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  Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 8  |  Issue : 4  |  Page : 254-256  

Multiple verrucous hemangiomas: A case report with new therapeutic insight


1 Department of Dermatology, Venereology and Leprosy, Post Graduate Institute of Medical Education and Research (PGIMER) and Dr. Ram Manohar Lohia Hospital, New Delhi, India
2 Department of Pathology, Vardhman Mahavir Medical College and Safdarjung Hospital, New Delhi, India

Date of Web Publication5-Jul-2017

Correspondence Address:
Sidharth Tandon
Department of Dermatology, Venereology and Leprosy, Post Graduate Institute of Medical Education and Research (PGIMER) and Dr. Ram Manohar Lohia Hospital, New Delhi
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/idoj.IDOJ_313_16

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   Abstract 


Verrucous hemangioma is an uncommon congenital vascular malformation, which may clinically masquerade angiokeratoma, lymphangioma circumscriptum, or malignant melanoma. Differentiation is essential owing to varied therapeutic and prognostic implications. We present a rare case of multiple verrucous hemangiomas in a teenage girl who presented with multiple warty lesions over the dorsal aspect of the left foot since birth. Magnetic resonance imaging (MRI) scan was suggestive of a vascular malformation, and skin biopsy showed ectatic blood vessels extending from the papillary dermis into the subcutaneous tissue, diagnostic of verrucous hemangioma. A combination of 0.05% halobetasol propionate with 3% salicylic acid ointment was advised. This therapeutic intervention resulted in significant resolution of the warty lesions over a period of 2 months, following which surgical excision was performed. The implication is that we can use a combination of super potent topical steroid with salicylic acid as an adjunct to surgical resection.

Keywords: Hemangioma, topical steroid, verrucous


How to cite this article:
Singh J, Sharma P, Tandon S, Sinha S. Multiple verrucous hemangiomas: A case report with new therapeutic insight. Indian Dermatol Online J 2017;8:254-6

How to cite this URL:
Singh J, Sharma P, Tandon S, Sinha S. Multiple verrucous hemangiomas: A case report with new therapeutic insight. Indian Dermatol Online J [serial online] 2017 [cited 2021 Jun 25];8:254-6. Available from: https://www.idoj.in/text.asp?2017/8/4/254/209615


   Introduction Top


Verrucous hemangioma is an uncommon, localized, congenital vascular malformation which presents as a bluish red lesion in the early stages and eventually acquires a warty surface as a protective phenomenon.[1] Clinically, lesions may simulate angiokeratoma, lymphangioma circumscriptum, verrucous epidermal nevus, or even malignant melanoma.[2] Although thorough clinicoradiological examination helps in narrowing the differential diagnosis, histomorphological assessment of this masquerader is imperative owing to therapeutic and prognostic implications.

We describe a rare case of multiple verrucous hemangiomas in a teenage girl with numerous warty lesions over the left foot. Our case highlights the clinical, radiological, histopathological, and immunohistochemical features of verrucous hemangioma along with utility of topical steroids as a therapeutic intervention.


   Case Report Top


A 15-year-old female presented to the outpatient dermatology department with chief complaints of brownish painful warty lesions over the dorsum of the left foot since birth. Initially, the lesions were red, soft, and gradually increased in size and number up to the age of 5 years to become warty, brownish black, hard, and tender. History of bleeding usually following minor trauma was noted. General physical examination was within normal limits. Cutaneous examination revealed multiple hyperpigmented, hyperkeratotic plaques consisting of closely-set warty papules varying from 2 cm × 2.5 cm to 3 cm × 3.5 cm involving the dorsum of the left foot and lateral malleolus [Figure 1]. Surface of the lesion was firm and verrucous with no ulceration, bleeding, or atrophy. The lesion was noncompressible and diascopy was negative. No other hair, nail, or mucosal abnormality was noted. There was no palpable regional lymphadenopathy. Radiographic films of the left foot showed normal findings. Color Doppler revealed ill-defined heteroechoic soft tissue densities at the anterior, superior, and lateral aspect of the left foot showing few echogenic foci and slow venous flow. Magnetic resonance imaging (MRI) scan showed mild delayed enhancing lesion in the skin and subcutaneous plane of the left foot. A probable diagnosis of vascular malformation, capillary venous type, was given and skin biopsy was performed. Hematoxylin and eosin (H and E) sections from skin biopsy showed hyperkeratosis, parakeratosis, acanthosis, and papillomatosis of the epidermis. Underlying papillary dermis showed multiple thin-walled and ectatic blood filled spaces with mild perivascular mononuclear cell inflammatory infiltrate [Figure 2]a. Similar vascular channels were also seen in the mid dermis and subcutaneous tissue [Figure 2]b. These channels were lined by flattened endothelial cells. On immunohistochemistry (IHC), the endothelial cells showed positivity for cluster of differentiation 34 (CD 34) [Figure 2]c, CD 31, and Wilm's tumor 1 (WT-1) [Figure 2]d. Based on the immunomorphological features a final diagnosis of verrucous hemangioma of the left foot was rendered. Patient was started on a combination of topical 0.05% halobetasol propionate with 3% salicylic acid ointment twice daily. The lesions reduced in size and became less warty over a period of 2 months [Figure 3]. Patient was then referred to plastic surgery for excision. Postoperative period was uneventful with no recurrence after 1-year follow-up.
Figure 1: A teenage girl with multiple hyperpigmented, hyperkeratotic plaques consisting of closely set warty papules involving the dorsum of the left foot and lateral malleolus

