Successful Treatment of Macroglossia Due to Lymphatic Malformation With Sirolimus

Ann Otol Rhinol Laryngol. 2015 Oct;124(10):820-3. doi: 10.1177/0003489415583330. Epub 2015 Apr 22.

Abstract

Objective: To evaluate the effectiveness and safety of sirolimus therapy in a child with macroglossia due to lymphatic malformation.

Methods: Sirolimus treatment was applied to the patient with an initial dosing of 0.8 mg/m2 per dose, administered orally, twice daily at approximately 12-hour intervals.

Results: After 9 months of sirolimus therapy, there was a nearly complete resolution of lymphatic malformation. The last evaluation was performed 6 months after withdrawal of treatment, and the lesion had almost completely resolved.

Conclusion: This article presents a novel approach to the treatment of lymphatic malformation of the tongue using sirolimus, which appears to be safe and effective for the management of complex cases.

Keywords: lymphatic malformation; macroglossia; sirolimus.

Publication types

  • Case Reports

MeSH terms

  • Administration, Oral
  • Antibiotics, Antineoplastic / administration & dosage
  • Dose-Response Relationship, Drug
  • Drug Administration Schedule
  • Drug Monitoring
  • Female
  • Humans
  • Infant
  • Lymphatic Abnormalities* / complications
  • Lymphatic Abnormalities* / diagnosis
  • Lymphatic Abnormalities* / physiopathology
  • Lymphatic Abnormalities* / therapy
  • Macroglossia* / etiology
  • Macroglossia* / physiopathology
  • Macroglossia* / therapy
  • Sirolimus / administration & dosage*
  • Treatment Outcome

Substances

  • Antibiotics, Antineoplastic
  • Sirolimus