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Brown Tumor a Rare Presentation of Maxillary Cyst with Pathological Fracture of Femur OPEN ACCESS

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Abstract

Brown tumor is a giant cell lesion associated with hyperparathyroidism. It is a benign condition and represents the terminal stage of the remodeling process in the hyperparathyroid state. This severe parathyroid bone disease is a rare clinical presentation of primary hyperparathyroidism which is most often due to a parathyroid adenoma, secreting Parathormone (PTH). Elevated PTH levels cause bone resorption, the formation of polyostotic lesions and a reduction in bone mineral density, predisposing to pathological fractures [1]. Here we report the case of middle aged female having primary hyperthyroidism due to a parathyroid adenoma with osteolytic cystic lesions of the pelvis, the femur, and right maxillary bone. She was treated with surgical excision of the affected parathyroid gland, followed by open reduction and internal fixation with intra medullary nailing of the fractured femur.
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World Journal of Surgery and Surgical Research
2021 | Volume 4 | Article 1316
1
Brown Tumor a Rare Presentation of Maxillary Cyst with
Pathological Fracture of Femur
OPEN ACCESS
*Correspondence:
Aluru Jayakrishna Reddy, Department
of Surgical Gastroenterology, Lifeline
Institute of Minimal Access, No. 47/3
New Avadi Road, Kilpauk, Chennai
600010, India, Tel: 8754589739;
E-mail: doc_jk@yahoo.in
Received Date: 29 Jun 2021
Accepted Date: 16 Jul 2021
Published Date: 19 Jul 2021
Citation:
Reddy AJ, Shreya R, Anirudh R,
Rajkumar JS, Senthil Kumar S. Brown
Tumor a Rare Presentation of Maxillary
Cyst with Pathological Fracture of
Femur. World J Surg Surgical Res.
2021; 4: 1316.
Copyright © 2021 Aluru Jayakrishna
Reddy. This is an open access
article distributed under the Creative
Commons Attribution License, which
permits unrestricted use, distribution,
and reproduction in any medium,
provided the original work is properly
cited.
Case Report
Published: 19 Jul, 2021
Abstract
Brown tumor is a giant cell lesion associated with hyperparathyroidism. It is a benign condition
and represents the terminal stage of the remodeling process in the hyperparathyroid state. is
severe parathyroid bone disease is a rare clinical presentation of primary hyperparathyroidism
which is most oen due to a parathyroid adenoma, secreting Parathormone (PTH). Elevated PTH
levels cause bone resorption, the formation of polyostotic lesions and a reduction in bone mineral
density, predisposing to pathological fractures [1]. Here we report the case of middle aged female
having primary hyperthyroidism due to a parathyroid adenoma with osteolytic cystic lesions of the
pelvis, the femur, and right maxillary bone. She was treated with surgical excision of the aected
parathyroid gland, followed by open reduction and internal xation with intra medullary nailing of
the fractured femur.
Keywords: Primary hyperparathyroidism; Brown tumor; Excision
Aluru Jayakrishna Reddy*, Rajkumar Shreya, Rajkumar Anirudh, Rajkumar JS and Senthil
Kumar S
Department of Surgical Gastroenterology, Lifeline Institute of Minimal Access, India
Introduction
Primary hyperparathyroidism most oen is due to a parathyroid adenoma secreting PTH.
e severity of hypercalcemia is proportional to the size of the adenoma. In patients with primary
hyperparathyroidism, 85% are caused by solitary parathyroid adenoma, 13% have hyperplasia,
1% to 2% has double adenoma and 1% has carcinoma [2]. e PTH hypersecretion causes excess
calcium reabsorption from kidneys, phosphaturia, and increased vitamin D synthesis and bone
reabsorption. PTH increases osteoclastic activity which predisposes to pathologic fractures [1,2-4].
Accumulation of erythrocytes and their pigments give a reddish/brown hue to the lesions, hence
the name “brown tumor”. ese may be the rst sign of hyperparathyroidism [2]. In this report we
have discussed the rare presentation of a patient having parathyroid adenoma with maxillary cyst,
femoral pathological fracture and pelvic bone skeletal lesions. Brown tumors are more commonly
seen in the mandible than in the maxilla. e reported prevalence of brown tumors is 0.1%.
Case Presentation
A 53-year old female came with complaints of pain over the le thigh following trauma for the
past 2 weeks, with no co-morbidities. On examination: e right lower limb appeared externally
rotated with painful range of motion over the right hip joint, restricted mobility and a swelling
over the right maxilla which was rm in consistency (Figure 1). No nodule was palpable over the
neck. Other systems were normal. Laboratory analysis revealed serum calcium - 11.5 mg/dl, alkaline
phosphatase - 956 IU/l, phosphorus- 1.5 mg/dl, serum T3 – 3.43 ng/ml, serum T4 – 4.7 µg/dl, serum
TSH – 2.45 µIU/ml and serum parathyroid hormone level was 1900 pg/ml.
USG neck: A large heterogeneously hypoechoic lesion measuring 4.4 cm × 2.2 cm × 2.5 cm
with increased vascularity seen posterior to the right lobe of the thyroid - neoplastic lesion of the
parathyroid may be considered.
X-ray pelvis: Comminuted and displaced fracture seen involving the proximal 1/3rd of the right
femur (Figure 2).
CT pelvis: Diuse osteoporotic changes with thinning of the cortex and the trabeculae involving
all the pelvic bones and the visualized portion of both the femurs. Multiple mildly expansile lytic
lesions seen involving the le iliac bone, both the femurs in the proximal aspect and the transverse
process of L5 vertebrae on the right side-suggestive of brown tumor (Figure 3).
