Agranulocytosis is a rare but fatal side effect of antithyroid drugs (ATDs) with incidence reported at 0.1%-1%. Agranulocytosis is defined as a granulocyte count <500 cells/μL following the use of ATDs and is an absolute contraindication to the use of these drugs; in this case, radioactive iodine (RAI) or surgery are therapeutic options. A 28-year-old female patient was on follow-up at our clinic after she presented with anterior neck swelling of 4 years. The patient was started on propylthiouracil (PTU) and propranolol based on clinical symptoms of hyperthyroidism and low thyroid stimulating hormone (TSH) levels. After taking the ATDs for 7 months, she came to the clinic for her regular follow-up. At this point, she was declared euthyroid and booked for surgery. Investigations were sent and the complete blood count (CBC) result showed leucopenia with agranulocytosis, even though she was completely asymptomatic. The offending ATD was immediately discontinued. The patient was kept inpatient for monitoring, and lugol's iodine and propranolol were initiated. Eight days after discontinuing the ATD, the CBC profile was determined once again, showing normalized total leukocyte, as well as, absolute neutrophil count. Eventually, the multinodular goiter (MNG) was managed with subtotal thyroidectomy. Despite the fact that agranulocytosis is an extremely rare side effect of ATDs, most often PTU; it is a potentially fatal complication when it occurs. Patient education at the time of prescription should not be overlooked, and systematic programs should be put in place. The baseline granulocyte count should be determined and monitored on a regular basis.