[Interferon alpha-2a in the treatment of diffuse thoracic angiomatosis in adulthood--a ase report]

Pneumologie. 2000 Jun;54(6):243-8. doi: 10.1055/s-2000-8994.
[Article in German]

Abstract

A diffuse angiomatosis affecting thoracal organs is a rare, frequently fatal disorder occurring mainly in childhood. The definite diagnosis is usually made by autopsy. A specific treatment does not exist. In the case report presented here we describe the course of an 37-year old patient presenting initially with dyspnea and hemoptysis. Radiological investigations showed a mass lesion in the anterior superior mediastinum, bihilar masses and a periesophageal mass with soft tissue density and a pericardial effusion. Bilateral interstitial infiltrations were also present. CT-guided mediastinal biopsies and bronchial mucosal biopsies were nonspecific. Open-lung biopsy revealed the diagnosis of an extensive diffuse angiomatosis affecting mediastinum, pericardium and pleura. During the 18-month monotherapy with interferon-alpha 2a the patient showed significant improvements of clinical as well as laboratory and radiological parameters. The presented case confirms the experience of several authors, that the diagnosis of a diffuse thoracal authors can only be made by open lung biopsy or autopsy. The presented case also underlines the possibility of a palliative therapy of aggressive angiomatous diseases with interferon-alpha 2a.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Angiogenesis Inhibitors / therapeutic use*
  • Angiomatosis / diagnosis
  • Angiomatosis / drug therapy*
  • Dyspnea
  • Hemoptysis
  • Humans
  • Interferon alpha-2
  • Interferon-alpha / therapeutic use*
  • Male
  • Recombinant Proteins
  • Thoracic Diseases / diagnosis
  • Thoracic Diseases / drug therapy*

Substances

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