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La tunisie Medicale - 2017 ; Vol 95 ( n°06 ) : 453-454
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Summary

Abstract:
A 57 year-old, male presented with a chronic unilateral nasal obstruction and epistaxis. Intranasal endoscopy showed multiple polypoid lesions. The computed tomography exam revealed a heterogeneous mass that occupied the right nasal cavity with osteolysis of the middle and lower cone causing fluid retention of the right maxillary sinus. He underwent resection of these lesions. Pathological examination revealed malignant transformation of nasal inverted papilloma into sarcomatoid carcinoma. This case report highlights the importance of considering malignant transformation in the differential diagnosis of polypoid lesions.

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Article


Introduction
Nasal inverted papilloma is a benign epithelial tumor that develops from schneiderian mucosa lining the nasal cavity [1]. His incidence is estimated at 0.4 cases per 100 000 people. It typically corresponds to an endophytic proliferation of transitional cells. It has the potential to recur and the tendency towards malignant transformation [2]. 
The aim was to determine clinical and histological features of malignant inverted papilloma and to discuss the differential diagnosis.
Case report
We present the case of a 57 year-old male, who presented with a chronic unilateral nasal obstruction. He complained of epistaxis and headaches. The clinical examination revealed a right nasal tumor. Intranasal endoscopy showed multiple polypoid lesions. The computed tomography exam revealed a heterogeneous mass of tissue density that occupied the right nasal cavity with osteolysis of the middle and lower cone causing fluid retention of the right maxillary sinus. The treatment was decided to consist in surgical resection. Frozen section showed a benign inverted papilloma. But the definitive microscopic examination revealed malignant areas. It showed hyperplastic aggregates of well-demarcated squamous and respiratory epithelium that growth endophytically [Figure 1]. These lesions were merely associated with carcinomatous component. This component showed squamous differentiation [Figure 2]. It was composed by nests and sheets of cells with pearl formation and intercellular bridges. Some other tumoral areas revealed undifferentiated component with infiltrative sheets of spindle shaped cells with markedly atypical nuclei and numerous mitotic figures [Figure 3]. Immunohistochemical study revealed that the sarcomatous area expressed Vimentin antigen. Actine, desmine, caldesmone and PS100 were negative revealing an   undifferentiated sarcomatous component. The diagnosis of malignant transformation of nasal inverted papilloma into sarcomatoid carcinoma was made.
Discussion:
Inverted papilloma specifically affects male subjects with a sex ratio of 3/1. The median age is 50 to 60 years [2]. Nasal obstruction is the most common presenting symptom [1]. The case presented with nasal obstruction and epistaxis.
This tumor has the potential to recur and exhibit malignant transformation [3]. The latter is possible in 3-15% of cases [4]. Malignant transformation is characterized by the presence of the following features: cellular pleomorphism, atypical mitoses, keratin pearls and stromal invasion [5]. Based on the literature, the rates of synchronous and metachronous carcinoma are 7,1 and 3,6% respectively. Most cases are infiltrating squamous cell carcinomas, followed by transitional cell carcinomas. Transformation into sarcomatoid carcinoma is a rare condition which represents approximately 5% of malignant cases. This tumor has a sarcomatous morphology and aggressive behavior [6]. The Wnt signaling pathway plays an important role in embryogenesis and carcinogenesis [7]. The differential diagnosis include nasal polyp with squamous metaplasia and adenoid epithelial respiratory hamartoma.
The complete resection is difficult and the prognosis is poor. Complete endoscopic resection and close follow up are recommended in nasal inverted papilloma. In case of malignant transformation, post-operative radiation or chemotherapy will be needed, especially in case of sarcomatoid carcinoma.
Conflict of interest: The authors declare that they have no conflict of interests

Figures


Figure 1: Pathological examination (Hematoxylin eosin x 10):  Inverted papilloma characterized by respiratory epithelium that growth endophytically


Figure 2: Pathological examination (Hematoxylin eosin x 20): Malignant transformation of the inverted papilloma


Figure 3: Pathological examination (Hematoxylin eosin x 40): Sarcomatoid component with infiltrative sheets of spindle shaped cells with markedly atypical nuclei and numerous mitotic figures

Reference
  1. Vorasubin N, Vira D, Suh JD, Bhuta S, Wang MB. Schneiderian papillomas: comparative review of exophytic, oncocytic, and inverted types. Am J Rhinol Allergy. 2013; 27: 287-92.
  2. Terada T. Malignant transformation of exophytic Schneiderian papilloma of the nasal cavity. Pathol Int. 2012; 62: 199-203.
  3. Nicolae V, Sabău M, FăgeŢan MI, Petrescu Seceleanu V, Iorgulescu D, Dumitra DE, Sabău AD, Ibric-Cioranu S, Sava A, Ibric-Cioranu V. Clinical and histological aspects of malignant inverted sinonasal papilloma. Two case reports. Rom J Morphol Embryol. 2016; 57: 289-94.
  4. Nygren A, Kiss K, von Buchwald C, Bilde A. Rate of recurrence and malignant transformation in 88 cases with inverted papilloma between 1998-2008. Acta Otolaryngol. 2016; 136: 333-6.
  5. El-Naggar AK, Chan JKC, Grandis JR, Takata T, Slootweg PJ. World Health Organization Classification of Head and Neck Tumours. Lyon: IARC Press; 2017. p.28
  6. Yu HX, Liu G. Malignant transformation of sinonasal inverted papilloma: A retrospective analysis of 32 cases. Oncol Lett. 2014; 8: 2637-41.
  7. Jung YG, Lee HW, Kim MG, Dhong HJ, Cho KS, Roh HJ. Role of Wnt signaling pathway in progression of sinonasal inverted papilloma to squamous cell carcinoma. Am J Rhinol Allergy. 2015; 29: e81-6.
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