Dentomaxillofacial Radiology (2009) 38, 53–58

’ 2009 The British Institute of Radiology

http://dmfr.birjournals.org

CASE REPORT
Brown tumour of the maxilla and mandible: a rare complication of
tertiary hyperparathyroidism
F Selvi*,1, S Cakarer1, R Tanakol2, SD Guler3 and C Keskin1
1
Department of Oral and Maxillofacial Surgery, Faculty of Dentistry, Istanbul University, Turkey; 2Department of Endocrinology
and Metabolism, Istanbul Faculty of Medicine, Istanbul University, Turkey; 3Department of Tumour Pathology and Oncology
Cytology, Istanbul University, Turkey

Hyperparathyroidism is nowadays diagnosed early and asymptomatically with the

improvements in routine biochemical tests and radiological procedures. The late bony
complications of the disease have therefore started to decline rapidly. Brown tumours are one
of the bony complications of hyperparathyroidism. The mandible is the predominantly
affected site in the maxillofacial area. Maxillary involvement is rare. Here, an extremely rare
case of a 19-year-old male patient with brown tumours in his maxilla and mandible
associated with tertiary hyperparathyroidism is presented. A thorough diagnostic work-up
was carried out and treatment options for both hyperparathyroidism and brown tumours
were discussed. The importance of different radiological evaluation methods and the
consultation between the oral and maxillofacial surgeons, general practitioner dentists,
endocrinologists and radiologists are emphasised.
Dentomaxillofacial Radiology (2009) 38, 53–58. doi: 10.1259/dmfr/81694583

Keywords: brown tumour; maxilla; mandible; hyperparathyroidism; panoramic radiograph

