Revisiting the Case of Sarah Newbury's Death from Mollities Ossium.

Multiple myeloma and its precursor and variant types represent some of the most common hematologic malignancies in adults. These plasma cell dyscrasias are well-known in modern medicine. There are well-established clinical, laboratory, and pathologic criteria for diagnosis and staging. There is debate about the diagnosis of some of the earliest cases of myeloma described in the literature. We present a critical review of one such case.

A patient with femoral osteitis fibrosa cystica mimicking bone neoplasm: a case report.

BACKGROUND: Osteitis fibrosa cystica is a rare, benign and osteolytic 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 and persistent pain in the right knee joint. Imaging studies were suspicious for a benign tumor in 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 results 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.

Osteitis Fibrosa Cystica and pathological fractures-the classic but neglected skeletal manifestation of primary hyperparathyroidism: a case report.

BACKGROUND: Osteitis fibrosa cystica is the classic manifestation of primary hyperparathyroidism (PHPT), occurs after prolonged exposure of bone to high serum parathyroid hormone (PTH) level. It has become increasingly rare due to early detection of PHPT. CASE PRESENTATION: A 37-year-old woman was referred to our institution for fixation of multiple fractures of upper and lower extremities that had been reoccurring in the past 5 years. Her medical history showed right-shoulder, left-elbow, and right-femur fractures after a fall 5 years previously. One month ago, she sustained fractures of the right distal humerus, left tibia, and left femur without history of trauma. Upon arrival to our hospital, a thorough review of her plain radiographs demonstrated brown tumors at multiple sites, along with a salt-and-pepper appearance of the skull and a rugger-jersey spine, compatible with osteitis fibrosa cystica. Patient was diagnosed with PHPT, confirmed by high-corrected serum calcium (13.6 [8.6-10.0] mg/dl), low serum phosphate (2.2 [2.5-4.5] mg/dL), high serum alkaline phosphatase (1482 [35-105] U/L), and significantly elevated parathyroid hormone (PTH 3850 [15-65] pg/mL). A histologically confirmed, 2.5-cm parathyroid adenoma was removed by parathyroidectomy. Ten days later, closed reduction and internal fixation of the left proximal femoral shaft was performed. Pain and ambulation were significantly improved 6 months postoperatively. At the 1.5-year follow-up, fracture unions and complete mineralization of brown tumors were noted; the patient could ambulate with neither pain nor an assistive device. CONCLUSIONS: PHPT has become more asymptomatic in countries where routine calcium screening is performed. Nevertheless, the classic skeletal involvement, osteitis fibrosa cystica, should not be overlooked, particularly in young patients who present with a low-energy fracture.

Osteitis fibrosa cystica mimicking bone tumor, a case report.

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.

Giant parathyroid adenoma diagnosed by brown tumour, a clinical manifestation of primary hyperparathyroidism: A case report.

Brown tumour (BT) is the pathological expression of osteitis fibrosa cystica which is caused due to primary and secondary hyperparathyroidism (HPT). It is a rare benign lesion of skeletal system that usually affects the facial bones, clavicles, ribs, pelvis and extremities. The purpose of this case report is to present the clinical, pathological and radiological findings of BT - rarely seen in adults - originating from the giant parathyroid adenoma and emerging as the first clinical sign of HPT. The patient underwent a successful parathyroidectomy and was discharged on the first postoperative day without any complications. Histopathologically, it was confirmed as parathyroid adenoma. With the increased use of biochemical tests, HPT is diagnosed and treated early and so the frequency of symptomatic HPT due to parathyroid adenoma has decreased in developed countries, but we still sometimes encounter cases of primary hyperparathyroidism (PHPT) occurring as BT. With multidisciplinary preoperative evaluation, surgical resection of bones is refrained from in order to prevent unnecessary morbidity and mortality, unless these osteolytic bone lesions cause a pathological fracture.

Severe hypercalcemia and a pelvic brown tumor in an adolescent with primary hyperparathyroidism: a case report.

