
We report a case of a 69-year-old woman with primary hyperoxaluria type I, who developed a severe hypercalcemia despite controlled secondary hyperparathyroidism. Bone scintigraphy showed diffuse increased uptake in axial and peripheral skeleton. 18F-FDG PET/CT showed countless striking hypermetabolic foci, interesting 2 types of lesions (joint calcifications and periosteal resorptions). Bone biopsy demonstrated inflammatory changes around many calcium oxalate crystals; hypercalcemia was then related to oxalate osteopathy. Immunotherapy with denosumab was thus initiated. Eighteen months later, a second PET/CT showed decreased 18F-FDG uptake, reflecting treatment efficacy on inflammatory reaction secondary to calcium oxalosis skeletal deposits.
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