Treatment of a tongue lymphangioma with sirolimus after failure of surgical resection and propranolol

Pediatr Blood Cancer. 2014 May;61(5):931-2. doi: 10.1002/pbc.24866. Epub 2013 Nov 22.

Abstract

Lymphangiomas of the tongue are rare, and their treatment is problematic. A 10 year-old patient with tongue lymphangioma who was previously treated with surgery and propranolol with no response was treated with sirolimus in our department. We used sirolimus with a dose of 1.6 mg/m(2)/day. After 3 months of treatment, the mass had decreased by more than 60%. We continued the treatment for 1 year with a maximum response of 70% decrease in mass. Disease remained stable 6 months after stopping therapy, the latest time of follow-up. Sirolimus appears to be effective in lymphangioma but requires further study.

Keywords: lymphangioma; outcome; sirolimus; treatment.

Publication types

  • Case Reports

MeSH terms

  • Adrenergic beta-Antagonists / adverse effects
  • Antibiotics, Antineoplastic / therapeutic use*
  • Child
  • Combined Modality Therapy
  • Humans
  • Lymphangioma / drug therapy*
  • Lymphangioma / etiology
  • Lymphangioma / surgery
  • Male
  • Postoperative Complications*
  • Propranolol / adverse effects*
  • Sirolimus / therapeutic use*
  • Tongue Neoplasms / drug therapy*
  • Tongue Neoplasms / etiology
  • Tongue Neoplasms / surgery
  • Treatment Outcome

Substances

  • Adrenergic beta-Antagonists
  • Antibiotics, Antineoplastic
  • Propranolol
  • Sirolimus