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British Journal of Oral and Maxillofacial Surgery 50 (2012) e33–e35

Short communication
Not all radiolucencies of the jaw require enucleation: a case
of brown tumour
Srikanth Gangidi a,∗ , Robert Dyer b , David Cunliffe a
a Department of Oral and Maxillofacial Surgery, Torbay Hospital, Torbay, Devon, United Kingdom
b Department of Diabetes and Endocrinology, Torbay Hospital, Torbay, Devon, United Kingdom

Accepted 22 August 2011


Available online 13 September 2011

Abstract

We describe a case of brown tumour from primary hyperparathyroidism, which presented with radiolucency in the jaw. It was treated by
parathyroidectomy, which resulted in complete resolution of the lytic lesion without any surgery to the jaw. It is important to be aware of
endocrine causes of a common radiological sign to avoid unnecessary local surgical intervention.
© 2011 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

Keywords: Brown tumour; Primary hyperparathyroidism; Enucleation; Mandibular radiolucency; Hypercalcaemia

Introduction multinodular thyroid goitre, and generalised body aches that


interfered with mobility.
Radiolucencies of the jaw are common findings in maxillo- Oral examination showed an edentulous maxilla and
facial clinics. They are often of odontogenic origin, and are mandible, which were otherwise normal, but radiological
usually managed with enucleation. Rare causes of radiolu- imaging showed a radiolucency in the right mandibular body
cencies include brown tumour from hyperparathyroidism; that was unilocular and well defined, but not corticated
this diagnosis is important, as local surgical intervention such (Fig. 1). In view of her systemic symptoms and thyroid signs
as enucleation is not necessary. (thyroid disease is present in 40% of patients with primary
We describe such a case and highlight the importance of hyperparathyroidism) biochemical investigations were car-
taking a comprehensive history and focusing the investiga- ried out, which showed marked hypercalcaemia (corrected
tions so that appropriate treatment can be given. level 3.60 mmol/L, range 2.10–2.55), and associated hyper-
parathyroidism (parathyroid hormone (PTH) 62.8 pmol/L,
range 1.6–6.9).
A subsequent radiological skeletal survey showed lytic
Case report
lesions that affected the left glenoid fossa and lateral part of
the left iliac blade, which were in keeping with osteitis fibrosa
An 83-year-old woman presented to the maxillofacial depart-
cystica. Iliac trephine biopsy examination confirmed a giant
ment with mandibular pain beneath her lower denture.
cell tumour consistent with a brown tumour, and a consequent
Systematic history and examination showed a longstanding
sestamibi scan showed the presence of a functioning right
inferior parathyroid adenoma. In this context, together with
its radiological characteristics, the mandibular radiolucency

Corresponding author. Tel.: +01752 763202. was considered to be a brown tumour, so no further biopsy
E-mail addresses: sgangidi@nhs.net (S. Gangidi), robert.dyer@nhs.net
examination was carried out.
(R. Dyer), david.cunliffe@nhs.net (D. Cunliffe).

0266-4356/$ – see front matter © 2011 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
doi:10.1016/j.bjoms.2011.08.009
e34 S. Gangidi et al. / British Journal of Oral and Maxillofacial Surgery 50 (2012) e33–e35

Fig. 1. Orthopantogram showing a well-defined radiolucency consistent Fig. 3. Orthopantogram taken 6 months after the parathyroid adenoma had
with a brown tumour affecting the right mandibular body. been removed showing complete resolution of the radiolucency in the right
mandibular body with no local operative intervention.