Introduction

Parathyroid hormone (PTH) is produced and secreted
by the parathyroid glands, whose activity is controlled
by the free (ionized) serum calcium level.1 Increased
PTH secretion results in a condition called hyper-
parathyroidism (HPT). HPT is divided into primary,
secondary and tertiary types. Primary HPT is char-
acterized by increased parathyroid hormone secretion
occurring as a result of abnormality in one or more
of the parathyroid glands.2 Adenomas are the main
cause in about 85% of primary HPT cases. Most
cases of primary HPT are identified by the presence
of hypercalcaemia and hypophosphataemia on
routine multipanel serum testing.3
Secondary HPT is caused by hypocalcaemia or
vitamin D deficiency acting as a stimulus for excessive
PTH production. Chronic renal failure is the main
cause of secondary HPT.3 It results in hypocalcaemia
and the parathyroid glands over-function to compen-
sate for this low serum calcium level.
*Correspondence to: Firat Selvi, Istanbul University, Faculty of Dentistry,
Department of Oral and Maxillofacial Surgery, Istanbul, Turkey; E-mail:
firatselvi@gmail.com
Received 31 December 2007; revised 11 February 2008; accepted 15 February
2008
Sometimes, in long-standing secondary HPT cases,
the parathyroid glands gain an autonomous character.4
This phenomenon is known as tertiary HPT. Some
authors also report a fourth type of HPT which is
thought to arise from increased PTH levels synthesized
in patients with a malignant disease.4,5
Because of the progress in routine biochemical
screening, today HPT can be diagnosed much earlier,
mainly in the asymptomatic stage, whereas it was
generally diagnosed as an overt disease with predomi-
nantly skeletal manifestations in the 1970s.6 Late bony
manifestations of the disease include generalized osteo-
porosis, multiple focal areas of demineralization of the
skull, and osteitis fibrosa cystica (brown tumour).7
Brown tumours are non-neoplastic lesions resulting
from abnormal bone metabolism in HPT that creates a
local destructive phenomenon.8,9 The ribs, femora and
pelvis are the most commonly seen sites of brown
tumours.2 This bony lesion of the HPT is caused by
increased circulating levels of parathyroid hormone,
which result in increased osteoclastic bone resorption,
primarily in cortical bone.5 This may explain why the
mandible, a cortical bone, is the most commonly
affected site,10 whereas maxillary involvement is less
common3,7,9,11,12 in the maxillofacial area. Here, a rare
 Brown tumours in tertiary hyperparathyroidism
54 F Selvi et al
case of a young male patient with brown tumours in his
maxilla and mandible associated with tertiary hyper-
parathyroidism, will be presented and the importance
of a thorough diagnostic work-up, as well as the
contemporary treatment options, will be emphasised.
Case report
In July 2007, a 19-year-old male was referred to the
Department of Oral and Maxillofacial Surgery of the
Faculty of Dentistry, Istanbul University, by his
endocrinologist for consultation regarding large swel-
lings of his mandible and maxilla. The patient’s medical
history disclosed that he had had chronic renal failure
for about 10 years and was on a regular haemodialysis
programme three times a week for the past 9 years.
3 years prior to his referral, he was diagnosed with
hyperparathyroidism. Since then, he was treated with
vitamin D therapy. Family history revealed that his
younger sister died due to renal insufficiency at 12 years
of age. The patient’s older and younger brothers, along
with his parents, were healthy.
On clinical oral examination, a severe exophytic mass
was found in the mandible starting at the left canine
tooth and extending to the right second molar tooth,
causing displacement of the related teeth and forcing
the tongue into the pharynx, nearly obstructing the
airway (Figure 1). The related teeth were also mobile
(Miller’s grade II/III). The patient had difficulty in
eating and speaking. There was a minor ulcerated area
on the occlusal side of the lesion, probably due to
chewing with the antagonist maxillary teeth. The
maxillary enlargement was smaller in size, presenting
as a swelling on the right side of the palate at the level
of the apices of the molar teeth (Figure 2). Both of the
lesions were non-tender and firm on palpation. They
appeared to be attached to the bone; the overlying
Figure 1 Severe enlargement of the mandible. Note displacement of
the teeth in the related area and the tongue being forced to the
pharynx, nearly obstructing the airway. Minor ulceration visible on
the dorsum of the lesion
Dentomaxillofacial Radiology
Figure 2 Maxillary enlargement. Expansive mass at the level of the
apices of the right maxillary molar teeth
mucosa was freely mobile. The mucosa over the
mandibular lesion was highly vascular. The mandibular
lesion caused asymmetry of the face on the right side.
The patient indicated that these lesions had been
present for about 3 years, but had enlarged rapidly in
the last year.
On physical examination the patient was thin and
short in stature, despite his age (height 1.35 meters/
49 4.40 and weight 25.5 kg/56.1 lbs; body mass index:
14.4 kg m22). The patient complained of generalized
weakness and difficulty in performing daily domestic
work. A deficiency of secondary male sex charac-
teristics, such as lack of pubic and axillary hair and a
deep voice, was also observed. The thyroid gland was
minimally enlarged.
On the panoramic radiograph, a well-demarcated
radiolucent area starting at the left mandibular canine
tooth and extending to the right second molar tooth
was observed (Figure 3). The maxillary lesion was not
clearly visible on the panoramic radiograph, but upon
close observation of the radiograph, another radiolu-
Figure 3 Panoramic radiograph of the maxillary and mandibular
lesions. A well-demarcated radiolucent area, starting at the left
mandibular canine tooth and extending to the right second molar
tooth is shown. Another osteolytic area is visible at the apex of the left
mandibular molar teeth. The maxillary lesion is not clearly
distinguishable on this panaromic radiograph

