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Pleomorphic adenoma of the nasal cavity: a case report.

Abstract

Pleomorphic pleomorphic adjective Referring to a variable appearance or morphology  adenomas of the nasal cavity are rare. We describe the case of a middle-aged white man who presented to our clinic with a long history of unilateral nasal obstruction. Preoperative imaging was performed, and the patient was taken to the operating room for endoscopic excision of the lesion. Surgical margins were negative. The patient's nasal obstruction resolved, and he was doing well at his first postoperative visit. The pathologic diagnosis was a pleomorphic adenoma. We discuss the features and management of this entity.

Introduction

Pleomorphic adenoma is the most common benign neoplasm of the major salivary glands, accounting for approximately 65% of all salivary gland neoplasms. (1) In the salivary glands, 80% of all cases of pleomorphic adenoma occur in the parotid gland, and 6.5 to 8% of cases occur in the minor salivary glands. (2,3) Pleomorphic adenomas of the upper respiratory tract are very rare in the literature. When they have occurred, the most common site of origin is the nasal cavity; other cases have been documented in the maxillary sinus, nasopharynx, larynx, and trachea. (3,4) We report a new case of a pleomorphic adenoma of the nasal cavity.

Case report

A 44-year-old white man presented to the otolaryngology clinic with a complaint of a unilateral (left-sided) nasal obstruction that had slowly worsened over the previous 5 years. He denied associated pain, epiphora epiphora /epiph·o·ra/ (e-pif´or-ah) [Gr.] overflow of tears due to obstruction of lacrimal duct.

e·piph·o·ra
n.
, epistaxis, rhinorrhea, and episodes of sinusitis. He also denied occupational exposures to wood and heavy metals. He reported no fevers, chills, night sweats, lymphadenopathy, or recent change in weight. His medical history was significant for obstructive sleep apnea,hypertension, and the removal of a bone cyst from his right humerus as a teenager. He was a nonsmoker and drank socially.

On examination, anterior rhinoscopy revealed that the septum septum /sep·tum/ (sep´tum) pl. sep´ta   [L.] a dividing wall or partition.

alveolar septum  interalveolar s.
 was mildly deviated to the right. On the left, the inferior turbinate extended from the floor of the nose to the nasal roof. Neither rigid nor flexible endoscopy on the left was possible in the office because of the enlargement of the inferior turbinate.The tympanic membranes were normal, and findings on examination of the oral cavity, oropharynx, and neck were within normal limits.

Computed tomography (CT) detected a soft-tissue mass with calcification that was obscuring the left nasal cavity and causing osseous osseous /os·se·ous/ (os´e-us) of the nature or quality of bone; bony.

os·se·ous
adj.
Composed of, containing, or resembling bone; bony.
 displacement, but no bony destruction or invasion was evident (figure 1). Magnetic resonance imaging magnetic resonance imaging (MRI), noninvasive diagnostic technique that uses nuclear magnetic resonance to produce cross-sectional images of organs and other internal body structures.  (MRI) demonstrated a heterogeneous 3-cm mass arising from the inferior turbinate; again, no destruction or invasion was evident (figure 2).

The patient was counseled about endoscopic removal of the mass and a possible lateral rhinotomy or medial maxillectomy. Intraoperatively, the mass was found to be confined to the anteroinferior turbinate. It was removed without complication intranasally with the endoscope. Negative margins after wide local excision were confirmed by frozen-section histopathology. Postoperatively, the nasal obstruction resolved and the patient did well. Pathology identified the mass as a pleomorphic adenoma.

Discussion

Approximately 80% of intranasal pleomorphic adenomas are found in the septum. (3) Only about 20% are found in the lateral wall or turbinate, (3) despite the fact that there is a significantly higher proportion of mucous and serous glands located on the lateral nasal wall. (5) Several theories have been proposed to explain why there is a higher incidence ofpleomorphic adenomas in the nasal septum than in the gland-rich tissue of the lateral nasal wall, but none has been proven.

[FIGURE 1 OMITTED]

The differential diagnosis of an intranasal mass is broad; it includes inverting papilloma, benign nasal polyp, squamous cell carcinoma squamous cell carcinoma
n.
A carcinoma that arises from squamous epithelium and is the most common form of skin cancer. Also called cancroid, epidermoid carcinoma.
, cartilaginous cartilaginous /car·ti·lag·i·nous/ (kahr?ti-laj´i-nus) consisting of or of the nature of cartilage.

car·ti·lag·i·nous
adj.
1. Chondral.

