OtolaryngologyHead and Neck Surgery
Case Reports
renee rate with radiotherapy and a 60% recurrence rate
with conservative management. Radical surgery failed
to cure the disease in only 4.5% of cases." Results
from other studies are comparable."
The case we report is interesting from several viewpoints. The lack of prior dental history may lend support to the theory that these lesions arise from nonneoplastic or dentigerous cysts. Furthermore, the unique
presentation of unilateral nasal obstruction, together
with intermittent epistaxis, only occurs in previously
treated patients. Finally, the location of this normally
aggressive tumor in the maxilla with relatively little
bony destruction allowed for an en bloc resection that
did not violate adjacent anatomic fields and perhaps
diminished the possibility of local spread and recurrence.
Conservative local or radiation therapy should not be
considered when dealing with mandibular or maxillary
ameloblastomas. In accordance with Shatkin and
Hoffmeister," "Radical treatment conserves the patient's appearance, function, and life. "
CONCLUSION
A case of a maxillary ameloblastoma presenting as
unilateral nasal obstruction and epistaxis of long duration has been presented. The aggressive nature of this
lesion has been stressed and the necessity of extirpation
with adequate margins to obtain a cure has been emphasized.
REFERENCES
1. Sedhev MK, Huvos AG, Strong EW, Gerold FP, Willis GW:
Ameloblastoma of maxilla and mandible. Cancer 33:324-333,
1974.
2. Ivey RH, Churchill HR: The need of a standardized surgical and
pathological classification of tumors and anomalies of dental
origin. Am Assoc Dent Sch Trans 7:240-245, 1930.
3. International histological classification of tumors. Histological
typing of odontogenic tumours, jaw cysts and allied lesions
(Monograph No.5). Geneva, 1971, World Health Organization.
4. Bhaskar SN: Synopsis of oral pathology. St Louis, 1981, The
CV Mosby Co, pp 252-256.
5. Batsakis JB: Tumors of the head and neck. Baltimore, 1980, The
Williams & Wilkins Co, pp 531-536.
6. Hertz J: Adamantinoma: Histopathologic and prognostic studies.
Acta Chir Scand 102:405-432, 1952.
7. Hinds EC, Pleasants JE, Snyder PL: Management of ameloblastoma. Oral Surg 7:1169-1177,1954.
8. Godin RJ, Chaudry AP, Pinborg 11: Odontogenic tumors.
Cancer 14:73-101, 1961.
9. Frankel KA, Smith JD, Frankel LS: Soft tissue ameloblastoma in
a 92-year-old woman. Arch Otolaryngol 103:499-500, 1977.
10. Young DR, Robinson M: Ameloblastomas in children-report
of a case. Oral Surg 15:1155-1162, 1962.
11. Godin RJ, Meskin LH, Brodey R: Odontogenic tumors in man
and animals: Pathological classification and clinical behavior.
Ann NY Acad Sci 108:722-771, 1963.
12. Becker R, Pertl A: Zur Therapie des Ameloblastomas. Dtsch Z
Mund Kiferheilkd 49:423-436, 1967.
13. Mehlisch DR, Dahlin DC, Masson JK: Ameloblastoma: A
clinicopathologic report. J Oral Surg 30:9-22, 1972.
14. Shatkin S, Hoffmeister FS: Ameloblastoma-a rational approach to therapy. Oral Surg 16:1100-1101, 1963.
Pleomorphic adenoma of the nasal septum
BARRY L. WENIG. MD. JAMES J. SCIUBBA. DMD. PhD. ALFRED COHEN, MD, and ALLAN L.ABRAMSON, MO.
New Hyde Park, New York
From the Department of Otolaryngology and Communicative Disorders (Drs. Wenig, Cohen, and Abramson), and the Departments of
Dentistry and Laboratories (Dr. Sciubba), the Long Island Jewish-Hillside Medical Center.
Presented at the Annual Meeting of the American Academy of
Otolaryngology-Head and Neck Surgery (ScientificPoster), New
Orleans, La., Oct. 17-21, 1982.
Submitted for publication Oct. 16, 1982; revision received April 22,
1983; accepted May 3, 1983.
