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Case Report
An unusual case of brown tumor of
hyperparathyroidism associated with ectopic
parathyroid adenoma
Mathan Mohan1, Ravana Sundaram Neelakandan1, D. Siddharth1, Ravi Sharma1
ABSTRACT
Brown tumor is a giant cell lesion associated with hyperparathyroidism. It is a nonneoplastic condition and represents terminal stage of
the remodeling process in hyperparathyroid state. We report a case of brown tumor with multiple lesions in craniofacial region associated
with ectopic parathyroid adenoma revealed after acute Lthyroxine poisoning. This case report emphasizes on the need for routine
biochemical investigations along with serum calcium, phosphorus and parathyroid hormone levels in patients on thyroxine therapy.
Key words: Brown tumor, ectopic parathyroid gland, giant cell lesions, hyperparathyroidism, parathyroid hormone, thyroxine
How to cite this article: Mohan M, Neelakandan RS, Siddharth D, Sharma R. An unusual case of brown tumor of hyperparathyroidism associated with
ectopic parathyroid adenoma. Eur J Dent 2013;7:500-3.
mobility, loss of vitality or tenderness was elicited of ectopic(intrathoracic) parathyroid gland with
with any teeth in the left quadrant. neoplastic changes in left submandibular salivary
gland region[Figure2a]. Ultrasound of the abdomen,
Biochemical assay and blood analysis revealed renal function tests were unremarkable.
an increased value of serum alkaline phosphatase
(420 u/l; normal range: 100172 u/l), serum CT scan of the chest was taken, which revealed a
PTH (370.40 pg/ml; normal range: 50300 pg/ml) moderate size nodular lesion showing heterogeneous
and serum calcium (13.3 mgs/dl; normal range: enhancement in right prevascular space measuring
8.511.0mg/dl). The serum phosphorus level was 3.0cm2.4cm[Figure2b]. The history, biochemical
decreased(2.4mg/dl; normal range: 2.74.5mg/dl). values and imaging reports corroborated with the
clinical features of the brown tumor of HPT associated
Cone beam computed tomography(CT) scan of with pathologic ectopic parathyroid gland.
the facial region revealed a welldefined soft tissue
lesion within the left body of the mandible of An incisional biopsy of the mandibular lesion was
approximately 3.5cm2.8cm[Figure1a and d]. It performed under local anesthesia that revealed fibro
also represented welldefined hypodense lesions in collagenous tissue containing plenty of osteoclastic
relation to the right body of the mandible measuring giant cells dispersed throughout the lesion with small
2.5cm1.0cm[Figure1d] and anterior maxilla, fragments of reactive bone, the features consistent
measuring 3.5cm3.6cm[Figure1b]. There was also with reparative giant cell granuloma.
lytic lesion on the left side skull bone with generalized
reduction in bone density[Figure1c]. AFull body The patient was then prepared for surgery under
skeletal survey was also performed, which revealed general anesthesia. Thoracotomy was carried out
no such lesions in long bones. and ectopic intrathoracic parathyroid gland was
excised[Figure3a]. An intraoral buccal vestibular
Ultrasound of neck failed to reveal any pathology incision was used to access the mandibular lesion.
in thyroid and parathyroid glands. To further assess Acombination of sharp and blunt dissection was
the parathyroid gland status 99mTc sestamibiSPECT used to excise the mass and it was delivered per
parathyroid scintigraphy was carried out, which oral in total[Figure3b]. The wound was closed in a
demonstrated relatively prominent flow of activity single layer. Postoperative recovery was uneventful.
toward left submandibular salivary gland region. Postoperative serum calcium level was 10.6mg/dl
Abnormal ovoid region of intense tracer concentration and PTH level 11.90pg/mg.
in the anterior mediastinum in right paratracheal
region was noted. The findings were suggestive The intrathoracic mass and mandibular specimen
was sent for histopathological examination.
The intrathoracic mass revealed parathyroid
neoplasm suggestive of atypical parathyroid
adenoma[Figure4a]. The mandibular lesion presented
a giant cell proliferation in the background of a
a b
c d
a b
Figure1:(a) Computed tomography scan(axial section) showing
welldefined soft tissue lesion within the left body of the mandible. Figure2:(a)99m
Tc sestamibisingle photon emission computed
(b) Axial section showing maxillary lesion.(c) Coronal section showing tomography parathyroid scintigraphy image showing intense tracer
skull bone lesion on the left side.(d) 3D reconstruction showing concentration in right paratracheal region and left submandibular
bilateral lytic lesion within the body of the mandible and generalized region.(b) Computed tomography scan(axial section) showing well
reduction in the bone density(arrow indicating the lesion) defined mass in intrathoracic prevascular space
a b
Figure4:(a) Histopathological section of mandibular lesion showing
a b giant cell proliferation with variable areas of haemorrhage and
Figure3: Intraoperative view of(a) Ectopic parathyroid gland. (b) Left hemosiderin deposition(HandE, 20).(b) Histopathological section
mandibular lesion of parathyroid neoplasm composed of nuclear pleomorphism with
cystic changes, suggestive of parathyroid adenoma(HandE, 20)
The exact mechanism of the interaction of thyroid absence of any pathology of anatomic parathyroid,
and PTHs is unclear. Few authors have reported ectopic sites should be assessed. This case emphasizes
that prolonged thyroidstimulating hormone(TSH) the need for periodic biochemical investigations in
stimulation may lead to HPT or a state of HPT in the hypothyroid patients on exogenous thyroxine
hypothyroidism and vice versa.[7,8] This case report therapy.
establishes a credible support for this hypothesis
and brings out a definite correlation between TSH ACKNOWLEDGMENTS
inhibition and onset of HPT.
The authors would like to acknowledge the support of
The treatment of brown tumor mainly focuses on Dr.Joseph Arnold and Dr.Vinod Krishna for their sincere
correction of the underlying disorder and maintenance efforts in development of this article.
of normal PTH and serum calcium levels. Use of
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Our patient represents a rare case of brown tumor Access this article online
with a wide spectrum of associated clinical findings. Quick Response Code:
Website:
In conclusion, the management of the brown tumor www.eurjdent.com
should involve early diagnosis, complete biochemical
assay and full body skeletal survey followed by Source of Support: Nil.
normalization of PTH, serum calcium and phosphorus Conflict of Interest: None declared
levels and parathyroidectomy, if indicated. In the