Jonnae Y Barry, MDa,*, Christopher H Le, MDa, Rihan Khan, MDb, Abraham Jacob, MDa,

Alexander G Chiu, MDa,1

a Department of OtolaryngologyeHead and Neck Surgery, Banner University Medical Center, 1501 N Campbell Ave., PO Box 245074, Tucson, AZ 85724,

United States

b Department of Radiology, Banner University Medical Center, 1501 N Campbell Ave., PO Box 245067, Tucson, AZ 85724, United States

a r t i c l e i n f o

Article history:

Received 9 September 2016

Accepted 4 January 2017

Available online 16 February 2017

Keywords:

Inverted papilloma

Sinonasal tumor

Schneiderian

a b s t r a c t

Introduction: Inverting papillomas (IPs) represent the most common benign neoplasm of the sinonasal cavity and are known for local invasion, proclivity for recurrence, and risk of malignant transformation IP

of the temporal bone (TBIP) is exceptionally rare, with 32 reported cases We present a new case of multifocal IP of the sinonasal cavity and temporal bone

Methods: Case report and review of the literature

Results: A 45-year-old man presented with a left sided biopsy proven IP and associated left sided hearing loss Imaging demonstrated a left sided nasal mass and a separate non-contiguous soft tissue massfilling the left middle ear without involvement of the eustachian tube He underwent an endonasal endoscopic gross total resection of the sinonasal lesion and biopsy of the middle ear mass with pathology showing IP

He subsequently underwent a left sided transtemporal resection of the TBIP Review of the literature, revealed 32 TBIP cases, with 59% having history of associated sinonasal IP and 41% with isolated temporal bone disease Over half of the patients demonstrated recurrence In comparison to patients with history

of sinonasal IP, isolated TBIP occurred in younger patients, was more common in females, and had less association with HPV and malignant transformation

Conclusion: TBIP is extraordinarily rare and usually presents with a history of sinonasal IP Isolated TBIP may be a distinctly different disease process Disease recurrence is common and risk of malignant transformation is present, so aggressive initial surgical treatment with gross total resection is advocated

© 2017 Published by Elsevier Inc This is an open access article under the CC BY license (http://

creativecommons.org/licenses/by/4.0/)

1 Introduction

Inverting papilloma within the temporal bone (TBIP) is

extremely rare with only 32 reported cases in the literature[1]

Sinonasal inverted papilloma (IP) is a benign, but locally aggressive

neoplasm, which most often originates from the lateral nasal wall

and represents between 0.5% and 4% of all sinonasal tumors[2]

There is a risk for transformation into squamous cell carcinoma and

recurrence is common [3e7] TBIP has a higher rate of

transformation to SCC at 36% compared to IP within the nasal cavity which has been estimated to have rates between 5% and 21%[8] Involvement of the temporal bone is hypothesized to occur via several mechanisms including transmission of cells via the eusta-chian tube, direct extension through the eustaeusta-chian tube, iatrogenic implantation or seeding, or from stimulation or conversion of re-sidual Schneiderian mucosa within the middle ear by such triggers

as chronic otitis media[1,9] Of the 32 cases previously reported, 13 had isolated TBIP without sinus involvement[1] The mean age of presentation was 52 and most commonly patients presented with hearing loss and otorrhea[1] We present a case of TBIP in a patient with concurrent IP of the sinonasal cavity

2 Case report

A 45-year-old man presented with two-year history of left-sided nasal obstruction, anosmia and diminished hearing The referring

* Corresponding author Department of Otolaryngology-Head and Neck surgery,

The University of Arizona College of Medicine, 1501 N Campbell Ave., PO Box

245074, Tucson, AZ 85724, United States.

E-mail address: jonnaeb@oto.arizon.edu (J.Y Barry).

1 Present address: Department of OtolaryngologyeHead and Neck Surgery

Uni-versity of Kansas Medical Center 3901 Rainbow Boulevard Kansas City, KS 66160,

United States.

Contents lists available atScienceDirect Otolaryngology Case Reports

j o u r n a l h o me p a g e :w w w o t o l a r y n g o l o g y c a s e r e p o r t s c o m

http://dx.doi.org/10.1016/j.xocr.2017.01.004

2468-5488/© 2017 Published by Elsevier Inc This is an open access article under the CC BY license ( http://creativecommons.org/licenses/by/4.0/ ).

