scholarly journals A Patient With Femoral Osteitis Fibrosa Cystica Due To Hyperparathyroidism: A Case Report

Author(s):  
Xiaolong Xu ◽  
Cuiping Yang ◽  
Shengjun Lu ◽  
Hong Pei ◽  
Shunguang Chen ◽  
...  

Abstract Background: Osteitis fibrosa cystica is a rare benign, lytic bone lesion attributed to hyperparathyroidism. The high level of parathyroid hormone cause rapid bone loss.Case presentation: The patient is a 50-year-old male complaining of severe sustained pain of the right knee joint. Imaging studies were suspicious for a benign tumor of the right distal femur. Biopsy under CT guidance showed numerous osteoclast aggregation and hemosiderin deposition around the bone trabeculae. Blood tests disclosed significantly elevated parathyroid hormone, serum calcium, serum alkaline phosphatase. Parathyroid ultrasonography and CT scan showed a solid mass in front of the trachea at the thoracic entrance plane. After resection of the mass, the clinical symptoms were relieved and the radiological findings were significantly improved, which further confirmed the diagnosis.Conclusions: Metabolic diseases-associated bone lesions require a comprehensive diagnosis of multiple inspection items.An interprofessional team approach to the diagnosis and treatment of Osteitis fibrosa cystica will provide the best outcome.

2021 ◽  
Vol 22 (1) ◽  
Author(s):  
Maya L. Nasser ◽  
Serge Medawar ◽  
Tonine Younan ◽  
Halim Abboud ◽  
Viviane Trak-Smayra

Abstract Background We report a case of osteitis fibrosa cystica, a rare benign resorptive bone lesion caused by hyperparathyroidism, that presented on imaging as an aggressive bone tumor. Case presentation The patient is a 51-year-old male complaining of severe sustained pain of the right hip region. Imaging studies were suspicious for a malignant tumor of the right iliac bone. Biopsy under CT guidance was performed and showed remodeled bone trabeculae with numerous osteoclasts, excluding bone tumor and raising the possibility of osteitis fibrosa cystica. Complementary tests disclosed elevated blood level of parathyroid hormone and a partially cystic enlarged left inferior parathyroid gland consistent with adenoma. After parathyroidectomy, the clinical symptoms were relieved and the radiological findings were significantly improved, which confirmed the diagnosis. Conclusions Metabolic diseases-associated bone lesions should always be considered in the differential diagnosis of bone tumors, to avoid unnecessary surgeries and treatments.


2021 ◽  
Vol 5 (Supplement_1) ◽  
pp. A207-A208
Author(s):  
Patricia Luengo Pierrard ◽  
Laura M Tortolero Giamate ◽  
Belén Porrero Guerrero ◽  
Joaquín Gómez Ramírez ◽  
Jordi Nuñez Nuñez

Abstract Introduction: Osteitis fibrosa cystica (OFC) is the most serious bone involvement of primary hyperparathyroidism (PHPT), it is characterized by subperiosteal resorption, lytic lesions and the appearance of brown tumors; this is why, in some cases, this condition can easily be mistaken for a malignant neoplasm. Its prevalence in developed countries is only 5%. Clinical Case: We present a 58-year-old woman, with no relevant personal history, who came to the emergency room with pain in her right shoulder after an accidental fall on the bus. The humerus radiograph shows a pathological fracture of the right humerus, with significant osteopenia. In the emergency analysis, serum Calcium 13.3 mg / d), Ionic Calcium 7.03 mg / dL, Phosphorus 2.4 mg / dL, Alkaline Phosphatase 248 U / L and normal kidney function stand out. With a diagnosis of severe hypercalcemia, treatment was started in the emergency room with serum therapy (1000 ml of 0.9% physiological saline in 4 hours) and intravenous diuretic treatment (furosemide 40mg) with a decrease in calcemia to 12.8mg / dL. Later, she was admitted to the Internal Medicine hospital ward to perform a differential diagnosis of hypercalcemia secondary to a primary tumor, Multiple Myeloma or Primary Hyperparathyroidism. The study findings are: Calcium metabolism: PTH 660 pg / ml (12 - 65), 25 Hydroxyvitamin D: 14.00. Thyroid ultrasound: Posterocaudal to right thyroid lobe, an area of ​​echogenicity slightly lower than the thyroid is identified, of dimensions not estimated by endothoracic component, which could correspond to a parathyroid adenoma. Body CT: Neck: Heterogeneous nodule dependent on the posterior region of the right thyroid nodule with endothoracic extension. Skeleton: Lytic lesions with a tumor aspect in the humerus, right scapula and bilateral seventh rib and right pubic branch. Skull: Diffuse increase in bone density of the calvaria, showing multiple punctate lytic lesions with a permeative appearance. Bone densitometry: Femur neck: - <1.5, Lumbar spine: - <3.0 With the diagnosis of PHPT causing osteitis fibrosa cystica, surgical intervention was decided. Under general anesthesia, a selective right approach was performed, finding a large parathyroid adenoma weighing 17 grams. PTH fell to 36 pg / ml after surgery. At 9 months after surgery, the patient presented calcium levels of 9 mg / dl and PTH 146 pg / ml with clear radiological improvement. Discussion: Osteitis fibrosa cystica is rare in our environment, it is often confused with other neoplasms. After parathyroidectomy, patients with PHPT have a marked and sustained recovery from OFC, although in some cases this recovery can only be achieved after several years. We consider this case of interest, since it illustrates the importance of evaluating the study of phospho-calcium metabolism and parathyroid function in all patients with bone lesions to rule out Primary Hyperparathyroidism with OFC.


