The aim of this case report is to highlight an exceptional presentation of primary hyperparathyroidism.

Asymptomatic maternal hyperparathyroidism manifesting in the neonate as hypocalcemic convulsions is exceedingly rare.

Case report: Primary hyperparathyroidism has got a varied presentations. We present a case report of neonatal hypocalcemic convulsions secondary to asymptomatic maternal hyperparathyroidism. Here, the challenge in diagnosing this condition and management will be discussed in the context of available literature.

In the event of nonfebrile seizures in the neonate due to hypocalcemia, the mother has to be evaluated for primary hyperparathyroidism.

Clinical significance: Physicians’ awareness of this unique manifestation of primary hyperparathyroidism is essential for the appropriate management of both the mother and the neonate.

Timely identification and treatment of this condition will prevent complications in both.

Teja VS, Ramakrishnan R. Unusual Presentation of Primary Hyperparathyroidism: A Rare Case Report. World J Endoc Surg 2017;9(1):13-15.