Endocrine Abstracts

Purpose: Primary hyperparathyroidism (PHPT) is the third common endocrine disorder which often manifest with skeletal or renal disfunction. In developed countries asymptomatic form is the most common and such severe manifestations as osteofibrosis cystica are very rare.

Clinical case: We present the case of 49-years-75 × 66 old caucasian woman, with history of urolithiasis, nephrolithotripsy in 2014. In November 2016 she was diagnosed with left femur fracture. The X-ray showed multiple lesions of bone resorption in the right radius, left and right femurs, left humerus and pelvic bones. Locked intramedullary osteosynthesis was performed. In spring 2017 limitation of mobility in right forearm and severe bone pain appeared. The PTH level was 1859 pg/ml (15–65), serum ionized calcium level was 2.20 (1.11–1.32). Bone scan showed lesions of increased uptake in skull, pelvic, right forearm bones and in femurs. CT–scan showed multiple massive tumor-like deformities of the left ilium and nadacetular region measuring 75 × 66 × 42 mm (+22 HU), of the right pubic bone and the right acetabulum measuring 40 × 37 × 31 mm, of the right radius distal metaepiphysis measuring 28 × 25 × 20 mm ( + 41 HU), in the proximal right ulna measuring 27 × 20 × 16 mm, multiple lesions of bone rarefaction in the proximal right and left femurs. The DXA showed decreased BMD (Z-score L1–L4 –1.8 SD, Neck –2.3 SD, Radius 33% –5.0 SD). Sestamibi scintigraphy showed increased uptake behind the sternum with dimention of 5 cm. Parathyroidectomy was performed, the severe hungry bone syndrome with muscle twitches, impair temperature sensivity, panic attacks, loss of taste and insomnia appeared on the fifth day after with PTH level of 23 pg/ml, total calcium level of 2.38 mmol/l. The patient started taking calcium and vitamin D supplementation and anti–resorptive treatment (denosumab). Three months after parathyroidectomy hypocalcemia was detected with normal 25(OH)D level, PTH level wasn’t measured. Six months later serum PTH level increased to 280.2 pg/ml with normal calcium and 25(OH)D levels. Neck ultrasound didn’t show any pathological formation. 11C-methionine PET/CT scan was also negative. The control DXA showed improving of bone remodeling (Z-score L1–L4 –0.6SD, Neck 0.1SD, Radius 33% –1.7SD).

Conclusion: The classical presentation of PHPT isn’t frequent nowadays because of serum calcium screening. Brown tumors appears through rapid osteoclast turnover and can significantly decreased QoL due to the multiple fragility fractures. It’s likely that the patient has a reccurent PHPT, and negative imaging data requires a more detailed analysis.