Two patients presenting with mandible-jaw neoformations were referred to us for endocrine evaluation. A 51-year-old male presented with painful T9 vertebral fracture and multiple osteolytic areas in spine, pelvis, and right mandible branch (Fig. 1A). Metastatic disease was excluded with 18fluorine-fluorodeoxyglucose positron emission tomography (18F-FDG-PET) showing multiple uptakes corresponding to osteolytic areas. Mandible lesion biopsy was not conclusive. Biochemical workup was consistent with hypercalcaemic primary hyperparathyroidism (HPH) (calcium 13.4 mg/dl, range: 8.5–10.5 mg/dl; PTH 667 pg/ml, range 12–88 pg/ml) and vitamin D deficiency (7.8 ng/ml). Neck ultrasound (US) performed at our Unit showed a hypoechoic area under the left thyroid lobe suspected for enlarged parathyroid. This lesion was also identified after CT images re-evaluation (Fig. 1B). Pathology review of the previously provided sample integrated with clinical and radiological information was finally concordant with brown tumor (BT) of the jaw. Spine magnetic resonance was performed and confirmed multiple osteolytic spine lesions typical of BT (Fig. 1C). Left inferior parathyroidectomy was performed and histology showed a parathyroid adenoma. After surgery, serum calcium levels rapidly dropped. High-dose oral and parenteral calcium supplementation as well as vitamin D active metabolite administration was needed in order to contrast hungry bone syndrome development. Concerning vertebral fractures, a corset was applied for at least 1 month and therapy with cholecalciferol was subsequently started. In addition, further treatment with zoledronic acid (5 mg) was administered in order to prevent further fractures.