Volume 3, Issue 3 , Pages 124-127, September 2008
An inverted papilloma isolated to the sphenoid sinus in a pediatric patient
Article Outline
Summary
A sinonasal inverted papilloma is a benign tumor which has been reported up to 4%. However, an inverted papilloma originating in the sphenoid sinus is even rarer, and a pediatric case has never before been reported. We report a case of pediatric inverted papilloma originating in the sphenoid sinus that presented with symptoms of sinusitis. Endoscopic sinus surgery was performed using a computer-aided image-guided system. The tumor was removed without any surgical complications or recurrence for 2 years.
Keywords: Papilloma, Sphenoid sinus, Young, Sinusitis
Introduction
An inverted papilloma (IP) is a rare benign tumor that constitutes only 0.5–4% of all nasal neoplasms [1]. An IP most commonly arises from the lateral nasal wall; the sphenoid sinus, however, is rarely the primary site of involvement, and pediatric patients have a very low incidence of IPs originating from the sphenoid sinus [2], [3]. In this report, we briefly describe the first case of an isolated IP originating from the sphenoid sinus in a pediatric patient presenting with sinusitis. This report received approval from the Institutional Review Board of the Yonsei University College of Medicine and informed consent for publication was obtained from the patients’ parents.
Case report
A 15-year-old boy presented with a 5-year history of nasal obstruction. He had also been suffering from purulent nasal discharge and post-nasal drip. The patient had been treated repeatedly with oral antibiotics at local clinics but did not show any signs of improvement. Anterior rhinoscopy and endoscopic examination revealed bilateral hypertrophy of the inferior turbinates and accumulation of purulent secretions in the middle meatus. The sphenoethmoidal recess and superior meatus did not show any abnormal lesions. Plain paranasal radiography showed haziness in both maxillary sinuses, and a paranasal computed tomography (CT) was therefore ordered. The CT scan demonstrated near-total opacification of both maxillary sinuses and the left sphenoid sinus (Fig. 1). Based on the patient's age, presenting symptoms, and radiographic findings, the initial diagnosis was chronic sinusitis of both maxillary sinuses and the left sphenoid sinus. The patient underwent conventional endoscopic sinus surgery, which consisted of a bilateral middle meatal antrostomy and a left sphenoidotomy. Purulent discharge was drained from both maxillary sinuses and the left sphenoid sinus, in which a polypoid mass was observed. We assumed that it was only a mucosal polypoid change and removed it. However, postoperative histopathologic results reported that the mass was an IP (Fig. 2). Because of these findings, revision endoscopic surgery was performed 1 month later. The second operation was performed with computer-aided image-guided endoscopic sinus surgery (StealthStation® TREON™ navigation system) to avoid injury to surrounding vital structures (Fig. 3). The lower half of the superior turbinate was resected so that a wide opening could be created in the anterior wall of the sphenoid sinus. The anterior wall was then widened to the lateral wall and lengthened inferiorly to the level of the sphenoid sinus floor. Openings were made in these areas because the remnant tumor was located deeply at the pneumatized inferolateral portion of the sphenoid sinus, with attachment to the floor and lateral wall of the sinus. The entirely visible tumor could be resected safely with the adjacent normal mucosa and intersinus septum using the navigation system. The histopathological result of the second surgery was also reported as an inverted papilloma without malignancy. A 2-year endoscopic follow-up has shown no recurrences (Fig. 4).
Fig. 1.
A preoperative coronal (A and B) and axial (C) computed tomography scan demonstrates near-total opacification of both maxillary sinuses and the left sphenoid sinus.
Fig. 2.
Histological appearance demonstrating an inverted papilloma with an endophytic or inverted growth pattern. A thickened squamous epithelium grows downward into the myxoid stroma (hematoxylin and eosin; 100×).
Fig. 3.
Intraoperative imaging from the computer-aided image-guided navigation system shows the most superior and posterior site of the left sphenoid sinus.
Fig. 4.
A postoperative endoscopic view shows no recurrence of the inverted papilloma in the sphenoid sinus. Asterisk (*) indicates a part of the intersinus septum that has been resected: FL: floor; OC: optic canal.