2.
 tumors, adenocarcinoma, melanoma, chondroma chondroma /chon·dro·ma/ (kon-dro´mah) pl. chondromas, chondro´mata   a benign tumor or tumor-like growth of mature hyaline cartilage. , encephalocele, and adenoid cystic carcinoma adenoid cystic carcinoma
n.
A carcinoma characterized by large epithelial masses containing round glandlike spaces or cysts, frequently containing mucus, that are bordered by layers of epithelial cells. Also called cylindromatous carcinoma.
. (5-9) Most intranasal pleomorphic adenomas arise in whites from the third to sixth decades of life; there is a slight female preponderance. (1) The most common presenting symptom (75% of cases) is unilateral nasal obstruction. (5) Less often, the chief complaint is epistaxis, sinusitis, or a mass within the nasal cavity. (1,5) Sinusitis can occur as a result of either (1) an extension of a primary neoplasm into the maxillary sinus or (2) obstruction of the osteomeatal complex by the tumor. Occasionally, pleomorphic adenomas arising from the nasopharynx obstruct the eustachian tubes and cause symptoms of eustachian tube dysfunction. (10) Generally, affected patients present within 1 year of the onset of symptoms. (5)

[FIGURE 2 OMITTED]

Most intranasal pleomorphic adenomas range in size from 0.5 to 7 cm, and most have a broad base (a few have a pedicled base). (5) These neoplasms have been described as exophytic, polypoid, oval, dome-shaped, firm, and gray. (5) Grossly, these masses have a polypoid, smooth, lobulated appearance with a well-defined capsule. (1) No destruction of surrounding tissue is seen.

Histologically, pleomorphic adenomas of the nasal cavity are not identical to those found in the salivary glands. In the nasal cavity, these neoplasms have greater cellularity and they contain more epithelial components than the chondroid, myxoid myxoid /myx·oid/ (mik´soid) mucoid.

myx·oid
adj.
Containing or resembling mucus; mucoid.



myxoid

resembling mucus.

myxoid adjective 1.
, and collagenous stromal components that are seen in pleomorphic adenomas of the parotid gland. (5) These small, oval epithelial cells are densely packed "back to back" in masses or cords, a pattern that is associated with benign neoplasms. (5) There is usually minimal or no evidence of mitotic activity.

Malignant transformation of pleomorphic adenoma of the nasal cavity has been reported in 2.4 to 10% of cases. (10) The most common variant is carcinoma ex pleomorphic adenoma carcinoma ex pleomorphic adenoma
n.
A carcinoma developing in a benign mixed tumor of a salivary gland and characterized by rapid growth and pain.
, which is capable of metastasis. A less common form of malignant transformation is the malignant mixed form, in which both the epithelial and stromal components transform and metastasize together. Bone is the most common site of metastasis, followed by the liver, lung, and regional lymph nodes.

The radiographic findings with pleomorphic adenomas of the nasal cavity are nonspecific, much like the clinical presentation. CT typically shows a well-defined soft-tissue mass; calcification is seen in rare cases. CT allows the clinician to assess bony involvement or destruction, which may occur when a neoplasm has been left untreated over time. (1) MRI is used to assess the epithelial and stromal components of the neoplasm, as well as the surrounding soft tissue. MRI of the stromal components reveals a low signal intensity on T1-weighted imaging and an intermediate to high signal intensity on T2-weighted imaging; epithelial components have a low signal intensity on T2-weighted imaging.

Treatment entails wide local excision with clear margins. (5) If these neoplasms are allowed to progress untreated, their expansion may cause local destruction of the paranasal sinuses. Four approaches to the removal of intranasal pleomorphic adenomas have been reported in the literature: a transpalatal approach, a lateral rhinotomy, a Le Fort I osteotomy osteotomy /os·te·ot·o·my/ (os?te-ot´ah-me) incision or transection of a bone.

cuneiform osteotomy  removal of a wedge of bone.
, and an endoscopic intranasal approach. (1,11) The transpalatal approach provides good exposure of the posterior nasal cavity and nasopharynx, but considerable palatal pal·a·tal
adj.
Palatine.


palatal (pal´t
 dysfunction from soft-palate retraction is a known complication. (1) The lateral rhinotomy, which requires an external approach, is typically not favored for this benign neoplasm because it leaves a facial scar. A facial scar can be avoided by using the Le Fort I osteotomy, but this technique requires fixation of the alveolar ridge. The endoscopic intranasal approach allows for good visualization without external scarring and is therefore the preferred approach.

