Manifestations of hyperparathyroidism in the jaws: Concepts, mechanisms, and clinical aspects.

Hyperparathyroidism is one of the most common endocrine disorders worldwide. In countries where routine biochemical screening is not common, symptomatic hyperparathyroidism predominates. Its manifestations include skeletal alterations, calcification of soft tissues, kidney stones, and functional alterations in other systems. Notably, jaw alterations can be the first clinical sign of hyperparathyroidism, including brown tumor, renal osteodystrophy, osteitis fibrosa, and leontiasis ossea, and knowing such conditions is of core importance for the multidisciplinary diagnosis and management of hyperparathyroidism. We aimed to perform a concise review, systematizing the concepts and mechanisms underlying hyperparathyroidism and associated gnathic alterations. In addition, a detailed description of the clinical aspects of the jaw manifestations is presented.

Diabetes and Bone Involvement in Primary Hyperparathyroidism: Literature Review and Our Personal Experience.

Background: Primary hyperparathyroidism (PHPT) and type 2 diabetes mellitus (T2DM) are common endocrine disorders impacting on skeletal health, whose concomitant occurrence is becoming more frequent. Patients and Methods: We searched the PubMed database from the National Library of Medicine about the relationship between T2DM and its treatment and bone manifestations of PHPT. Thereafter, we retrospectively evaluated a consecutive series of 472 PHPT patients. Among them 55 were also affected by T2DM. At the diagnosis of PHPT we compared bone turnover markers and bone densitometry between 55 patients with and 417 without T2DM and in the former group according to antidiabetic treatment. Results: Few data are available about T2DM and PHPT bone involvement, studies about T2DM treatments and PHPT bone manifestations are lacking. Among patients with PHPT of our series, those with T2DM were older, had a lower prevalence of osteitis fibrosa cystica, higher lumbar and femoral T-scores than the remaining patients. No difference was disclosed among the diabetic patients according to ongoing antidiabetic treatment, even though modern treatments were under-represented. Conclusions: No clinical study specifically evaluated the impact of T2DM on bone involvement in PHPT. In our experience, diabetic patients resulted more frequently "mild asymptomatic" than non-diabetic patients and showed a lower prevalence of radiological PHPT bone manifestations. The treatment of T2DM does not seem to affect the biochemical or clinical features of PHPT in our series. Further studies are needed to fully disclose the influence of T2DM and antidiabetic treatment on bone health in patients with PHPT.

18F-Fluorocholine PET/CT Imaging of Brown Tumors in a Patient With Severe Primary Hyperparathyroidism.

Brown tumors are rare skeletal anomalies occurring in patients with hyperparathyroidism and exposing patients to pathological fractures. We report the case of a 26-year-old woman with severe primary hyperparathyroidism (calcemia, 2.9 mmol/L; parathyroid hormone, 59 pmol/L) who underwent F-fluorocholine (FCH) PET/CT before parathyroidectomy. FCH PET localized the hyperfunctioning parathyroid gland and showed multiple foci in correspondence with bone lytic lesions on CT. Those lesions were not visible on the Tc-MIBI dual-phase scintigraphy. The pathology of one of the FCH-positive bone lesions corresponded to a brown tumor related to hyperparathyroidism.

Brown tumor of the thoracic spine presenting with paraplegia in a patient with peritoneal dialysis.

Secondary and tertiary hyperparathyroidism is an important problem of chronic kidney disease. Brown tumor is a benign, unusual, reactive lesion as a result of disturbed bone remodeling, from long-standing increase in parathyroid hormone level. Brown tumors may cause morbidity due to pressure symptoms on neural structures and spontaneous bone fractures. Herein, we presented a peritoneal dialysis patient with tertiary hyperparathyroidism under calcand calcitriol treatment for 4 years due to refusing of the parathyroidectomy operation. She admitted to hospital for sudden onset back pain with difficulty in gait and walking, and imaging studies showed an expansile mass lesion in the thoracic spine. She was operated for mass and diagnosed with brown tumor. After operation, she lost the ability of walking than become paraplegic and she underwent rehabilitation program. Preventive measures including calcitriol and cinacalcet may cause a modest decrease in parathyroid hormone levels but it should be remembered for the development of bone complications such as brown tumor formation in patients with moderate elevated PTH levels, especially those with tertiary hyperparathyroidism. Parathyroidectomy should be performed without delay in these cases.

