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Mas Takashima, MDBaylor College of MedicineEmail: takashim@bcm.eduPhone: 713-798-7217Website: www.bcm.edu/oto

Objectives1) Understand the relationship of the opticocarotid recess to the surrounding structures. 2) Explain the pathophysiology and natural history of an inverting papilloma (IP) in the sphenoid sinus. 3) Examine the significance of a neoplasmoriginating from the anterior clinoid.

MethodsThis case report was conducted in a tertiary hospital setting in 6/2011. The subject, a 49 year-old female, presented with an inverting papilloma originating from the opticocarotid recess causing optic nerve dehiscence. The intervention wasendoscopic excision. Outcome was measured by clinical examination. Follow-up patient analysis is ongoing.

ResultsThe patient was found to have an inverting papilloma (IP) of the sphenoid sinus. During endoscopic sinus surgery, the inverting papilloma was visualized originating from the sphenoid sinus and causing bony erosion in the area of theopticocarotid recess. An area of optic nerve dehiscence was identified, and the sheath of the optic nerve was clearly visualized once the tumor was peeled off the optic canal. Pathology confirmed the diagnosis of inverting papilloma and was negative for carcinoma. Follow-up clinical examination of the patient revealed that endoscopic excision effectively removed the neoplasm without local complications or recurrence.

ConclusionInverting papillomas originating from the region of the opticocarotid recess are an atypical entity, and have the potential to cause rare, but serious complications. The authors are unaware of any reported cases of an IP originating from this area.Early, effective management is crucial given the vital structures at risk.

Inverted Papilloma Originating from the Opticocarotid RecessRamya Srinivasan, MD1,2; Elyse Portillo, MPH1,2; Mas Takashima, MD1,2

1Baylor College of Medicine, 2The Texas Medical Center

A 49-year-old female with an 80-pack-year smoking history was referred from an outside facility with a computed tomography (CT) scan demonstrating a mass in the right sphenoid sinus. The patient’s history was significant for a previous resection of an IP in the right sphenoid sinus several years before, and her new CT scan was suspicious for recurrence of the disease. The patient’s only main complaint was retrobulbar pain on the right. Rigid nasal endoscopy revealed right septal deviation with a papillomatous mass protruding from the right sphenoid sinus. A repeat CT scan of the sinuses without contrast revealed almost complete opacification of the right sphenoid sinus, an area of dehiscence along the right optic nerve, and a thin wall of bone separating the sphenoid sinus from the adjacent internal carotidartery (ICA) [images 3-5].The mucosa of the skull base was taken down using a suction curette, and the tumor was carefully peeled off the walls of thesphenoid sinus. The area of optic nerve dehiscence was identified in the area of the opticocarotid recess. Tumor was seen invaginating into the opticocarotid recess, causing dehiscence of the bony wall in this region. The sheath of the optic nerve was clearly visualized once the tumor was peeled off the optic canal and optic sheath with a suction curette [image 2]. Gross pathology of the specimen showed tan-pink tissue that was minimally fibrous. Microscopic sections showed deeply invaginated nests of ciliated epithelium with underlying loose stroma [image 6]. The final pathology report confirmed the diagnosis of inverted papilloma and was negative for carcinoma. The patient was seen in clinic after discharge from the hospital. At her last visit, which was two months after surgery, the right optic nerve was intact and no complications were noted. In addition, rigid nasal endoscopy did not reveal a recurrence of the tumor.

We present this case to emphasize the need for rapid identification and management of inverting papillomas of the sphenoid sinus, especially in light of the potential for opticocarotid recess dehiscence, which has not been previously described in the literature. Early management is crucial given the challenging anatomic location of the sphenoid sinus and the vital structures at risk.

