Download Knee brown tumor revealing a primary hyperparathyroidism: a case

Current practice
Ann Biol Clin 2014; 72 (2): 245-8
Knee brown tumor revealing a primary
hyperparathyroidism: a case report
Copyright © 2017 John Libbey Eurotext. Téléchargé par un robot venant de 88.99.165.207 le 17/06/2017.
Tumeur brune du genou révélant une hyperparathyroïdie primitive :
à propos d’un cas
Aoulia Dekaken1
Adel Gouri2
Ahmed Aimen Bentorki1
Amina Yakhlef3
1 Department of internal medicine and
cardiology, El Okbi Hospital, Guelma,
Algeria
<[email protected]>
2 Laboratory of medical biochemistry,
IBN Zohr Hospital, Guelma, Algeria
Abstract. Primary hyperparathyroidism is a common endocrine disorder,
asymptomatic and diagnosed through a fortuitous hypercalcemia. Brown tumors
are exceptional but severe hyperparathyroidism bone complications. We report
in this paper an original observation of hyperparathyroidism due to a parathyroid adenoma presenting as a brown tumor. A 28 year-old girl admitted for
a bone tumor of the knee, the blood test shows hypercalcemia with hyperparathyroidism, bone biopsy revealed giant cell lesions characteristic of brown
tumors.
Key words: hyperparathyroidism, brown tumor, giant-cell tumor
3
Department of hematology, IBN Zohr
Hospital, Guelma, Algeria
doi:10.1684/abc.2014.0934
Article received November 07, 2013,
accepted November 12, 2013
Résumé. L’hyperparathyroïdie primitive est une endocrinopathie fréquente,
le plus souvent asymptomatique et diagnostiquée à l’occasion d’une hypercalcémie de découverte fortuite. Les tumeurs brunes sont des complications
osseuses exceptionnelles mais sévères de l’hyperparathyroïdie. Nous rapportons ici une découverte originale d’une hyperparathyroïdie due à un adénome
parathyroïdien devant une tumeur brune. Il s’agit d’une femme âgée de 28
ans admise pour une tumeur osseuse du genou, l’examen biologique montre
une hypercalcémie avec une hyperparathyroïdie, la biopsie osseuse révèle des
lésions osseuses à cellule géantes caractéristiques des tumeurs brunes.
Mots clés : hyperparathyroïdie, tumeur brune, tumeur à cellule géante
Primary hyperparathyroidism (PHP) is a common
endocrine disorder characterized by an excessive secretion
of parathyroid hormone (PTH) and consequent hypercalcaemia [1]. The frequency of bone disease has been reported
to be around 10-15%. Brown tumor is a rare aspect of its
manifestations (2 -3%) and is exceptionally revealing of the
disease [2-4].
Brown tumor is not neoplastic bone lesion caused by rapid
osteoclastic activity due to hyperparathyroidism (HPT)
resulting in a locally destructive phenomenon. In regions
where bone loss is rapid, hemorrhage, hemosiderin deposition, and vascularized fibrous tissue replace the normal
bone contents, resulting in a reddish-brown appearance [5].
Common sites of brown tumor are the ribs, clavicle, tibia,
femur and pelvic girdle.
We report an unusual case in which primary hyperparathyroidism was diagnosed upon investigation of
knee bone manifestations caused by brown tumor. We
also summarize several critical radiological and clinical points that are easily missed and should be kept in
mind.
Case report
A 28-year-old woman married and mother of four children
was admitted at the internal medicine department exploring
a bone tumor discovered through radiographs. The patient
had a history of pain in the left knee for several months
prompted radiographs of the knees, which showed lytic
lesions (figure 1). Here medical history was unremarkable.
At admission, mild asthenia and polydipsia-polyuria
syndrome were noted. Mechanical diffuse pain, lower
extremity motor weakness and arthritis of the left knee were
the only musculoskeletal abnormalities. No other significant clinical abnormality was observed.
To cite this article: Dekaken A, Gouri A, Bentorki AA, Yakhlef A. Knee brown tumor revealed primary hyperparathyroidism: a case report. Ann Biol Clin 2014; 72(2):
245-8 doi:10.1684/abc.2014.0934
245
Copyright © 2017 John Libbey Eurotext. Téléchargé par un robot venant de 88.99.165.207 le 17/06/2017.
Current practice
B. Plain radiograph of femur
llustrate abnormal bony architecture in lateral
femoral condyle.
A. Plain radiograph of knee joint
llustrate abnormal bony architecture in patella
and tibial tuberosity.
Figure 1. Radiographs of the left knee. A. Plain radiograph of knee joint (illustrate abnormal bony architecture in patella and tibial tuberosity).
B. Plain radiograph of femur (Illustrate abnormal bony architecture in lateral femoral condyle).
Laboratory analysis showed an elevated serum calcium
level of 3.04 mmol/L (reference range, 2.15-2.50 mmol/L)
and a low serum phosphate level of 0.63 mmol/L (reference
range: 0.77-1.32 mg/L), and normal tests for inflammation.
Urinary phosphate excretion was increased to 26 mmol/24h
(reference range: 10 à 20 mmol/24h) and urinary calcium
excretion to 10 mmol/24 h (reference range, 3.5 à 7.5
mmol/24h).The serum alkaline phosphatase level was 890
IU/L (reference range, 50-240 IU/L). Subsequent testing
revealed normal renal function tests and a high intact PTH
level of 137 pmol/L (reference range: 1.2-5.8 pmol/L),
confirming primary hyperparathyroidism. Both liver and
thyroid function were normal, serology for viral hepatitis
B and C was negative (table 1).
