Benign neonatal hemangiomatosis with early regression of skin lesions: A case report and review of the published work.

Abstract

A 2-day-old Japanese male infant was referred to our outpatient clinic for multiple cutaneous hemangiomas present since birth. Physical examination revealed 14 small, well-circumscribed red papules, scattered over the head, face, dorsum of the right hand, trunk, lower extremities, buttocks and penis. Ultrasound examination revealed no evidence of visceral involvement. Histological examination of a cutaneous lesion was consistent with infantile hemangioma, resulting in the final diagnosis of benign neonatal hemangiomatosis (BNH). The hemangiomas enlarged by 1month of age and began to resolve at 2months of age. Within the next month, the lesions had almost completely disappeared. BNH is a rare, non-heritable, self-limited, benign disease characterized by multiple cutaneous infantile hemangiomas and no or unremarkable visceral lesions. Generally, BNH lesions spontaneously regress within the first 1-2years of life or within 4months of onset. However, there have been no detailed reports about the time course of BNH. To our knowledge, 31 cases of BNH without hepatic hemangiomas, excluding this case, have been reported so far. Twenty-one of these cases demonstrated spontaneous regression of the cutaneous hemangiomas without treatment. In all cases, the cutaneous hemangiomas were present at birth. The median age at the beginning of spontaneous regression was 6.0months (range, 1-12) and the median age at complete or almost complete regression was 15months (range, 3-28). Cutaneous hemangiomas in BNH without hepatic hemangiomas undergo spontaneous regression within the first year of life.