BACKGROUND: Primary hyperparathyroidism may present in a myriad of manners, varying from an incidental asymptomatic biochemical finding to gastrointestinal, psychiatric, renal, and bone manifestations. While hyperparathyroidism remains a rare diagnosis in the pediatric population, the initial approach to diagnosis and management of hypercalcemia in children is imperative for the general pediatrician. Herein, we describe an adolescent who presented with a lytic bone lesion and severe, symptomatic hypercalcemia due to primary hyperparathyroidism. CASE PRESENTATION: A 14-year-old male presented with vomiting, constipation, abdominal pain, and lethargy. He had an elevated total corrected calcium of 4.3 mmol/L. He was found to have a large pelvic lytic tumor consistent with a brown tumor due to primary hyperparathyroidism. He received pharmacologic therapy for stabilization of his hypercalcemia, including intravenous saline, intravenous bisphosphonates, and calcitonin. He subsequently received definitive therapy via parathyroidectomy and his post-operative course was complicated by hungry bone syndrome. Long-term follow-up has found full resolution of the lytic lesion and restored calcium homeostasis. CONCLUSIONS: We present this case to highlight the possible presentations of hypercalcemia and hyperparathyroidism that are essential for a general pediatrician to recognize to ensure prompt diagnosis and management. Evaluation for hypercalcemia should be considered in patients with suggestive symptoms and physical exam findings. To our knowledge, this patient represents the first reported pediatric case of a pelvic brown tumor in an adolescent. While the multi-systemic complications of hyperparathyroidism may be quite severe, swift and appropriate management may mitigate these clinical outcomes.

Primary Hyperparathyroidism: Experience from a Tertiary Care Centre in Pakistan.

OBJECTIVE: To study the clinical, biochemical and radiological features and management outcomes of patients with primary hyperparathyroidism. METHODS: This retrospective study was conducted at the Aga Khan University Hospital, Karachi, Pakistan and comprised data of patients with primary hyperparathyroidism from 2008 to 2017. RESULTS: Out of 103 patients, 83(80.6%) were female. Overall mean age was 59.3±16.2 years. Musculoskeletal manifestations were seen in 60(58.3%) patients and renal manifestations in 28(27.2%). Ostieits fibrosa cystica was found in 04(3.88%) patients. Overall, Ultrasound neck and sestamibi scan localized the lesion in 66 (64.1%) and 77 (76.2%) patients respectively. Among 79 patients who underwent surgery, 67 (84.8%) patients had an adenoma, 05 (6.3%) had hyperplasia and 02(2.53%) patients had parathyroid carcinoma whereas histopathology was inconclusive in 5 (6.32%) out of the 79 surgically treated patients. Disease recurrence was seen in 13 out of 79(16.45%) patients who underwent surgery. CONCLUSION: Primary hyperparathyroidism is associated with significant morbidity in our population. Targeted measures like improving patient awareness, routine calcium screening, vitamin D supplementation and a high index of suspicion by the clinician may help in early diagnosis of the condition and thus reduce morbidity.

A hemodialysis patient with bone disease after pregnancy: a case report.

BACKGROUND: Pregnancy is rare in women on hemodialysis. Recommendations for the treatment of secondary hyperparathyroidism (sHPT) and preservation of bone health in pregnant dialysis patients are lacking. CASE PRESENTATION: We present the case of a young woman with end-stage kidney disease (ESKD) due to lupus nephritis, who developed multiple brown tumors while on hemodialysis during her second pregnancy. During her first pregnancy sHPT was well controlled and no skeletal complications occurred. Before the second pregnancy she developed severe sHPT. During pregnancy, dialysis time was increased to 24 h per week, the patient was given oral calcitriol, and the dialysate calcium concentration was set at 1.5 mmol/l. In week 20 the patient complained about bone pain in her left hip. Magnetic resonance imaging revealed a cystic lesion compatible with a brown tumor. The baby was delivered in the 36th week by cesarean section. Further assessment identified multiple brown tumors of her skeleton, including the acetabulum, tibia, ribs, skull, thoracic spine and thumb. She required multiple orthopedic surgeries. Three months after pregnancy, etelcalcetide was started, which brought about a gradual improvement in her sHPT. CONCLUSIONS: This case demonstrates that the combination of pregnancy and severe sHPT in dialysis patients can have deleterious consequences for bone health.

Systematic review of oral manifestations related to hyperparathyroidism.

