Inverted Nasal Papilloma Affecting Ears and Pulmonary Parenchyma: Case Report.

Inverted papilloma is a rare sinonasal neoplasm. It can be locally invasive and potentially degenerate to a malignant tumor. We present a case report of a 36-year-old woman who was treated for nasal inverted papilloma for over 10 years and presented bilateral temporal bone, and pulmonary involvement. Several procedures were performed to completely remove the tumor. Even without evidence of malignant degeneration, the patient continued battling tumor recurrences. To the best of our knowledge, this report presents the first case of a multicentric inverted papilloma with nasal, bilateral temporal bone, and pulmonary metachronous localization. Laryngoscope, 131:E2640-E2642, 2021.

Pediatric naso-sinusal inverted papilloma: report of a case and literature review.

Inverted papilloma is a rare nasosinusal tumor that mainly occurs in adults during the 5th decade. The occurrence in children is exceptional and only few cases have been reported in the litterature. Clinical and radiological findings mimic other benign nasosinusal pathologies; therefore, diagnosis is based on histopathology either via biopsy or following surgical excision. Here we present a rare case of pediatric inverted papilloma in a 11-year-old child and we discuss clinical, radiological, therapeutic and evolutionary features through a literature review.

Clinical Implications of Carcinoma In Situ in Sinonasal Inverted Papilloma.

OBJECTIVE: Sinonasal inverted papilloma (IP) is a typically benign sinonasal tumor with a tendency to recur and the potential for malignant transformation. Varying degrees of dysplasia may be present, of which carcinoma in situ (CIS) is most advanced. We hereby describe the biological and clinical behavior of IP with CIS (IPwCIS). STUDY DESIGN: Retrospective cohort. SETTING: Tertiary academic referral center. SUBJECTS AND METHODS: Patients who underwent surgical resection for IP between 2002 and 2017. Pertinent clinical data were obtained, and all IPwCIS cases were histologically confirmed. RESULTS: In total, 37 of 215 cases (17.2%) were identified with IPwCIS. Mean age was 57 years and 86.5% of patients were male. Median follow-up was 82 months, and the recurrence rate was 27%. The maxillary sinus was the most common primary site (37.8%) and 14 tumors (37.8%) demonstrated multifocal attachment, which was associated with recurrence (odds ratio [OR], 9.7; 95% confidence interval [CI], 1.4-112.8; P = .028). IPwCIS was also associated with multiple recurrences (OR, 2.71; 95% CI, 1.246-5.814; P = .021). Most patients were treated with surgery alone (89.1%) and 4 patients received adjuvant radiotherapy (8.1%). Only 1 patient (2.7%) demonstrated malignant transformation after definitive surgery. CONCLUSIONS: IPwCIS represents the most severe degree of dysplasia prior to malignant transformation and is associated with higher recurrence rate and multifocal involvement but low rate of conversion to invasive carcinoma. The need for adjuvant therapy remains controversial, and further research into the etiology of the disease is warranted.

Better surgical outcome by image-guided navigation system in endoscopic removal of sinonasal inverted papilloma.

PURPOSE: The purpose of this study was to validate and compare treatment outcomes for endoscopic resection of sinonasal inverted papilloma (IP) with or without the use of a navigation system. MATERIALS AND METHODS: A total of 58 patients who underwent endoscopic resection of sinonasal inverted papilloma by a single surgeon from 2007 to 2016 at our institution were retrospectively reviewed. Depending on the use of the navigation system, subjects were divided into two groups: a conventional endoscopic resection group without navigation system (CER group) and a navigation-assisted endoscopic resection group (NER group). RESULTS: There were 24 patients (41.4%) in the CER group and 34 patients (58.6%) in the NER group. Treatment outcomes showed that navigation-assisted endoscopic resection was a more beneficial surgical technique than conventional endoscopic resection for sinonasal IP. Post-surgical recurrence was noted in seven cases (29.2%) in the CER group and two cases (5.9%) in the NER group. Accordingly, the recurrence rate was significantly less in the NER group compared to the CER group (p = 0.026). There were two cases of complications (8.3%) in the CER group comprising cerebrospinal fluid leak and periorbital fat exposure, while no complications were noted for the NER group (p = 0.167). CONCLUSION: This study demonstrated that navigation-assisted endoscopic removal of sinonasal IP is helpful for reducing recurrence and avoiding surgical complications. Therefore, navigation systems should be always considered when performing endoscopic removal of sinonasal IP.