1998 ◽  
Vol 116 (5) ◽  
pp. 1790-1797 ◽  
Author(s):  
Maria Eugênia Leite Duarte ◽  
Ana Lúcia Passos Peixoto ◽  
Andréa da Silva Pacheco ◽  
Angela Vieira Peixoto ◽  
Rodrigo Dezerto Rodriguez ◽  
...  

INTRODUCTION: Renal osteodystrophy includes the complete range of mineral metabolism disorders that affect the skeleton in patients with chronic renal failure. PATIENTS AND METHODS: 200 patients with end-stage renal disease and on dialysis were investigated regarding the clinical, biochemical and histological findings of bone disease. RESULTS: The spectrum of renal osteodystrophy consisted mainly of high turnover bone lesions (74.5%), including osteitis fibrosa in 57.5%. Patients with mild bone disease were on dialysis for shorter periods of time and were mostly asymptomatic. Patients with aluminum-related bone disease (16.5%) had the greatest aluminum exposure, either orally or parenterally, and together with patients with high turnover mixed disease, were the most symptomatic. Although on a non-regular basis, the vast majority of the patients (82.5%) had been receiving vitamin D. The incidence of adynamic bone disease was high (n=8) among parathyroidectomized patients (n=12). Significantly higher serum levels of alkaline phosphatase were observed in osteitis fibrosa. CONCLUSIONS: The use of calcitriol and phosphate-binding agents on a non-regular basis seems to be the reason for the apparent reduced response to the treatment of secondary hyperparathyroidism. Alkaline phosphatase has been shown to be a fair marker for bone turnover in patients with osteitis fibrosa. The severity of the clinical manifestations of bone disease correlates with the histological features of bone lesion and to the time spent on dialysis.


2021 ◽  
Vol 11 (12) ◽  
pp. 128-133
Author(s):  
Hela Zouaghi ◽  
Dorsaf Touil ◽  
Raouaa Belkacem Chebil

