Total parathyroidectomy with autotransplantation of parathyroid tissue is one of the treatment modalities for primary hyperparathyroidism in Multiple endocrine neoplasia type 1 (MEN1). Many of these patients are young and recurrence may take decades to emerge. We present a case of a 45-year-old woman who was diagnosed with MEN syndrome type 1 at the age of 12. Over the years, she underwent multiple surgical interventions including pituitary macroprolactinoma resection resulting in panhypopituitarism, pancreatectomy for insulinoma resulting in insulin dependent diabetes and parathyroidectomy. In order to avoid future hypoparathyroidism, she had 3 parathyroid glands removed with autotransplantation of 4th parathyroid gland in the flexor surface of the left arm. Ten years following autotransplantation, she developed depression with recurrent vague abdominal pain and constipation which necessitated referral to departments of psychiatry and gastroenterology. She underwent extensive investigations and treated with antidepressants. Mild hypercalcemia was observed which triggered areferral to endocrine clinic. Further assessment revealed persistently elevated parathyroid hormone level of 15.6 pmol/l. Corrected serum calcium was elevated (2.68 mmol/l) with low phosphorous level of 0.77 mmol/l. 25-hydroxy-vitamin D and 1,25-dihydroxyvitamin D were both in the normal range. Clinical and laboratory data confirmed the diagnosis of recurrent hyperparathyroidism. Once the diagnosis was established, she had an uneventful surgical removal of the autotransplanted parathyroid gland and she maintained normocalcaemic state. Careful history exploration and surgical resection are two important components of successful management of this patient.