The atypical parathyroid adenoma is a histological diagnosis. It is a parathyroid tumor with atypical histological features different from an adenoma and not similar enough to be considered as a carcinoma. It has an uncertain malignant potential. We report the case of a 55‐year‐old woman, diagnosed with severe hypercalcemia during her follow‐up with the rheumatologist for osteoporosis. The laboratory testing led to the detection of a very important level of parathormone PTH 1325.62 pg/ml (20 fold the normal level), confirming the diagnosis of primary hyperparathyroidism. Investigations of the potential causes led to the presence of a large left inferior parathyroid adenoma. The patient was operated and on the examination of the removed tissue, the histopathological examination concluded to an atypical parathyroid adenoma, and the post‐operatory PTH level dropped significantly to 152 pg/ml. The atypical parathyroid adenoma is a very rare tumor, and the diagnosis is still a challenge, the outcome of patients is not well known yet, there for the surveillance is important and must be regularly.

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