Treatment of recurrent suprahyoid cervicofacial lymphangioma with intravenous cyclophosphamide.

Purpose: Surgical resection of cervicofacial cystic hygromas and lymphangiomas rarely effects complete reduction because of severe anatomic restrictions.

Patients and methods: With prior knowledge of cyclophosphamide activity against lesions of this type, a formal trial of cyclophosphamide was initiated.

Results: Overall dose escalation therapy resulted in 50% reduction in mass without recurrence after cessation of therapy and with minimal and readily reversible toxicity.

Conclusions: The favorable responses to cyclophosphamide in this study suggest that a prospective randomized trial should be initiated. Certainly, children who have airway and/or esophageal compromise who have failed surgical therapies should be considered for cyclophosphamide treatment.