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CASE REPORT
Year : 2019  |  Volume : 20  |  Issue : 2  |  Page : 174-176

Congenital acral verrucous hemangioma: A rare entity


Department of Pathology and Paediatric Surgery, Pt. B.D. Sharma PGIMS, Rohtak, Haryana, India

Date of Web Publication29-Mar-2019

Correspondence Address:
Dr. Renuka Verma
Pt. B.D. Sharma PGIMS, Rohtak, Haryana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijpd.IJPD_45_17

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  Abstract 


Verrucous hemangioma is an uncommon capillary or cavernous hemangioma with reactive epidermal changes of hyperkeratosis, acanthosis, and papillomatosis. Lesions are mostly seen at birth or appear during childhood. These growths are usually located on the lower extremities, and involvement is generally unilateral. Diagnosis of this unusual presentation can only be confirmed following histopathology. Surgical excision resulted in a satisfactory functional and cosmetic outcome.

Keywords: Angiokeratoma, hemangioma, vascular disease, verrucous


How to cite this article:
Rattan KN, Kalra R, Verma R, Singh S, Gupta M, Ralli M. Congenital acral verrucous hemangioma: A rare entity. Indian J Paediatr Dermatol 2019;20:174-6

How to cite this URL:
Rattan KN, Kalra R, Verma R, Singh S, Gupta M, Ralli M. Congenital acral verrucous hemangioma: A rare entity. Indian J Paediatr Dermatol [serial online] 2019 [cited 2019 Oct 19];20:174-6. Available from: http://www.ijpd.in/text.asp?2019/20/2/174/255206




  Introduction Top


Verrucous hemangioma is a rare variety of angiomatous nevi usually involving the lower extremities. It has been reported under a diversity of names such as angiokeratoma circumscriptum neviforme, unilateral verrucous hemangioma, and keratotic hemangioma.[1]

They do not regress spontaneously and require a large, deep excision; in contrast, angiokeratomas respond to various means of therapy (e.g., cryotherapy, electrocautery, and argon laser).[2]


  Case Report Top


A 6-month-old child presented in the pediatric outpatient department with painless and gradually progressive warty lesion of size 2.5 cm × 2.0 cm on the second finger of the left leg. The lesion was present since birth. On examination, it was solitary hyperpigmented, hyperkeratotic plaque involving the pulp space. The surface of the lesion was firm and verrucous with no ulceration, bleeding, or atrophy. The lesion was noncompressible. Regional lymph nodes were not enlarged. Systemic examination was normal. Routine laboratory investigations and X-ray chest were also normal. While, radiograph of foot revealed a soft-tissue lesion of approx.2.0 cm in diameter. Underlying bone was normal [Figure 1].
Figure 1: Dorsal-plantar X-ray, showing lesion of approx. 2.5 cm × 2.0 cm on the second finger of the left foot

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Clinically differential diagnosis of tuberculosis verrucosa cutis and angiokeratoma was kept. The entire growth was excised by a vertical excision, and the excised material was subjected to histopathological examination.

Hematoxylin- and eosin-stained section of the specimen showed marked hyperkeratosis, parakeratosis, papillomatosis, and elongation of the rete ridges [Figure 2]. In deeper dermis, numerous small-to-large vascular channels were seen lined by flattened endothelial cells and filled with blood [Figure 3]. Intervening dermal fibrosis and chronic inflammatory infiltrate were also present [Figure 4].
Figure 2: Epidermis revealing marked hyperkeratosis, parakeratosis, papillomatosis, and elongation of the rete ridges (H and E, ×100)

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Figure 3: Deeper dermis revealing numerous small-to-large vascular channels lined by flattened endothelial cells (H and E, ×200)

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Figure 4: Dermis revealing intervening dermal fibrosis (Van Gieson, ×200)

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These findings were different from angiokeratoma as it involves only the papillary dermis without any extension to the deep dermis and the subcutaneous fat. Ziehl–Neelsen staining for acid-fast bacilli using 20% H2SO4 was negative. A final diagnosis of verrucous hemangioma was confirmed.


