Neonatal thyrotoxicosis is mostly caused by the transplacental passage of maternal thyroid stimulating hormone (TSH) receptor antibodies (TRAb) to the fetus. Although rare and transient, this condition may be associated with significant morbidity and mortality if not diagnosed and treated in an appropriate and timely manner. Anti-thyroid drugs are the main therapy. Here, we report a preterm newborn that presented with two uncommon presentations of neonatal thyrotoxicosis, cholestasis and pulmonary hypertension that significantly improved following the administration of anti-thyroid medications.

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