[Treatment of multiple angiomatosis involving the skeleton and the abdominal and thoracic cavities with interferon alpha, thalidomide and zoledronate].
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 alpha (initial dose of6 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 alpha, thalidomide and zoledronate in this specific case of multiple angiomatosis.
Adam
Z
Z
Interní hematoonkologická klinika Lékarské fakulty MU a FN Brno. z.adam@fnbrno.cz
Krikavová
L
L
Krejcí
M
M
Mechl
M
M
Pour
L
L
Moulis
M
M
Vanícek
J
J
Neumann
C
C
Navrátil
M
M
Veselý
K
K
Vorlícek
J
J
D000328:Adult; D020533:Angiogenesis Inhibitors / Q000008:administration & dosage*; D000798:Angiomatosis / Q000175:diagnosis / Q000188:drug therapy* / Q000473:pathology; D003937:Diagnosis, Differential; D004164:Diphosphonates / Q000008:administration & dosage*; D004359:Drug Therapy, Combination; D006801:Humans; D007093:Imidazoles / Q000008:administration & dosage*; D016898:Interferon-alpha / Q000008:administration & dosage*; D008297:Male; D008477:Mediastinal Diseases / Q000188:drug therapy*; D010532:Peritoneal Diseases / Q000188:drug therapy*; D013122:Spinal Diseases / Q000188:drug therapy*; D013792:Thalidomide / Q000008:administration & dosage*; D000077211:Zoledronic Acid
D020533:Angiogenesis Inhibitors; D004164:Diphosphonates; D007093:Imidazoles; D016898:Interferon-alpha; D013792:Thalidomide; D000077211:Zoledronic Acid