Improvement of disseminated lymphangiomatosis with recombinant interferon therapy

Pediatr Pulmonol. 2000 Apr;29(4):321-4. doi: 10.1002/(sici)1099-0496(200004)29:4<321::aid-ppul13>3.0.co;2-c.

Abstract

Disseminated lymphangiomatosis is a rare disorder with a poor prognosis. We present a case involving a 3-year-old boy who presented with pulmonary infiltrates, multiple lytic lesions of the ribcage, and small cystic lesions in the spleen. Open-lung and bone biopsies revealed disseminated lymphangiomatosis. Significant clinical and radiologic improvement were observed and persisted after 28 months of treatment with recombinant interferon alpha-2b (IFN alpha-2b). No significant toxicity has been observed.

Publication types

  • Case Reports

MeSH terms

  • Angiogenesis Inhibitors / therapeutic use*
  • Antineoplastic Agents / therapeutic use*
  • Child, Preschool
  • Humans
  • Interferon alpha-2
  • Interferon-alpha / therapeutic use*
  • Lung Neoplasms / diagnostic imaging
  • Lung Neoplasms / drug therapy*
  • Lymphangioleiomyomatosis / diagnostic imaging
  • Lymphangioleiomyomatosis / drug therapy*
  • Male
  • Recombinant Proteins
  • Tomography, X-Ray Computed

Substances

  • Angiogenesis Inhibitors
  • Antineoplastic Agents
  • Interferon alpha-2
  • Interferon-alpha
  • Recombinant Proteins