|Year : 2015 | Volume
| Issue : 3 | Page : 666
Inverted papilloma of atypical origin with unusual extension into the oropharynx
M Panduranga Kamath1, S Vijendra Shenoy1, Vishnu Prasad1, Kiran Bhojwani1, Radha Pai2, Neethu Mary Mathew1
1 Department of ENT and Head and Neck Surgery, Kasturba Medical College, Manipal University, Mangalore, Karnataka, India
2 Department of Pathology, Kasturba Medical College, Manipal University, Mangalore, Karnataka, India
|Date of Web Publication||9-Oct-2015|
S Vijendra Shenoy
Department of Otorhinolaryngology, Kasturba Medical College Hospital, Manipal University, Attavar, Mangalore - 575 001, Karnataka
Source of Support: None, Conflict of Interest: None
Inverted papilloma is a benign tumor with malignant potential that commonly arises from the lateral wall of the nose. We present a case of inverted papilloma, in a premalignant stage, of unusual origin and with oropharyngeal extension. The patient presented with complaints of a nasal mass arising from the left nasal cavity and progressively increasing in size since 2 years. On examination a fleshy, firm, sensitive mass was noted, which was attached to the medial wall, and extending posteriorly and inferiorly into the oropharynx. Diagnostic nasal endoscopy and complete excision of the mass was performed with a stable postoperative period. Histopathology of the mass revealed an inverted nasal papilloma of the septum with carcinoma in situ.
Keywords: Inverted papilloma, oropharynx, nasal septum
|How to cite this article:|
Kamath M P, Shenoy S V, Prasad V, Bhojwani K, Pai R, Mathew NM. Inverted papilloma of atypical origin with unusual extension into the oropharynx. J Can Res Ther 2015;11:666
|How to cite this URL:|
Kamath M P, Shenoy S V, Prasad V, Bhojwani K, Pai R, Mathew NM. Inverted papilloma of atypical origin with unusual extension into the oropharynx. J Can Res Ther [serial online] 2015 [cited 2020 May 31];11:666. Available from: http://www.cancerjournal.net/text.asp?2015/11/3/666/140830
| > Introduction|| |
Inverted papilloma of the nasal cavity is a benign neoplasm where the epithelium grows inwards, exhibiting certain aggressive features such as recurrence, and localized destruction of tissues and the possibility of malignant degeneration. It usually arises from the lateral nasal wall and seldom involves the frontal or sphenoid sinuses. Inverted papillomas comprise 0.5-4% of primary nasal tumors, with no specific side predilection.  The incidence of inverted papilloma on the nasal septum is further less. Inverted papilloma of the nasal septum accounts for between 5.5%  and 16.7%  of inverted papillomas. Here, we report a case of an inverted papilloma of the nasal septum with changes of carcinoma in situ, managed by endoscopic resection of the tumor along with a portion of the septum.
| > Case report|| |
An 83-year-old male patient with no comorbid illnesses presented with complaints of left sided nasal obstruction for 2 years and a nasal mass arising from the left nasal cavity since 1 year. The nasal obstruction was insidious in onset, gradually progressing along with a mass within the left nasal cavity, which is filling the entire cavity and minimally protruding out of the anterior nares [Figure 1]. He also had a hyponasal voice and persistent mucopurulent, nonblood stained nasal discharge on the same side. There was no associated ear, throat, or ophthalmological complaints. The patient also had no history of weight loss or appetite loss.
|Figure 1: Nasal mass seen arising from and filling the left nasal cavity, extending until the anterior nares|
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On examination, the vitals and systemic examination were within normal limits. Examination of the nose revealed a fleshy polypoidal firm mass protruding out from the left nares, the bulk of the mass filling the entire left nasal cavity. On probing, the mass was fleshy and firm in consistency. The mass was insensitive to touch and not bleeding on touch. On examination of the oral cavity and oropharynx, the fleshy mass from the nasal cavity was seen hanging from the nasopharynx, and it was up to the level of the tip of the uvula within the oropharynx [Figure 2].
