Background. While the clinical benefits of terlipressin (TP) have been reported in adults and children with refractory hypotension, data in neonates are limited. Case. Herein, we report a case of off-label rescue TP therapy in a neonate with septic shock and persistent hypotension. The patient’s blood pressure was normalized, and tissue perfusion improved without serious adverse reactions. However, genetic testing revealed mitochondrial gene defects in the patient, and the parents subsequently elected to stop treatment after 25 doses of TP (20 μg/kg/min every 4 h for 100 h). Conclusion. While TP treatment appeared to help control hypotension and may prolong the survival time, there are no conclusive data regarding the safety and efficacy of TP in neonates.
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