Remission of infantile generalized myofibromatosis after interferon alpha therapy.

Abstract

Infantile myofibromatosis is the most common fibrous tumor of infancy. Solitary or generalized myofibromas without visceral involvement usually regress within a few months. The multifocal disease infantile generalized myofibromatosis, with visceral involvement, is associated with a significant mortality due to the effect of tumors on vital organs. We report a case of infantile generalized myofibromatosis with visceral involvement, including 2 right atrium tumors. The infant expressed high circulating vascular endothelial growth factor and fibroblast growth factor-2 levels, and interferon alpha-2b was started as antiangiogenic treatment, aimed at triggering regression of the life-threatening cardiac lesions. The tumors regressed and vascular endothelial growth factor and fibroblast growth factor-2 levels were reduced after treatment discontinuation.

• Authors: Cinzia Auriti, Mark W Kieran, Giovanni Deb, Rita Devito, Luciano Pasquini, Olivier Danhaive
• Journal: Journal of pediatric hematology/oncology (J Pediatr Hematol Oncol) Vol. 30 Issue 2 Pg. 179-81 (Feb 2008) ISSN: 1077-4114 [Print] United States
• PMID: 18376275 (Publication Type: Case Reports, Journal Article)

Chemical References

• Interferon alpha-2
• Interferon-alpha
• Recombinant Proteins
• Vascular Endothelial Growth Factor A
• Fibroblast Growth Factor 2

Topics

• Female
• Fibroblast Growth Factor 2 (blood, urine)
• Humans
• Infant
• Interferon alpha-2
• Interferon-alpha (therapeutic use)
• Myofibromatosis (drug therapy)
• Recombinant Proteins
• Vascular Endothelial Growth Factor A (blood, urine)