Association of capillary haemangioma with bilateral hydronephrosis in an infant *

Introduction Capillary (or strawberry) haemangiomas are benign localized tumours of blood vessels, usually occurring in the head and neck region. They have a malformational, hamartomatous basis and spontaneously disappear within the first few years of life. They occur in 1-2% of all neonates and have a female: male ratio of 3:1. They typically arise early in life, grow rapidly during a proliferative phase and then slowly regress in an involutional phase. We present a case of capillary haemangioma of face, neck and upper part of chest associated with bilateral hydronephrosis.


Introduction
Capillary (or strawberry) haemangiomas are benign localized tumours of blood vessels, usually occurring in the head and neck region 1 .They have a malformational, hamartomatous basis and spontaneously disappear within the first few years of life 2 .They occur in 1-2% of all neonates and have a female: male ratio of 3:1 1 .They typically arise early in life, grow rapidly during a proliferative phase and then slowly regress in an involutional phase 1 .We present a case of capillary haemangioma of face, neck and upper part of chest associated with bilateral hydronephrosis.

Case report
A 7 month old boy came to the outpatient clinic with his parents.He was conscious and playful, immunized according to age, with normal development.His systemic examination was unremarkable.His mother had developed gestational diabetes mellitus and she gave a history of swollen kidneys in the antenatal ___________________________________________ 1  ultrasound examination of fetus.Infant had developed a reddish skin rash (which later became hypertrophied) on lower face including left parotid region, some anterior part of neck and extending to upper part of chest from the 18 th day of life, which was diagnosed as a capillary haemangioma (Figures 1A and 1B).

*Permission given by parents to publish photograph
He was treated with an oral β-blocker, propranolol 1.5 mg/kg and Timolol drops for local application on affected site.No side effects were recorded.On further evaluation, his ultrasound scan of abdomen revealed a right kidney 5.4×3.0 cm in size and a left kidney 6.1×4.1 cm in size at 4 months.Ultrasound examination did not reveal any intra-abdominal mass.Hence, to minimize radiation hazard to the infant, CT scan or MRI of the abdomen was not performed to rule out the possibility of intra-abdominal mass lesion.Later, bilateral hydronephrosis was confirmed by intravenous pyelography, which suggested left sided vesicoureteral junction (VUJ) obstruction and right sided pelvicalyceal ureteric junction (PUJ) obstruction (Figure 1C).Patient was surgically managed with ureteric reimplantation on left side and pyeloplasty was performed on the right side.The post-surgical course of infant was uneventful with normal renal function.

Discussion
Most haemangiomas are solitary; when multiple (with or without associated lesions in internal organs) or affecting a large segment of body, condition is known as multifocal angiomatosis 1 .Kasabach and Merrit reported a case of haemangioma involving skin and deep soft tissues of thigh in 1940 3 .Haemangiomas may present as small isolated lesions or as large masses that cause systemic symptomatology, impair vital or sensory functions or cause disfigurement, despite their self-limited course 1 .Their pathogenesis and optimal management remain unknown Department of Regenerative Medicine and Department of Paediatrics, 2 Department of Pathology, Laboratory Medicine, Transfusion Services and Immunohaematology, 3 Department of Nephrology and Transplantation Medicine, 4 Department of Anaesthesiology and Critical Care Medicine, 5 Department of Urology and Transplantation, G. R. Doshi and K.M. Mehta Institute of Kidney Diseases & Research Centre -Dr.H. L. Trivedi Institute of Transplantation Sciences *Correspondence: umangpaedia@yahoo.co.in (Received on 01 March 2016: Accepted after revision on 22 April 2016) The authors declare that there are no conflicts of interest Personal funding was used for this project.Open Access Article published under the Creative Commons Attribution CC-BY License.
7.There was no external compression observed due to any mass producing (PUJ) obstruction and VUJ) obstruction in right and left kidney respectively in this patient.Corticosteroids are the first line of treatment for infantile capillary haemangiomas.Other options include laser therapy, interferon alfa-3, vincristine and propranolol which can inhibit the growth of these haemangiomas7.