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Available online 24 March 2014

Hypercalcemia;

Parathyroid adenoma 2014 Production and hosting by Elsevier B.V. on behalf of King Saud University.

or secondary hyperparathyroidism. It is a component of a met-

abolic bone disease recognized as osteitis brosa, cystica gen-

bones, pelvic girdle, clavicle, ribs, and the mandible. Brown tu-

adenocarcinoma (Thompson et al., 1982). In most cases, ade-

arise in an ectopic site and could be found anywhere betweenthe angle of the jaw and the pericardium. As a result of vari-

thyroid adenoma is found inside the mediastinum.anifesting as ais rare. Our case

brown atinal para

A 55-year-old Saudi woman was admitted to the Oral and

Maxillofacial Surgery unit at the King Abdulaziz UniversityHospital (Jeddah) with a complaint of anterior maxilla massof one month duration. Her medical history was

unremarkable.Initial examination revealed a painful mass in the anterior

maxilla (Fig. 1). An anesthesia consultation was requested,

* Address: King Abdulaziz University, P.O. Box 21943, Jeddah

13042, Saudi Arabia. Tel.: +966 (5) 05677905; fax: +966 (2) 6408315.

E-mail address: faiza_qari@yahoo.co.uk.

Peer review under responsibility of King Saud University.

Production and hosting by Elsevier

The Saudi Dental Journal (2014) 26, 7477

King Saud

The Saudi De

www.ksuwww.sciencePrimary hyperparathyroidism in 80% of the cases is due toa parathyroid adenoma; in over 15% of the cases it is due to a 2. Case reportmor is rarely involved in the maxilla.The reported prevalence of brown tumors is 0.1%, with a

male to female ratio of 1:3 (Proimos et al., 2009). Symptomscan occur at any age; however, the disease is more commonin persons older than 50 years

Primary hyperparathyroidism initially mbrown tumor located in the anterior maxilla

report presents a 55-year-old woman with amaxilla tumor arising in the ectopic mediasadenoma.1013-9052 2014 Production and hosting by Elsevier B.V. on behalf of King Saud University.http://dx.doi.org/10.1016/j.sdentj.2013.12.009nteriorthyroideralisata or Recklinghausens disease of bone (Neville et al.,2009; Pinar Sumer et al., 2004). It usually occurs in the long

ability in glandular tissue migration during embryologic life

(Weller, 1933), in more than 80% of the cases the ectopic para-Brown tumor is one of the clinical manifestations of primary nomas are located in the neck, while in 13% of the cases they1. Introduction glandular hyperplasia, and extremely rare due to a parathyroidKEYWORDS

Brown tumor;

Maxilla;

Mediastinum;wAbstract Brown tumors are uncommon focal giant-cell lesions that arise as a direct result of the

effect of parathyroid hormone on bone tissue in some patients that have hyperparathyroidism. Pri-

mary hyperparathyroidism could be caused by ectopic mediastinal parathyroid adenomas. The

occurrence of lesions is explainable on embryologic basis. We present a 55-year-old Saudi woman

ith a rare case of brown tumor of the maxilla due to ectopic mediastinal parathyroid adenoma.CASE REPORT

Brown tumor in a patient witmediastinal parathyroid aden

Faiza A. Qari *

Department of Endocrinology, King Abdulaziz University Hospit

Received 20 March 2013; revised 7 December 2013; accepted 2ectopica: A case report

Jeddah, Saudi Arabia

ecember 2013

University

ntal Journal

.edu.sadirect.com

sections showed multiple giant cells consistent with Brown tu-

Figure 1 Preoperative image of the maxillary tumor.

A Case of Brown Tumor 75and surgery (including the excision of the lesion and curettage

of the bone under general anesthesia) was offered; however,surgery was not undertaken because routine laboratory inves-tigations revealed hypercalcemia.

The endocrinologist was urgently consulted for investiga-tion and treatment of hypercalcemia in this patient. She alsoreported a six-month history of asthenia, generalized muscle

pain, polyuria and constipation. The patient appeared welland had an unremarkable physical examination.

Initial laboratory tests performed on admission showed thefollowing: alkaline phosphates 143 IU/L (reference range, 50

136 IU/L), corrected serum calcium 3.2 mmol/L (referencerange, 2.122.52 mmol/L), serum phosphate 0.55 mmol/L (ref-erence range, 0.81.58 mmol/L), intact parathyroid hormone

120 pmol/L (1.66.9 pmol/L).A provisional diagnosis was made for hypercalcemia due to

hyperparathyroidism. Further investigations were performed.

These included technetium thallium (99mTc-201Th) subtrac-tion scintigraphy (Sestamibi scanning), which demonstrated asingle, ectopic anterior mediastinal parathyroid adenoma

(Fig. 2). Magnetic resonance imaging (MRI) of the thoraxshowed a mediastinal parathyroid adenoma (Fig. 3).