Postoperative follow-up is necessary for all patients with an intranasal pleomorphic adenoma because as many as 10% of patients may experience a recurrence despite wide local excision with clear margins. (5) Even so, this recurrence rate is much lower than the 50% recurrence rate seen in patients with pleomorphic adenoma of the parotid gland. (5) Histologically, recurrence is most associated with the myxoid stromal pattern. Freeman et al reported the discovery of a metastasis 17 years after the excision of a primary intranasal pleomorphic adenoma. (12)

In conclusion, intranasal pleomorphic adenoma is rare. The most common site of this benign neoplasm is the parotid gland. Compared with pleomorphic adenomas of the parotid, intranasal lesions are associated with greater cellularity (more epithelial components), a more benign course overall, and a lower rate of recurrence (10 vs. 50%). (5) Intranasal pleomorphic adenomas have no definitive clinical or radiographic findings. Despite the fact that intranasal pleomorphic adenomas have a nonspecific appearance on CT and MRI CT and MRI
Two high technology methods of creating images of internal organs. Computerized axial tomography (CT or CAT) uses x rays, while magnetic resonance imaging (MRI) uses magnet fields and radio-frequency signals. Both construct images using a computer.
, it is advantageous to use these two modalities to assess bony destruction and soft-tissue components, respectively. The treatment of choice is local excision with clear margins, usually via an intranasal approach. Recurrence and metastasis are uncommon, but patients need to be followed postoperatively nevertheless.

References

(1.) Clark M, Fatterpekar GM,Mukherji SK, Buenting J. CT of intranasal pleomorphic adenoma. Neuroradiology 1999;41(8):591-3.

(2.) Shaheen OH. Benign salivary gland tumors Salivary Gland Tumors Definition

A salivary gland tumor is an uncontrolled growth of cells that originates in one of the many saliva-producing glands in the mouth.
. In: Kerr AG, ed. Scott Brown's Otolaryngology. 6th ed. London: Butterworth-Heinemann; 1997:5/20/1-5/20/18.

(3.) Batsakis JG. Tumors of the major salivary gland major salivary gland
n.
Any of three salivary glands, the parotid gland, the submandibular gland, and the sublingual gland, which are the largest of the oral cavity and secrete the most saliva.
. In: Batsakis JG. Tumors of the Head and Neck: Clinical and Pathological Considerations. 2nd ed. Baltimore: Williams & Wilkins; 1979.

(4.) Mackle mack·le   also mac·ule
n.
A blurred or double impression in printing.

v. mack·led also mac·uled, mack·ling also mac·ul·ing, mack·les also mac·ules

v.tr.
 T, Zahirovic A, Walsh M. Pleomorphic adenoma of the nasal septum. Ann Otol Rhinol Laryngol 2004;113(3 Pt 1):210-11.

(5.) Compagno J, Wong RT. Intranasal mixed tumors (pleomorphic adenomas): A clinicopathologic study of 40 cases. Am J Clin Pathol 1977;68(2):213-18.

(6.) Ersner MS, Saltzman M.A mixed tumor of the nasal septum. Report of a case. Laryngoscope 1944;54:287-96.

(7.) Stevenson HN. Mixed tumor of the septum. Ann Otol Rhinol Laryngol 1932;41:563-70.

(8.) Evans RW, Cruickshank AN. Epithelial tumors of the salivary glands. In: Evans RW, Cruickshank AN. Major Problems in Pathology. Vol. 1. Philadelphia: W.B. Saunders; 1970.

(9.) Motoori K,Takano H, Nakano K, et al. Pleomorphic adenoma of the nasal septum: MR features. AJNR Am J Neuroradiol 2000;21(10): 1948-50.

(10.) Fushiki H, Morijiri M, Maruyama M, et al. MRI of intranasal pleomorphic adenoma. Acta Otolaryngol 2006;126(8):889-91.

(11.) Roh JL, lung BJ, Rha KS, Park CI. Endoscopic resection of pleomorphic adenoma arising in the nasopharynx. Acta Otolaryngol 2005;125(8):910-12.

(12.) Freeman SB, Kennedy KS, Parker GS, Tatum SA. Metastasizing pleomorphic adenoma of the nasal septum. Arch Otolaryngol Head Neck Surg 1990;116(11):1331-3.

Jason L. Acevedo, MD; Jennifer Nolan, MD; J. Kevin Markwell, MD, JD; David Thompson, MD, FACS

From the Otolaryngology/Head and Neck Surgery Service, Waiter Reed Army Medical Center, Washington, D.C.

Corresponding author: Jason L. Acevedo, MD, 6900 Georgia Ave. N.W., Washington, DC 20307. E-mail: Jasag00@yahoo.com

Disclaimer: The opinions expressed in this article are those of the authors and do not necessarily reflect those of the U.S. Army or the Department of Defense.
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Title Annotation:ORIGINAL ARTICLE
Author:Acevedo, Jason L.; Nolan, Jennifer; Markwell, J. Kevin; Thompson, David
Publication:Ear, Nose and Throat Journal
Article Type:Case study
Date:May 1, 2010
Words:1744
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