Otolaryngology Case Reports 2 (2017) 33e36

community ENT had obtained a biopsy of a left-sided nasal mass,

which was consistent with IP A computed tomography (CT) scan

demonstrated a large mass filling the entire left nasal cavity,

ethmoid, and sphenoid sinuses, extension into nasopharynx with

thinning of the medial orbital wall and cribiform plate Magnetic

resonance imaging (MRI) demonstrated characteristicfindings of IP

including frond-like heterogeneous enhancement of the lesion

(Fig 1A and B) Additionally, a similar soft tissue mass was seen in

the left middle ear surrounding the ossicles (Fig 1C, E, F) The nasal

mass appeared separate from the soft tissue massfilling the left

middle ear, and there was no involvement of the Eustachian tube

(Fig 1D)

The patient underwent an endonasal endoscopic gross total

resection of the sinonasal tumor The lesion was pedicled off of the

posterior superior septum and did not extend into the Eustachian

tube (Fig 2) The mass abutted but did not erode through the

cri-biform plate or involve the mucosa of the anterior skull base, and

final pathology was consistent with inverted papilloma having

high-grade dysplasia (Fig 4B) At the end of the endonasal

endo-scopic surgical resection, the left ear was examined and

myr-ingotomy performed revealing a polypoid mass within the middle

ear space A biopsy of this mass was obtained and demonstrated IP

The patient subsequently underwent left transotic resection of the

extradural TBIP approximately two months after his sinonasal

resection, allowing him time to heal from his endonasal operation

and come to terms with the multifocal nature of his disease (Fig 3)

Unfortunately, in these intervening two-months, the patient had

progression of disease in his temporal bone with new symptoms of

worsening hearing and vertigo/disequilibrium To remove the

entire lesion, which was found to extend into the vestibule and

cochlea, the ear canal was closed, a fallopian bridge technique was utilized, and complete labyrinthectomy was performed The Eustachian tube was also dissected 6 mm from its protympanic orifice prior to obliteration; here biopsies obtained that were negative for IP Final pathology of the main tumor mass demon-strated inverted papilloma with high-grade dysplasia (Fig 4A), similar to the specimens previously obtained from the nose The patient has healed well from his operation and has been disease free for 9-months

3 Discussion Wardfirst described sinonasal inverting papilloma in 1854 The pathogenesis of the disease, however, remains largely unclear Historically, risk factors for IP were smoking, allergy, occupational exposures, or viral infection, particularly HPV[10e12] Although extensively studied, HPV infection as an etiology is still contro-versial A recent study by Roh and colleagues argues against its involvement This study utilized PCR to target HPV DNA from sinonasal IP specimens, which was then genotyped They found only 14.8% of their study participants tumors contained HPV DNA, and none of the HPV positive patients were found to have recur-rence with mean follow-up of 34.1 months[13] Their study group population contained 13% smokers Interestingly, of the patients with recurrent IP, 42.9% of them were smokerse suggesting that smoking may be a greater risk factor than HPV[13] A recent meta-analysis examined HPV infection and risk for malignant trans-formation in sinonasal IP and found a significant association be-tween the two, especially with those infected with HPV-18 types

[14]

Fig 1 A Axial T1 MRI with contrast and B Axial T2 MRI demonstrate a large intranasal mass (asterisk) A portion of the mass at the sphenoid sinuses (circle) demonstrates frond like heterogeneous enhancement with a “mini-brain” appearance characteristic of inverted papilloma More anteriorly the heterogeneity is lost consistent with pathologic findings

of degeneration to high grade dysplasia C Axial T2 MRI demonstrating the large intranasal mass (asterisk) and trapped secretions in the left maxillary sinus (short arrow) as well as effusion of the left mastoid (long arrow), inverted papilloma (circle) with a more dark appearance D Coronal T1 MRI with contrast enhancing inverted papilloma throughout the middle ear cavity, surrounding the ossicles (tip of arrow) E DWI shows bright signal (circle) and ADC map (F.) Show dark signal (circle) indicative of restricted diffusion from tumor