can present with polyuria, polydipsia, and renal calculi,


as well as with tiredness, poor concentration, and gener-
alised body aches. Other causes of parathyroid-mediated
hypercalcaemia are primary hyperparathyroidism (parathy-
roid adenoma, parathyroid hyperplasia, and parathyroid
carcinoma), tertiary hyperparathyroidism, familial hypocal-
ciuric hypocalcaemia, and lithium therapy. Causes of
non-parathyroid-mediated hypercalcaemia are malignancy-
associated hypercalcaemia, granulomatous diseases (for
example, sarcoidosis and tuberculosis), endocrinopathies
(such as hyperthyroidism and adrenal insufficiency), drugs
(thiazides, calcium supplements, vitamin D), and immobili-
sation.
Hyperparathyroidism, the third commonest endocrine dis-
order after diabetes mellitus and thyroid disease, can appear
Fig. 2. Right inferior parathyroid adenoma. in primary, secondary, or tertiary forms. Primary hyper-
parathyroidism is the commonest, with a reported incidence
In accordance with National Institute of Health of 4/100 000/year.2 It has an overall prevalence of 2–3%,3
guidelines for the surgical management of primary affects women more than men between the ages of 50 and
hyperparathyroidism,1 our patient had right inferior parathy- 60 years, and in 40% of cases there is concurrent thyroid
roidectomy (Fig. 2) with simultaneous total thyroidectomy disease.4 Its most common cause is a single parathyroid ade-
for the multinodular goitre. Intraoperatively, PTH concentra- noma (80%). Other causes include double adenoma (5%),
tion (t1/2 = 3–5 min) taken 15 min after the adenoma had been multigland parathyroid hyperplasia (15%), and parathyroid
removed confirmed a level within the normal range, which carcinoma (less than 1%).5
indicated successful removal of the affected gland. Chronically raised PTH increases bony remodelling (with
Postoperatively, biochemical variables monitored accord- greater osteoclastic than osteoblastic effects), which leads to
ing to hospital protocol for thyroid and parathyroid surgery osteopenia and osteoporosis. These bony effects can lead to
showed mild hypocalcaemia, which was managed with cal- brown tumours, which present as lytic, focally well-defined
cium supplements. Diagnosis of parathyroid adenoma was lesions. In the jaws they are a rare cause of radiolucency
confirmed histologically. (roughly 1%).6
At 6-month review, the mandibular pain had resolved Mandibular radiolucencies are common in patients attend-
completely and the patient’s mobility had improved consid- ing maxillofacial clinics. Although the incidence of brown
erably. An orthopantogram showed complete regression of tumours from primary hyperparathyroidism is rare, this report
the mandibular radiolucency (Fig. 3). highlights the importance of taking a comprehensive history
to guide appropriate investigations and aid diagnosis.
If a brown tumour is suspected, an initial test to measure
Discussion calcium would first rule out hyperparathyroidism. If raised,
then a subsequent PTH test would indicate which differential
Hypercalcaemia from raised PTH is often chronic and is usu- group to investigate further.
ally found on routine biochemical testing, as patients are Our case also shows that after surgery to remove
commonly asymptomatic or minimally symptomatic. They the parathyroid adenoma, subsequent normalisation of the
S. Gangidi et al. / British Journal of Oral and Maxillofacial Surgery 50 (2012) e33–e35 e35

metabolism led to complete resolution of the brown tumour 3. Akerstrom G, Ljunghall S, Lungren E. Natural history of untreated pri-
without local operative intervention. mary hyperparathyroidism. In: Clark OH, Duh QY, editors. Textbook of
endocrine surgery. Philadelphia: Saunders; 1997. p. 303–10.
4. Cheng D, Jacob LA, Scoutt L. Parathyroid imaging. In: Oertli D, Udels-
man R, editors. Surgery of the thyroid and parathyroid glands. Berlin:
References Springer; 2007. p. 248.
5. Holt EH, Inzucchi SE. Physiology and pathophysiology of the parathyroid
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Endocrinol Metab 2002;87:5353–61. 6. Duran C, Ersoy C, Bolca N, et al. Brown tumors of the maxillary sinus
2. Wermers RA, Khosla S, Atkinson EJ, Hodgson SF, O’Fallon WM, and patella in a patient with primary hyperparathyroidism. The Endocri-
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