Primary hyperparathyroidism (PHPT) is an endocrine disorder with high serum calcium and parathyroid hormone (PTH) levels. Excessive secretion of PTH can cause bone loss and may have potential damage to implant fixation after total knee arthroplasty (TKA). However, there is currently no research or literature reporting prosthesis loosening after TKA associated with PHPT./r/nThis case report describes a TKA failure of tibial prothesis loosening associated with PHPT. The patient faced aggravated pain and difficulty in walking 6 months after the primary TKA. Further examinations showed high serum calcium and PTH levels indicating the presence of PHPT. After the surgery of parathyroidectomy, the patient received revision TKA with 3D-printed metaphyseal cone. Knee symptoms and function were finally improved./r/nPHPT is a risk factor for prosthesis loosening after TKA. Preoperative screening test of serum calcium and PTH levels is important to diagnose PHPT. For patients with PHPT undergoing TKA, preoperative endocrinotherapy and parathyroidectomy are recommended to improve the survival of prosthesis.