Introduction Primary hyperparathyroidism (PHPT) is an endocrine disorder, characterized by excessive secretion of parathyroid hormone (PTH) from one or more parathyroid glands 1 . The clinical spectrum of primary hyperparathyroidism has undergone a striking change in developed countries, with the predominant form being asymptomatic hyperpara-thyroidism, detected on routine biochemical screening for other causes. 2-6 However, the disease continues to present with classic florid skeletal and renal manifestations in subcontinent. 7-10 Primary hyperparathyroidism is increasingly being recognized because of the widespread use of general multiphasic screening. Several reports have stated that in experienced hands, some 90%-95% of parathyroid adenomas will be localized during an initial neck exploration. Here we report a 36-year-old lady of PHPT presented with multiple fractures, nephrolithiasis, and nephrocalcinosis. All of the imaging techniques to localize the cause were negative. She underwent surgical neck exploration with detection of left parathyroid adenoma. Her post-operative period was uneventful with significant clinical and biochemical improvement. Abstract Primary hyperparathyroidism typically presents with skeletal and renal manifestations. In most (80–85%) cases, the cause is a single parathyroid adenoma. We present a case of 36-year-old lady of primary hyperparathyroidism presented with multiple fractures, nephrolithiasis, and nephrocalcinosis. All of the imaging techniques to localize the cause were negative. She underwent surgical neck exploration with detection of left parathyroid adenoma. Her post-operative period was uneventful with significant clinical and biochemical improvement.