OBJECTIVES: We sought to identify oral symptoms found in hyperparathyroidism and compare their rate of occurrence, as well as potential variations in sequelae between primary, secondary, and tertiary hyperparathyroidism. MATERIALS AND METHODS: Database searches were performed through EMBASE and PubMed, with a continual handsearch for relevant articles. PRISMA guidelines were followed. RESULTS: Two hundred five articles including 245 patients were analyzed with data extraction. The average age was 34.02 years old (age range 1-83), with 91 male and 154 female patients (1:1.7 M/F ratio). Patients presented with symptoms including facial asymmetry or swelling (167/214 cases; 78.0%), oral pain (30/214; 14.0%), systemic symptoms (25/214; 11.7%), referrals or incidental findings (16/214; 7.5%), and neuropathy (6/214; 2.8%) independently and in combination together. Bony pathology occurred most often in the mandible (100/245 cases; 40.8%), while 72 cases were in the maxilla (29.4%) and 73 cases in both jaw bones (29.8%). CONCLUSIONS: Our data collection identifies a wide variation in the presentation of hyperparathyroidism. In order to be more certain of oral maladies from hyperparathyroidism, studies with large patient populations need to be conducted at healthcare centers to clarify the oral outcomes of hyperparathyroidism. CLINICAL RELEVANCE: What was thought to be a characteristic finding of HPT, mandibular radiolucency occurred in only a minor portion of cases. Furthermore, the pathognomonic sign of HPT on radiograph, loss of lamina dura, was only the third most common presentation. Bone pathology was most commonly reported in literature, but should not be assumed the only oral sequelae of hyperparathyroidism.

Maxillofacial brown tumors in secondary hyperparathyroidism: A case report and literature review.

Parathyroid glands produce parathyroid hormone (PTH). PTH has a main role in bone formation. Hyperparathyroidism (HPT) is explained as primary, secondary and tertiary types defined as overproduction of PTH. The brown tumor or osteitis fibrosa cystica is a benign bone lesion that is caused by HPT. This complication has been decreased by diagnosis and successful treatment of secondary hyperparathyroidism. Pelvis, ribs, clavicle, mandible and the extremities are most commonly affected bone in brown tumor, whereas maxillary involvement is rare. The present article report a 29-year-old man with chief complaints of bone pain, swelling cheeks and teeth displacement with secondary HPT. Parathyroidectomy was done due to bone disorder. It is important for dentists and endocrinologists to understand maxillofacial manifestation of secondary HPT to prevent its complication.

Primary Hyperparathyroidism Mimicking Skeletal Metastasis - A Diagnostic Dilemma.

Introduction: Primary hyperparathyroidism (PHPT) is an intrinsic abnormality of the parathyroid glands in which there is an inappropriate secretion of parathormone (PTH), resulting in skeletal resorption and bone loss. The characteristic bony changes of fibrotic cystic lesions are called Brown's tumors. Clinical dilemmas exist due to the varied clinical presentation of hypercalcemia with multiple lytic lesions mimicking metastatic bone disease. The 99 mTc sestamibi scanning is the imaging modality of choice used for the preoperative localization of parathyroid adenomas. Surgery provides a definitive treatment, and the bony lesions resolve completely over a period of time. Case Report: We present four cases of PHPT where they presented with multiple lytic lesions and were evaluated for metastatic deposits. The diagnosis was confirmed with a biopsy. They were successfully treated by excision of the parathyroid gland. A high index of suspicion will avoid misdiagnosis and inappropriate treatment. Conclusion: PHPT must be considered as a differential diagnosis for multiple osteolytic bone lesions. Diagnosis can be aided by a thorough clinical examination, including an assessment for neck swelling, and laboratory testing of serum calcium levels and PTH levels. Surgical excision of the hyperactive gland serves as the definitive treatment for this condition, with bony lesions regressing gradually over time.

Unusual body weight loss due to primary hyperparathyroidism: A case study with literature review.