|Figure 2: Nasal mass noted to be extending posteriorly and hanging in the oropharynx|
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A contrast-enhanced computed tomography scan of the nose, paranasal sinuses and nasopharynx was taken, which revealed a soft tissue density filling bilateral nasal cavities with mild heterogeneous enhancement causing complete obliteration of the left nasal cavity and partial obliteration of the right nasal cavity with extension into the posterior nasal choana, and nasopharynx up to the upper limit of the oropharynx with obliteration of the nasopharynx [Figure 3] and [Figure 4]. The patient was posted for endoscopic excision of the mass under general anesthesia. The patient was induced with general anesthesia, and a throat pack was secured. The nasal cavity was topically decongested with cottonoids soaked in 4% lignocaine mixed with adrenaline. Local infiltration of the septum was done with 2% lignocaine mixed with adrenaline. A 4 mm 0° rigid nasal endoscope connected to the camera was passed into the nasal cavity, and the site of origin of the mass was localized. Intraoperatively, the mass was noted to be arising from the posterior end of the bony nasal septum on the left side and extending into the oropharynx through the nasopharynx. The attachment of the mass was released along with a margin of the posterior nasal septum. The scope was removed from the nasal cavity. The oral cavity was kept open using a Boyle-Davis mouth gag. The entire mass was then delivered in toto through the oral cavity. Hemostasis was achieved by the topical application of cottonnoids soaked in 4% lignocaine with adrenaline at the sites of bleeding. The nasal cavity was then packed with Merocel ® bilaterally and an external bolster was applied. The pack was removed on the 2 nd postoperative day, and the patient was started on topical oxymetazoline nasal drops.
|Figure 3: Contrast-enhanced computed tomography image of the nose and paranasal sinuses revealing the mass within the left nasal cavity with haziness of the sinuses|
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|Figure 4: Contrast-enhanced computed tomography image of the nose and paranasal sinuses revealing the extent of the nasal mass into the oropharynx through the nasopharynx|
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The specimen was sent for histopathological evaluation [Figure 5]. Grossly, the specimen consisted of large, bulky, translucent, membranous mass of 8.5 × 5.0 × 3.0 cm. The surface showed polypoidal projections. On microscopy, the sections showed respiratory mucosa with a tumor consisting of invaginated, endophytic growth of no keratinizing (transitional) squamous epithelial cells giving it a lobulated appearance. Glycogenated squamous cells were present. In one section, carcinoma-in situ change was seen with nuclear hyperchromatism and increased mitosis. There was an extension of the papilloma into the seromucinous glands. The stroma was edematous and showed minimal inflammation. The surface epithelium was merging with the transitional squamous epithelium. Initially, six sections were studied. In view of the presence of carcinoma-in situ in one of the sections, further evaluation was done. Six more sections from difference areas were studied and found to be negative for malignancy. The final histopathological evaluation was reported as inverted squamous papilloma (Schneiderian type) with a single focus of carcinoma in one low power field. There was no evidence of dysplasia or invasive malignancy in any of the studied sections [Figure 6] and [Figure 7].
|Figure 5: Excised nasal mass prior to dispatch for histopathological evaluation|
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|Figure 6: Inverted Schneiderian papilloma showing an endophytic growth pattern of no keratinizing squamous epithelium (H and E, low power)|
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|Figure 7: Inverted papilloma showing the single focus of carcinoma-in situ on the surface (H and E, high power)|
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On follow-up, endoscopic cleaning of the nasal cavities was done, which showed no evidence of residual or remnant tumor, and the patient felt symptomatically better. The patient has been on regular follow-up for 6 months, and no recurrence has been noted.
| > Discussion|| |
The World Health Organization defines inverted papilloma as a benign epithelial tumor composed of well-differentiated columnar or ciliated respiratory epithelium having variable squamous differentiation.  They classify inverted papilloma as a subgroup of Schneiderian papillomas. The term inverted papilloma describes the histological appearance of the epithelium that is, inverting into the stroma, with a distinct and intact basement membrane that separates and defines the epithelial component from the underlying connective tissue stroma. 
