Vnitr Lek 2008, 54(6):653-664

Treatment of multiple angiomatosis involving the skeleton and the abdominal and thoracic cavities with interferon α, thalidomide and zoledronate

Z. Adam^1,*, L. Křikavová², M. Krejčí¹, M. Mechl², L. Pour¹, M. Moulis³, J. Vaníček⁴, Č. Neumann⁵, M. Navrátil¹, K. Veselý⁶, J. Vorlíček¹

¹ Interní hematoonkologická klinika Lékařské fakulty MU a FN Brno, pracoviště Bohunice, přednosta prof. MUDr. Jiří Vorlíček, CSc.

² Radiologická klinika Lékařské fakulty MU a FN Brno, pracoviště Bohunice, přednosta prof. MUDr. Vlastimil A. Válek, CSc., MBA

³ Ústav patologie Lékařské fakulty MU a FN Brno, pracoviště Bohunice, přednosta prof. MUDr. Jirka Mačák, CSc.

⁴ Klinika zobrazovacích metod Lékařské fakulty MU a FN u sv. Anny Brno, přednosta doc. MUDr. Petr Krupa, CSc.

⁵ Chirurgická klinika Lékařské fakulty MU a FN Brno, pracoviště Bohunice, přednosta prof. MUDr. Zdeněk Kala, Ph.D.

⁶ I. patologicko-anatomický ústav Lékařské fakulty MU a FN u sv. Anny Brno, přednostka doc. MUDr. Markéta Hermanová, Ph.D.

Multiple angiomatosis is a very rare disease formed by histologically benign angiomas spreading beyond single organ or tissue. In the case reported herein, hemangiomas affected several vertebrae of a young man and spread through his peritoneal cavity projecting to his stomach and causing recurrent hematemesis. Also affected was the mediastinum. The patient suffered from bone pain and digestive problems. Initial treatment involved 2 drugs with antiangiogenic effect: interferon α (initial dose of 6 million units 3 times a week, later reduced to 3 million units 3 times a week due to adverse effects) and zoledronate (4 mg i.v. every 28 days). Even though the therapy eliminated bone pain after 2 months, CT check at a later stage showed but little regression of the mass of the angiomas in the abdominal cavity and the mediastinum. Substantial reduction in the mass of the angiomas to merely residual quantity, i.e. partial remission of the disease, was achieved only after the addition of 100 mg/day thalidomide (Myrin) to the above mentioned doses of interferon and zoledronate administered on a regular basis. However, the disease recurred after the therapy was interrupted, and the above triple combination therapy has had to be restored. Maintenance therapy will succeed to repeated achievement of remission of angiomas. A very good therapeutic effect was recorded for combined interferon α, thalidomide and zoledronate in this specific case of multiple angiomatosis.

Keywords: angiomatosis; osetolysis; multiple myeloma; zoledronate; thalidomide; interferon α

Received: January 3, 2008; Accepted: March 19, 2008; Published: June 1, 2008  Show citation