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Aim: To highlight the management of huge multinodular goiters and report a rare case of intrathyroidal parathyroid adenoma. Background: Goiter is an abnormal enlargement of thyroid gland. Huge goiters are a surgical challenge due to possible difficulty in intubation, tracheomalacia, retrosternal extension and distorted anatomy. Goiters associated with congenital hypothyroidism undetected at birth leads to permanent neurological deficits. This case report highlights a case of huge multinodular goiter with a rare intrathyroidal parathyroid adenoma detected on histopathology. Case description: This case report deals with a 27-year-old male patient, presenting with huge hypothyroid multinodular goiter weighing 750 g with no compression symptoms. He had low IQ probably due to undetected hypothyroidism at birth. His postoperative histopathology report showed an intrathyroidal parathyroid adenoma which is an extremely rare tumor. Conclusion:This report aims to emphasize the existence of congenital hypothyroidism and also that huge goiters when dissected meticulously may be removed without any major complications. We also report an incidental finding of rare intrathyroidal parathyroid adenoma. Clinical significance: Neonatal screening of hypothyroidism and its subsequent treatment can prevent mental retardation. We would also like to emphasize the importance of preoperative serum parathormone and calcium levels in all patients undergoing thyroidectomy to detect asymptomatic parathyroid adenomas.

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