Clinical features of nasal and sinonasal inverted papilloma associated with malignancy.

OBJECTIVE: Nasal and sinonasal inverted papilloma (IP) are rare benign tumors and have the potential to exhibit malignancy in approximately 10% of cases. This study aimed to analyze the clinical features of IP associated with malignancy. Furthermore, we reviewed our therapeutic strategy and the clinical course of malignant IP. METHODS: Overall, 70 patients with IP at our institution were retrospectively analyzed from April 2006 to December 2015; of these, six (9%) had associated malignancy. Data was collected on sex, age, presenting symptoms (nasal bleeding, rhinorrhea, facial or cheek pain, and nasal obstruction), bone destruction, and extent of disease on CT and MRI. Categorical data of patients with and without malignancy were compared using the chi-square test. A p value of <0.05 was considered statistically significant. Our therapeutic strategy for IP with malignancy, particularly the surgical procedure, i.e., the external incision or the endoscopic nasal approach, varied based on when the carcinoma was detected. In addition, we considered postoperative radiation therapy depending on histological examination. RESULTS: Nasal bleeding (p<0.001), pain (p=0.040), bone destruction (p<0.001), and extent of disease (p=0.026) on CT and MRI findings were significantly associated with malignancy. Carcinoma was diagnosed preoperatively in two (33%) and postoperatively in four patients (67%). We operated five patients (one case was not treated because of end-stage pancreatic cancer). Two patients underwent endoscopic sinus surgery (ESS) alone, two ESS plus Denker's method, and one ESS plus anterior craniotomy. Three patients underwent surgery only, and two patients received postoperative radiotherapy. The median follow-up period was 69.3 months. One patient died of the disease and the remaining patients are alive without recurrence. CONCLUSION: For IP patients exhibiting these clinical findings preoperatively, we should suspect complication with malignancy and plan a treatment. Even if postoperative histology does not confirm malignancy, we should ensure careful observation because of metachronous malignant transformation or the possibility to overlook small malignant lesions. Our result suggests that our strategy for malignant IP could be a reasonable option.

Outcomes of sinonasal squamous cell carcinoma with and without association of inverted papilloma: A multi-institutional analysis.

INTRODUCTION: Sinonasal squamous cell carcinoma (SCC) accounts for <1% of all malignancies but represents 70% of sinonasal cancer. Up to 10% of SCCs are associated with inverted papilloma (IPSCC). Studies that compare patients, treatment, and outcomes of SCC and IPSCC are absent in the literature. METHODS: A retrospective review of patients with SCC and those with IPSCC at Cleveland Clinic and Indiana University from 1995 to 2015. The records were analyzed for demographics, tumor characteristics, treatment, and outcomes. RESULTS: The study comprised 117 patients with SCC, of whom, 29 had IPSCC. The mean age at diagnosis was similar: 63 and 64 years for patients with SCC and patients with IPSCC, respectively; with female patients representing 36% and 34%, respectively (p > 0.99).Smokers represented 64% of the patients with SCC and 55% of patients with IPSCC (p = 0.3); excessive alcohol intake was noted in 16% of the patients with SCC and 21% of the patients with IPSCC (p = 0.56).The maxillary sinus was most commonly involved, followed by the nasal cavity (51% versus 35% SCC, 45% versus 38% IPSCC). Frontal ethmoid and sphenoid sinuses contained primary tumors only in patients with SCC. Upfront treatment was surgery in 84% of patients with SCC and 97% of patients with IPSCC (p = 0.18); 68 and 55% received radiation, respectively, and 25 and 21% received chemotherapy, respectively.Overall survival averaged 5.5 and 3.4 years for patients with SCC and patients with IPSCC, respectively (p = 0.12); disease-free survival was 4.8 and 2.9 years, respectively (p = 0.18). Nodal metastasis was more likely in patients with SCC (18 versus 0%; p = 0.02). When divided into high- and low-stage disease: more common nodal metastases were demonstrated in high-stage SCC than in low-stage disease (p = 0.03). Overall survival was decreased between high- and low-grade disease but not when subdivided between SCC and IPSCC. CONCLUSION: Although SCC with and without IP association are considered different diseases, their demographics and outcomes seem similar. Nodal metastasis was noted to be higher in the SCC cohort, which may indicate different tumor biology. Further study is warranted.