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Figure 2: (a) Photomicrograph showing skin biopsy with variable sized dilated blood vessels (arrow) extending from the superficial papillary dermis down to the mid dermis (H and E, ×100) (b) Photomicrograph showing ectatic blood vessels in the reticular dermis and subcutaneous fat (arrow) (H and E, ×100). On immunohistochemistry, the endothelial cells show positivity for (c) CD 34 (Immunostain ×200) and (d) WT-1 (Immunostain ×200)

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Figure 3: Significant resolution of lesions two months after halobetasol and salicylic acid ointment application

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   Discussion Top


Verrucous hemangioma is an uncommon, congenital vascular malformation first denominated by Imperial and Helwig in 1967.[3] These are most frequently located on the lower extremities with rare cases reported on the head, trunk, upper limbs, and glans penis. Although lesions are usually solitary and localized, a rather uncommon variant of multiple disseminated lesions without systemic involvement has been described.[4] Our case revealed similar multiple verrucous lesions over the dorsal aspect of the left foot. However, there was no disseminated pattern or systemic involvement.

Clinically, lesions of verrucous hemangioma may closely simulate angiokeratoma, and differentiation is essential because incomplete excision of verrucous hemangioma would lead to recurrence of the lesion from the deeper components.[5] MRI is the diagnostic modality of choice because it delineates the dermal and subcutaneous plane of these lesions and guides biopsy.[6] Histopathological examination remains the gold standard for diagnosis with presence of ectatic channels extending into the deeper dermis vis-à -vis angiokeratoma circumscriptum [Table 1]. Immunohistochemical positivity of endothelial cells lining the vascular channels for CD 34 and CD 31 antibodies acts as an adjunct.
Table 1: Comparison between verrucous hemangioma and angiokeratoma circumscriptum

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The accurate categorization of verrucous hemangioma still remains elusive. To clarify the nosology of this vascular anomaly, Trindade et al. first reported WT-1 positivity in 13 cases of verrucous hemangiomas.[7] Similar immunohistochemical results were obtained in our case. However, the recently identified genetic mutation in the Ang1-TIE2 (angiopoietin 1-tunica internal endothelial cell kinase) pathway confirms that verrucous hemangioma has genetic features consistent with a venous malformation, despite having an immunoprofile similar to vascular neoplasms.[8] This also opens the door for targeted therapy.

Verrucous hemangiomas do not resolve spontaneously and early diagnosis is important for timely surgical excision and better cosmetic result. Superficial ablative procedures such as electrocautery and cryosurgery invariably lead to recurrence of the lesion.[9] Thus, surgery remains the mainstay of treatment. While topical steroids have a promising result in most superficial hemangiomas, the therapeutic role of this treatment modality has never been studied in the setting of verrucous hemangiomas. Numerous theories explaining the mechanism of steroids have been published with the concept of vasoconstriction being widely accepted.[10] A combination of halobetasol propionate with salicylic acid ointment used in our case resulted in significant resolution in 2 months following application. Salicylic acid aids in the penetration of the topical steroid in this warty hyperkeratotic lesion owing to its keratolytic action. The implications of this therapy is that we can use a combination of super potent topical steroid with salicylic acid to reduce the size of these lesions, which would aid in the ease of subsequent surgical resection. However, due to paucity of literature, the potential utility and outcome of this therapeutic modality cannot be ascertained.


   Conclusion Top


To conclude, this case was presented for its atypical presentation as multiple warty lesions. Moreover, our aim was to diminish the size of the warty lesions using a combination of halobetasol propionate with salicylic acid, with the intention of reducing the amount of bleeding at surgery and better cosmetic results. Further studies are recommended to study the response of these lesions to this combination therapy.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Wang G, Li C, Gao T. Verrucous hemangioma. Int J Dermatol 2004;43:745-6.  Back to cited text no. 1
    
2.
Jain VK, Aggarwal K, Jain S. Linear verrucous hemangioma on the leg. Indian J Dermatol Venereol Leprol 2008;74:656-8.  Back to cited text no. 2
[PUBMED]  [Full text]  
3.
Imperial R, Helwig EB. Verrucous hemangioma: A clinicopathologic study of 21 cases. Arch Dermatol 1967;96:247-53.  Back to cited text no. 3
[PUBMED]    
4.
Cruces MJ, de la Torre C. Multiple eruptive verrucous hemangiomas: A variant of multiple hemangiomatosis. Dermatologica 1985;171:106-11.  Back to cited text no. 4
[PUBMED]    
5.
Mani MZ, Feierabend TC. Verrucous Hemangioma, Report of a case treated by skin grafting. Ind J Dermatol Venereol Leprol 1982;48:116-7.  Back to cited text no. 5
    
6.
Nagarajan K, Banushree CS. Usefulness of MRI in delineation of dermal and subcutaneous verrucous hemangioma. Indian J Dermatol 2015;60:525.  Back to cited text no. 6
[PUBMED]  [Full text]  
7.
Trindade F, Torrelo A, Requena L, Tellechea Ó, Del Pozo J, Sacristán F, et al. An immunohistochemical study of verrucous hemangiomas. J Cutan Pathol 2013;40:472-6.  Back to cited text no. 7
    
8.
Knöpfel N, Hoeger P. Verrucous Hemangioma or Verrucous Venous Malformation? Towards a Classification Based on Genetic Analysis. Actas Dermosifiliogr 2016;107:427-8.  Back to cited text no. 8
    
9.
Yang CH, Ohara K. Successful surgical treatment of verrucous hemangioma: A combined approach. Dermatol Surg 2002;28:913-20.  Back to cited text no. 9
    
10.
Gangopadhyay AN, Sharma SP, Gopal SC, Gupta DK, Panjawani K, Sinha JK. Local steroid therapy in cutaneous hemangiomas. Indian Pediatr 1996;33:31-3.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]
 
 
    Tables

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