Two infants with life‐threatening diffuse neonatal hemangiomatosis treated with cyclophosphamide

Abstract

Diffuse neonatal hemangiomatosis (DNH) is a rare, frequently fatal disorder characterized by multiple cutaneous and visceral hemangiomas. Complications include high-output cardiac failure, hemorrhage, hepatic failure, and consumption coagulopathy. We present two cases of neonates with DNH. We prescribed cyclophosphamide after failure of corticosteroids. Cyclophosphamide induced a fast regression of the lesions with no side effects in both infants. We were able to stop treatment after 3-4 courses of cyclophosphamide in both infants because of the excellent response. Cyclophosphamide seems to be a safe and effective treatment in patients suffering from life-threatening DNH unresponsive to corticosteroids.

Propranolol therapy for infantile haemangiomas: Review of the literature

“…In cases of life-threatening haemangiomas and haemangiomatosis cyclophosphamide was also reported to offer promising results [7]. However, the serious side effects of cyclophosphamide like avascular necrosis, cardiomyopathy, pulmonary fibrosis, gonadal damage, and subsequent malignancies [8] have to be kept in mind and therefore the application of cyclophosphamide in the therapy of infantile haemangiomas needs to be carefully considered. Reported successful invasive treatments, especially for airway haemangiomas, include intralesional steroid injection, endoscopic and open excision, laser therapy, and tracheotomy.…”

Screening for Brain Involvement in Infants with Multifocal Cutaneous Infantile Hemangiomas

“…A literature search revealed 15 other cases described with multifocal IHs and brain involvement [5,6,7,8,9,10,11,12,13,14,15,16,17,18]. Table 2 summarizes the data reported for these cases including our patient.…”

“…This high frequency of central nervous system involvement is unprecedented, and does not reflect the reports in the literature thereafter. Most of the infants described in this study and in other case reports [5,6,7,8,9,10,11,12,13,14,15,16,17,18] were symptomatic, with symptoms such as increased head circumference, flaccid paralysis, cranial nerve palsy, decreased rectal tone, areflexia, and others. Radiological findings in these cases include IHs, but also hemorrhages and hydrocephalus.…”

An experimental study on the therapy of infantile hemangioma with recombinant interferon γ

“…Steroid therapy also has its disadvantages. About 20% of hemangiomas are steroid resistant [5]. Steroids also have limited efficacy in the management of pulmonary hemangiomatosis and diffuse neonatal hemangiomatosis.…”