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Clinical practice

Schneiderian papilloma occasionally found in the lacrimal sac

DONG, Xu-ran; LIU, Cong; WANG, Wei; TIAN, Yan-jie

Editor(s): WANG, De

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doi: 10.3760/cma.j.issn.0366-6999.2012.16.031
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Schneiderian papilloma is a kind of benign tumor that generally arises in the nasal and paranasal sinus mucosa. Here we present a case of Schneiderian papilloma found in the lacrimal sac.

A 38-year-old woman with half-a-year history of right eye keratoleukoma applied to the clinic. Examinations found a central corneal ulcer, and penetrating keratoplasty was scheduled as a means of relieving vision problems in the right eye. However, penetrating keratoplasty was delayed because of a purulent discharge effusing from the right inferior punctum during irrigation of the lacrimal passages.

Focusing on the patient's past history, we found that she had undergone a right nasal polypectomy 13 years ago due to continuous rhinobyon. Having been relieved of rhinobyon, the woman began to complain of epiphora and a slowly enlarging swelling on the right side of her nose a few months after surgery. The swelling would compress when it was pressed and a purulent discharge effused from the right inferior punctum. The symptom persisted for the past 13 years without significant changes.

Based upon this aspect, an external dacryocystorhinostomy was planned before preceeding with penetrating keratoplasty. During dacryocystorhinostomy, the lacrimal sac was found to be half filled with pale multilobulated papillary tissue. A piece of tissue was removed from the lacrimal sac and was sent for histological examination. Dacryocystectomy and lacrimal duct construction with intubation of the canaliculi was performed. Histological examination of the specimen suggested an inverted Schneiderian papilloma with regions of mild atypical hyperplasia of epithelial cells (Figure 1). No evidence of invasive carcinoma was seen.

Figure 1.
Figure 1.:
Histological examination of the specimen. A: Inverted papilloma, low magnification. Histology revealed multiple finger-like inverted papillomatous fragments of tissue covered by a variable layer of non-keratinizing (Schneiderian) transitional and squamous cell epithelium. The epithelial islands are well demarcated from the stroma (HE staining, Original magnification ×20). B: Microscopic finding shows a single focus of inversion. The tissue is endophytic with multilayer epithelium inverting into the underlying vascularized stroma. Inflammatory cells, especially neutrophils can be found in the stroma. Basement membrane is thin and delicate (HE staining, Original magnification ×200).

In order to evaluate the extent of the disease, MRI was ordered for the patient post discharge, after the penetrating keratoplasty. Enhanced T1 signals showed unilateral opacification of the maxillary or ethmoid sinus and a mass in the nasal fossa (Figure 2). At the time of MRI the woman had no complaints of rhinobyon, epiphora or purulent discharge.

Figure 2.
Figure 2.:
Enhanced T1 signals showing unilateral opacification of the maxillary or ethmoid sinus and a mass in the nasal fossa. A: coronal plane. B: sagital plane.

Tumors of the lacrimal sac are relatively rare. The epithelial lining of the lacrimal sac is the same as the rest of the upper respiratory tract. Tumors, therefore, are similar to those found elsewhere in the upper respiratory system.

Inverted papilloma, a type of Schneiderian papilloma, is a type of benign tumor that generally arises in the nasal and paranasal sinus mucosa, characterized by a potentially invasive nature and relatively high recurrence rates.1,2 It has been suggested that ectopic migration of the Schneiderian membrane during embryogenesis could account for the aberrant papilloma in sites contiguous with the sinonasal tract.3 Recurrence rates quoted in the literature can be as low as 2%4 to as high as 80%.5 This depends mostly on the extent of the original disease, the completeness of the resection, the tumor location and the biological variability of the tumor.6 The malignant transformation rate ranges from 6% to 14.5%.7,8 CT imaging or MRI can determine the extent of the disease preoperatively for proper planning of treatment. More recently, MRI has been used to aid in the assessment and guide surgery as it can accurately distinguish between secretions and the soft tissue of a tumor.6,9

Complete surgical removal is the first option for the treatment of inverted papilloma. A variety of surgical retained techniques have been developed in otolaryngology. However, methods of managing the neoplasm in the lacrimal sac are still controversial. Dacryocystectomy, with complete surgical excision of the tumor, is the preferred treatment.10 But an adjunct therapy is still to be developed. Parmar and Rose reported using dacryocystorhinostomy, then further surgical excision and an argon laser for rhinostomy after the recurrence of a papilloma.11 In 53 months of follow-up there was no further recurrence. In another case, interferon alpha-2b had been successfully used.12

Recently, Woodcock et al13 presented a case with recurrent Schneiderian papilloma of the nasolacrimal sac, which underwent an external dacryocystorhinostomy with irrigation of the nasolacrimal system with 0.02% mitomycin C. Since mitomycin C has been used effectively in the treatment of superficial bladder cancer and inverting papillomas of the urinary bladder,14 they applied this method as a way of reducing the risk of recurrence. In the 18 months of follow-up, no recurrence was found.

As mentioned before, the majority of these tumors arise from the nasal and paranasal sinus mucosa. In our case, the patient's MRI which was taken after dacryocystectomy now shows unilateral opacification of the maxillary or ethmoid sinus. This highlights both the high recurrence rate and the need for suspicion of inverted papilloma when a patient shows signs of chronic dacryocystitis and has a history of unilateral nasal obstruction. Usually, tumor in the lacrimal sac is due to extension from the site of the primary tumor. However in our case, since the histology of the mass causing the patient's rhinobyon a decade ago is uncertain, we cannot exclude the possibility of the lacrimal sac as a primary site of the tumor. A long-term follow-up will be required. Although histologically benign, inverted papilloma has an unlimited growth potential and, if neglected, can cause considerable morbidity or even death by extending into contiguous structures.15

REFERENCES

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Keywords:

lacrimal duct obstruction; dacryocystitis; dacryocystectomy; inverted papilloma; Schneiderian papilloma

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