Funding sources This work is supported by grants from the Gillies McIndoe Foundation and the Wellington Regional Plastic Surgery Unit Research & Education Trust.
Treatment of infantile haemangioma with captopril
Version of Record online: 25 JUL 2012
© 2012 The Authors. BJD © 2012 British Association of Dermatologists
British Journal of Dermatology
Volume 167, Issue 3, pages 619–624, September 2012
How to Cite
Tan, S.T., Itinteang, T., Day, D.J., O’Donnell, C., Mathy, J.A. and Leadbitter, P. (2012), Treatment of infantile haemangioma with captopril. British Journal of Dermatology, 167: 619–624. doi: 10.1111/j.1365-2133.2012.11016.x
Conflicts of interest None declared.
Parts of this work were presented at the Joint Annual Scientific Meeting of the American Society of Plastic Surgeons and the Canadian Society of Aesthetic Plastic Surgeons, Toronto, Canada, 1–5 October, 2010; the 80th Annual Scientific Congress of the Royal Australasian College of Surgeons, Adelaide, Australia, 2–6 May, 2011; the 16th World Congress of the International Confederation for Plastic, Reconstructive and Aesthetic Surgery, Vancouver, Canada, 21–27 May, 2011; the Plastic Surgery Congress, and the 19th International Society for the Study of Vascular Anomalies Workshop, Malmo, Sweden, 16–19 June 2012, Gold Coast, Australia, 7–10 July, 2011
- Issue online: 27 AUG 2012
- Version of Record online: 25 JUL 2012
- Accepted manuscript online: 25 APR 2012 11:43AM EST
- Accepted for publication 16 April 2012
Background Infantile haemangioma (IH) has recently been reported as an aberrant proliferation and differentiation of a primitive mesoderm-derived haemogenic endothelium regulated by the renin-angiotensin system (RAS), leading us to propose angiotensin converting enzyme (ACE) as a potential therapeutic target.
Objectives To present initial results of our open-labelled observational clinical trial using captopril, an ACE inhibitor (ACEi), in the treatment of problematic proliferating IH.
Methods After initial screening investigations, infants with problematic IH were admitted for initiation of captopril with a 0·1 mg kg−1 test dose orally, followed by 0·15 8-hourly over 24 h. This was then followed by dose escalation to 0·3 mg kg−1 8-hourly for another 24 hours. The dosage was increased to 0·5 mg kg−1 8-hourly 1 week later, if a noticeable involution had not already occurred. The response of IH to captopril was documented clinically and photographically before and after treatment and any side-effect was recorded.
Results Two boys and six girls aged 5–22 weeks (mean 12·9) with problematic IH were recruited with the lesions located in nasal tip (n = 1), cervicofacial (n = 3), periorbital (n = 1) and perineal (n = 2) areas, and shoulder (n = 1). Transient mild renal impairment occurred in one subject but resolved spontaneously. No other complication was observed. The IHs in all patients responded to captopril at a dosage of 1·5 mg kg−1 daily which led to a dramatic response in three, moderate response in two, and slow response in three patients. Continued involution of IHs was observed during the follow-up period of 8–19 months (mean 15·8) in all subjects. Treatment was ceased at 14 months of age in seven patients with no rebound growth. In the remaining patient, rapid healing occurred with ongoing gradual reduction in the size and colour of a large ulcerated retroauricular lesion following 5·5 months of treatment. The lesion was excised to address its persistent distortion of the ear.
Conclusions The response of IH to an ACEi supports a critical role for the RAS in IH and represents a paradigm shift in the understanding and treatment of this enigmatic condition.