Malignant transformation of sinonasal inverted papilloma and related genetic alterations: a systematic review.

Schneiderian papillomas are uncommon tumors which may develop within the nasal cavity and comprise three well-defined histological types: sinonasal inverted papilloma (SNIP), exophytic papilloma, and oncocytic papilloma. It is well known the rate of Schneiderian papilloma may also present a malignant degeneration and SNIP represents the most important subgroup in consideration of its frequency and malignant propensity. Although HPV infection is always considered the first event favoring the development of SNIP, however, it is not established as an eventual connection between viral actions and malignant transformation. In fact, different molecular mechanisms are suspected to play a crucial role in this process and, currently, many authors agree that only by improving our knowledge about these mechanisms it will be possible to achieve new and effective targeted therapies. So the aim of this study was firstly to systematically review the literature focusing on different biomarkers that could be implicated in the stages of SNIP malignant degeneration. Secondly, a systematic review with meta-analysis was performed to better define the incidence of sinonasal malignancies originating from Schneiderian papilloma (SNIP, exophytic papilloma, and oncocytic papilloma). Twenty-nine studies comprising a total of 3177 patients were statistically analyzed. Results showed a 9% (95% CI = 7-11) overall rate of malignant transformation from Schneiderian papilloma. In conclusion, this analysis confirmed that the potential malignancy of Schneiderian papilloma should not be underestimated. On the other hand, our review showed the paucity of studies investigating the molecular alterations which may be related with the malignant transformation of SNIP.

Clinical management of squamous cell carcinoma associated with sinonasal inverted papilloma.

OBJECTIVE: The aim of this study was to investigate the clinical features and prognosis of patients with squamous cell carcinoma (SCC) associated with sinonasal inverted papilloma (IP). METHODS: The medical records of 95 patients who were diagnosed with IP or SCC associated with IP were retrospectively reviewed. Out of 95 patients, 15 were diagnosed with SCC associated with IP. The clinical characteristics, treatment modalities, and survival outcomes of the patients were analyzed. RESULTS: The incidence of SCC associated with IP was 15.8%. Although differential diagnosis between IP and SCC associated with IP is difficult, epistaxis may be the specific symptom in SCC associated with IP cases. The 3-year disease-specific survival rate was higher in cases with T1, 2 and 3 than in cases with T4. There was no significant difference in survival rate between maxillary sinus and other primary sites. On the other hand, there was a significant difference in survival rate between the microscopic SCC with IP cases and the other cases. In addition, the patients with <70 years old better than those with >70 years old with a 3-year disease free survival of 80% versus 0%. CONCLUSIONS: Some T4 patients were found to have a highly aggressive disease. Therefore, complete surgical resection followed by chemo-radiation therapy is the recommended treatment for patients with T4 disease to control of the primary tumor site.

Inverting papilloma of the temporal bone: Report of four new cases and systematic review of the literature.

OBJECTIVES/HYPOTHESIS: Inverting papillomas (IPs) are benign locally invasive tumors that most commonly present within the sinonasal cavity. Temporal bone involvement is exceedingly rare, with fewer than 30 cases reported within the English literature to date. STUDY DESIGN: Case series and systematic review of the literature. METHODS: Four consecutive subjects with temporal bone inverting papilloma (TBIP) were treated, and an additional 28 previously published cases were identified in the literature. Main outcome measures were disease presentation, diagnostic evaluation, management strategy, and outcome. RESULTS: A total of 32 cases were analyzed. The median age at diagnosis was 54 years (mean 54.1; range 19-81 years). Nineteen (59%) patients had synchronous or metachronous sinonasal IP, whereas 13 (41%) had isolated temporal bone disease without sinus involvement. Over half of the patients undergoing microsurgical resection experienced at least one recurrence. Compared to patients with a history of sinus IP, subjects with primary TBIP were younger at time of presentation (44 vs. 58 years; P=0.012); were more commonly female (62% vs. 32%; P=0.15); and were less likely to have intracranial spread (8% vs. 26%; P=0.36), cranial neuropathy (8% vs. 26%; P=0.36), human papillomavirus positivity (11% vs. 57%; P=0.11), or associated carcinoma (0% vs. 47%; P=0.004). CONCLUSIONS: Inverting papilloma of the lateral skull base is rare and can pose a significant therapeutic challenge. Primary lesions of the temporal bone appear to follow a less aggressive clinical course when compared to those arising in association with sinonasal disease. Gross total resection is the preferred method of treatment, when feasible, given the high rate of recurrence with subtotal resection and risk of associated malignancy. LEVEL OF EVIDENCE: 4.

