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E-IJD CORRESPONDENCE
Year : 2016  |  Volume : 61  |  Issue : 1  |  Page : 124
Multiple infantile hemangiomas treated successfully with oral propranolol


1 Department of Paediatric Medicine, NRS Medical College and Hospital, 138, AJC Bose Road, Kolkata, West Bengal, India
2 Department of Radiology, NRS Medical College and Hospital, 138, AJC Bose Road, Kolkata, West Bengal, India

Date of Web Publication15-Jan-2016

Correspondence Address:
Radheshyam Purkait
Department of Paediatric Medicine, NRS Medical College and Hospital, 138, AJC Bose Road, Kolkata, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0019-5154.174171

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How to cite this article:
Purkait R, Basu R, Das R, Bhadra R. Multiple infantile hemangiomas treated successfully with oral propranolol. Indian J Dermatol 2016;61:124

How to cite this URL:
Purkait R, Basu R, Das R, Bhadra R. Multiple infantile hemangiomas treated successfully with oral propranolol. Indian J Dermatol [serial online] 2016 [cited 2019 Jun 18];61:124. Available from: http://www.e-ijd.org/text.asp?2016/61/1/124/174171


Sir,

Infantile hemangiomas (IH) are the most common benign vascular tumors, characterized by a period of growth after birth and eventual spontaneous involution. [1] Although most hemangiomas are benign and require no intervention, but intervention is warranted for large disfiguring lesions or lesions that impair vital functions. Propranolol, a nonselective beta-blocker, has been used sporadically as a safe and effective treatment modality for IH since its discovery by Lιautι-Labrθze et al. in 2008. [2],[3] Here, we report such a case.

An otherwise healthy 1-month-old female infant was brought to our outpatient clinic with the complaint of inability to open her left eye since day 15 of her life. Cutaneous examination revealed that there was a large brightly erythematous, lobulated, soft plaque on the left side of the forehead, involving the left upper eyelid obscuring the visual axis. A similar lesion was also noted on the left upper lip [Figure 1]a. Ocular examination revealed normal anterior and posterior segments. Examination of oral and nasal cavity was non-contributory. Laboratory investigations including the complete blood count and routine blood chemistry did not reveal any abnormality. Magnetic resonance imaging (MRI) of the orbit showed a large soft tissue lesion measuring 36 mm × 32 mm [Figure 2]a. Abdominal ultrasonography and echocardiography were unremarkable. Colour Doppler study as well as MRI of lip confirmed the diagnosis of capillary hemangiomas [Figure 2]b. For histopathological confirmation, an excision biopsy of the lesions was planned but parents did not consent. The treatment was initially started with low-dose propranolol (0.17 mg/kg per dose three times daily) under careful monitoring of heart rate and blood glucose levels. Gradually the dose was augmented over 3 days to 2 mg/kg/day. After 4 weeks of treatment, the lesions regressed by 50% and the child was able to open her left eye [Figure 1]b. By the end of the third month, the lesions reduced to three forth of their original size [Figure 1]c. Propranolol was continued up to the end of first year of life and tapered over next 2-3 weeks. At the end of therapy, the child showed complete regression of all the lesions [Figure 1]d. Now she is 3 year old and enjoying a happy life without recurrence of tumors.
Figure 1: (a) Photograph of patient at the time of diagnosis (b) Post-treatment photograph at 1month (c) at 3 months (d) and at 15 months

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Figure 2: (a) T2W Coronal MRI of orbit showing fairly large hyperintense soft tissue lesion involving lateral and superior portion of the left orbit (b) T2 STIR sequence axial MRI of lip showing well defined large hyperintense soft tissue lesion involving left paramedian region of upper lip

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Although multiple theories have been proposed regarding mechanism of action of propranolol on IH but the exact mechanism is still obscure. One of most accepted theories suggested by Storch and Hoeger [4] is that propranolol might interfere with endothelial cells, vascular tone, angiogenesis and apoptosis. According to their hypothesis, early effects are characterized by brightening of the hemangioma surface within 1-3 days after start of therapy as a result of vasoconstriction due to decreased release of nitric oxide. This is followed by intermediate effects which are mainly due to the blocking of proangiogenic factors, i.e. vascular endothelial growth factor, basic fibroblast growth factor, matrix metalloproteinase-2, -9 and result in growth arrest of hemangioma. Finally, long-term effects of propranolol are attributed by induction of apoptosis in proliferating capillary endothelial cells that results in tumor regression.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

 
   References Top

1.
Frieden IJ, Haggstrom AN, Drolet BA, Mancini AJ, Friedlander SF, Boon L, et al. Infantile hemangiomas: Current knowledge, future directions. Proceedings of a research workshop on infantile hemangiomas, April 7-9, 2005, Bethesda, Maryland, USA. Pediatr Dermatol 2005;22:383-406.  Back to cited text no. 1
    
2.
Léauté-Labrèze C, Dumas de la Roque E, Hubiche T, Boralevi F, Thambo JB, Taïeb A. Propranolol for severe hemangiomas of infancy. N Engl J Med 2008;358:2649-51.  Back to cited text no. 2
    
3.
Sans V, de la Roque ED, Berge J, Grenier N, Boralevi F, Mazereeuw-Hautier J, et al. Propranolol for severe infantile hemangiomas: Follow-up report. Pediatrics 2009;124:e423-31.  Back to cited text no. 3
    
4.
Storch CH, Hoeger PH. Propranolol for infantile haemangiomas: Insights into the molecular mechanisms of action. Br J Dermatol 2010;163:269-74.  Back to cited text no. 4
    


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