J.Y Barry et al / Otolaryngology Case Reports 2 (2017) 33e36 34

The pathogenesis of multifocal IP is perplexing A recent

sys-tematic review by Carlson et al revealed 32 TBIP cases, with 59%

having history of associated sinonasal IP and 41% with isolated

temporal bone disease[1] Of the patients with TBIP, over half of the

patients demonstrated recurrence of disease following

microsur-gical resection[1] They also found that in comparison to patients

with history of sinonasal IP, isolated TBIP occurred in younger

pa-tients, was more common in females, and had less association with

HPV and malignant transformation[1] Patients with secondary

TBIP had an associated carcinoma 47% of the time in their review

[1] Attempts to identify differences between sinonasal and

tem-poral bone IP have also failed to differentiate them as separate

pathologic entities[15] Proposed mechanisms for development of

multifocal disease include direct extension, multicentric primary

development, and rests of ectopic Schneiderian mucosa; however,

no dominant hypothesis has emerged

The mainstay of treatment for IP of the paranasal sinuses is aggressive primary resection with surveillance given the proclivity for recurrence[16] Although surgical resection should serve as the primary treatment option for IP, there may be a role for radiation therapy Some recommend the consideration of radiation therapy for those patients with malignant conversion, multiple recurrent IPs, or in those in whom complete resection is not possible[17] Unlike head and neck malignancies, there are no established guidelines for surveillance IP, although benign in and of itself, does carry the risk of transformation to squamous cell carcinoma and also has a high recurrence rate for incompletely resected lesions As such, surveillance for early detection of recurrence is critical As recommended by Suh and Chiu in 2014, it may be judicious to base surveillance for IP on the more standardized recommendations for squamous cell carcinoma of the head and neck[18] Though IP can recur> 5 years after initial treatment, the majority of recurrences will occur within the first 2 years following surgery [19]

Fig 2 A Characteristic irregular grey to brown gelatinous and frond like mass of inverted papilloma filling the left nasal cavity B Tumor site of attachment (asterisk) to the left superior septum (arrow) C Left nasopharynx with mass (asterisk) and uninvolved eustachian tube (arrow).

Fig 3 A Inverted papilloma (arrow) seen extending into the external auditory canal through a perforation in the left tympanic membrane (asterisk) B Tumor (asterisks) involving the epitympanum and mastoid antrum (asterisk), with extension to the tegmen tympani (arrow) C Tumor extending into the cochlea through the round window (long arrow) The protympanic eustachian tube orifice (short arrow) is free of disease.

Fig 4 H&E stained specimens consistent with inverted papilloma in both (A) the middle ear and (B) The intranasal mass C Immunostaining for p16 was negative.

J.Y Barry et al / Otolaryngology Case Reports 2 (2017) 33e36 35

Additionally, Mirza and coworkers identified 7.1% synchronous

carcinoma and 3.6% metachronous carcinomas and that the mean

time to metachronous lesion development was 52-months, further

supporting that long-term follow-up and close surveillance is

prudent[20] An example surveillance regimen may include

com-plete examination including nasal endoscopy every 1e3 months for

thefirst year, every 2e6 months for the second year, every 4e8

months for years 3e5 and yearly thereafter[21] Post-treatment

imaging may also serve an important role in surveillance

espe-cially when patients develop concerning signs or symptoms or

when sites previously involved are difficult to visualize e frontal

sinuses or middle ear spaces A contrast enhanced magnetic

reso-nance image (MRI) is thought to be the best imaging modality to

detect recurrence [18] Formal recommendations for imaging or

surveillance when the temporal bone is involved are also lacking,

even with risk for malignant transformation being much higher

than for isolated sinonasal IP

4 Conclusion

TBIP is exceptionally rare and typically presents with a history of

sinonasal IP Clinicians should consider the possibility of multifocal

disease, especially in patients presenting with seemingly unrelated

symptoms such as hearing loss Isolated TBIP may be a distinct

disease process, and its pathogenesis is not clear Disease

recur-rence is common and risk of malignant transformation is high;

therefore, aggressive surgical treatment with diligent and

long-term surveillance is prudent

Source of funding

None

Conflicts of interest

None

Presentations

This manuscript was presented as a poster at the American

Rhinologic Society Spring meeting in Chicago, Illinois on September

16 and 17, 2016

Acknowledgements

The authors thank Dr Zahra Aly MD, PhD for her technical

assistance and provision of pathologic specimen photographs

contained within this manuscript

References [1] Carlson ML, Sweeney AD, Modest MC, Van Gompel JJ, Haynes DS, Neff BA Inverting papilloma of the temporal bone: report of four new cases and sys-tematic review of the literature Laryngoscope 2015;125:2576e83 [2] Mitchell CA, Ebert CS, Buchman CA, Zanation AM Combined transnasal/ transtemporal management of the eustachian tube for middle ear inverted papilloma Laryngoscope 2012;122:1674e8