Aluru Jayakrishna Reddy, et al., World Journal of Surgery and Surgical Research - Surgical Gastroenterology
2021 | Volume 4 | Article 1316
2
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Surgical treatment
Findings: right superior parathyroid was well encapsulated
measuring about 5 cm × 3 cm which was displacing the thyroid
medially. Right recurrent laryngeal nerve was spared, and the
adenoma was excised into (Figure 4).
Repeat PTH levels: 14.9 pg/ml. Following this she was posted
for ORIF, with intramedullary nailing of the fractured femur 2 weeks
aer surgery. e patient was given oral calcium and vitamin D for
2 months. Histopathological report conrmed the diagnosis of a
parathyroid adenoma. We followed up the patient every month for 6
months, and there were no other complications till the last follow up.
Discussion
e prevalence of primary hyperparathyroidism may range from
1% to 4%, with a male:female ratio of 1:3. In most cases (around 80%)
a solitary adenoma is the cause and in 20% of cases, it is due to a
glandular hyperplasia. Serum calcium, ionized calcium and PTH levels
must be obtained to conrm the diagnosis [3,5]. Brown tumors are
relatively rare benign lesions, resulting in abnormal osteoclastic and
osteoblastic activity resulting in resorption of the bone. e incidence
of brown tumors in patients with primary hyperparathyroidism
is 1.5% to 1.7%. At present, hyperparathyroidism is usually treated
before such lesions develop; therefore they have become extremely
uncommon [6,7]. Radiological examination showed osteopenia of
the whole skeleton and multiple localized lytic lesions with a benign
aspect. Hyperparathyroidism aects mainly cortical bone. e bone
marrow in these cysts may be replaced by vascularized brous tissue
and giant cell reaction. Brown tumors represent foci of hemorrhage
within an enlarged brotic marrow space. Organization of these
lesions results in the release of hemosiderin and the accumulation of
macrophages, broblasts and giant cells. In our case the patient had
multiple cystic lesions in the skeleton [3]. Sestamibi scan, however, is
indicated if ectopic PTH producing adenoma is suspected or if the CT
scan and the USG failed in localizing the PTH producing lesion [3,8].
Aer parathyroidectomy, the serum PTH level falls dramatically, and
bone resorption stops, resulting in hungry bone syndrome [3,5,8].
Brown tumors are the late manifestation of hyperparathyroidism.
ey should only be removed if they persist even aer the removal of
the parathyroid adenoma, when functional problems are detected, or
if the tumors are too large. However the regression can take several
months.
Conclusion
Any patient suspected of having a pathological fracture should
undergo serum PTH level assessment, along with whole skeleton
screening. Parathyroidectomy is the treatment of choice, coupled with
other orthopedic interventions to address the pathological fractures.
References
1. Scott SN, Sato Y, Graham SM, Robinson RA. Brown tumor of the palate in
a patient with primary hyperparathyroidism. Ann Otol Rhinol Laryngol.
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2. Keyser JS, Postma GN. Brown tumor of the mandible. Am J Otolaryngol.
1996;17(6):407-10.
3. Patwa J, Patel K, Patel P, Patel N, akor P, akor P. Brown tumor: Rare
skeletal presentation secondary to hyperparathyroidism. Int J Biomed Adv
Res. 2015;6(2):169.
4. Usta A, Alhan E, Cinel A, Türkyılmaz S, Erem C. A 20-year study on
190 patients with primary hyperparathyroidism in a developing country:
Turkey experience. Int Sug. 2015;100(4)648-55.
5. Khan AA, Hanley DA, Rizzoli R, Bollerslev J, Young JEM, Rejnmark L,
et al. Primary hyperparathyroidism: Review and recommendations on
Figure 1: RT-Maxillary cyst.
Figure 2: RT-Femur fracture.
Figure 3: CT-Pelvis: Comminuted and displaced fracture seen involving
the proximal 1/3rd of the right femur with over-riding of the bony fragments-
suggestive of pathological fracture.
Figure 4: Intra operative picture, RT-para thyroid adenoma.
Aluru Jayakrishna Reddy, et al., World Journal of Surgery and Surgical Research - Surgical Gastroenterology
2021 | Volume 4 | Article 1316
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evaluation, diagnosis, and management. A Canadian and international
consensus. Osteoporos Int. 2017;28(1)1-19.
6. Som PM, Lawson W, Cohen BA. Giant-cell lesions of the facial bones.
1983;147(1):129-34.
7. Kar DK, Gupta SK, Agarwal A, Mishra SK. Brown tumor of the palate
and mandible in association with primary hyperparathyroidism. J Oral
Maxillofac Surg. 2001;59(11):1352-4.
8. Scheible W, Deutsch AL, Leopold GR. Parathyroid adenoma: accuracy of
preoperative localization by high-resolution real-time sonography. J Clin
Ultrasound. 1981;9(6)325-30.
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Article
Full-text available
The manifestation of primary hyperparathyroidism as skeletal disease has nearly disappeared in the last 2 decades. Cases are now most often diagnosed by the incidental finding of asymptomatic hypercalcemia. Improved screening techniques have made clinical evidence of bone disease uncommon. Classic radiographic findings of subperiosteal bone resorption, bone cysts, brown tumors, and generalized osteopenia now occur in fewer than 5% of cases.<SUP>1-3</SUP> Brown tumors may occur in the head and neck, with the mandible being the most frequent site. Involvement of the maxilla is exceptionally rare.<SUP>4,5 </SUP>A brown tumor most commonly presents as a slowly enlarging, painful mass. These tumors can be locally aggressive, but do not possess metastatic potential. Histologically, they are similar to giant cell lesions. Treatment initially focuses on correction of the hypercalcemic state. Brown tumors frequently show regression after normocalcemia is achieved, but surgical resection is necessary in some patients. We present a case of a brown tumor involving the hard palate and nasal cavity in a young woman.
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