Solid aneurysmal bone cyst of the humerus mimics metastasis or brown tumor.

Solid aneurysmal bone cyst (ABC) is a rare subtype of ABC that most commonly involves the small bones of the hands or feet. We present a case of a solid ABC of the distal humerus in a 52-year-old man with a history of chronic kidney disease and renal cell carcinoma. On imaging with plain radiographs, CT, and MRI, this expansile lucent lesion with solid internal enhancement had an appearance that overlapped with metastasis or brown tumor of hyperparathyroidism. On 18F-FDG PET-CT, this lesion was hypermetabolic with an SUVmax of 9.9. Only 37 cases of solid ABC have previously been reported to involve the long bones in the literature, and only 4 in the humerus. We review the clinical, imaging, and histopathological findings and differential diagnosis of solid ABC, and highlight the usefulness of identifying the USP6 gene rearrangement on FISH to distinguish this lesion from other lesions with secondary ABC formation.

Osteitis fibrosa cystica masquerading as bone neoplasm.

A 50-year-old female patient with no significant medical history presented with left knee pain. Radiographs of the knee showed a circumferential swelling of the distal femur suggestive of neoplasia. Further evaluation revealed multiple lesions in the left iliac bone and proximal femur. Biopsy was suggestive of a reparative granuloma or an aneurysmal bone cyst. Laboratory assessment showed hypercalcaemia and elevated parathyroid hormone consistent with severe primary hyperparathyroidism. Osseous survey was significant for salt and pepper appearance of the skull. Ultrasound of the neck and 99mTc-sestamibi parathyroid scintigraphy localised a left parathyroid adenoma/carcinoma. Parathyroidectomy was successful, and a large parathyroid adenoma was excised. Six months later, the patient was doing fine with her gait returning to normal. On follow-up 2 years later, she had no recurrence of the lesions.

Brown tumor of the jaws as a manifestation of tertiary hyperparathyroidism: A literature review and case report.

Brown tumor of the jaws is a manifestation of hyperparathyroidism consisting of osteolytic lesions that show proliferation of multinucleated giant cells in the maxilla and/or mandible. Differential diagnosis of these lesions from local central giant-cell granuloma is mandatory for the correct treatment of the patient. Radiographic and histopathological exams of the jaw lesion are not sufficient to determine the diagnosis, which requires laboratory tests including serum levels of calcium, alkaline phosphatase, parathyroid hormone (PTH) and phosphate, and radiographic examination of other bones as well, such as hand-wrist, pelvis, and femur. We present here a brief literature review focusing on the clinical and radiographic features, diagnostic criteria and treatment of brown tumor and also report a case of the disease affecting the jaw.

Giant mediastinal parathyroid adenoma presenting as bilateral brown tumour of mandible: a rare presentation of primary hyperparathyroidism.

Hyperparathyroidism (HPT) is becoming increasingly common endocrinopathy in clinical practice. Nowadays, it is mostly diagnosed in subclinical or early clinical stage. Bony involvement in HPT has seen significant fall in incidence. Brown tumour of bone is exceptionally rare as a first manifestation of primary HPT (PHPT). Its radiological and histopathological features may be mistaken for other bony pathologies. If possibility of underlying HPT is overlooked the disease is bound to recur after surgery adding to morbidity of the patient. Here we present a case of bilateral brown tumour of mandible which was mistakenly treated as giant cell granuloma by surgical curettage. That the patient was harbouring an ectopic parathyroid adenoma with hypercalcemia causing non-specific symptoms was missed by the referring physician. This led to recurrence of the lesion. On subsequent evaluation, a giant mediastinal parathyroid adenoma causing PHPT was detected at our centre and was removed via mini sternotomy approach.