  Discussion Top


Vascular malformations are present at birth in 60% of cases, becoming apparent by the time the child reaches 2 years of age in 90% of cases. Regression is not generally spontaneous.[3] According to the 1996 classification of the International Society for the Study of Vascular Anomalies/Mulliken, vascular anomalies may be classified as simple (capillary, cavernous, lymphatic, venous, and arterial) or combined.[4] In 1967, Imperial andHelwig introduced the term “verrucous hemangioma” and defined it as a congenital vascular malformation comprising a capillary or cavernous hemangioma in the dermis and subcutaneous tissue associated with reactive epidermal acanthosis, papillomatosis, and hyperkeratosis, distinguishing it from angiokeratoma.[2]

Verrucous hemangioma has been reported in the literature with a variety of names including hemangioma unilateralis neviforme, unilateral verrucous hemangioma, angiokeratoma circumscriptum neviforme, nevus vascularis and nevus unius lateris, keratotic hemangioma, nevus angiokeratoticus, nevus keratoangiomatosus, and papulous angiokeratoma.[1]

Verrucous hemangioma usually involves the lower extremities, and involvement is generally unilateral. In our case, the lesion had been present since birth and involved his left leg. Initially, the lesions are nonkeratotic, soft, and bluish-red in color, but with time, they progressively enlarge and become hyperkeratotic and verrucous.[5] Secondary bleeding and infection are frequent complications, which often result in reactive papillomatosis and hyperkeratosis, and thus, the older lesions acquire a warty surface.[6]

The diagnosis of verrucous hemangioma is mainly made by histopathological examination, although clinical correlation is required to make a correct diagnosis. Histologically, verrucous hemangioma shows hyperkeratosis, variable epidermal acanthosis, and papillary telangiectasias overlying a deep cavernous or capillary hemangioma.

Clinically, verrucous hemangioma mimics angiokeratoma, but histopathologically, they can be clearly differentiated as the former involves the dermis and subcutaneous fat and the latter involves only the papillary dermis.[7] Hence, deep excisional surgery is the proper treatment for verrucous hemangioma as a superficial therapeutic approach would be unsuccessful because of the deep angiomatous proliferation. Superficial ablative procedures such as electrocautery, cryosurgery, and laser ablation almost invariably lead to recurrence of the lesion. A combined approach using surgery and erbium laser has been reported with good results.[8]

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.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Calduch L, Ortega C, Navarro V, Martínez E, Molina I, Jordá E, et al. Verrucous hemangioma: Report of two cases and review of the literature. Pediatr Dermatol 2000;17:213-7.  Back to cited text no. 1
    
2.
Imperial R, Helwig EB. Verrucous hemangioma. A clinicopathologic study of 21 cases. Arch Dermatol 1967;96:247-53.  Back to cited text no. 2
    
3.
Welsh O, Vera-Cabrera L, Salinas-Carmona MC. Mycetoma. Clin Dermatol 2007;25:195-202.  Back to cited text no. 3
    
4.
Revankar SG. Dematiaceous fungi. Mycoses 2007;50:91-101.  Back to cited text no. 4
    
5.
Loria PR, Derbes VJ, Krafchuk JD. Keratotic hemangiomas. AMA Arch Derm 1958;77:216-9.  Back to cited text no. 5
    
6.
Wang G, Li C, Gao T. Verrucous hemangioma. Int J Dermatol 2004;43:745-6.  Back to cited text no. 6
    
7.
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. 7
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8.
Yasar A, Ermertcan AT, Bilac C, Bilac DB, Temiz P, Ozturkcan S, et al. Verrucous hemangioma. Indian J Dermatol Venereol Leprol 2009;75:528-30.  Back to cited text no. 8
[PUBMED]  [Full text]  


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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