Brown tumors (osteitis fibrosa cystica) are rare pathognomonic signs that occur in patients with primary hyperparathyroidism (PHPT). Brown tumors can exist in multiple bones and can easily be misdiagnosed as a metastatic tumor or multiple myeloma. It is also localized in the forearm, humerus, and leg. The symptoms of hypercalcemia, pathologic fracture, and bodyweight loss may increase the diagnostic difficulty of brown tumors because multiple myeloma and bone metastasis also show the same symptoms. We studied a 68-year-old woman who had experienced unusual bodyweight loss in the past 6 months (56kg-40kg) and bone pain. She went to the hospital after a fall with a complaint of bone pain. An X-ray revealed a left bubbly-like cystic change and multiple fractures at the left ulna midshaft. Upon investigation, the level of intact parathyroid hormone was ascertained to be 1800 (normal: 10-60) pg/ml. Microscopically, the tumor demonstrated a benign bone lesion and was compatible with osteitis fibrosa cystica due to PHPT. The parathyroid scan (Tc-99 m sestamibi) indicated right parathyroid hyperplasia, which was later confirmed by a parathyroidectomy. She was diagnosed with osteitis fibrosa cystica associated with PHPT due to a parathyroid adenoma. PHPT can be presented with multiple fractures, bone pain, and bodyweight loss. Therefore, if a patient presents these symptoms, PHPT should be considered.

Fragility Fractures Unveil the Hidden Dragon: A Case of Osteitis Fibrosa Cystica and Secondary Hyperparathyroidism in End-Stage Renal Disease Post-Trauma.

Secondary hyperparathyroidism is a prevalent complication of end-stage renal disease (ESRD), arising from chronic renal insufficiency leading to disturbed calcium metabolism. This disruption triggers hypersecretion of the parathyroid gland, characterizing the condition. Osteitis fibrosa cystica (OFC), a rare complication of untreated secondary hyperparathyroidism, results in benign resorptive bone lesions and the formation of cystic cavities within bones. Our case report describes a 46-year-old incarcerated Hispanic male with a 17-year history of end-stage renal disease and secondary hyperparathyroidism. The patient initially presented with a traumatic right elbow injury. Further diagnostic evaluation revealed an 8 cm destructive process involving the distal humerus, initially suspected as malignancy but confirmed as OFC through bone biopsy. Management involved orthopedic surgery performing an open reduction and internal fixation (ORIF) of the affected limb, with subsequent consideration for inpatient parathyroidectomy. Imaging studies, including magnetic resonance imaging (MRI) and computed tomography (CT) scans, elucidated a 6 × 5.5 cm soft tissue mass, further confirmed as a brown tumor. The case underscores the complexities of diagnosing OFC, often misinterpreted in radiologic studies, and highlights the multidisciplinary approach involving orthopedics, otolaryngology, and nephrology in managing this intricate scenario. The objective is to explore clinical manifestations and treatment challenges of OFC and secondary hyperparathyroidism triggered by trauma in end-stage renal disease, emphasizing the need for continued awareness and precise diagnostic strategies in resource-rich areas.

Primary Hyperparathyroidism With Brown Tumor of the Mandible Misdiagnosed as a Giant Cell Tumor: A Case Report.

In this case study, we present an incidentally discovered giant cell granuloma, which, upon detailed investigation, led to an unexpected diagnosis. A 36-year-old woman exhibited a bone lesion in the right retromolar trigone area, initially suspected of being malignant. However, histopathological examination revealed a giant cell tumor of bone. Further biochemical profiling, including serum calcium, phosphorus, and parathyroid hormone (PTH) levels, showed elevated PTH and hypercalcemia, prompting consideration of primary hyperparathyroidism and the diagnosis of a brown tumor due to this condition. This case underscores the importance of considering brown tumors associated with primary hyperparathyroidism as a potential differential diagnosis in patients with lytic bone lesions.

Hyperparathyroidism-jaw tumour syndrome detected by aggressive generalized osteitis fibrosa cystica.

Severe hyperparathyroidism can affect bone metabolism and be in the origine of multiple brown tumours (generalized osteitis fibrosa cystica). When associated with fibro-ossifying tumours of the jaw, it realizes a rare genetic syndrome referred as Hyperparathyroidism-jaw tumour HPT-JT. We report the case of a patient we treated for HPT-JT, and literature review.

Brown Tumors of the Jaws: A Retrospective Study.

