Inverted Papilloma Arising Primarily from the Lacrimal Sac

Tia Y.E. Raemdonck, MD, Caroline M. Van den Broecke, MD, Ilse Claerhout, PhD, MD, Christian E. Decock, MD Orbit, 28, 181184, 2009

Introduction

Inverted papilloma

benign but aggressive epithelial neoplasm due to malignant potential (a high recurrence rate) Most of cases arise from the nasal wall and paranasal sinuses

Introduction

Inverted papilloma of lacrimal sac

Secondarily involved through a process of local extension a. the tumor eroding through the orbital bones b. following the nasolacrimal duct

Case

24-year-old female Chief Complaint

a. b. epiphora of the right eye, without any purulent or sanguineous discharge firm, nontender swelling within the right lacrimal fossa (present for 6 months)

Past medical history

a. b. no episodes of daryocystitis General medical history was unremarkable

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Physical examination
a. b. c. d. VA : 20/20 Slit lamp examination, fundoscopy, ocular motility : normal A firm mass, not adherent to the overlying skin obstruction of the lacrimal duct, without any purulent discharge.

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Imaging work-up
a. Computed tomography a homogenous mass within the lacrimal fossa without contrast enhancement There was no bony invasion toward the nasal cavities or paranasal sinuses b. Dacryocystorhinostomy (To explore the mass)

a firm lacrimal sac

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Operation
Frozen section : Inverted papilloma dacryocystectomy under frozen section control of the margins ENT surgeon performed an endonasal scopy, which was unremarkable A Bowman probe was placed into the lacrimal duct and connected with a monopolar cautery destroy its epithelium in order to prevent local spread

Case

Macroscopic evaluation
a. protruding, well circumscribed mass

Pathological examination
a. b. c. d. e. superficial exophytic, papillary tumor basis had an inverted, lobular growth pattern The papillae were covered by a squamous, nonkeratinizing epithelium Goblet cell number decreased There was a sharp transition between the papilloma and the normal lacrimal

duct epithelium as the latter is thinner and contains more goblet cells
lesion was solely located within the lacrimal sac

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HPV
a. b. Koilocytes (typical for HPV infection) were not found immunohistochemistry was positive for p16 suggesting HPV infection c. d. Staining for high risk subtypes was negative an increased staining for MIB-1 low-grade atypia

No recurrence during a follow-up period of 24 months

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Discussion-papilloma

a benign tumor resulting from an overgrowth of epithelial tissue on papillae of vascularized connective tissue (as the skin and mucosa) three growth patterns
exophytic, inverted, mixed types Subdivided histologically into sqameus , transitional, mixed cell papillomas

Exophytic papilloma : affect the whole of the epithelium of the nasolacrimal system, particularly when they are of the transitional cell type Inverted papilloma : more prone to developing focally invasive carcinoma, usually of a lowgrade variety Mixed papillomas : mixed clinical and histopathologic features of exophytic and inverted types

Discussion -inverted papilloma

locally invasive, infiltrate and destroy adjacent bones c malignancy

suspected to carry a risk for malignant transformation

invade the orbit : association with a high incidence of malignancy

early, accurate and wide excision of the lesion is desired due to the tendency to invaginate the stroma more difficult to achieve complete resection augmenting its recurrence rate (most often found within the first 2 years after incomplete surgery)

Discussion complete excision

combined approach for complete excision of the lacrimal drainage apparatus 1. open excision of the superior part of the lacrimal drainage apparatus 2. direct manipulation of the nasolacrimal duct
(guided by nasal endoscopy)

adjunct treatment for recurrent, incompletely resectable and malignant tumors 1. endoscopic ablation with argon laser, radiotherapy or chemotherapy
could be considered in recurrent, incompletely resectable and malignant tumors

Discussion primary site

deep spreading and invading nature primary origin is often unclear. lacrimal sac was secondarily involved in several reports some reports indicated the lacrimal sac as primary site of origin all these cases had extensions beyond the lacrimal fossa into the nasal cavity, so the primary location remained unclear Some describing large series of epithelial tumours of the lacrimal sac clinicopathologic evidence that the primary location of inverted papillomas could be found within the lacrimal sac.