Treatment was initiated by hydration with normal salineand intravenous biophoshante until normalization of the pa-

tients serum calcium level.Initially the patient underwent extirpation of the mass and

curettage of the bone under general anesthesia. HistologicalFigure 2 Sestamibi scan showing the ectopic mediastinal para-

thyroid adenoma.mor of primary hyperparathyroidism (Fig. 4).The patient was readmitted after two weeks, and she under-

went mediastinal parathyroidectomy by median sternotomy.The histopathological diagnosis of the lesion was parathyroidadenoma (Fig. 5). The post-operative course was uneventful

and on the sixth post-operative day, the patient was dischargedwithout complications. The results of postoperative laboratorytests were normal (Graphs 1 and 2).

3. Discussion

Hyperparathyroidism was rst diagnosed by Sylvanus, while

Recklinghausen was credited with the rst description of theassociated bone changes known as osteitis brosa cystic (Nev-ille et al., 2009).

The brown tumor is mainly due to secondary hyperparathy-roidism in patients with renal insufciency, but it has also beenknown as a rare manifestation of calcium malabsorption andsome forms of osteomalacia (Pinar Sumer et al., 2004). These

days, brown tumors are an extremely infrequent manifestationof primary hyperparathyroidism because of routine screeningof hypercalcemia and early diagnosis of hyperparathyroidism.

Inour patient, the histological diagnosiswas indicative of a giantcell bone expanding lesion with a brown tumor being the mostprobable diagnosis. Blood tests established diagnosis of primary

hyperparathyroidism. The localization of primary hyperpara-thyroidism revealed a big ectopic mediastinal parathyroid ade-noma with overproduction of parathyroid hormone.

The treatment of hyperparathyroidism by resection of para-

thyroid adenoma is the initial step in the management of thebrown tumor. Brown tumor regression and healing are pre-dicted after the correction of hyperparathyroidism. However,

many case reports of brown tumor in the litutre showed thatthe tumors grew parathyroidectomy or normalization of para-thyroid hormone level. The treatment of choice in these cases

should be brown tumor resection (Yamazaki et al., 2003).The incidence of bone lesions in patients with hyperpara-

thyroidism has reduced from 80% to 15%, this reduction

has attributed to better hypercalcemia monitoring in asymp-tomatic patients and to the greater use of biochemical analyses(Praveen and Thriveni, 2012).

Brown tumors are one of the osseous manifestations of

hyperparathyroidism. Brown tumors appear in around 10%of the cases in the advanced stages of the chronic kidney dis-ease. It could be found in any part of the skeleton, most com-

monly seen on the ribs, clavicle and pelvis. 4.5% has beenlocated in the mandibul (Pahlavan and Severin, 2006; Siaet al., 2012).

Parathyroid tumors are usually located on the posteriorcapsule of the thyroid but may be in other ectopic locations.Ectopic parathyroid adenomas report for 13% of all parathy-roid tumors. Most of the tumors are located in the anterior

mediastinum (Al-Mashat et al., 2009). An enlarged gland des-cends into the mediastinum because of its higher mass as wellas a result of intrathoracic negative pressure and esophageal

movement. Ectopic locations are certainly connected to themigratory pathways of embryologic parathyroid tissue to theadult positions (Weigel et al., 2005).

The most frequent location for ectopic parathyroid tumorsis the mediastinum. Approximately 20% of parathyroid

Figure 3 Axial magnetic images of the

Figure 4 Hematoxylin and eosin staining revealed multinucle-

ated giant cells, mononuclear stromal cells, osteoblastic and

osteoclastic areas, and deposits of hemosiderin at 100magnication.

Figure 5 Parathyroid adenoma containing chief and clear cells

as seen after hematoxylin and eosin staining at 100magnication.

76 F.A. Qaritumors are situated in the mediastinum. Seventy percent of the

ectopic parathyroid glands are situated in the thymus. Theseglands can be excised through a cervical approach with orwithout a sternotomy (Mantar et al., 2012; Soundarya et al.,

2011).This is the only case reported of a brown tumor situated in

the anterior part of the maxilla presented as the rst sign ofprimary hyperparathyroidism due to an ectopic parathyroid

mediastinal parathyroid adenoma.

Graph 1 Normalization of calcium levels post

parathyroidectomy.

Graph 2 Normalization of parathyroid hormone levels after

parathyroidectomy.

adenoma situated in the anterior mediastinum (Akinoso et al.,1975).

Financial/nonnancial disclosures

The authors have reported that no signicant conicts of inter-est exist with any companies/organizations whose products or

services may be discussed in this article.

Funding support

None.

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A Case of Brown Tumor 77

Brown tumor in a patient with ectopic mediastinal parathyroid adenoma: A case report1 Introduction2 Case report3 DiscussionFinancial/nonfinancial disclosuresFunding supportReferences