Home About us Editorial board Search Ahead of print Current issue Archives Submit article Instructions Subscribe Contacts Login 
Home Print this page Email this page Small font size Default font size Increase font size Users Online: 996

 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 21  |  Issue : 1  |  Page : 45-46

Angiokeratoma circumscriptum of children: A case of postinjury with a lesion on the foot


Department of Plastic and Cosmetic Surgery, Daping Hospital, Army Medical University, Chongqing, China

Date of Submission11-Jun-2019
Date of Decision23-Oct-2019
Date of Acceptance27-Nov-2019
Date of Web Publication24-Dec-2019

Correspondence Address:
Dr Yuangang Lu
Department of Plastic and Cosmetic Surgery, Daping Hospital, Army Medical University, 10 Changjiang Branch Road, Chongqing 400042
China
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijpd.IJPD_58_19

Rights and Permissions
  Abstract 


Angiokeratoma circumscriptum vascular lesions are defined by ectasia of the papillary dermal vessels, and the etiology is unknown. We report a rare case of angiokeratoma circumscriptum following the excision of a lipoma, from the same area, on the right foot 3 years prior. Therefore, this case indicates that the injury could be involved in the development of angiokeratoma circumscriptum.

Keywords: Angiokeratoma, angiokeratoma circumscriptum, Post injury angiokeratoma


How to cite this article:
Tan L, Li X, Lu Y. Angiokeratoma circumscriptum of children: A case of postinjury with a lesion on the foot. Indian J Paediatr Dermatol 2020;21:45-6

How to cite this URL:
Tan L, Li X, Lu Y. Angiokeratoma circumscriptum of children: A case of postinjury with a lesion on the foot. Indian J Paediatr Dermatol [serial online] 2020 [cited 2020 Jan 21];21:45-6. Available from: http://www.ijpd.in/text.asp?2020/21/1/45/273834

Author Liuchang Tan and Xinying Li contributed equally to this work





  Introduction Top


Since the first description of angiokeratomas in 1890, angiokeratomas can be classified into five types: (1) angiokeratoma of Mibelli; (2) angiokeratoma of Fordyce; (3) angiokeratoma corporis diffusum; (4) angiokeratoma circumscriptum; and (5) solitary or multiple angiokeratomas.[1],[2] Angiokeratoma circumscriptum was first described by Fabry in 1915, which is the least frequent among the five types of angiokeratomas, and the etiology is unknown, and its lesions are usually present at birth and may be associated with cavernous hemangioma, nevus flammeus, or hemangiectic hypertrophy.


  Case Report Top


A 5-year-old Chinese child presented with a 1-year history of plaques and red to black-colored papules on the right foot. The child accepted the excision of a lipoma on the same area 3 years ago. Examination revealed plaques and black-colored papules, measuring 0.2–10 mm, forming a coalescent plaque predominantly on the medial malleolus of the right foot [Figure 1]. Initially, the lesion was small red plaque confined to the excision scar. Subsequently, the plaque areas became hyperkeratotic with age. The lesion was asymptomatic except for symptom of pruritus. There was no relevant family history. A dermoscopic pattern presented by dark lacunae, whitish veil, and peripheral erythema with hemorrhagic crust [Figure 2]. The biopsy specimen from a representative papule showed dilated thin-walled capillary capillaries with hyperkeratosis and acanthosis [Figure 3]. According to the clinical and histopathological features, angiokeratoma circumscriptum was diagnosed. No recurrence has been observed during a 6 months' follow-up period after treated with an intense pulse light source.
Figure 1: Plaques and black-colored papules forming a coalescent plaque predominantly on the medial malleolus of the right foot

Click here to view
Figure 2: (a) A dermoscopic pattern presented by dark lacunae, a whitish veil, and peripheral erythema with a hemorrhagic crust. Dilated vascular spaces in the papillary dermis with thrombosis presented as dark lacunae. Hyperkeratosis and acanthosis appeared as a whitish veil. (b) In the dermoscopic view, the presence of well-demarcated, round, and dark lacunae which histologically represent dilated vessels with thrombosis

