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A Child with Prostaglandin I2-associated Thyrotoxicosis: Case Report

Yuri Sonoda1, Kenichiro Yamamura1, Kanako Ishii1

  • 1Kyushu University Graduate School of Medical Sciences, Department of Pediatrics, Fukuoka, Japan

Journal of Clinical Research in Pediatric Endocrinology
|October 17, 2018
PubMed

Insights

Prostaglandin I2 (PGI2) therapy for pulmonary arterial hypertension (PAH) can cause hyperthyroidism in children with congenital portosystemic venous shunt syndrome (CPSVS). Prophylactic thyroid monitoring is recommended during PGI2 treatment.

Area of Science:

  • Pediatric Endocrinology
  • Cardiology
  • Hepatology

Background:

  • Pulmonary arterial hypertension (PAH) is a serious complication in children with congenital portosystemic venous shunt syndrome (CPSVS).
  • Prostaglandin I2 (PGI2) therapy is used for PAH, but its effects on thyroid function in CPSVS patients are not well understood.
  • Hyperthyroidism is a known complication of PGI2 in adult PAH patients.

Observation:

  • A pediatric case of CPSVS with PAH developed thyrotoxicosis during PGI2 treatment.
  • Literature review identified 12 additional pediatric PAH patients treated with PGI2 who developed hyperthyroidism.
  • Hyperthyroidism during PGI2 treatment was associated with a high mortality rate in pediatric PAH patients.

Findings:

  • PGI2 therapy is linked to hyperthyroidism in children with CPSVS and PAH.
  • The onset of hyperthyroidism can occur years after initiating PGI2 treatment.
  • Hyperthyroidism poses a significant risk, potentially leading to fatal PAH crises.

Implications:

  • Prophylactic thyroid function monitoring is crucial for children with CPSVS receiving PGI2 therapy.
  • Early detection and management of hyperthyroidism may improve outcomes in this vulnerable population.
  • This study highlights the need for careful risk-benefit assessment of PGI2 in pediatric CPSVS patients.

Keywords:
Prostaglandin I2pulmonary arterial hypertensioncongenital portosystemic venous shunt syndromehyperthyroidism

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