Objectives: (1) Describe the clinical spectrum, imaging features, management, and outcome of brown tumors (BT) of the jaws. (2) Determine via a literature review the facts and controversies around the characteristics and management of this lesion. Materials and methods: Our study was approved by the institutional committee of Sahloul University Hospital in accordance with the ethical standards of the Declaration of Helsinki. Sixteen patients with BT in the context of a primary, secondary, and tertiary hyperparathyroidism were treated and followed up during their recovery. Results: This study reports 16 patients with a mean age of 48 years old. Brown tumor lesions were associated with primary hyperparathyroidism in 6 cases (38%), secondary hyperparathyroidism to chronic kidney failure in 5 cases (31%), and tertiary hyperparathyroidism in the context of a long lasting CRI in 5 cases. Their location was maxillary in 7 cases, mandibular in 5 cases, and bimaxillary in 4 cases. The treatment consisted of parathyroidectomy in 13 patients, maxillary resection in 3 cases, and vitamin D treatment in 2 cases. Favorable outcomes, characterized by tumor regression, were reported in 9 cases, whereas unfavorable evolution was observed among 7 patients. Conclusion: Parathyroidectomy is the gold standard treatment for primary hyperparathyroidism. It resulted in a total regression in all our cases. Regarding secondary hyperparathyroidism, blood screening and chronic renal insufficiency follow-up are critically valuable to detect this condition at an early stage, hence increasing the success rate of brown tumor regression. Our secondary and tertiary hyperparathyroidism results were miscellaneous; although it is important to emphasize the importance of the chronic renal insufficiency management to ensure a success. Clinical relevance: Brown tumors should be included in the differential diagnosis of giant cell lesions. Parathyroidectomy is usually sufficient to induce the total regression of the lesion in primary hyperparathyroidism cases. A more delicate approach is needed regarding secondary hyperparathyroidism. Meticulous control of chronic renal insufficiency is mandatory in addition to parathyroidectomy.

Secondary Hyperparathyroidism Presenting as a Brown Tumor: A Case Report and Review of the Literature.

Severe secondary hyperparathyroidism in advanced stages of chronic kidney disease (CKD) is associated with CKD-related mineral and bone disease (CKD-MBD). A 70-year-old woman was admitted at the Hospital for generalized bone pain and peripheral edema. Initial laboratory tests revealed normocytic anemia and severe renal dysfunction, and further tests evidenced severe secondary hyperparathyroidism. Full-body computerized tomography showed an osteolytic lesion in the right iliac wing. The iliac bone lesion was biopsied and histological examination was consistent with the diagnosis of a brown tumor of hyperparathyroidism. Brown tumors are a rare variant of osteitis fibrosa cystica that results from sustained high levels of parathyroid hormone in CKD. This case sheds light on rare complications that are experienced today in CKD. The clinical and biochemical setting, as well as the clinical suspicion, are essential to the diagnosis.

Multiple Brown Tumors in Primary Hyperparathyroidism Causing Pathological Fracture: A Case Report of a 21-Year-Old Adult Male.

Multiple brown tumors are more common in females and older age groups and an unlikely site is the long bones. We report a case of a 21-year-old male presenting with a pathological fracture at the left neck of the femur. Laboratory investigations showed elevated parathyroid hormone (PTH) and serum calcium levels (PTH-dependent hypercalcemia). A CT scan revealed multiple osteolytic lesions in the pelvis and femurs, and a Tc-99m sestamibi scan showed a solitary parathyroid adenoma. We demonstrate this rare case and illustrate the importance of the consideration of multiple brown tumors in young males presenting with multiple osteolytic lesions at the long bones in the differential diagnosis. Every physician needs to have a high clinical suspicion of primary hyperparathyroidism innovation, in those who present with osteolytic lesions, with respect to the patient's age and gender.

Widespread presentation of brown tumors mimicking multiple myeloma.

Brown tumors or osteitis fibrosa cystica has become a rare presentation of primary hyperparathyroidism in up-to-date clinical practice. Here, we describe a case of longstanding untreated hyperparathyroidism presenting itself with brown tumors in a 65-year-old patient. During the diagnostic work-up of this patient, bone SPECT/CT and 18F-FDG-PET/CT revealed multiple widespread osteolytic lesions. Differentiating from other bone tumors such as multiple myeloma is challenging. In this case, the final diagnosis was made by integrating the medical history, biochemical diagnosis of primary hyperparathyroidism, pathological findings and medical imaging.