The diagnosis of osteolytic lesions of the jaws can be challenging. Case Reports: Two cases of brown tumor of hyperparathyroidism were reported. A 76- year-old female patient presented with indolent swelling of her right lower jaw measuring approximately 5 cm /6 cm. The panoramic radiograph showed a well-defined osteolytic radiolucency involving the entire mandibular symphysis. Blood investigations revealed High level of parathyroid Hormone (PTH): 102pg/ml. The diagnosis of a brown tumor of hyperparathyroidism was suspected. A parathyroid technetium scintiscan revealed abnormally high uptake at the lower pole of the thyroid lobe interpreted as hyperplasia of right inferior parathyroid gland with possible brown tumor of the mandible. Second case: A 36- year-old female patient presented for the replacement of her missing teeth. Her medical history revealed chronic renal failure and a recent surgical excision of an Osteitis fibrosa cystica of her fifth left proximal phalange. Panoramic radiograph showed multiple well defined osteolytic lesions of the mandible. The diagnosis of a brown tumor of the mandible secondary to hyperparathyroidism was suspected. Laboratory investigations showed increased PTH level, serum hypocalcemia and hyperphosphatemia and vitamin D deficiency. The patient was referred to the department of endocrinology for further investigation and the correction of PTH level. At Six months follow up all the lesions disappeared on radiological control. Discussion: Brown tumor of hyperparathyroidism is a metabolic disorder causing bone resorption that can affect the jaw bones. Clinical symptoms depend on the size and the location of the lesion. Radiographically, it appears as radiolucent unique or multiple well-defined intra-osseous radiolucency. Biological examination is the key to the diagnosis and it is marked by high level of parathyroid hormone (PTH). Key words: Jaw, Tumors, Osteitis Fibrosa Cystica, Hyperparathyroidism, Diagnosis.


2020 ◽  
Vol 4 (Supplement_1) ◽  
Author(s):  
Suruchi K Gupta ◽  
Runhua Hou ◽  
Harold Rosen

Abstract Background: Brown tumors are a part of the complex “osteitis fibrosa cystica” which is a diffuse resorptive process of the bone resulting from uncontrolled hyperparathyroidism. Although these brown tumors were fairly common in the past1, the incidence of brown tumors is now extremely rare in the United States due to early diagnosis and treatment of hyperparathyroidism. Here we describe an unusual presentation for osteitis fibrosa cystica. Clinical Case: A 38 year old woman was admitted to the hospital with rapidly growing facial swelling in June 2019. The swelling initially appeared in January 2019 and had rapidly increased in size since April 2019. The mass was malodorous, painful and interfered with oral intake. The patient smoked 1 pack per day for 13 years. A CT scan of the head and neck with contrast showed a 3.6 x 4.5 x 3.3 cm mass destroying the right mandibular body, involving the right platysma muscle and displacing the central and lateral incisor teeth and was concerning for malignancy. The initial impression was that this was likely a squamous cell carcinoma with local and perhaps distant metastases, complicated by humoral hypercalcemia of malignancy and she was admitted for management. Upon admission, her calcium level was 14 mg/dL, albumin 4.1 g/dL, phosphorus 1.4 mg/dL PTH level was 890 pg/mL and vitamin D level was 22 ng/mL. Since the high PTH did not fit with hypercalcemia of malignancy, further evaluation was undertaken. A thyroid ultrasound showed a 4.6 x 1.8 x 1.5 cm isoechoic, heterogeneous lesion in the left lower pole, and Tc-99 sestamibi scan suggested parathyroid origin. The biopsy of the mouth lesion showed fibro histiocytic proliferation with multiple giant cells, negative for malignancy and consistent with a brown tumor. CT scan of the chest, abdomen and pelvis did not reveal any other masses or lytic lesions. . A skeletal survey showed another 2 cm lytic lesion in the proximal left humeral metaphysis which was not biopsied. On parathyroidectomy, the patient was found to have an enlarged left inferior parathyroid gland measuring 3.3 x 3.0 x 0.8 cm weighing 7.093 grams. Intraoperatively PTH level decreased from 890 pg/mL to 69 pg/mL. Her calcium levels returned to normal post-operatively and she was discharged home. Pathologic examination report revealed the mass to be a parathyroid adenoma. The patient reports a reduction in the size of her mandible mass since the surgery. Conclusion: Although uncommon, brown tumors can be seen in uncontrolled primary hyperparathyroidism. The mandible is a common site, though, as in this case, it is less common for it to be the only site affected Reference: 1. Rosenberg, E. H. (1962). Hyperparathyroidism. A review of 220 proved cases with special emphasis on findings in the jaws. Oral Surg Oral Med Oral Pathol Oral Radiol Endod, 15(2), 84–94.


2021 ◽  
Vol 21 (1) ◽  
Author(s):  
Shuai Lu ◽  
Maoqi Gong ◽  
Yejun Zha ◽  
Aimin Cui ◽  
Wei Tian ◽  
...  