[Treatment and prognosis of the carcinoma arising from nasal and sinonasal inverted papilloma: report of 62 cases and systematic review].

OBJECTIVE: To analyze the factors affecting prognosis of the carcinoma arising from nasal and sinonasal inverted papilloma. METHODS: The clinicopathological data of sixty-two patients treated in our hospital from January 1974 to February 2012 were retrospectively analyzed. Of the 62 cases, 10 were at stage I or II, 24 at stage III, and 28 at stage IV. Twenty-six patients were treated with surgery alone, and 36 with surgery combined with radiation therapy. Kaplan-Meier method and log-rank test were used for the survival analysis. RESULTS: The overall 5-years survival rate was 39.6%. The 5-years survival rate was 67.5% in the stage I or II patients,51.3% in the stage III patients, and 18.3% in the stage IV patients (P<0.05). The 5-years survival rate was 65.7% in patients who had no surgical history, and 29.9% in patients who had surgery (P<0.05). The 5-year survival rate was 17.6% in the group who relapsed after the treatment, and 49.6% in the non-relapsed patients (P<0.05). The 5-year survival rate was 23.4% in the patients who had involvement of cranial base and (or) orbit, and 47.6% in the patients whose cranial base and (or) orbit were clean (P<0.05). Of the patients at the same stage (III-IV), the 5-year survival rate of patients treated with surgery alone was 32.4%, and those treated with combination therapy was 36.2%(P=0.89). The univariate analysis showed that clinical stage, surgical history before malignization, involvement of the cranial base and (or) orbit organs, and post-operative relapse are significantly correlated to prognosis of the patients (P<0.05 for all). Multivariate analysis showed that age, clinical stage, and previous history of surgery were independent factors affecting the prognosis of the patients. Distant metastasis was the major cause of death, mostly lung metastases. CONCLUSIONS: Age, clinical stage and surgical history are the main factors affecting the prognosis of the patients. The history of recurrence and involvement of cranial base or orbit also play an important role for the prognosis. Distant metastasis is the main cause of death in the patients with carcinoma arising from nasal and sinonasal inverted papilloma.

Inverted papilloma of lacrimal sac invading into the orbit: case report and review of literature.

Inverted papilloma (IP) is a sinonasal tumor of benign etiology with local invasion and malignant potential. IP arising in lacrimal sac invading the orbit is extremely rare with only one case reported so far. The presented case appears to be the second such case reported in the literature. A 60-year-old Caucasian male presented with a medial canthal mass and epiphora. Incisional biopsy confirmed a transitional neoplasm. The lesion was completely excised enbloc with clear margins by using a Weber-Ferguson incision; orbital clearance and nasolacrimalfossa clearance was achieved via a medial maxillectomy. Enbloc resection of orbital and nasolacrimal parts of the tumor with clear margins is recommended.

Long-term outcome of primary Papillary Urothelial Neoplasm of Low Malignant Potential (PUNLMP) including PUNLMP with inverted growth.