Reprint requests: Barry L. Wenig, MD, Department of Otolaryngology and Communicative Disorders, the Long Island JewishHillside Medical Center, 270-05 76 Ave., New Hyde Park, NY
11042.
432
Pleomorphic adenoma is the most common type of
benign epithelial salivary tumor. It most frequently presents in the major salivary glands. The appearance of
this tumor in the upper respiratory tract is highly unusual. The few scattered reports indicate that the nasal
cavity is the favored site of this tumor, followed distantly by the maxillary sinus and the nasopharynx.
We present a case of a young white man who developed progressive unilateral nasal obstruction. The
pathologic diagnosis was a benign mixed tumor, or
pleomorphic adenoma. The importance of this disease
Volume 93 Number 3
June 1985
Case Reports 433
Fig. 1. Areas of pseudocartilage separated from cords of cells exhibiting ductal differentiation by
mature collagenous stroma. (Hematoxylin-eosin stain; x72.)
entity lies in the necessity of making a correct diagnosis. Pleomorphic adenoma can easily be confused
with generally more aggressive destructive lesions of
the nasal septum. Treatment consists of conservative
surgical excision, which will often suffice as definitive
therapy.
CASE REPORT
A 21-year-old white man was referred because of a history
of unilateral left nasal obstruction. The patient noted that the
symptoms began 2 years prior to admission. Several months
prior to admission he sustained a traumatic blow to the nose
and 1 month prior to admission was scheduled for elective
rhinoplasty to correct the nasal deformity sustained by the
trauma. At the time of surgery a unilateral left nasal mass was
noted and a biopsy was performed. The pathologic diagnosis
was pleomorphic adenoma.
On examination a grayish white mass was noted in the left
nostril that completely occluded the vestibule. There was no
evidence of epistaxis or rhinorrhea. The right nostril was free
of any obstruction.
Treatment consisted of surgical excision of the mass
through a lateral rhinotomy approach with excision of the
mass down to and including the mucoperichondrium. The
surgical margins of the specimen were free of tumor.
The postoperative course was uneventful and the patient
was discharged on the sixth postoperative day. He has been
followed up for 18 months since treatment and there has been
no recurrence of disease.
Pathologic findings. On gross examination the lesion
measured 1.8 x 1.3 x 0.2 em, was polypoid in outline,
grayish white in color, and firm to focally mucinous in consistency. The surrounding mucosa over the nasal septum was of
normal character. On cut section the mass was pale yellow to
white in color with a translucent quality and glistening appearance.
Microscopically, small islands and comma-shaped cords of
epithelial cells were loosely arranged within a supportive
stroma that displayed significant heterogeneity. In areas of
highest epithelial concentration, stromal elements were
primarily collagenous in nature, with mature, elongated
fibroblasts evenly distributed throughout. Sparsely cellular
zones demonstrated an amphophilic to halophilic, mucinous
434 Case Reports
OtolaryngologyHead and Neck Surgery
Fig. 2. Myoepithelial cells of plasmacytoid type and ductal elements are juxtaposed within loose
connective tissue stroma. (Hematoxylin-eosin stain; x180.)
to myxochondroid background with unevenly scattered stellate to spindle-shaped cells that were singly grouped (Fig. 1).
Occasional clear vacuoles were present within the stroma,
especially within the mucoid material (Fig. 2). Growth patterns of epithelial cells included solid sheets, while elsewhere
ductal differentiation was evident. An incomplete or pseudocapsule was noted at the tumor margins, with the most
peripheral epithelial elements growing in narrow cords parallel to the outer margin of the lesion (Fig. 3).
DISCUSSION
The literature dealing with the mixed tumor, or
pleomorphic adenoma, is vast, yet the presence of this
neoplasm in the upper respiratory tract is rare.v" Batsakis! states that within the upper respiratory tract the
most favored site of origin is the nasal cavity, followed
by the maxillary sinus and the nasopharynx. Isolated
reports documenting the presence of this tumor along
the nasal septum exisr'r"; however, there is only one
large series that affords precise information regarding
the clinical and histopathologic features of these intranasal mixed tumors. 2
The nasal septum is sparsely populated by salivary
glands. The vast majority of minor mucous and serous
glands are located on the lateral nasal wall, thereby
emphasizing the unique nature of the septal origin for
these benign mixed tumors. Among all reported cases
of pleomorphic adenomas in the nasal cavity, 80%
arose from the septum and only 20% originated from
the lateral wall or involved a turbinate.'
