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Prostaglandin I (PGI2) causes hyperthyroidism, a critical complication in patients with pulmonary arterial hypertension (PAH). However, it remains unknown whether PGI2 may have unfavorable effects on thyroid functions in children with congenital portosystemic venous shunt syndrome (CPSVS). We present a boy with CPSVS who developed PAH at 7 years of age. During the PGI2 therapy, he experienced thyrotoxicosis at 17 years of age. The literature review showed that the past 12 patients with PAH (median 11 years of age) developed hyperthyroidism during 1 to 11 years of PGI2 treatments. Only one patient survived the acute PAH crisis due to hyperthyroidism. These data provide evidence that prophylactic intervention against hyperthyroidism is indicated for children with CPSVS during the PGI2 treatments. Keywords: Prostaglandin I2, Pulmonary arterial hypertension, Congenital portosystemic venous shunt syndrome, and Hyperthyroidism.

A pediatric case with prostaglandin I2-associated thyrotoxicosis: Case report and the literature review