Abstract Background Multiple pathological manifestations are rarely present in patients with primary hyperparathyroidism (PHPT). Here we described a case of a young woman who presented with multiple skeletal destructions and received an unclear diagnosis at several hospitals. Case presentation A 30-year-old woman was admitted to our hospital due to pain in both knees and walking difficulty that lasted for 6 and 2 years, respectively. Her laboratory test results revealed a high parathyroid hormone level (822 pg/ml) and hypercalcemia (2.52 mmol/L) in the blood. Parathyroid imaging revealed a lumpy concentration of radioactive uptake detected at the lower pole in the right lobe of the thyroid, and was nearly 2.2 cm * 2.4 cm in size. Next, the patient was treated with parathyroidectomy that resulted in a significant improvement in physiological and clinical symptoms. Moreover, the skeletal destruction and bone mineral density were significantly improved after a 5-years follow-up period. Conclusions Multiple skeletal destructions can be caused by PHPT that should be taken into consideration in young patients with complex bone lesions.


2020 ◽  
Vol 26 (9) ◽  
pp. 983-989
Author(s):  
Elena Castellano ◽  
Roberto Attanasio ◽  
Alberto Boriano ◽  
Valentina Borretta ◽  
Marta Gennaro ◽  
...  

Objective: Clinically overt symptomatic bone disease in primary hyperparathyroidism (PHPT) is rarely seen today, and osteoporosis is the dominant finding. Subperiosteal bone resorption in the fingers and skull mottling are typical bone PHPT findings, the contemporary prevalence of which is unknown. We evaluated these mild lesions and investigated the impact of their occurrence on PHPT clinical management. Methods: We evaluated retrospectively a monocentric series of 363 PHPT patients classified in Group 1 (n = 100) or Group 2 (n = 263) according to the presence or absence of bone involvement, respectively. Patients belonging to Group 1, in turn, were subdivided into Group 1A, with severe and symptomatic lesions (n = 48), and Group 1B, with milder signs of osteitis fibrosa cystica (OFC) without brown tumors or fractures (n = 52). Results: Serum total and ionized calcium, parathyroid hormone, osteocalcin, alkaline phosphatase, and its bone fraction levels were higher in Group 1 than in Group 2, while no gender or age differences were observed between the groups. Osteoporosis prevalence was similar in Group 1B and Group 2 patients but lower than in Group 1A. Mild OFC findings did not modify the surgical indication in any patient of Group 1B. Conclusion: Minor radiologic signs of OFC are not uncommon in the modern PHPT phenotype and occur in patients with more active disease. These signs could identify PHPT patients in an earlier stage of typical bone involvement. However, these features do not seem to upgrade either the clinical classification of asymptomatic PHPT patients or the propensity toward surgical choice. Abbreviations: 25OHD = 25-hydroxyvitamin D; ALP = alkaline phosphatase; bALP = bone-specific alkaline phosphatase; BMD = bone mineral density; eGFR = estimated glomerular filtration rate; OFC = osteitis fibrosa cystica; PHPT = primary hyperparathyroidism; PTH = parathyroid hormone; VF = vertebral fracture


2021 ◽  
Vol 5 (Supplement_1) ◽  
pp. A899-A899
Author(s):  
Maria G Mejia ◽  
Hospital San José de Bogotá, C ◽  
Luz Amaya Veronesi ◽  
Diana Carolina Concha