The apparent discrepancy between the relative paucity of glandular elements on the septum and the predilection of the mixed tumors to originate on the septum has lead several authors to postulate the etiology of
these lesions. Matthew et a1. 3 thought that misplaced
embryonic ectodermal epithelial cells, carried via the
nasal pits into the septal region, could account for
tumors in this location. On the other hand, Stevenson"
stated that the true rarity of embryonic septal growths
excludes the possibility of these tumors arising in this
Volume 93 Number 3
June 1985
Case Reports 435
f'..
-, I-
•
.'.,.
....-.1'
'*, ' . \
t" -., "
4#
••••
" ....
•
,
..
.
, " -\.
..\ "
'1\
Fig. 3. Pseudocartilaginous areas are intersected by arcades of myoepithelial cells. Clusters of
polygonal plasmacytoid myoepithelial cells are present on left portion of field. (Hematoxylin-eosin
stain; x360.)
area. He believed that the remnants of the vomeronasal
Jacobson organ give rise to mixed tumors in this area.
Evans and Cruickshank," however, thought that all
mixed tumors are of epithelial origin and that they arise
from fully developed salivary gland tissue rather than
from embryonic remnants.
The range of age of presentation of these lesions is
large, spanning early childhood to old age, with the
majority occurring from the third through the sixth decades." There appears to be no predominance by sex.
Almost uniformly the chief complaint is nasal
obstruction, usually unilateral. Rarely patients will present with a "mass" in the nose or with epistaxis. Patients usually seek medical attention within I year of
the onset of symptoms.'
The mixed tumor is a homogeneous, lobular mass
that is often polypoid in outline and translucent. Compagno and Wong" reported that the pleomorphic adenoma of the nasal septum differs on a cellular level
from mixed tumors commonly seen in the major salivary glands. They thought that the cellularity is much
greater in these tumors, such that the epithelial rather
than the stromal elements predominate. Batsakis ,!
while concurring with this viewpoint, emphasized that
at times the tumor may be composed almost entirely of
epithelial cells with little or no stroma.
Polypoid masses presenting as unilateral nasal obstruction should be approached judiciously in a diagnostic sense. When dealing with a unilateral nasal mass
one must consider the benign polyp as the most likely
entity. However, the various histologic cellular components of the nose can develop into a wide variety of
tumors.
Congenital lesions such as dermoid cysts, external
encephaloceles, and external nasal gliomas must be
considered as causes of unilateral nasal obstruction in
children." Adolescents may present with the highly
vascular, locally invasive juvenile angiofibroma.' The
OtolaryngologyHead and Neck Surgery
Case Reports
more destructive lesions presenting in adults include
papillomas (fungiform type)," chordomas,'? plasmacytomas,'! fibrous histiocytomas," oncocytomas," esthesioneuroblastomas," hemangiopericytomas, 15 chondromyxofibrosarcomas, and various other tumors. 16 All
of these require aggressive surgical intervention that is
usually combined with some sort of adjuvant therapy.
Treatment of the pleomorphic adenoma by local or
wide surgical excision without the necessity of radical
extirpation differentiates this lesion from its more malignant counterparts. Compagno and Wong! cite a 10%
recurrence rate following conservative local treatment.
They attribute this to the cellular nature of this lesion,
stating that the marked histologic difference between
the nasal variant and the other salivary gland types in
which the myxoid stroma predominates accounts for
the difference in recurrence rates Y Excision of parotid
or intraoral mixed tumors allows for spillage of stroma
into the surgical field, thereby providing a focus for
recurrence.
Although the pleomorphic adenoma of the nasal septum is a relatively uncommon tumor, its importance
lies in its histologic differentiation from other salivary
gland tumors, its generally benign biologic behavior,
and its low rate of recurrence following conservative
management.