The most common nasal sites involved by inverted papilloma, in order of descending prevalence, are: Lateral nasal wall, ethmoid cells, maxillary sinus, and less often, the frontal and sphenoid sinuses and nasal septum.  The lateral nasal wall represents the most common site of origin, whereas paranasal sinuses are quite frequently found to be involved by extension. There have been no reported incidences of oropharyngeal extension of an inverted papilloma.
Inverted papilloma poses many challenges clinically, pathologically, and surgically in terms of management. Various surgical techniques have been advocated for the treatment of the same. Traditional lateral rhinotomy is performed, which provides good access to the tumor and ensures complete removal. Despite achieving complete surgical removal, recurrences tend to occur. Recurrence rates of inverted papilloma, when treated surgically are as high as 71%. , Persistent disease is unacceptable due to the possibility of malignant transformation in squamous cell carcinoma, the incidence of which is as high as 10-15%, according to one study by Roh et al.  In a literature review by von Buchwald and Bradley the risk of transformation of cases to squamous cell carcinoma was quoted as 10% with the rates of synchronous and metachronous malignancy in inverted papilloma being 7.1 and 3.6%, respectively.  Lawson et al., in their series of 112 patients with inverted papilloma, describes the overall rate of squamous carcinoma to be 5%.  In yet another study by Saha et al.,  23% of the patients had recurrence without any specific histological patter, 12% had recurrence with focal dysplasia and 4% had recurrence with malignant transformation into squamous cell carcinoma. The time from presentation with inverted papilloma to malignant transformation is reported to be between 6 and 180 months (mean 52 months).  Hence, it is mandatory to perform a close follow-up of all cases, with timely biopsies when indicated. Lifelong follow-up is recommended.
Immunohistochemical studies have shown increased expression of p21 and p53 in inverted papillomas associated with dysplasia and squamous cell carcinoma. Human papilloma virus (HPV) family 6/11 and 16/18 DNA has been observed in inverted papilloma associated with severe dysplasia and squamous cell carcinoma and HPV infection may be an early step in the process of malignant transformation.  Arndt et al.  demonstrated the presence of HPV through polymerase chain reaction in 69% of their cases with inverted papilloma. HPV 6/11 was seen in 48%, HPV 16 in 65% and both HPV 6/11 and 16 in 45%, and HPV 16 was implicated to be involved in the malignant transformation of inverted papilloma. Similar results were demonstrated in a study by Hwang et al.,  suggesting a relation between malignant transformation and recurrence of inverted papillomas with the presence of HPV infection. The analytical overview by Lawson et al.  hypothesized that low risk HV may induce the formation of inverted papilloma; however, they are lost as the infected cells are shed, in a "hit and run" phenomenon. Dysplasia in inverted papilloma however has been attributed to either primary of secondary infection with high-risk HPV. However, in one recent study by Jenko et al., it was illustrated that the presence of HPV DNA was not a statistically significant predictor of the recurrence of inverted papilloma, nor was it a statistically significant risk factor for associated squamous cell carcinoma and when compared with the control cases represented an incidental colonization rather than a causative factor for inverted papilloma. 
Lawson et al. in 1995  in a study on inverted papilloma described septectomy as a method of treatment of isolated septal inverted papillomas. Our patient underwent transnasal endoscopic resection of the tumor with posterior septectomy. Transnasal endoscopic surgery avoided aggressive surgery and facial scarring in this patient, especially in view of the limited growth within the nasal cavity. Kelly et al.  described inverted papillomas of the nasal septum and the advantages of the various methods of excision of the lesion. Their review suggested that cautery of septal papillomas or superficial excision and cautery of the base was insufficient treatment. A diagnosis of inverted papilloma necessitated a well-conceived, wide, local excision. Small lesions are amenable to be excised through a transnasal approach, but with the inclusion of a generous margin of mucoperichondrium. If cartilage invasion is questionable, then cartilage must also be removed with the placement of a skin or mucosal graft.
