Introduction: Thyroid gland, brownish red and highly vascular endocrine gland, is placed anteriorly in the neck, extending from 5th cervical to 2nd thoracic vertebra. In present case the isthmus of thyroid gland was absent. It’s a developmental anomaly of thyroid gland. Observations: The two lobes of thyroid gland were separate from each other. There was agenesis of thyroid isthmus. Discussion: The agenesis of thyroid isthmus is the complete and congenital absence of the thyroid isthmus. The absence of isthmus can be associated with other types of dysorganogenesis, such as the absence of a lobe or the presence of ectopic thyroid tissue autonomous thyroid nodule ; thyroiditis; primary carcinoma; neoplastic metastases; and infiltrative diseases such as amyloidosis Knowledge regarding thyroid anomalies as agenesis of isthmus of thyroid gland can help the surgeon for surgical interventions.