Correspondence

Treatment of Life-Threatening Infantile Hemangiomas with Vincristine

N Engl J Med 1995; 333:69July 6, 1995

Article

To the Editor:

We describe two cases of corticosteroid-resistant, life-endangering hemangioma treated with vincristine: a benign hemangioendothelioma in a patient with Kasabach-Merritt syndrome and a liver hemangioendothelioma. Mortality rates among patients with these kinds of hemangioma can be as high as 40 percent and may be more than 50 percent if poor prognostic factors are present.1,2 Corticosteroids are the medical treatment of choice, except for resistant cases,3 which are not infrequent.

The first patient was a nine-month-old baby who presented with a tumor 10 cm in diameter in the left supraclavicular region and a platelet count of 7400 per cubic millimeter. Biopsy of the mass showed a benign hemangioendothelioma; Kasabach-Merritt syndrome was diagnosed. Corticosteroids at a dose of 2 mg per kilogram of body weight per day were begun. After 55 days of treatment and several platelet transfusions, the size of the tumor was unchanged and thrombocytopenia persisted. We therefore started vincristine at a weekly dose of 2 mg per square meter of body-surface area for five weeks. The size of the tumor diminished, and the platelet count increased to 522,000 per cubic millimeter within five weeks. Two months later, the platelet count was 500,000 per cubic millimeter and the maximal diameter of the tumor, verified by computed tomography (CT), was 0.5 cm. One year later the tumor had completely disappeared (as verified by CT).

The second patient was a 20-day-old baby who presented with respiratory distress. Echography and CT disclosed a 7.5-cm tumor of the left hepatic lobe. Arteriography suggested a hemangioendothelioma. Treatment with corticosteroids at a dose of 2 mg per kilogram per day was started. Ten days later, respiratory distress was still present and the size of the tumor was unchanged. Vincristine, at a weekly dose of 1 mg per square meter, was given for two weeks. After the first week, respiratory distress disappeared and the tumor decreased in size to 4.5 cm. By the second dose the tumor was reduced by more than 50 percent, and a surgical excision was carried out. Histologic examination revealed an extensively necrotic and calcified hemangioendothelioma.

We suggest that vincristine is effective for the treatment of corticosteroid-resistant, life-threatening infantile hemangiomas.

Jaime Perez Payarols, M.D.
Jose Pardo Masferrer, M.D., Ph.D.
Cristina Gomez Bellvert, M.D.
Son Dureta University Hospital, 07014 Palma de Mallorca, Spain

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