Surgical Science, 2011, 2, 344-347
doi:10.4236/ss.2011.26074 Published Online August 2011 (
Copyright © 2011 SciRes. SS
Vertebral Brown Tumor in Childhood (Case Report)
Csenge Szeverényi1, Balázs Dezső2, Tamás Deményi3, Zoltán Csernátony1
1Department of Orthopedic Surgery, Debrecen, Hungary
2Departmen t o f Pathology, Debrecen, Hungary
3University of Debrecen Medical and Health Science Center, General Practitioner, Debrecen, Hungary
Received February 23 , 20 11; revised May 27, 2011; accepted June 9, 2011
One form of renal osteodystrophy secondary to chronic renal failure is the osteitis fibrosa cystica, which is
the complication of secondary hyperparathyroidism. Osteitis fibrosa cystica is a histologically benign, tu-
mor-like lesion of bones. Since early diagnosis and treatment of hyperparathyroidism is available nowadays,
the development of osteitis fibrosa cystica has become rare. It can only be found in neglected cases and in
those where the treatment of chronic renal failure is unsuccessful. We present the case of a boy, who deve-
loped osteitis fibrosa cystica as a complication of secondary hyperparathyroidism. Our article is the first to
report a case about an osteitis fibrosa cystica found in a child’s vertebra and this tumor was the only one,
which reached a size of approximately 10 × 15 × 15 cm. We present the CT films and X-ray pictures, the
surgical solution and the results of the histological examination.
Keywords: Vertebral Localization, Osteitis Fibrosa Cystica, Secondary Hyperparathyroidism, Chronic Renal
1. Introduction
Brown tumor (i.e. osteitis fibrosa cystica, osteoclastoma,
giant cell reparative granuloma) is a lytic bony lesion in
non-treated or uncontrollable hyperparathyroidism. Ostei-
tis fibrosa cystica may develop in primary and in secon-
dary hyperparathyroidism as well. The most frequent
cause of secondary hyperparathyroidism is chronic renal
failure. Since the early diagnosis and treatment of hy-
perparathyroidism are available nowadays, the occur-
rence of brown tumor became very rare [1-7].
Our main objective with this case report is to draw at-
tention to the possible occurrence of brown tumors in
rare locations throughout the body and to provide an in-
sight on the diverse manifestation and localizations of
these lesions and the comprehensive treatment of this
disease and its complications.
2. Materials and Methods
Case Report
The 9-year-old male was taken to the doctor because of
enuresis. His renal failure was diagnosed at that time.
The results of the kidney biopsy performed were unable
to clarify the cause of the disease since it was in an ad-
vanced stage. Histology revealed severe glomerular,
nodular sclerosis. Conservative therapy (calcium carbon-
ate, sodium bicarbonate) was started in order to treat the
renal failure and it was recommended to keep a strict diet.
However, regular haemodialysis became necessary two
years later. Elevated parathyroid hormone levels were
detected 6 months after diagnosing the child’s renal fail-
ure {PTH: 27.0 pmol/L (Reference range: 0.8 - 5.2 pmol/L;
Primary hyperparathyroidism: 45.0 - 58.6 pmol/L; chro-
nic renal failure: 0.9 - 418.0 pmol/L)}, which was the
result of his chronic renal failure. Parathyroid scintigra-
phy was performed four years later because of his ex-
tremely high PTH levels. This study excluded the possi-
bility of a parathyroid adenoma in the cervical region and
in the mediastinum as well. Cervical CT scan revealed a
persistent thymus and calcifications in the soft tissue in
the retrosternal region and near the spinal processes of
the vertebrae. Meanwhile, the patient was excluded from
the waiting list of renal transplantation candidates, be-
cause of his severe obesity. Subtotal parathyroidectomy
was performed four months later due to high PTH levels
(PTH: 188.5 pmol/L). Histopathology of the parathyroid
revealed hyperplasia. A few months later, the child was
admitted to our Department, in order to remove the large
(15 × 15 × 10 cm) tumor on the rear side of his neck
(Figure 1). The abnormality was detected a half year
before the operation for the first time and it was only 2 ×
1 × 3 cm large at that time.
Chest CT scan verified calcifications in the soft tissue
at the levels of the C7-Th4 vertebrae in the deep back
muscles of the left paravertebral region, the destruction
and calcification of the spinous process of the 7th cervi-
cal vertebra and a 10x15x15 cm large cystic tumor at the
level of C1-Th2 vertebrae, whose caudal part was calci-
fied (Figure 1).
During the surgery the cervical cyst was prepared and
removed with multiple cysts and solid tumors spreading
into the muscles of the back. There was no communica-
tion between the cyst and the liquor space, but the cyst
contained thick, white-yellowish fluid (Figure 2).
3. Results
According to the histological findings, the removed tu-
mor was diagnosed as osteitis fibrosa cystica, which was
the complication of the chronic hyperparathyroidism
(Figure 3).
