Treatment of generalized infantile myofibromatosis with sorafenib and imatinib: A case report

Pediatr Blood Cancer. 2020 Jun;67(6):e28288. doi: 10.1002/pbc.28288. Epub 2020 Apr 19.

Abstract

Infantile myofibromatosis (IM) is characterized by solitary musculoskeletal nodules presenting during infancy but can manifest as multiple lesions with visceral involvement. Multicentric IM with visceral involvement carries a high risk of mortality and there is no consensus on treatment. We present a case of a patient with multicentric IM and pulmonary involvement who progressed on several chemotherapeutic regimens and subsequently had a complete response to sorafenib and later imatinib. This report describes the novel use of sorafenib and imatinib to treat generalized IM and the role of continued tyrosine kinase inhibitor therapy to maintain remission.

Keywords: imatinib; infantile myofibromatosis; soft tissue tumor; sorafenib; targeted therapy.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Antineoplastic Combined Chemotherapy Protocols / therapeutic use*
  • Female
  • Humans
  • Imatinib Mesylate / administration & dosage
  • Infant
  • Myofibromatosis / congenital*
  • Myofibromatosis / drug therapy
  • Myofibromatosis / pathology
  • Prognosis
  • Sorafenib / administration & dosage

Substances

  • Imatinib Mesylate
  • Sorafenib

Supplementary concepts

  • Fibromatosis, Congenital Generalized