[3] Liu ZW, Walden A, Lee CA Sinonasal inverted papilloma involving the tem-poral bone via the eustachian tube: case report J Laryngol Otol 2013;127: 318e20

[4] Ramey SJ, Russo JK, Condrey 3rd JM, Coulter B, Sharma AK Synchronous bilateral inverted papilloma of the temporal bone: case report and review of the literature Head Neck 2013;35:E240e5

[5] Dingle I, Stachiw N, Bartlett A, Lambert P Bilateral inverted papilloma of the middle ear with intracranial involvement and malignant transformation: first reported case Laryngoscope 2012;122:1615e9

[6] Shen J, Baik F, Mafee MF, Peterson M, Nguyen QT Inverting papilloma of the temporal bone: case report and meta-analysis of risk factors Otol Neurotol 2011;32:1124e33

[7] Gaio E, Marioni G, Blandamura S, Staffieri A Inverted papilloma involving the temporal bone and its association with squamous cell carcinoma: critical analysis of the literature Expert Rev Anticancer Ther 2005;5:391e7 [8] 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: 555e9

[9] Kainuma K, Kitoh R, Kenji S, Usami S Inverted papilloma of the middle ear: a case report and review of the literature Acta Otolaryngol 2011;131:216e20 [10] Buchwald C, Franzmann MB, Tos M Sinonasal papillomas: a report of 82 cases

in Copenhagen County, including a longitudinal epidemiological and clinical study Laryngoscope 1995;105:72e9

[11] Scheel A, Lin GC, McHugh JB, Komarck CM, Walline HM, Prince ME, et al Human papillomavirus infection and biomarkers in sinonasal inverted pap-illomas: clinical significance and molecular mechanisms Int Forum Allergy Rhinol 2015;5:701e7

[12] d'Errico A, Zajacova J, Cacciatore A, Baratti A, Zanelli R, Alfonzo S, et al Occupational risk factors for sinonasal inverted papilloma: a case-control study Occup Environ Med 2013;70:703e8

[13] Roh HJ, Mun SJ, Cho KS, Hong SL Smoking, not human papilloma virus infection, is a risk factor for recurrence of sinonasal inverted papilloma Am J Rhinol Allergy 2016;30:79e82

[14] Zhao RW, Guo ZQ, Zhang RX Human papillomavirus infection and the ma-lignant transformation of sinonasal inverted papilloma: a meta-analysis J Clin Virol 2016;79:36e43

[15] Blandamura S, Marioni G, de Filippis C, Giacomelli L, Segato P, Staffieri A Temporal bone and sinonasal inverted papilloma: the same pathological en-tity? Arch Otolaryngol Head Neck Surg 2003;129:553e6

[16] Sharma J, Goldenberg D, Crist H, McGinn J Multifocal inverted papillomas in the head and neck Ear Nose Throat J 2015;94:E20e3

[17] Rutenberg M, Kirwan J, Morris CG, Werning JW, Mendenhall WM Radiation therapy for sinonasal inverted papilloma Pract Radiat Oncol 2013;3:275e81 [18] Suh JD, Chiu AG What are the surveillance recommendations following resection of sinonasal inverted papilloma? Laryngoscope 2014;124:1981e2 [19] Busquets JM, Hwang PH Endoscopic resection of sinonasal inverted papil-loma: a meta-analysis Otolaryngol Head Neck Surg 2006;134:476e82 [20] Mirza S, Bradley PJ, Acharya A, Stacey M, Jones NS Sinonasal inverted papil-lomas: recurrence, and synchronous and metachronous malignancy.

J Laryngol Otol 2007;121:857e64 [21] Network NCC Head and neck cancers 2016 J.Y Barry et al / Otolaryngology Case Reports 2 (2017) 33e36

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