Discussion
A sinonasal IP is a benign neoplasm usually observed in adults (fifth to seventh decade) but rarely originating from the sphenoid sinus [1], [2], [3]. The most common presenting symptom of a sinonasal IP is unilateral nasal obstruction and epistaxis. However, patients with an IP confined to the sphenoid sinus often present with obscure, nonspecific symptoms [4]. Early disease usually manifests with headache or retro-orbital pain, and subsequent extension to the sinus walls compromises cranial nerves, resulting in neurologic symptoms [2], [4]. In this case, the patient did not have headaches but did have sinusitis symptoms such as nasal obstruction and chronic rhinorrhea. This made it difficult for early detection of the IP in the sphenoid sinus. Although paranasal CT imaging showed opacification in the sphenoid sinus, this observation was not sufficient to warrant suspicion of a tumor, especially since no erosion of the bony wall or intratumoral calcification was seen. In addition, CT findings of the near-total opacification of both maxillary sinuses and the young age of the patient led us to believe that the lesion of the sphenoid sinus was a polyp or mucocele. The characteristic radiographic finding of an IP is a unilateral polypoid lesion occupying the lateral nasal wall or paranasal sinuses along with internal hyperdensity or intratumoral calcification [5], [6]. Seventy percent of cases reportedly show bony erosion on CT imaging [7]. However, one study showed that 66% of patients diagnosed with inflammatory nasal polyps who had undergone traditional sinus surgery needed revision surgery for a recurrent or residual IP [8]. Sinonasal soft-tissue lesions, which have been observed in the pediatric population, include nasal polyps, hemangiomas, choanal polyps (antrochoanal and sphenochoanal polyps), angiofibromas, encephaloceles, mucoceles, and IPs [9]. In this case, at the time of operation, a nasal polyp and an IP could not be differentiated according to gross appearance. If the preoperative radiological workup had prompted suspicion for an IP, frozen specimens might have been obtained for intraoperative identification of the IP.
An IP is a unique benign neoplasm characterized by a high-recurrence rate, an association with malignancy, and an ability to destroy bone [1]. These biological characteristics of IPs have been observed in pediatric patients, who receive treatment similar to that for adults with IPs [3]. Moreover, an IP in childhood can exhibit disturbing characteristics such as a high-recurrence rate and associated malignancies [10]. Several early reports used an external approach for wide surgical excision; however, recurrence rates reportedly range from 14% to 71% depending on the type of operation [11], [12]. A suitable surgical method for an IP originating from the sphenoid sinus has not yet been established. Surgical management of the sphenoid sinus entails dangerous complications because of its anatomical proximity to vital structures such as the optic nerve, internal carotid artery, trigeminal nerve, and cavernous sinus. Recently, computer-aided image-guided endoscopic sinus surgery has been used for atypical sphenoid diseases because of the surrounding vital neurovascular structures [13]. In this case, because of the patient's young age, surgical morbidity, and tumor location, computer-aided image-guided endoscopic sinus surgery was an optimal treatment choice. The system provided us with real-time images of complex, three-dimensional anatomical landmarks. The high accuracy of localization facilitated a safer surgery, with a favorable postoperative outcome for 2 years without any recurrences.
Conclusion
The symptoms and CT findings of an IP confined to the sphenoid sinus can be very obscure; however, an IP should always be included in the differential diagnosis of a sinonasal mass, even in pediatric populations. Endoscopic treatment of an isolated IP arising from the sphenoid sinus can be a safe and beneficial method when performed with the computer-aided image-guided system. Any tissues excised during surgery should be carefully analyzed for accurate histopathological confirmation.
Acknowledgements
This study was supported by a grant of the Korea Health 21 R&D Project, Ministry of Health & Welfare, Republic of Korea (A06-0561-AA1018-06N1-00010A).
References
- . Management of inverted papilloma. Laryngoscope. 1990;100:481–490
- . Inverting papilloma of the sphenoid sinus. Otolaryngol. Head Neck Surg. 1981;89:710–712
- . Inverted papilloma of the nose and paranasal sinuses in children. Ear Nose Throat J. 1992;71:264–266
- . Isolated sphenoid lesions: diagnosis and management. Otolaryngol. Head Neck Surg. 1999;120:730–736
- . Schneiderian papilloma. J. La. State Med. Soc. 1997;149:310–315
- . Optimum imaging for inverted papilloma. J. Laryngol. Otol. 2000;114:891–893
- . Inverted papilloma of the nasal vault and paranasal sinuses: spectrum of CT findings. Am. J. Roentgenol. 1994;162:419–423
- . Radiographic assessment of inverted papilloma. Acta Otolaryngol. 2007;127:515–520
- . Recurrent inverted papilloma of a pediatric patient: clinico-radiological considerations. Int. J. Pediatr. Otorhinolaryngol. 2005;69:861–864
- . Inverted papilloma of the nose and paranasal sinuses in childhood and adolescence. Laryngoscope. 1985;95:17–23
- . The clinical behavior of inverting papilloma of the nose and paranasal sinuses: report of 112 cases and review of the literature. Laryngoscope. 1990;100:463–469
- . Inverting papilloma of the head and neck: the UCLA update. Otolaryngol. Head Neck Surg. 1993;109:71–76
- . Computer-aided image-guided endoscopic sinus surgery in unusual cases of sphenoid disease. Rhinology. 2002;40:179–184
PII: S1871-4048(08)00005-1
doi:10.1016/j.pedex.2008.01.003
© 2008 Elsevier Ireland Ltd. All rights reserved.
Volume 3, Issue 3 , Pages 124-127, September 2008
