Inverted papilloma (IP) is an uncommon, benign yet aggressive epithelial neoplasm, characterised by a high recurrence rate and its propensity to turn malignant. IP originates from the mucosal epithelial lining of the sinonasal tract which is in continuity with the nasolacrimal duct (NLD) and lacrimal sac, with the majority of cases originating in the ethmoid region, lateral wall of the nasal fossa, and maxillary sinus [
A 69-year-old gentleman presented to the ophthalmology outpatient department with a varying lump in his right medial canthal region for two years. The patient reported chronic epiphora and discharge bilaterally, denying bloody tearing, pain, or diplopia. A nontender 1.5 cm smooth dome-shaped mass was palpable below the level of the right medial canthal ligament. Syringing revealed 90% flow and 10% reflux following dilatation of the puncta with no mucopurulent regurgitation. Visual acuity was 6/9 and 6/6 in the right and left eyes, respectively, without correction and there was no relative afferent pupillary defect or proptosis. The patient thirty years previously had conjunctival papilloma excised from the right eye with no extension into the medial canthus. He is otherwise fit and well.
Computed Tomography (CT) of the orbits revealed a 2 cm round well-defined, heterogeneously enhancing soft tissue mass within the proximal right NLD causing pressure erosion and thinning of the bony canal, protruding medially into the middle meatus and laterally into the right orbit (Figures
CT orbits: coronal section showing a well-defined heterogeneous lesion protruding into the middle meatus and right orbit.
CT sinuses: axial section showing the lesion within the right nasolacrimal duct with evidence of bony remodelling.
Magnetic Resonance Imaging (MRI) of the head confirmed a mass arising from the proximal NLD, mildly hypointense on T2 weighted images, isointense on T1 weighted images, and avidly enhancing on postcontrast images eroding the anterior lamina papyracea and bulging into the medial aspect of the right orbit. Right nasal bones were eroded with the mass extending into the nasal cavity to lie in the middle meatus anteriorly (Figure
Endoscopic view demonstrating a pale, polypoid-like mass, covered by a papillary surface, protruding from the middle meatus.
An endoscopic biopsy under general anaesthesia revealed a benign squamous papilloma of the lacrimal duct with no evidence of dysplasia or malignancy.
Following discussion at the Neuroradiology and Head and Neck Multidisciplinary meetings, complete resection as a joint case between Otorhinolaryngology, Oral and Maxillofacial Surgery, and Ophthalmology was planned.
A lateral rhinotomy approach was utilized to gain access to the tumour, followed by dissection along the medial wall of orbit and around tumour. The tumour was dissected free from its remodelled lacrimal fossa and the common canaliculus divided laterally to the tumour. The anterior buttress of the frontal process of maxilla overlying the remodelled NLD was drilled away. Right inferior turbinate, middle turbinate, and its posterior stump were removed endoscopically to expose the ostium. The tumour was thereby excised en bloc using a combined external and endoscopic approach. The lacrimal drainage pathway was not reconstructed on this occasion.
Histological examination revealed hyperplastic ribbons of basement membrane-enclosed epithelium growing endophytically into the underlying stroma (Figures
(a) Hyperplastic squamous epithelium growing endophytically. The inverted papilloma is seen to arise from nasolacrimal duct epithelium (arrow). Haematoxylin and Eosin, 20x. (b) Foci of atypia amounting to moderate dysplasia. No invasive malignancy is seen. Haematoxylin and Eosin, 40x. (c) Inverted pattern of growth. Haematoxylin and Eosin, 20x. (d) Immunohistochemistry for P16 showing block positivity. P16 IHC Syntech antibody, 20x.
The patient recovered with unchanged visual acuity and normal extraocular movements following surgery with no evidence of recurrence clinically at 15-month follow-up.
The Schneiderian papilloma was first described in 1854 by Ward and Billroth [
The lacrimal drainage system is an uncommon [
Lacrimal system papillomas largely present with epiphora and a medial canthal mass if detected early during the course of the disease. In advanced cases, proptosis, nonaxial globe displacement, skin ulceration, and invasion of adjacent structures can occur [
Preoperative radiological planning is an important step in determining the extent of the lesion and invasion into adjacent structures. CT and MRI together are the recommended technique for pretreatment staging in IP as they are complementary in the accurate evaluation of disease extension and in differentiating tumour from inflamed mucosa, retained secretions, and granulation tissue [
IP has the ability to destroy adjacent bone even in the absence of malignant transformation; therefore, an early, well-planned wide excision of the lesion, diseased mucosa, and the underlying mucoperiosteum [
In the last 25 years, treatment strategy for sinonasal IP has evolved due to advancements in endoscopic surgery. Traditionally, medial maxillectomies via lateral rhinotomy or midfacial degloving was considered gold standard [
A review of the literature by Lund et al. [
Woodworth et al. [
Tumour location is an important factor influencing the choice of approach [
A meta-analysis suggested that piecemeal resection of IP via an endoscopic approach did not compromise tumour control rates compared to en bloc external techniques. The authors advocate careful identification and complete removal of the site of attachment of the lesion [
Adjuvant treatment with radiotherapy could be considered for patients with early or multiple recurrence, advanced stage lesions, aggressive tumours, incompletely resected lesions, lesions with positive margins, and tumours associated with malignancy [
Several studies since the early 1980s have postulated the aetiological role of HPV in the development of IP. Syrjänen 2003 [
Topical interferon alpha-2b therapy was first used successfully in 2002 on a 10-year-old patient presenting with a recurrent lacrimal system papilloma, with no recurrence at 12 months [
Long-term follow-up for IP is essential. Most recurrences are seen within 9 months but can occur many years later. An analysis of 3058 cases by Mirza et al. [
The case presented demonstrates the variability of location within the sinonasal tract that IP can develop, and the individuality of each case necessitates tailored operative techniques for complete excision whilst minimising recurrence rates. We also present a combined endoscopic approach for the en bloc resection of a NLD IP with no clinical recurrence at 15-month follow-up.
Written consent obtained from the patient is available upon request.
This paper was presented at the Association of Otolaryngologists in Training Conference, Birmingham, 14 May 2015.
The views expressed in the submitted paper are the authors’ own and not an official position of the institution.
The authors declare no conflict of interests.
The authors would like to acknowledge Dr. Anurag Joshi from the Department of Cellular Pathology in the University Hospital of Wales for arranging additional tests on histology specimens and photomicrographs for publication.