Interferon alpha-2a therapy for disseminated intravascular coagulation in a patient with blue rubber bleb nevus syndrome - A case report

被引:34
作者
Kunishige, M
Azuma, H
Masuda, K
Shigekiyo, T
Arii, Y
Kawai, H
Saito, S
机构
[1] First Dept. of Internal Medicine, School of Medicine, University of Tokushima, Tokushima City
[2] First Dept. of Internal Medicine, School of Medicine, University of Tokushima, Tokushima City 770
关键词
D O I
10.1177/000331979704800312
中图分类号
R6 [外科学];
学科分类号
1002 ; 100210 ;
摘要
The authors present a sixteen-year-old girl with blue rubber bleb nevus syndrome (BRBNS) associated with disseminated hemangiomas involving the skin, oral cavity, skeletal muscle, and cerebrum. Although she denied neurologic symptoms, magnetic resonance imaging of the brain demonstrated dilatated cerebral veins and the Chiari I malformation. Examination of hemostasis revealed disseminated intravascular coagulation (DIG) manifesting as Kasabach-Merritt syndrome, with the potential for life-threatening bleeding or thrombosis in the central nervous system. Since successful management of life-threatening hemangiomas with interferon alpha-2a (IFN alpha-2a) has been reported, the authors administered IFN alpha-2a with an improvement in hemostasis. These findings suggest that IFN alpha-2a therapy is beneficial for relieving the life-threatening consumptive coagulopathy associated with BRBNS.
引用
收藏
页码:273 / 277
页数:5
相关论文
共 13 条
  • [1] Bean WB., 1958, VASCULAR SPIDERS REL, P178
  • [2] INHIBITION OF CELL MOTILITY BY INTERFERON
    BROUTYBOYE, D
    ZETTER, BR
    [J]. SCIENCE, 1980, 208 (4443) : 516 - 518
  • [3] INTERFERON ENHANCES PROSTACYCLIN PRODUCTION BY CULTURED VASCULAR ENDOTHELIAL-CELLS
    ELDOR, A
    FRIDMAN, R
    VLODAVSKY, I
    HYAM, E
    FUKS, Z
    PANET, A
    [J]. JOURNAL OF CLINICAL INVESTIGATION, 1984, 73 (01) : 251 - 257
  • [4] INTERFERON ALFA-2A THERAPY FOR LIFE-THREATENING HEMANGIOMAS OF INFANCY
    EZEKOWITZ, RAB
    MULLIKEN, JB
    FOLKMAN, J
    [J]. NEW ENGLAND JOURNAL OF MEDICINE, 1992, 326 (22) : 1456 - 1463
  • [5] SUCCESSFUL MANAGEMENT OF AN INFANT WITH A GIANT HEMANGIOMA OF THE RETROPERITONEUM AND KASABACH-MERRITT SYNDROME WITH ALPHA-INTERFERON
    HATLEY, RM
    SABIO, H
    HOWELL, CG
    FLICKINGER, F
    PARRISH, RA
    [J]. JOURNAL OF PEDIATRIC SURGERY, 1993, 28 (10) : 1356 - 1359
  • [6] BLUE RUBBER-BLEB NEVUS SYNDROME - REPORT OF A CASE WITH CONSUMPTION COAGULOPATHY COMPLICATED BY MANIFEST THROMBOSIS
    HOFHUIS, WJD
    ORANJE, AP
    BOUQUET, J
    SINAASAPPEL, M
    [J]. EUROPEAN JOURNAL OF PEDIATRICS, 1990, 149 (08) : 526 - 528
  • [7] Oranje A P, 1986, Pediatr Dermatol, V3, P304, DOI 10.1111/j.1525-1470.1986.tb00530.x
  • [8] ORCHARD PJ, 1989, LANCET, V2, P565
  • [9] CENTRAL-NERVOUS-SYSTEM INVOLVEMENT IN BLUE-RUBBER-BLEB-NEVUS SYNDROME
    SATYAMURTI, S
    NAVADA, S
    EAMES, F
    [J]. ARCHIVES OF NEUROLOGY, 1986, 43 (11) : 1184 - 1186
  • [10] SIDKY YA, 1987, CANCER RES, V47, P5155