LESSA, SAKAE, TSUJI, ARAUJO FILHO, VOEGELS, BUTUGAN

432 ENT-Ear, Nose & Throat Journal

July 2005

Brown tumor of the facial bones:

Case report and literature review
ORIGINAL ARTICLE
From the Department of Otorhinolaryngology, University Hospital,
University of So Paulo Medical School.
Reprint requests: Dr. Flavio Akira Sakae, Av. Dr. Enas de Carvalho
Aguiar, 255-6 Andar, Sala 6021, So Paulo, SPBrazil, 05403-
000. Phone: 55-11-3069-6288; fax: 55-11-3088-0299; e-mail:
sakaeavio@yahoo.com.br
Marcus M. Lessa, PhD; Flavio A. Sakae, PhD; Robinson K. Tsuji, PhD;
Bernardo C. Arajo Filho, PhD; Richard L. Voegels, MD; Ossamu Butugan, MD
Abstract
Brown tumor, an uncommon focal giant-cell lesion, arises
as a direct result of the effect of parathyroid hormone on
bone tissue in patients with hyperparathyroidism. The initial
treatment involves the correction of hyperparathyroidism,
which usually leads to tumor regression. We report a case
of brown tumor of the right nasal fossa in a 71-year-old
woman. The tumor had caused nasal obstruction and
epistaxis. Laboratory evaluation revealed that the patient
had primary hyperparathyroidism. Anatomicopathologic
investigation revealed the presence of a giant-cell tumor.
We performed a partial parathyroidectomy, but the tumor
in the right nasal fossa failed to regress. One year later, we
performed surgical resection of the lesion. The patient re-
covered uneventfully, and she remained asymptomatic and
recurrence-free at the 1-year follow-up. Facial lesions with
histologic features of a giant-cell tumor should be evaluated
from a systemic standpoint. Hyperparathyroidism should
always be investigated by laboratory tests because most
affected patients are asymptomatic. Surgical resection of
a brown tumor should be considered if the mass does not
regress after correction of the inciting hyperparathyroid-
ism or if the patient is highly symptomatic.
Introduction
Brown tumor, an uncommon focal giant-cell lesion, arises
as a direct result of the effect of parathyroid hormone on
bone tissue in some patients with hyperparathyroidism.
Brown tumors can affect the mandible, maxilla, clavicle,
ribs, and pelvic bones.
1
Radiographic and histologic
changes associated with the presence of this lesion may
be very similar to those of other bone lesions of the face.
Therefore, the diagnosis requires a systemic investigation
for lesion differentiation.
We report a case of brown tumor in an elderly woman,
and we discuss the clinical history, differential diagnosis,
diagnosis, and treatment of this type of lesion.
Case report
A 71-year-old woman was referred to our otorhinolaryngol-
ogy service for evaluation of a painful mass in the right
nasal fossa that had developed 1 month earlier. The size of
the mass had progressively increased and had eventually
caused nasal obstruction and epistaxis. Six years earlier, the
patient had presented with a nodule in the right mandibular
region. At that time, she underwent an excisional biopsy
and hemimandibulectomy, and the mass was diagnosed
histologically as a giant-cell tumor. Until the development
of the new mass, she had remained asymptomatic. She
had not been evaluated for hyperparathyroidism during
her previous episode.
Otorhinolaryngologic examination revealed that the
mass had caused a septal deviation to the left. Oroscopy
identied a bulge in the right upper gingival-labial sulcus.
Computed tomography (CT) of the paranasal sinuses re-
vealed a dense area in the right nasal fossa (gure 1). No
bone erosion was evident. Biopsy of the mass revealed
that it was a giant-cell tumor.
Laboratory evaluation revealed that the patient had
primary hyperparathyroidism, as her parathyroid hormone
level was 988 pg/ml (normal: 12 to 72). Other labora-
tory measurements were total serum calcium, 11.2 mg/dl
(normal: 8.8 to 11.0); ionized calcium, 5.6 mg/dl (normal:
4.0 to 5.4); phosphorus, 2.0 mg/dl (normal: 2.5 to 4.8);
and alkaline phosphatase, 145 U/L (normal: 32 to 104).
Ultrasonography of the neck revealed an enlargement of
the lower left parathyroid gland (2.1 1.4 0.8 cm). The
nding of hyperparathyroidism conrmed the diagnosis
of brown tumor.
To correct the hormonal imbalance, we performed a
partial parathyroidectomy with a self-implantation of
the right lower parathyroid into the prethyroid muscles.
Histopathologic examination of the lower left parathyroid
revealed the presence of an adenoma.
During the rst year of postoperative follow-up, the mass
Volume 84, Number 7 433
BROWN TUMOR OF THE FACIAL BONES: CASE REPORT AND LITERATURE REVIEW
in the right nasal fossa failed to regress and the patients
nasal symptoms persisted. Therefore, she underwent
exeresis of the mass through a sublabial incision. Intra-
operatively, the mass appeared as a hard, brownish-white,
well-delimited, 2.5 1.2 1.1-cm lesion that extended
into the upper portion of the hard palate. Curettage of the
palatine bone was also performed.
Anatomicopathologic examination revealed that the
brous lesion contained numerous giant cells, again
conrming our diagnosis of brown tumor. The patient
recovered uneventfully, and she remained asymptomatic
and recurrence-free at the 1-year follow-up (gure 2).
Discussion
Brown tumor is an uncommon sequela of hyperparathyroid-
ism. The lesion localizes in areas of intense bone resorption,
and the bone defect becomes lled with broblastic tissue
that can deform the bone and simulate a neoplastic process.
2