Figure 4 CT scans of the related areas confirmed the presence of
intraosseous radiolucent lesions. The brown tumours on both sides of
the mandible are clearly visible
cent area was noticed on the left side of the mandible,
causing local destruction of the basal bone under the
area of the apices of the left mandibular molar teeth. A
generalized loss of lamina dura was noticeable on the
panoramic radiograph. CT scans of the mandible
confirmed the presence of these intraosseous radiolu-
cent lesions (Figure 4). According to the CT scans, the
right mandibular lesion measured 3.4565.4563.46 cm.
The CT scan also revealed that the maxillary lesion
(measuring 1.2861.4661.36 cm) had infiltrated into
the maxillary sinus (Figure 5).
The laboratory findings were as follows: intact PTH
2415 pg ml21 (15–65 pg ml21), calcium 11.0 mg dl21
(8.5–10.5 mg dl21), phosphate 6.7 mg dl21 (2.7–
4.5 mg dl21), alkaline phosphatase 1270 U L21
(90-260 L21).
Figure 5 CT scans of the maxillary lesion reveal the lesion to be
penetrating the right maxillary sinus
Brown tumours in tertiary hyperparathyroidism
F Selvi et al 55
Figure 6 Ultrasonography of the parathyroid glands revealed solid
hypoechogenic parathyroid lesions on both sides of the lower poles of
the thyroid lobes. Arrow indicates the hypoechogenic solid para-
thyroid lesion at the lower pole of the right thyroid lobe.
On parathyroid ultrasonography, bilateral thyroid
lobes showed no enlargement, whereas a hypoechogenic
solid lesion 6.3610 mm in size was observed in the left
thyroid lobe’s posteroinferior localization (Figure 6).
Other than that lesion, two more hypoechogenic solid
lesions with some punctate micro-calcifications were
observed: (i) in the region of the right thyroid lobe’s
posteroinferior region measuring 8.866.7 mm and
(ii) in the inferomedial of the latter, measuring
9.7617.3 mm.
Parathyroid technetium scintiscan (99Tcm sestamibi
scintigraphy; MIBI, methoxy-isobutyl-isonitrile)
showed abnormally high uptake at the lower and
superior poles of the left lobe of the thyroid, and also at
the lower pole of the right lobe of the thyroid. These
were interpreted as parathyroid hyperplasia. Other than
the thyroid gland, abnormally high uptake was also
shown on both sides of the mandible, at both of the
shoulders, the right anterior part of the ribs and the
sternum. These high uptakes were consistent with
brown tumours (Figure 7).
Fine needle aspiration biopsies were then performed
on the mandibular and maxillary lesions. On micro-
scopic examination, many multinucleated giant cells
arranged in groups adjacent to haemosiderin granules
within a fibrovascular haemorrhagic stroma were
observed (Figure 8). These findings were compatible
with a diagnosis of brown tumours of the jaws,
associated with hyperparathyroidism.
Finally, based on the thorough diagnostic work-up
including medical history, clinical manifestations,
radiographic findings and consecutive routine labora-
tory findings, the patient was diagnosed as having
tertiary hyperparathyroidism with brown tumours of
both jaws as a result of long-term renal disease.
Dentomaxillofacial Radiology
 Brown tumours in tertiary hyperparathyroidism
56 F Selvi et al
Figure 7 Parathyroid technetium scintiscan showing abnormally
high uptake in both sides of the mandible and shoulders as well as on
the sternum and right anterior part of the ribs. Abnormally high
uptake is also observed at the lower pole of the right and left thyroid
lobes
Figure 8 Giemsa stain. Histopathological examination showing a
multinucleated giant cell arranged within erythrocytes and some
connective tissue elements (magnification 6 200)
Discussion
With new and more objective diagnostic PTH radio-
immunoassay techniques, along with the successful
treatment of the disease, bony complications of HPT
have started to decline rapidly. The earliest and most
reliable changes seen with hyperparathyroidism are
subtle erosion of bone on the subperiosteal surfaces of
the phalanges.13 Osteitis fibrosa cystica is a late man-
Dentomaxillofacial Radiology
ifestation of severe HPT. Overt findings of osteitis
fibrosa cystica include generalized demineralization
of bone, ‘‘salt and pepper’’ appearance of the
skull, bone cysts and brown tumours.6,14
Brown tumours, or osteoclastomas, are caused by
localized, rapid, osteoclastic removal of bone secondary
to the direct effects of PTH on the bone. The name
‘‘brown tumour’’ derives from the colour, which is
caused by the vascularity, haemorrhage, and deposits of
haemosiderin.6,15,16 Brown tumour is actually a giant
cell lesion and often appears as an expansile osteolytic
lesion of the bone. The lesion usually presents as a
slight swelling in the jaw bones.8 However, similar to
the case presented by Dinkar et al,2 this case differs
from previously presented cases in its gross mandibular
destruction. Histological and radiographically, it is very
similar to the other giant cell lesions (true giant cell
tumour, reparative giant cell granuloma, cherubism and
aneurysmal bone cyst).15 On clinical examination and
using only routine panoramic radiography, the lesions
may resemble osteosarcoma, bone metastases of a
carcinoma, multiple myeloma, Langerhan’s cell histio-
cytosis, Paget’s disease, osteomyelitis or osteonecrosis
secondary to bisphosphonate therapy. The differential
diagnosis is based on the clinical findings and the
presence of hyperparathyroidism, which is confirmed
with biochemical tests including PTH level.
Even though the younger sister of the patient died of
renal failure at 12 years of age, this was not thought to
be a case of hyperparathyroidism–jaw tumour syn-
drome (HPT-JT syndrome), because this autosomal
dominantly inherited condition is only seen in primary
hyperparathyroidism.17
Secondary and tertiary HPT are mostly seen in
patients with chronic renal disease. Secondary HPT
usually affects older adults (50–80 years) with a 2:1
female predominance,16 so the patient in this report was
unusual for his young age and gender.
Radiographic findings in this case were similar to
those mentioned elsewhere.2,8 CT scans were especially
valuable for the determination of the exact borders and
size of the brown tumours. Ultrasound and parathyroid
scintiscans were also very useful in localizing the
abnormalities in the skeletal bones and parathyroid
glands.
The most significant point about the case described
here is the simultaneous appearance of brown tumours
in the maxilla and mandible. In the maxillofacial
region, brown tumours are most commonly seen in
the mandible; maxillary involvement is rarely reported.
However, as far as the authors have been able to
determine to date, there have only been five such cases
of brown tumours appearing simultaneously in the
maxilla and the mandible previously published in the
English literature.6,18–21
The various types of HPT manifest different labora-
tory findings. In primary HPT, the serum calcium level
is usually elevated, with low or normal serum phos-
phate level. Patients with secondary HPT, on the