Inverting papillomas (IPs) are rare, benign sinonasal neoplasms that tend to be locally aggressive. Histologically, the Schneiderian respiratory epithelium inverts and proliferatesinto the underlying stroma, while the basement membrane remains intact, to create an inverting papilloma (IP). Most IPs arise from the lateral nasal wall in the area of the middle meatus, and involve the paranasal sinuses by extension1,10. Unfortunately, IPs have a high rate of recurrence after treatment, which is most often due to incomplete resection of the tumor. In addition, there is an association with malignancy in 5-15% of cases of IPs—most commonly squamous cell carcinoma2,4,8. An isolated IP in the sphenoid sinus eroding into the opticocarotid recess with optic nerve dehiscience is an exceedingly rare entity, and to the authors’ knowledge, the only one reported in the literature.

INTRODUCTION

1. Eisen MD, Buchmann L, Litman RS, Kennedy DW. Inverted papilloma of the sphenoid sinus presenting with auditory symptoms: a report of two cases. Laryngoscope. 2002 Jul;112:1197-200.

2. Fakhri S, Citardi MJ, Wolfe S, Batra PS, Prayson RA, Lanza DC. Challenges in the management of sphenoid inverted papilloma. Am J Rhinol. 2005 Mar-Apr;19(2):207-13.

3. Kosugi EM, Santos Rde P, Ganança FF, Tangerina Rde P, Suguri VM, Yamaoka WY, Gregório LC. Inverted papilloma in the sphenoethmoidal recess. Braz J Otorhinolaryngol. 2008 Jan-Feb;74(1):151-4.

4. Lee JT, Bhuta S, Lufkin R, Castro DJ. Isolated inverting papilloma of the sphenoid sinus. Laryngoscope. 2003 Jan;113(1):41-4.

5. Liu SC, Lee JC, Chen JJ, Lin YS. Isolated inverted papilloma of the sphenoid sinus. J Chin Med Assoc. 2010 Sep;73(9):503-5.

6. Ozcan T, Yilmazlar S, Aker S, Korfali E. Surgical limits in transnasal approach to opticocarotid region and planum sphenoidale: an anatomic cadaveric study. World Neurosurg. 2010 Apr;73(4):326-33.

7. Peters BW, O'Reilly RC, Willcox TO Jr, Rao VM, Lowry LD, Keane WM. Inverted papilloma isolated to the sphenoid sinus. Otolaryngol Head Neck Surg. 1995 Dec;113(6):771-7.

8. Wong KK, Fenton RS. Endoscopic resection of isolated inverted papilloma of the sphenoid sinus. J Otolaryngol. 2004 Apr;33(2):125-8.

9. Yiotakis I, Gkoritsa E, Manolopoulos L, Kandiloros D, Korres S, Ferekidis E. Inverted papilloma of the sphenoid sinus: presentation of three cases. Rhinology. 2006 Jun;44(2):164-8.

10.Yiotakis I, Psarommatis I, Manolopoulos L, Ferekidis E, Adamopoulos G. Isolated inverted papilloma of the sphenoid sinus. J Laryngol Otol. 2001 Mar;115(3):227-30

11.Yiotakis J, Hantzakos A, Kandiloros D, Ferekidis E. A rare location of bilateral inverted papilloma of the nose and paranasal sinuses. Rhinology. 2002 Dec;40(4):220-2.

CONCLUSIONS

CASE STUDY

REFERENCES

Figure 4. Pre-operative CT scan (coronal view). Arrow points to tumor, circle shows area of

thinning of the bony wall.

Figure 2. Intra-operative endoscopic view of the opticocarotid recess after tumor resection. Optic nerve sheath is visible.

Figure 1. Pre-operative endoscopic view of poly-poid like mass. Figure 3. Pre-operative CT scan (axial view). The tumor occupies the right sphenoid sinus.

ABSTRACT

CONTACT

Figure 6. Histology of IP. Shows deeply invaginated nests of ciliated epithelium with underlying loose stroma.

Figure 5. Pre-operative CT scan (sagittal view). Arrow points to tumor in sphenoid sinus.