On the basis of the imagery the tumor’s origin was in
the spinous process of the 7th cervical vertebra and
starting from that location, it invaded the nearby soft
tissues. Despite of the parathyroidectomy performed 4
months earlier, the child’s PTH serum level remained
Figure 1. The tumor on the neck of the child grew to 15 × 5
× 10 cm large in half a year. On the chest CT scan image,
the deformity and calcification of the spinous process of the
7th cervical vertebra and the cystic tumor are visible.
Figure 2. During the surgical intervention, the cervical cyst
was removed with the other cystic and solid tumors
spreading into the muscles of the back.
Figure 3. Upper left section: the histopathology of the he-
matoxylin-eosin (HE) stain tissue section clearly shows that
the wall of the cyst is not covered by epithelium, therefore
the cyst is actually a pseudocyst. The lumen is covered by
necrotic debris with inflammatory cells, mainly macro-
phages (original magnification: 10x). Upper right section:
immunohistochemistry (IHC) for CD68 identifies macro-
phages (cells with brown membrane staining) including
foreign body giant macrophages (arrows). Note that osteo-
clast type of giant cells are CD68 negative (arrowheads).
(IHC with hematoxylin nuclear counter staining; original
magnification: 20x). Lower left section: Berlin blue staining
reveals the accumulation of hemosiderin laden macro-
phages (original magnification: 10x). Lower right section:
despite the huge size of the tumor, some remnant bone tra-
beculae (arrows) can be discovered in the histological sec-
tions. The big dark-blue (basophilic) precipitates corre-
spond to dystrophic calcifications (original magnification:
4. Discussion
In view of the fact that in chronic renal failure the matrix
of the kidneys shrinks, the production of the active vita-
min D will decrease, which has a primary etiological role
in the development of the brown tumor. Vitamin D fa-
cilitates the absorption of calcium from the intestines,
and it inhibits phosphate reabsorption in the kidneys.
Low levels of vitamin D lead to hypocalcaemia and
phosphate retention, which stimulate parathyroid hor-
mone secretion [4-6].
Parathyroid hormone facilitates the differentiation of
osteoclasts and these cells increase the serum calcium
level by bone resorption. In osteitis fibrosa cystica, reac-
tive fibrosis of the bone marrow can also be detected in
addition to the bone resorption caused by the activity of
the osteoclasts. The focal bone resorption and the pro-
gression of the fibrosis result in macroscopically visible
cysts, which build up the brown tumor [3,4,7-10 ]. During
the tumor growth the bone may become weak resulting
in pathologic fractures or spread into the soft tissues
Copyright © 2011 SciRes. SS
causing compressive symptoms [4,6]. Furthermore, as in
our case, the tumor has a potential to become so large
that it makes everyday life difficult.
PTH also has an effect on the kidneys as it increases
phosphate excretion. Therefore, hypersecretion of PTH
can be inhibited by eliminating phosphate excess. It can
be achieved by oral antacids, dietetic restrictions and
peritoneal dialysis or haemodialysis [4,10,11,12]. How-
ever, after a certain period of time, the enlarged parathy-
roid gland might not react to the fluctuant serum calcium
level, therefore some degree of autonomous glandular
function develops [5,13]. When this occurs, conservative
treatment cannot prevent progressive bone resorption,
hypercalcaemia and metastatic calcifications anymore. In
these cases subtotal parathyroidectomy may be effective,
although secondary hyperparathyroidism might recur,
due to the presence of any residual parathyroid gland
tissue [4]. In our case, even though parathyroidectomy
was performed, serum PTH levels remained high (PTH:
206.1 pmol/L). This arose the suspicion the presence of
an autonomous functioning, ectopic parathyroid tissue in
the persistent thymus or somewhere else in the body,
causing hypersecretion of PTH.
Based on the literature reviewed, the incidence of
brown tumor involving bones is between 1.5% - 13%
among patients suffering from chronic renal failure [4,6,
7]. Furthermore, the lesion develops most frequently in
the pelvic bones, costae, sternum, maxillary sinus, man-
dible and in the femur [3,4,7,8,12,13]. The presence of
brown tumor in the vertebral column is rare. After re-
viewing the literature since 1974, only 19 similar cases
were published, with 8 patients having primary and 11
having secondary hyperparathyroidism as the underlying
cause of the disease [1,4-7,12,14]. We did not find a sin-
gle case of brown tumor developing from a vertebra of a
child and reaching such an enormous size. The signifi-
cance of osteitis fibrosa cystica involving th e verteb rae is
that it may cause the pathologic fractures of the vertebrae
or it may invade the spinal canal causing the compres-
sion of the spinal cord [1,4-7,12,14].
5. Conclusions
Our publication focused on the diagnosis, adequate
treatment and the prevention of possible complications of
brown tumors, which manifested as a divers multi-local
disease in our case. Brown tumor is a complication of
chronic renal failure and even though brown tumor has
become very rare nowadays, one has to think of it in pa-
tients suffering from chronic renal failure, and especially
in those cases in which the metabolic balance cannot be
achieved by conservative methods. In certain cases sur-
gical excision or spine-stabilizing intervention may be-
come necessary due to the localization or the size of the
tumor. In our case, finding and removing the suspected
ectopic parathyroid tissue and completing the kidney
transplantation may bring significan t improvement to the
patient’s state of health.
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