other hand, usually present with hypocalcaemia and
hyperphosphataemia. These findings are variable in
tertiary HPT.16 This patient was slightly hypercalcae-
mic and definitely hyperphosphataemic. The level
of alkaline phosphatase was about five times the
normal level, which indicates high bone turnover.
In patients with a brown tumour, HPT should first
be treated before considering resection of the tumour.
In secondary and tertiary HPT, chronic renal failure
should be managed by means of haemodialysis or,
eventually, a renal transplant. In primary HPT,
removal of the autonomous parathyroid glands should
be performed.
Triantafillidou et al3 stated that the significant bone
disease associated with secondary HPT may be
prevented or reduced by medical treatment, such as
calcium carbonate, vitamin D and aluminum hydroxide
antacids for hyperphosphataemia. In contemporary
medical treatment alternatives, lanthanum carbonate,
sevelamer and calcimimetics such as cinacalcet are more
commonly preferred. Parathyroidectomy should be the
first choice of treatment in tertiary HPT when the
disease is resistant to medical therapy.
Normalization of PTH levels will often cause the
brown tumours to regress or sometimes even resolve
spontaneously.14 Systemic corticosteroids can be used to
reduce the size of the lesion; sometimes intralesional
corticosteroid injections also give satisfactory
results.11,22
Large lesions may resolve very slowly or may regress
with resultant asymmetry on the face. Surgery in the
form of excision of the brown tumour and recontouring
of the bone should therefore be done in such cases.5,13
In the case presented, parathyroidectomy was indi-
cated. With the removal of the autonomous parathyr-
oid glands, the PTH level would eventually return to
normal, thus leading to a spontaneous decrease in the
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F Selvi et al 57
sizes of the intraoral lesions within a few months.
Resection of the right mandibular lesion would enhance
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ment in his systemic condition. In the long run his renal
insufficiency must be treated, probably by means of a
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In conclusion, this dramatic entity therefore high-
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Panoramic radiographs, CT scans, ultrasonography,
and parathyroid scintiscan with 99Tcm sestamibi were
all useful radiographic methods for the correct diag-
nosis of this tumour. This case should attract the
attention of general practitioner dentists, oral and
maxillofacial surgeons, endocrinologists and radiolo-
gists whose consultation may be vital for patients with
hyperparathyroidism since the disease may result in
nearly fatal results if neglected. Dentists should also be
alert for the possible presence of brown tumours in the
jaws of patients who have previously been diagnosed as
having hyperparathyroidism.
Acknowledgments
The authors would like to thank Dr Daniel Laskin who
critically revised the manuscript, Dr Yakup Akyol, Dr Sema
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Dentomaxillofacial Radiology
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