These tumors have a brown or yellow hue.
3

Brown tumors arise secondary to both primary and sec-
ondary hyperparathyroidism. They have been reported to
occur in 4.5% of patients with primary hyperparathyroidism
and 1.5 to 1.7% of those with secondary disease.
1,4
Hyper-
parathyroidism is frequently caused by the development
of a parathyroid adenoma and less often by hyperplasia
or a carcinoma. Some parathyroid adenomas and hyper-
plasias are familial (5% of cases), and others are part of
multiple type I, IIa, and IIb endocrine neoplasias. The
size of a parathyroid adenoma is correlated with the level
of parathyroid hormone in blood.
5
Anatomicopathologic
study of the parathyroid in our patient revealed the pres-
ence of an adenoma.
Albright et al, in their classic description of primary
hyperparathyroidism, reported that 2 of 17 patients (11.8%)
had tumors involving the jaws.
6
In a review of 220 patients,
Rosenberg and Guralnick found that 10 patients (4.5%)
had a mass involving one or both jaws as their presenting
complaint.
7
Brown tumors arise more frequently in the
mandible than in the maxilla.
1,2
Our patient originally
had a brown tumor in the mandible and later in the right
nasal fossa.
The reported prevalence of brown tumor is 0.1%.
2
The
disease can manifest at any age, but it is more common
among persons older than 50 years.
2
It is three times
more common in women than in men.
2
Our patient was
65 years old the rst time she was diagnosed with a giant-
cell tumor.
Most patients with hyperparathyroidism are asymptom-
atic, as was our patient. Hypercalcemia is often discovered
incidentally during routine laboratory testing; hypophos-
phatemia and increased alkaline phosphatase levels in
blood may also be seen.
8
These metabolic changes were
detected in our patient.
Parathyroid adenomas are usually small and almost
never palpable in the neck. When clinical manifestations
are present, the signs and symptoms can be divided into
two types: urologic changes secondary to hypercalcemia
and skeletal changes. Urologic changes include polyuria,
Figure 1. CT of the paranasal sinuses shows the dense area in
the right nasal fossa.
Figure 2. One year following removal of the nasal fossa mass,
CT detects no sign of recurrence.
LESSA, SAKAE, TSUJI, ARAUJO FILHO, VOEGELS, BUTUGAN
434 ENT-Ear, Nose & Throat Journal

July 2005

polydipsia, and the development of kidney stones. Again,

hypercalcemia is usually asymptomatic, but in severe cases
it may cause anorexia, vomiting, constipation, fatigue,
weight loss, muscle weakness, psychiatric symptoms,
and pancreatitis.
5
Skeletal changes may represent the rst
manifestations of the disease, with a loss of cortical bone
and an increase in trabecular bone. Bone pain and arthralgia
are the most common symptoms. Bone demineralization is
uncommon in mild hyperparathyroidism, but cystic brous
osteitis is detected in some cases along with pathologic
fractures and brown tumors. Radiologically, parathyroid
adenomas may or may not exhibit well-dened margins;
they may also cause cortical expansions associated with
lytic lesions. Finally, patients may exhibit hyperparathy-
roidism-induced bone changes, such as medullary bone
demineralization of the mandible.
9

Brown tumors exhibit no pathognomonic histologic
changes. Examination will reveal a dense broblastic
stroma, areas of cystic degeneration, osteoid, microfrac-
tures, hemorrhage, macrophages with hemosiderin, and
multinucleated osteoclastic giant cells. Hemorrhage and
hemosiderin confer the lesions characteristic color. Similar
changes may occur in brous dysplasia, true giant-cell tu-
mors, and reparative granulomas.
2
Differentiating between
a brown tumor and other giant-cell tumors may be very
difcult, even with histology.
Fibrous dysplasia affects the bones of the face, and it is
most common among young women. Histology reveals
trabecular bone with a stroma rich in brous tissue and
multinucleated giant cells that are visible in areas of hem-
orrhage secondary to focal degeneration.
10

True giant-cell tumors are more inltrative than brown
tumors. Their cause is unknown. They are rare, but when
they do occur, they usually develop in older patients and
they primarily involve the long bones. They also have
a tendency to recur. Because of their inltrative nature,
true giant-cell tumors are difcult to remove surgically.
Histologic analysis reveals giant cells around a brous
stroma and some degree of cellular atypia.
2,10

Reparative granulomas are localized tumors detected
in young patients. They primarily involve the mandible.
Their cause is still unknown, but some investigators
believe that they are a result of trauma.
2
They can be eas-
ily removed by curettage or local excision. A reparative
granuloma can be differentiated from a brown tumor by
the absence of hyperparathyroidism. Histologically, they
contain giant cells, but their stroma is less dense and more
vascularized.
2,10
Therefore, patients with giant-cell tumors
should be investigated for the presence of hyperparathy-
roidism and hypercalcemia in order to differentiate these
granulomas from brown tumors. Our patient was tested
for hyperparathyroidism and hypercalcemia only during
her second episode.
The initial treatment of brown tumor involves control of
hyperparathyroidism, regardless of whether it is primary
or secondary. Treatment of primary hyperparathyroidism
requires a parathyroidectomy. Once hyperparathyroidism
is controlled, the tumor tends to regress, although in some
cases surgical removal is necessary, especially for patients
who have large, symptomatic tumors. Our patient required
surgical removal. Brown tumor can recur if hyperparathy-
roidism persists or recurs.
2

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