BACKGROUND: Few larger studies have evaluated the long-term outcome after a diagnosis of papillary urothelial neoplasm of low malignant potential (PUNLMP), demonstrating a broad range of recurrence and progression rates. Additionally, no study has addressed the outcome of PUNLMP exhibiting inverted growth. We evaluated the long term clinical outcome of primary papillary urothelial neoplasm of low malignant potential (PUNLMP), including PUNLMP with inverted growth in a large single center study. METHODS: We evaluated 189 primary PUNLMP (177 exophytic, 12 inverted), diagnosed from January 1, 2000 to December 31, 2009, in a centralized uropathology practice. We excluded PUNLMP diagnosed after a previous or with a concurrent urothelial neoplasm. Recurrence was defined as any subsequent urothelial neoplasm, regardless of the grade. Progression was defined as any subsequent higher-grade or invasive urothelial neoplasm. Recurrence and progression were established only if documented on a subsequent biopsy. Descriptive statistical analysis was performed using Microsof Excel software package. RESULTS: The location of PUNLMP included bladder (187) and renal pelvis and ureter (1 each). After a median follow-up of 61 months (range, 9-128 months), 20.1% patients developed a recurrence. Recurrence with PUNLMP only was found in 9% of patients. Subsequent low-grade urothelial carcinoma was documented in 9.5% of patients. Progression to high-grade urothelial carcinoma was found in 1.6% patients (1% with muscle invasion). No patients with recurrent PUNLMP or subsequent low-grade carcinoma demonstrated invasion. All patients with PUNLMP exhibiting an inverted growth had no recurrence or progression on follow-up. CONCLUSION: In this study, primary PUNLMP recurred primarily either as PUNLMP or low grade urothelial carcinoma. Primary PUNLMP rarely progressed to high grade or invasive carcinoma on long term follow-up. No recurrence or progression was documented on follow-up for PUNLMP that demonstrated exclusively inverted growth. VIRTUAL SLIDES: The virtual slide(s) for this article can be found here: http://www.diagnosticpathology.diagnomx.eu/vs/1332825572154074.

[Inverted papiloma and its rare forms]. / Invertovaný papilóm a jeho zriedkavé formy.

Authors address the issue of a frequent benign tumour of the nasal cavity and paranasal sinuses -  inverted papilloma. They analyse the available diagnostic methods and treatment options. On the background of selected case reports of a rare malignant transformation they emphasize the need for longterm dispensarization as a part of management plan for patients with this oncological disease.

Fibroblast growth factor receptor-1 as a potential therapeutic target in sinonasal cancer.

BACKGROUND: Despite multimodal treatment, sinonasal malignancies have an unfavorable prognosis. The purpose of this study was to elucidate if these tumors harbor amplifications of the fibroblast growth factor receptor 1 (FGFR1) gene, which has recently been identified as a potential therapeutic target in squamous cell lung cancer. METHODS: One hundred twelve primary tumors (including squamous cell carcinoma [SCC], carcinoma associated with an inverted papilloma, sinonasal undifferentiated carcinoma [SNUC], adenocarcinoma, adenoid cystic carcinoma [ACC], esthesioneuroblastoma, and 9 corresponding lymph node metastases) were assessed by fluorescence in situ hybridization (FISH) for FGFR1 copy number status. Human papillomavirus (HPV) status was assessed by p16 immunohistochemical as a surrogate marker. RESULTS: FGFR1 amplification was found in subsets of sinonasal SCCs (20%), carcinomas associated with an inverted papilloma (33%), and SNUCs (5%). In all cases, metastatic tumor samples shared the same FGFR1 amplification status as the corresponding primary tumor tissue. None of the FGFR1-amplified tumors expressed p16. CONCLUSION: FGFR1 amplification represents a potential molecular target in a subset of patients with sinonasal cancer. © 2014 Wiley Periodicals, Inc. Head Neck 36: 1253-1257, 2014.

Chapter 9: Benign sinonasal neoplasms.

Benign sinonasal neoplasms are a heterogeneous group of tumors that present with similar symptoms including nasal obstruction, anosmia, rhinorrhea, and epistaxis. The proper workup and accurate diagnosis is essential for these tumors so that the appropriate treatment plan can be established. In this article of benign sinonasal neoplasms, we discuss their typical clinical presentation, histological and radiographic findings, and treatment options.

Synchronous bilateral inverted papilloma of the temporal bone: Case report and review of the literature.

BACKGROUND: Temporal bone inverted papilloma (IP) is an extremely rare tumor. Its etiology is unknown and represents a source of debate. Only 2 previous cases of bilateral temporal bone IP have been reported. A case report and review of the literature via PubMed database search are presented. MATHODS AND RESULTS: A 52-year-old African-American man who initially underwent medial maxillectomy for right-sided nasal IP returned with bilateral temporal bone IP 7 months later without evidence of extension through the Eustachian tubes. Despite multiple resections and adjuvant radiation, the tumor transformed into squamous cell carcinoma and progressed to involve the intracranial dura, temporal lobe, and cervical dura. CONCLUSIONS: Multiple origins may exist for temporal IP: direct extension, iatrogenic seeding, or development from ectopic Schneiderian epithelium. Temporal bone IP appears to represent a much more aggressive tumor than its nasal counterpart, necessitating aggressive early surgical intervention to decrease recurrence and transformation risk.