Click here to view
Figure 3: Histopathology showing hyperkeratosis acanthosis. Papillary dermis showing dilated blood-filled vessels (H and E, ×100)

Click here to view



  Discussion Top


Angiokeratoma circumscriptum is typically situated on the lower legs or feet in a unilateral or otherwise asymmetrical distribution involving the thighs, buttocks, trunk or occasionally other areas. Clinically, angiokeratoma circumscriptum is characterized by aggregates of hyperkeratotic erythematous papules (deep red to blue–black in color) and nodules, which may coalesce to form verrucous plaques.[3] The lesions become increasingly studded with warty keratotic papules or nodules.[4] There is no tendency to spontaneous regression.

Angiokeratoma circumscriptum is often misdiagnosed as verrucous hemangioma, Cobb syndrome, angioma serpiginosum, lymphangioma circumscriptum, verrucae, malignant melanoma.[5] However, in the dermoscopic view, these above lesions hardly show whitish veils.[6] Dermatoscopy is useful for the diagnosis of angiokeratoma.[1]

Angiokeratoma circumscriptum's histopathology consist of dilated, thin-walled, congested papillary dermal vessels with secondary epidermal reaction changes, such as acanthosis and/or hyperkeratosis, without involvement of the deep dermis, which are thought to be a vascular lesion arising from local damage of papillary capillaries.

The angiokeratomas are rare verrucous vascular lesions that appear as dark red papules and plaques arranged either discretely or in clusters. As Imperial and Helwig wrote,[2] these lesions are not true angiomas but are merely telangiectasias of preexisting vessels. The exact etiology is unknown, although congenital cause, pregnancy, chilblains, trauma, and tissue asphyxia are given as causal factors.[7] There have been few reports of angiokeratoma circumscriptum following the excision of lipoma. We report a rare case of angiokeratoma circumscriptum following the excision of a lipoma, from the same area, on the right foot 3 years prior. Therefore, this case indicates that injury could be involved in the development of angiokeratoma circumscriptum.[3]

Consent

Informed consent from the parents obtained for publishing the photographs of the child.

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Schiller PI, Itin PH. Angiokeratomas: An update. Dermatology 1996;193:275-82.  Back to cited text no. 1
    
2.
Newton JA, McGibbon DH. The treatment of multiple angiokeratomata with the argon laser. Clin Exp Dermatol 1987;12:23-5.  Back to cited text no. 2
    
3.
Kwon NH, Kim SY, Kim GM. A case of post-injury angiokeratoma circumscriptum of the chest. Ann Dermatol 2010;22:66-8.  Back to cited text no. 3
    
4.
Kim JH, Kim MR, Lee SH, Lee SE, Lee SH. Dermoscopy: A useful tool for the diagnosis of angiokeratoma. Ann Dermatol 2012;24:468-71.  Back to cited text no. 4
    
5.
Campos-do-Carmo G, Ramos-e-Silva M. Dermoscopy: Basic concepts. Int J Dermatol 2008;47:712-9.  Back to cited text no. 5
    
6.
Zaballos P, Daufí C, Puig S, Argenziano G, Moreno-Ramírez D, Cabo H, et al. Dermoscopy of solitary angiokeratomas: A morphological study. Arch Dermatol 2007;143:318-25.  Back to cited text no. 6
    
7.
Saha M, Barlow R, Bunker CB. Angiokeratoma circumscriptum of the penis. Br J Dermatol 2006;154:775-6.  Back to cited text no. 7
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

Top
 
 
  Search
 
Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
Access Statistics
Email Alert *
Add to My List *
* Registration required (free)

 
  In this article
Abstract
Introduction
Case Report
Discussion
References
Article Figures

 Article Access Statistics
    Viewed110    
    Printed0    
    Emailed0    
    PDF Downloaded25    
    Comments [Add]    

Recommend this journal