CONCLUSION
A case of a young white man presenting with a unilateral nasal obstruction secondary to a septal pleomorphic adenoma has been presented. The histologic nature of this lesion in comparison to other salivary gland
tumors as well as its place in the differential diagnosis
of unilateral nasal masses has been stressed. Its low rate
of recurrence following conservative surgical management has been emphasized.
REFERENCES
I. Batsakis JB: Tumors of the head and neck. Baltimore, 1980, The
Williams & Wilkins Co, p 84.
2. Compagno J, Wong RT: Intranasal mixed tumors (pleomorphic
adenomas). Am J Clin Path 68:213-218, 1977.
3. Matthew S, Ersner MD, Saltzman M: A mixed tumor of the
nasal septum. Report of a case. Laryngoscope 54:287-296,
1944.
4. Gabrial GE: A case of mixed salivary tumor of the nasal septum.
J Laryngol Otol 66:329-330, 1952.
5. Majed MA: Pleomorphic adenoma of the nasal septum. J Laryngol Otol 85:975-976, 1971.
6. Stevenson HN: Mixed tumor of the septum. Ann Otol Rhinol
Laryngol 41:563·570, 1932.
7. Evans RW, Cruickshank AH: Epithelial tumors of the salivary
glands, vol I. In Major problems in pathology. Philadelphia,
1970, WB Saunders Co, p 167.
8. Sessions RB: Nasal dermal sinuses-new concepts and explanations. Laryngoscope (Suppl 29) 92:1-28, 1982.
9. Hyams VJ: Papillomas of the nasal cavity and paranasal sinuses.
Ann 0101 Rhinol Laryngol 80: 192-206, 1971.
10. Campbell WM, McDonald TJ, Unni KK, Laws ER: Nasal and
paranasal presentations of chordomas. Laryngoscope 90:612618, 1980.
II. Ben-David J, Fradis M, Podoshin L, Bartal AH: Plasmacytoma
arising in the vicinity of a foreign body in the nasal cavity.
Laryngoscope 90: 1686·1693, 1980.
12. Del Rey E, De la Torre FE: Fibrous histiocytoma of the nasal
cavity. Laryngoscope 90: 1686-1693, 1980.
13. DiMaio SJ, DiMaio VJM, DiMaio TM, Nicastri AD, Chen K:
Oncocytic carcinoma of the nasal cavity. South Med J 73:803806, 1980.
14. Cantrell RW, Chorayeb BY, Fitz-Hugh GS: Esthesioneuroblastoma: Diagnosis and treatment. Ann Otol Rhinol Laryngol
86:564-565, 1977.
15. Stout AP, Murray MR: Hemangiopericytoma; vascular tumor
featuring Zimmermann's pericytes. Ann Surg 116:26-33,1942.
16. Weber AL, Solis 0: Chondromyxofibrosarcoma of the nasal
cavity. Ann Otol Rhinol Laryngol 86:564-565, 1977.
17. Krolls SO, Boyers RC: Mixed tumors of salivary glands. Longterm follow-up. Cancer 30:276-281, 1972.
Nasal septal hemangioma
BARRY L. WENIG, MD. JAMES J. SCIUBBA, DMD. PhD. ALFRED COHEN, MD, and ALLAN L. ABRAMSON, MD.
New Hyde Park, New York
From the Department of Otolaryngology and Communicative Disorders (Drs. Wenig, Cohen, and Abramson), and the Departments of
Dentistry and Laboratories (Dr. Sciubba), the Long Island
Jewish-Hillside Medical Center.
Presented at the Annual Meeting of the American Academy of
Otolaryngology-Head and Neck Surgery (Scientific Poster), New
Orleans, La., Oct. 17-21, 1982.
436
Submitted for publication Oct. 16, 1982; revision received April 8,
1983; accepted May 3, 1983.
Reprints requests: Barry L. Wenig, MD, Department of Otolaryngology and Communicative Disorders, the Long Island JewishHillside Medical Center, 270-05 76 Ave., New Hyde Park, NY
11042.