The main differential diagnoses in this case are anterochoanal polyps, rhinosporidiosis, olfactory neuroblastoma and sinonasal carcinoma. Nasal polyps which are more frequent than inverted papilloma, can commonly present as a single, unilateral, nasal mass extending to the nasopharynx, and must be considered as one of the first main differential diagnosis. However, considering the advanced age of the patient and the nature of the mass on examination (fleshy versus smooth and polypoidal), inverted papilloma is more likely. Rhinosporidiosis is a commonly encountered condition in South India and can be considered a valid differential in this case. However, the patient did not elicit a history of nasal bleed nor was the mass friable or bleeding on probing, which led the author away from this differential. Olfactory neuroblastoma and other sinonasal carcinoma must also be kept in mind considering the patient's age, but the main point is diverting the examiner away from malignancy as a differential is the slow growing nature of the nasal mass.
| > Conclusion|| |
Nasal mass arising from the medial wall, that is the septum, should arouse the suspicion of a differential diagnosis of inverted papilloma of the septum. Complete excision of the growth with partial septectomy through transnasal endoscopy may be performed for control of the limited disease.
| > References|| |
Lane AP, Bolger WE. Endoscopic management of inverted papilloma. Curr Opin Otolaryngol Head Neck Surg 2006;14:14-8.
Kelly JH, Joseph M, Carroll E, Goodman ML, Pilch BZ, Levinson RM, et al.
Inverted papilloma of the nasal septum. Arch Otolaryngol 1980;106:767-71.
Shanmugaratnam K. Nasal cavity and paranasal sinuses (excluding nasal vestibule). In: Shanmugaratnam K, editor. Histological Typing of Tumours of the Upper Respiratory Tract and Ear. Berlin: Springer-Verlag; 1991. p. 20-1.
Mirza S, Bradley PJ, Acharya A, Stacey M, Jones NS. Sinonasal inverted papillomas: Recurrence, and synchronous and metachronous malignancy. J Laryngol Otol 2007;121:857-64.
Krouse JH. Endoscopic treatment of inverted papilloma: Safety and efficacy. Am J Otolaryngol 2001;22:87-99.
Ridolfi RL, Lieberman PH, Erlandson RA, Moore OS. Schneiderian papillomas: A clinicopathologic study of 30 cases. Am J Surg Pathol 1977;1:43-53.
Roh HJ, Procop GW, Batra PS, Citardi MJ, Lanza DC. Inflammation and the pathogenesis of inverted papilloma. Am J Rhinol 2004; 18:65-74.
Von Buchwald C, Bradley PJ. Risks of malignancy in inverted papilloma of the nose and paranasal sinuses. Curr Opin Otolaryngol Head Neck Surg 2007;15:95-8.
Lawson W, Ho BT, Shaari CM, Biller HF. Inverted papilloma: A report of 112 cases. Laryngoscope 1995;105:282-8.
Saha SN, Ghosh A, Sen S, Chandra S, Biswas D. Inverted papilloma: A clinico-pathological dilemma with special reference to recurrence and malignant transformation. Indian J Otolaryngol Head Neck Surg 2010;62:354-9.
Sandison A. Common head and neck cases in our consultation referrals: Diagnostic dilemmas in inverted papilloma. Head Neck Pathol 2009;3:260-2.
Katori H, Nozawa A, Tsukuda M. Markers of malignant transformation of sinonasal inverted papilloma. Eur J Surg Oncol 2005;31:905-11.
Arndt O, Nottelmann K, Brock J, Neumann OG. Inverted papilloma and its association with human papillomavirus (HPV). A study with polymerase chain reaction (PCR). HNO 1994;42:670-6.
Hwang CS, Yang HS, Hong MK. Detection of human papillomavirus (HPV) in sinonasal inverted papillomas using polymerase chain reaction (PCR). Am J Rhinol 1998;12:363-6.
Lawson W, Schlecht NF, Brandwein-Gensler M. The role of the human papillomavirus in the pathogenesis of Schneiderian inverted papillomas: An analytic overview of the evidence. Head Neck Pathol 2008;2:49-59.
Jenko K, Kocjan B, Zidar N, Poljak M, Strojan P, Zargi M, et al.
In inverted papillomas HPV more likely represents incidental colonization than an etiological factor. Virchows Arch 2011;459:529-38.
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]