Hemangioma is a most common tumor of infancy, and more common in preterm and female. Benign neonatal hemangiomatosis (BNH) is a type of infantile hemangioma and first reported by Stern et al. In BNH, multiple widespread superficial capillary hemangiomas are present without visceral involvement. We report a case of BNH presenting with the strikingly unilateral distribution of hemangiomas and reviewed literature.
A 26-day-old female neonate presented with multiple red small swellings on the left side of face and body. At birth, there were only two small red swellings on the left upper eyelid. After 15th day of life, new red swellings were appearing, almost daily and growing rapidly. Otherwise, the baby was well and on breastfeeding. On examination of the neonate, vitals were normal. There were multiple superficial, dome-shaped, dull red colored hemangiomas of various sizes on the left side of face and body; and associated with edema of left upper and lower eyelids [Figure 1]. Telangiectasias were also seen on the face. There was no mucosal involvement. Ophthalmological examination including intraocular pressure was normal. Other systemic examination was normal. Computed tomography of brain and orbit was normal. Ultrasonography of the abdomen was normal. Echocardiography was normal. Stool for occult blood was negative. Platelet count and tests for coagulation were normal. As there was no visceral involvement, we did diagnosis of BNH. In view of the rapid growth of lesions and severe lid edema, we treated our patient with oral prednisolon (2 mg/kg/day) for 5 days and then stopped. With this treatment, edema, the size of lesions and appearance of new lesions was also decreased [Figure 2]. The benign nature of the disease is explained to parents and advised for regular follow-up.
Benign neonatal hemangiomatosis is a rare, self-limiting condition, without complications, and spontaneous regression of lesions is likely. There are three phases of hemangioma; growth phase lasting up to a year, then stabilization phase, followed by involution phase. The similar aggressive growth of lesions in BNH is described in the previous study. The growth of lesions in infantile hemangioma is nonlinear and time of greatest growth is between 5.5 and 7.5 weeks of age. We found unilateral (left sided) distribution of multiple hemangiomas in BNH, which is very rare and not reported in literature till now.
Indications for intervention in infantile hemangioma are severe hemorrhage, ulceration, congestive cardiac failure, threatened interference with vital functions such as respiration, feeding, vision, hearing, and hemangiomas that may lead to permanent disfigurement. Drugs used for treatment are oral prednisolon, recombinant interferon-α 2a or 2b, vincristine, and propranolol. Propranolol is the most preferred treatment at present. For local therapy, clobetasol and triamcinolone are used. Controversy exists about the effectiveness of pulsed dye laser.
Facial hemangiomas involving the significant area of the face are frequently associated with PHACE syndrome and orbital hemangioma. Visceral hemangiomas are present in diffuse neonatal hemangiomatosis, and it is often associated with complications and mortality. All patients with multiple hemangiomas should be investigated for visceral hemangioma.
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