Sirolimus used for treatment of retroperitoneal arteriovenous malformation in a premature neonate.

Abstract

We describe a case of a premature infant with antenatally detected retroperitoneal arteriovenous malformation (AVM) with extensive intraspinal extension. Treatment of the malformation with embolectomy and sclerotherapy was not feasible in view of intraspinal extensions and small size of vessels of the lesion. During a trial of propranolol over 20 days, the lesion progressed in size, roughly doubling in volume and was accompanied with deranged coagulation parameters. Treatment was therefore switched to oral prednisolone and sirolimus. The steroid was stopped after 6 weeks and sirolimus was continued with serum drug level monitoring and serial imaging of the malformation. After 4 months of sirolimus, the AVM remained at the same size as at the start of treatment, thus we propose that the drug may have arrested the growth of the lesion. This case highlights the use of sirolimus in management of AVMs in infants.

• Authors: Chinmay Chetan, Suprabha Patnaik, Pradeep Suryawanshi, Reema Garegrat
• Journal: BMJ case reports (BMJ Case Rep) Vol. 15 Issue 3 (Mar 08 2022) ISSN: 1757-790X [Electronic] England
• PMID: 35260399 (Publication Type: Case Reports, Journal Article)
• Copyright: © BMJ Publishing Group Limited 2022. No commercial re-use. See rights and permissions. Published by BMJ.

Chemical References

• Prednisolone
• Sirolimus

Topics

• Arteriovenous Malformations (complications)
• Humans
• Infant
• Infant, Newborn
• Infant, Newborn, Diseases
• Intracranial Arteriovenous Malformations (complications)
• Prednisolone
• Sclerotherapy
• Sirolimus (therapeutic use)