CASE STUDY

19year-old male was referred for consultation regarding large swellings of his mandible and maxilla. The
patient's medical history disclosed that he had had chronic renal failure for about 10years and was on a
regular haemodialysis programme three times a week for the past 9years. 3years 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 12years 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 . 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. Both of the lesions were non-tender and firm on palpation. They appeared to be attached to the bone;
the overlying 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 1.35meters/4′4.4″
and weight 25.5kg/56.1lbs; body mass index: 14.4kgm−2). The patient complained of generalized weakness
and difficulty in performing daily domestic work. A deficiency of secondary male sex characteristics, 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 .The maxillary lesion was not clearly visible on
the panoramic radiograph, but upon close observation of the radiograph, another radiolucent 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 radiolucent lesions
According to the CT scans, the right mandibular lesion measured 3.45×5.45×3.46cm. The CT scan also
revealed that the maxillary lesion (measuring 1.28×1.46×1.36cm) had infiltrated into the maxillary sinus .
The laboratory findings were as follows: intact PTH 2415pgml−1 (15–65pgml−1), calcium 11.0mgdl−1 (8.5–
10.5mgdl−1), phosphate 6.7mgdl−1 (2.7–4.5mgdl−1), alkaline phosphatase 1270 UL−1 (90-260L−1).
On parathyroid ultrasonography, bilateral thyroid lobes showed no enlargement, whereas a hypoechogenic
solid lesion 6.3×10mm in size was observed in the left thyroid lobe's posteroinferior localization . 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 8.8×6.7mm and (ii) in the
inferomedial of the latter, measuring 9.7×17.3mm.
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 .
Fine needle aspiration biopsies were then performed on the mandibular and maxillary lesions. On microscopic
examination, many multinucleated giant cells arranged in groups adjacent to haemosiderin granules within a
fibrovascular haemorrhagic stroma were observed
picture below: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
whats ur diagnosis

based on the thorough diagnostic work-up including medical history, clinical manifestations, radiographic
findings and consecutive routine laboratory findings, the patient was diagnosed as having tertiary
hyperparathyroidism( HPT)with brown tumours(osteoclastomas) of both jaws as a result of long-term renal
disease.
Osteitis fibrosa cystica is a late manifestation 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
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. Brown tumour is actually a giant cell lesion and
often appears as an expansile osteolytic lesion of the bone.
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). On clinical examination and using
only routine panoramic radiography, the lesions may resemble osteosarcoma, bone metastases of a
carcinoma, multiple myeloma, Langerhan's cell histiocytosis, 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.
Secondary and tertiary HPT are mostly seen in patients with chronic renal disease. Secondary HPT usually
affects older adults (50–80years) with a 2:1 female predominance, 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.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.
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.
. 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.Systemic corticosteroids can be used to reduce the size of the lesion; sometimes intralesional
corticosteroid injections also give satisfactory results.
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.
With the removal of the autonomous parathyroid glands, the PTH level would eventually return to normal, thus
leading to a spontaneous decrease in the sizes of the intraoral lesions within a few months. Resection of the
right mandibular lesion would enhance the nutrition of the patient, resulting in an improvement in his systemic
condition. In the long run his renal insufficiency must be treated, probably by means of a renal transplant, to
prevent recurrent hyperparathyroidism.
In conclusion, this dramatic entity therefore highlights the importance of early diagnosis of hyperparathyroi
dism with a thorough diagnostic work-up. Panoramic radiographs, CT scans, ultrasonography, and
parathyroid scintiscan with 99Tcm sestamibi were all useful radiographic methods for the correct diagnosis
of this tumour. This case should attract the attention of general practitioner dentists, oral and maxillofacial
surgeons, endocrinologists and radiologists whose consultation may be vital for patients with hyperparathyroi
dism 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.