A thoracic vertebral brown tumor presenting with paraparesis in a patient with end-stage renal disease.

Vertebral brown tumors are rare, non-neoplastic bone lesions that occur in the setting of hyperparathyroidism. There are differences in the management of them in the literature. Because brown tumors usually resolve after a parathyroidectomy. We present a case of a thoracic vertebral brown tumor with paraparesis.

[Brown tumor as an exceptional origin of lumbocruralgia with favourable outcome]. / La tumeur brune : cause exceptionnelle de lombo-cruralgie à évolution favorable.

INTRODUCTION: We report a case of a particular lumbar and radicular pain revealing primary hyperparathyroidism and discuss its characteristics through a literature review. CASE REPORT: A 55-year-old woman was hospitalized for a nerve root pain associated with recent weight loss with normal physical examination. Biology showed no evidence for acute phase response and normal kidney and liver functions. However, hypercalcemia and hypophosphatemia were evidenced. Radiographs of the lumbar spine showed a lytic lesion occupying the body of L4. A lumbar spine CT scan confirmed the presence of a compressive nerve root brown tumor. High level of PTH and parathyroid mass raised the possibility of the diagnosis of primary hyperparathyroidism. After parathyroidectomy all signs of hyperparathyroidism resolved with complete disappearance of spinal brown tumors in a two year follow-up CT scan. CONCLUSION: Although uncommon, brown tumor should be a diagnosis to consider in their presence of a spinal tumor. The improvement after conservative treatment could be dramatic.

Skeletal lesions in primary hyperparathyroidism.

BACKGROUND: Osteitis fibrosa cystica (OFC), a relatively rare skeletal disorder caused by excess parathyroid hormone, is often misdiagnosed as a neoplasm. A summary of the diagnostic procedures and treatment protocols, especially the indications for orthopedic surgery, is helpful to avoid overtreatment. METHODS: Eight patients from the Orthopedic Department of Qilu Hospital of Shandong University diagnosed with OFC caused by primary hyperparathyroidism were treated, and the clinical manifestations, biochemical and radiography examination findings, surgeries and prognoses were recorded. RESULTS: All cases (5 female and 3 male) were admitted to our department with the complaint of bone pain (5/8) or fracture after mild trauma (3/8). Biochemical screening revealed hypercalcemia and high parathyroid hormone of varying levels. Two cases were misdiagnosed as primary bone lesion and metastasis. All cases were treated with parathyroidectomy and experienced spontaneous and progressive regression of the boney disease. Four cases underwent orthopedic surgery. Bone biopsies were necessary to exclude malignant tumors, especially with orthopedic procedures. CONCLUSIONS: OFC can easily be misdiagnosed in orthopedic patients because of a lack of radiological and histologic specificity. Reaching the correct diagnosis requires a combination of clinical manifestations, routine biochemical screenings, radiographic examinations of bone and parathyroid and bone biopsy. It is generally acknowledged that parathyroidectomy is effective, but orthopedic surgery is sometimes necessary after a correct diagnosis and parathyroidectomy. However, the indications for orthopedic surgery must be strictly controlled to avoid overtreatment.

Huge juxtacortical brown tumor in two patients with secondary hyper-parathyroidism due to chronic renal failure.

The brown tumor of the skeletal system is mainly caused by hyperparathyroidism (HPT), and HPT is divided into three categories according to its causes: primary, secondary and tertiary HPT. The secondary HPT patients with brown tumor caused by chronic renal insufficiency are rarely reported. The tumor occurs mostly in the bones such as metacarpals, phalanges, skull, pelvis, clavicle, ribs, femur and spine. We reported two cases of juxtacortical brown tumor in patients with HPT secondary to chronic renal insufficiency which has never been reported previously.