Generalized lymphatic anomaly successfully treated with long-term, low-dose sirolimus

Pediatr Dermatol. 2018 Jul;35(4):533-534. doi: 10.1111/pde.13494. Epub 2018 Mar 26.

Abstract

Generalized lymphatic anomaly is a rare, complex, lymphatic anomaly generally involving soft tissues, spleen, and bones. It can lead to focal skeletal fragility and pathologic effusions. A recent prospective trial of sirolimus for complicated vascular anomalies showed partial response in seven patients with generalized lymphatic anomaly treated with sirolimus with a target trough level of 10-15 ng/mL for 1 year (Adams et al). We describe successful treatment of generalized lymphatic anomaly with a lower-dose, long-term course of sirolimus.

Keywords: generalized lymphatic anomaly; mTOR inhibitor; sirolimus; vascular malformation.

Publication types

  • Case Reports

MeSH terms

  • Child, Preschool
  • Humans
  • Immunosuppressive Agents / administration & dosage*
  • Lymphatic Abnormalities / drug therapy*
  • Magnetic Resonance Imaging
  • Male
  • Sirolimus / administration & dosage*

Substances

  • Immunosuppressive Agents
  • Sirolimus