DISCUSSIONAn inverting papilloma (IP) is a rare, benign sinonasal tumor that tends to originate in the lateral nasal wall, in the area of the middle meatus. It usually involves the paranasal sinuses only by extension. Primary inverting papillomas (IPs) in the sinuses are rare, and they tend to occur in the maxillary sinus. A primary IP in the sphenoid sinus is exceedingly rare8. IPs are known for their unusual biological behavior, which makes management of the lesion difficult. They have the capacity to invade local anatomical structures and erode adjacent bony structures by pressure necrosis1,7. Of note, bony erosion caused by an IP of the sphenoid sinus can lead to the dehiscence of important structures, such as the internal carotid artery (ICA). Histologically, they are the result of a benign, downward endophytic growth of the Schneiderian respiratory epithelium. The basement membrane remains intact, which allows the subepithelial crypts to remain connected to the epithelial surface7. Although they are often regarded as a benign neoplasm, Fakhri et al (2005) present an alternate view that an IP is actually an inflammatory polyp with squamous metaplasia2. IPs of the lateral nasal wall tend to present with unilateral nasal obstruction, nasal discharge, epistaxis, hyposmia, facial pain, and pressure sensation7,8,10. An isolated lesion of the sphenoid sinus has absence of lateral nasal wall symptoms, and therefore, the clinical symptoms tend to be more non-specific. Diagnosing the lesion requires a high index of suspicion. The few symptoms that may be seen include headache,

pressure sensation, retro-orbital pain, visual deficits, and rarely, auditory symptoms such as tinnitus1,4,9. These non-specific symptoms can lead to an excessive delay in diagnosis, and worse prognosis for the patient. IPs of the sphenoid sinus are not only challenging cases to diagnose, but also to treat, because of the potential complications of surgery. There are many critical structures at risk, including the optic nerve, ICA, pituitary gland, orbit, and intracranial fossa. A few cases have been presented in the literature in which an IP led to dehiscence of the bony walls of the sphenoid sinus. Fakhri et al (2005) presented two cases in which an inverting papilloma led to dehiscence of the ICA; one of the cases also had absence of bony margins along the posterior, lateral, inferior, and superior boundaries of the sphenoid sinus2. Lee at al (2003) presented a patient who had dehiscence of the bony margins of the sphenoid sinus both inferiorly and laterally, but an intact base of skull4. In our patient, the CT scan revealed thinning of the wall separating the sphenoid sinus from the ICA thereby placing the artery at increased risk during surgery [image 4]. In addition, the tumor caused dehiscence of the bony wall in the opticocarotid recess, and optic nerve sheath was visualized once the tumor was peeled off [image 2]. At this time, there are no other cases in the literature describing involvement of the opticocarotid recess. The opticocarotid region is significant in sphenoid sinus surgery because it is where the ICA and the optic nerve are

closest to each other. The opticocarotid recess, which is caused by pneumatization of bone outside the sphenoid sinus, places the sphenoid sinus in very close proximity to both the ICA and the optic nerve6. Due to the paucity of cases reported in the literature, there is currently a lack of consensus on the appropriate treatment for this type of lesion. The locally aggressive nature of the lesion, and the potential for malignant transformation, make early and definitive surgical treatment crucial8. The current controversy lies in whether endoscopic versus external open approach is the best treatment. Endoscopic surgical management has several advantages, including avoidance of surgical scars, decreased post-operative pain, minimal blood loss, and shorter post-operative hospitalization10. In addition, it offers the advantage of direct visualization of the tumor, and possibly a more complete resection of the tumor. However, the surgeon must be prepared to change to an open approach if the tumor is extensive or bleeding complications obscure the view of the surgical field. Complete excision with negative margins is important due to the high recurrence rate associated with incomplete resection of the lesion. The recurrence rate of inverted papillomas after endoscopic surgery has been shown to be similar to the recurrence rate after an open procedure2. Our patient had a recurrence of her inverting sinus papilloma after previous endoscopic surgery at an outside hospital. We strived to avoid this complication by meticulously removing visible tumor and ensuring good margins.