Abstract Introduction: The relationship between the administration of recombinant TSH (rhTSH) and the progression of thyroid cancer has been described in some case reports (1). There are scant data suggesting that rhTSH acts as a growth factor that stimulates metastatic tumor expansion. Case Report: A 55 year old male patient who 3 years ago reported an evident mass at cervical level. The ultrasound documented a large thyroid mass with an endothoracic component. The FNA reported BETHESDA IV, suspicious for follicular neoplasia. Extension studies showed lytic left parietal bone lesion and liver metastatic compromise. The pathology of the total thyroidectomy showed a follicular carcinoma with a micronodular pattern and capsular invasion, of 19x9x7 cm, vascular and esophageal involvement T4BN0M1. He was sent to radioiodine therapy with 100 MCI with previous suspension of LT4 (Preablative TGL> 300 ng / ml (<50 ng / ml) Abs-TGL 2 IU / ml (<2 IU / ml) TSH 31.3 mIU / L (0.37-4.7 mIU / L) and body scan documenting uptake of the radiotracer at the left parietal, the anterior costal grating, and liver. A second dose of iodine was applied, with 150 MCI after stimulation with thyrotropin (Preablative TGL> 300 ng / ml, Abs-TGL 1.76 IU / ml, TSH 83 mIU / L) Immediately after the administration of rhTSH, the patient described the appearance of alopecic plaques on the scalp, sensation of mass at the right upper quadrant of the abdomen and bone pain in the right rib cage and at the right hip, which progressively worsened the following days until they were incapacitating. The scan report showed a new uptake in dorsal vertebrae, the MRI showed new bone injury at the innominate bone, as well as increase in the sum of its maximum dimensions by 22%, indicative of progression. The cerebral angio-MRI showed hypervascularized metastasis at the left parietal level. Bone scintigraphy documented foci of hyper uptake in both parietals, manubrium and sternal body, 5-8 right anterior costal arches, right scapula, both humeral heads, vertebral bodies T3-T5-T7L4, right acetabulum, left proximal femur and the entire length of the right femur. The clinical and imaging worsening of the bone lesions after the application of rhTSH was striking, for this reason radiotherapy and embolization of the liver lesion was performed. The patient was not a candidate for neurosurgical management due to the vascular component of the cranial lesions. Conclusion: rhTSH is a very well-tolerated method that avoids symptoms associated with severe hypothyroidism. However, it has been associated with neoplastic progression. This is one of the few cases documented in the literature where there is a direct association between the administration of rhTSH and the progression of bone metastases. References: (1)Braga M. Ringel M. Cooper DS. Sudden Enlargement of Local Recurrent Thyroid Tumor after Recombinant Human TSH Administration. J Clin Endocrinol Metab. 2001; 86(11):5148–51.


2021 ◽  
Vol 5 (Supplement_1) ◽  
pp. A307-A307
Author(s):  
Ellen Dauterive ◽  
Virginia Wright

Abstract A 40 yo African American female with pmhx of T2DM, DLD was admitted for worsening leg and arm pain that started a year prior but had worsened in the last 6 months. Pain started in the right arm and progressed to include the right leg and left leg. She had presented to the ER 3 times in the last 3 weeks with no diagnosis and prescribed anti-inflammatories. On ROS she had unintended weight loss of 50 lbs. Pain was not relieved with anti-inflammatories or narcotics. She was diagnosed with diabetes in the previous 5 years and had not been compliant with her medications. Plain x-rays showed OA of the hip. An osseus survey showed multiple expansile, bubbly, and lucent intramedullary lesions consistent with polyostotic fibrous dysplasia versus multiple myeloma. CT showed a radiolucent lesion of the left femur with absence of normal bone trabeculae. Her labs showed normal calcium, phosphorous, renal function, PTH and no evidence of monoclonal gammopathy. Vitamin D was low at 8.2 ng/ml (6.6–49 ng/ml). CT CAP showed no concern for malignancy in other organs. A lipid profile was done and showed elevated fasting triglycerides of 2617 mg/dL (<150 mg/dl) and LDL direct 54 mg/dl (<100 mg/dl). A1c was 11.2% on admission. She denied any use of alcohol, estrogens, SSRI’s. No history of pancreatitis. On physical exam she did not have tendinous xanthomas, eruptive xanthomas, palmar xanthomas, or lipemia retinalis. Family history not significant for lipid disorders. Patient was fasted for 24 hours and then started on intensive insulin regimen as well as fenofibrate for hypertriglyceridemia. Triglycerides came down to less than 500 over 7 days. She was evaluated by ortho for her bone lesions and underwent bone lesion biopsy as well as prophylactic IMN of her bilateral femurs for prevention of impending fragility fractures. Bone biopsy was significant for xanthoma of the bone. Following discharge, she remained on fenofibrate and fish oil as well as a basal/bolus insulin regimen. Triglycerides remained controlled. She has not followed up outpatient for further workup. This case highlights an atypical presentation of triglyceride deposition in the setting of hypertriglyceridemia. It shows that hypertriglyceridemia should be included in the differential for lytic lesions when preliminary workup is negative. It also highlights that complications other than pancreatitis and cardiovascular disease can significantly alter a patient’s life if triglycerides go untreated.


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