The radiographs of the left knee showed the previously
noted lytic lesions, as well as articular chondrocalcinosis,
which were subsequently found to be diffuse.
The parathyroid scintigraphy (Tc99 m sestamibi) clearly
locates a parathyroid adenoma (30 × 16 mm) in the lower
pole of the right lobe of the thyroid gland. Computed tomography (CT) scan of the left knee disclosed three osteolytic
lesions (lateral femoral condyle, patella and tibial tuberosity).
246
Table 1. Biological data on admission.
Laboratory exam
Hematocrit (%)
Erythrocyte sedimentation rate (mm)
Albumin (g/L)
Fasting blood sugar (mmol/L)
Total cholesterol (mmol/L)
Triglycerides (mmol/L)
Total bilirubin (␮mol/L)
Aspartate aminotransferase (UI/L)
Alanine aminotransferase (UI/L)
Alkaline phosphatase (UI/L)
Lactate dehydrogenase
Serum calcium (mmol/L)
Serum phosphate (mmol/L)
Urinary calcium (mmol/24 h)
Urinary phosphate (mmol/24 h)
Creatinine (␮mol/L)
Urea (mmol/L)
Parathyroid hormone (pmol/L)
Thyroid-stimulating hormone (mIU/L)
Free triiodothyronine (pmol/L)
Free thyroxine (pmol/L)
Result
(reference ranges)
34% (31-36)
15 (<20)
42 (35-50)
5,6 (3.3-6.1)
4,6 (3.4-6.4)
1.2 (0.4-1.9)
7 (1-12)
21 (5-45)
16 (5-45)
890 (50-240)
320 (230-460)
3.03 (2.1-2.6)
0.63 (0.80-1.61)
10 (3.5-7.5)
26 (10-20)
68 (53-97)
5.20 (2.49-7.49)
137 (1.2-5.8)
1.58 (0.5-4.70)
4.23 (3.5-6.5)
15.12 (10-23)
Ann Biol Clin, vol. 72, n◦ 2, mars-avril 2014
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Knee brown tumor and hyperparathyroidism
To arrive at conclusive diagnosis, pathological analysis
was performed. Biopsy specimens were obtained from
different zones of the lesion and at different depths.
Histopathological studies revealed the presence of a giant
cell granuloma with microscopic features suggest hyperparathyroidism.
The envisaged diagnosis was primary hyperparathyroidism
with brown tumor on the above of laboratory data and
characteristic histopathologic findings. Subtotal parathyroidectomia was performed and improvement in the
patient’s clinical condition and radiological findings was
marked in few months.
tests including PTH level will differentiate primary hyperparathyroidism from other giant cell lesions which have
similar histological appearances such as giant cell granuloma, aneurysmal bone cyst, cherubism, and giant cell
tumor [11].
The management of a brown tumor depends on the severity
of the lesion present and treatment of hyperparathyroidism
is the first step. Parathyroidectomy is the treatment of
choice, various studies have demonstrated regression of
skeletal and normalization of the biochemical aspects of
the disease promptly [12, 13].
Conclusion
Discussion
Primary hyperparathyroidism is an endocrine disease that
results from autonomous overproduction of the parathyroid
hormone; it is the third most common endocrine disorder
after diabetes and thyroid disorder [6, 7].
This case report illustrates the classic but rarely inaugural
bone manifestations of primary hyperparathyroidism. Due
to the improved laboratory methods and clinical practice in
the last 2 decades, most of the recent cases of primary hyperparathyroidism are diagnosed early and asymptomatically.
This makes advanced disease with bone lesions extremely
rare these days. Only 2% of all cases of primary hyperparathyroidism occur below the age of 30 years at the time
of diagnosis [8]. The patient reported here is in this rare
group because she was only 28 years old at the time of
diagnosis and the extent of her disease suggested that it had
been present for some time.
Increased levels of serum calcium and parathyroid hormones and reduced levels of serum phosphate as well
as increased urinary levels of phosphates and calcium
observed in this patient were sufficient to diagnose primary
hyperparathyroidism. Ultrasound, CT scan, or technetium
scan techniques can be used also to detect the diseased
parathyroid gland.
Brown tumors represent a serious complication of advanced
hyperparathyroidism with a frequency of 3-4% in primary
hyperparathyroidism [9]. It affects usually young people
especially females with varying degrees of aggressiveness
and risks of recurrence. The usual sites of these lesions
are the ribs, clavicles, pelvic girdle and facial bones and the
presentation may be a pathological fracture [10]. The lower
limb is rarely affected.
In our case the tumor is involving the left knee articulation; lateral femoral condyle, patella and tibial tuberosity.
The diagnosis of a brown tumor is the result of the sum of
the medical history of patients with clinical, radiographic,
laboratory findings and histological examination. Incisional
biopsy will show a giant cell lesion, and only biochemical
Ann Biol Clin, vol. 72, n◦ 2, mars-avril 2014
This case should attract the attention of clinical practitioner that brown tumor should be kept in mind during
daily practice and in the case of hypercalcemia and
radiographic evidence of multiple lytic lesion, primary
hyperparathyroidism should always be kept in differential
diagnosis and should be looked into once more common
causes.
Conflicts of interest:
none.
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