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CASE REPORT
Year : 2007  |  Volume : 52  |  Issue : 2  |  Page : 99-101
Late onset eccrine angiomatous hamartoma treated with intralesional sclerosant: A case report and brief review of literature


1 Department of Dermatology, Amrita Institute of Medical Sciences and Research Center, Kochi, Kerala, India
2 Department of Plastic and Reconstructive Surgery, Amrita Institute of Medical Sciences and Research Center, Kochi, Kerala, India
3 Department of Pathology, Amrita Institute of Medical Sciences and Research Center, Kochi, Kerala, India

Correspondence Address:
Feroze Kaliyadan
Department of Dermatology, Amrita Institute of Medical Sciences and Research Center, Elamakkara, Kochi - 26, Kerala
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0019-5154.33288

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   Abstract 

A 29-year-old male patient presented to us with multiple swellings on his left leg associated with localized sweating and occasional throbbing pain. Based on the clinical features and histopathology, a diagnosis of Eccrine angiomatous hamartoma was made. The patient showed good response with regression of lesions after four intralesional injections of ethoxysclerol (polidocanol). We present this as a case of late onset eccrine angiomatous hamartoma with multiple lesions, responding to treatment with intralesional ethoxysclerol.


Keywords: Eccrine angiomatous hamartoma, intralesional sclerosant, sudoriparous angioma


How to cite this article:
Kaliyadan F, Sundeep V, Hiran K R, Fouzia Z. Late onset eccrine angiomatous hamartoma treated with intralesional sclerosant: A case report and brief review of literature. Indian J Dermatol 2007;52:99-101

How to cite this URL:
Kaliyadan F, Sundeep V, Hiran K R, Fouzia Z. Late onset eccrine angiomatous hamartoma treated with intralesional sclerosant: A case report and brief review of literature. Indian J Dermatol [serial online] 2007 [cited 2018 Jul 20];52:99-101. Available from: http://www.e-ijd.org/text.asp?2007/52/2/99/33288



   Introduction Top


Eccrine angiomatous hamartoma (EAH) (also known as sudoriparous angioma) is a rare cutaneous lesion. Though EAH is a benign lesion, treatment in the form of excision or sclerosants may be required in some symptomatic cases. [1] We report a case of EAH treated with intralesional sclerosant injections.

A 29-year-old male patient, working in an offshore oil rig in Angola (Africa), presented to us with complaints of multiple swellings on the left leg, gradually increasing in size over the last 15 years. The patient had sought medical advise primarily because he noticed occasional throbbing pain in the lesions over the last few months. The pain according to the patient was worsened by prolonged standing and cold exposure. On examination multiple firm nodules were seen mainly over the posterior aspect of the lower leg. The nodules varied in size from 2 cm (centimeters) to 5 cm. Lesions had a bluish hue and were nontender on palpation [Figure - 1]. On firmly stroking the surface beaded drops of perspiration were visible over the lesion [Figure - 2]. Based on the history and clinical features a possibility of Eccrine angiomatous hamartoma was considered. This was confirmed by histopathological studies, which revealed circumscribed foci composed of a mixture of mature eccrine glands, capillary-sized blood vessels and lipomatous tissue [Figure - 3],[Figure - 4],[Figure - 5],[Figure - 6]. A Doppler study was done to assess the depth of the lesion. Doppler study revealed a diffuse subcutaneous predominantly hypoechoic lesion (greater than 1 cm thickness) seen in the subcutaneous plane of calf.

The case was discussed with the plastic surgeon to consider excision and grafting. However considering the diffuse nature and the depth of the lesion excision was deferred. Instead a trial with sclerosing agents was tried. The patient was given five sittings of intralesional sclerosant with 2 ml of 3% ethoxysclerol (polidocanol), with an interval of two weeks between the sittings. By the end of the fifth sitting, the lesion had regressed considerably [Figure - 7],[Figure - 8]. When the patient was reviewed a month after the last sitting the lesions had further regressed and the patient reported that his symptoms had significantly improved with the patient being free of pain even after prolonged standing or strenuous physical activity.


   Discussion Top


EAH is a rare cutaneous tumor, characterized histologically by proliferation of eccrine secretory coils and ducts, surrounding capillary angiomatous channels and occasionally other minor elements. [2] Typically EAH presents as a solitary flesh-colored or violaceous, papule or plaque appearing at birth or during childhood usually showing an acral prediliction. It may or may not be associated with hyperhidrosis and pain. [3],[4] EAH is considered to be a variant of the eccrine nevus. Though it classically presents in childhood, there have been reports of adult onset cases some presenting as late as 65 years. [5],[6]

Histopathology EAH typically demonstrates the presence of a mixture eccrine structures and vascular channels. [7] EAH has been seen to be associated with a number of other histological elements including adipose tissue, mucin and neural elements. [7],[8],[9] Immunohistochemical studies on EAH have been done demonstrating the presence of antigens commonly found in eccrine glands like carcinoembryonic antigen (CEA) and S-100 protein. [2] Other antigens demonstrated include CD34, CD44, human nerve growth factor receptor and Ulex europaeus antigens. [2]

Medical intervention is generally indicated either for the pain or for cosmetic reasons. The treatment of choice is surgical excision, [4] however in cases in which surgical removal may pose practical difficulties, other options like intralesional sclerosants and lasers have been tried. [5],[10]

Aethoxysclerol (also called aethoxysklerol, polidocanol) is a common sclerosant used in the treatment of varicose veins. It acts by provoking damage to the endothelium by attacking the lipids of the membrane, thereby causing cell damage and inflammation. Aethoxysclerol is also a local anesthetic and is therefore the injection process is relatively painless. [11] The use of sclerosants is generally safe though complications like necrosis and scarring can be rarely seen. An isolated case of cardiac arrest attributed to polidocanol sclerotherapy has been reported. [12]

In our case medical intervention was planned mainly because of the associated pain. Our case had certain uncommon features like the presence of multiple lesions, the presence of lipomatous tissue in histology and the relatively older age of onset. Because of the more diffuse and extensive distribution of the lesion, surgical excision posed practical difficulties. Hence,we opted for intralesional sclerosants which we found worked quite effectively in causing the lesion to regress.

 
   References Top

1.Cebreiro C, Sanchez-Aguilar D, Gomez Centeno P, Fernandez-Redondo V, Toribio J. Eccrine angiomatous hamartoma: Report of seven cases. Clin Exp Dermatol 1998;23:267-70.  Back to cited text no. 1      
2.Sulica RL, Kao GF, Sulica VI, Penneys NS. Eccrine angiomatous hamartoma (nevus): Immunohistochemical findings and review of the literature. J Cutan Pathol 1994;21:71-5.  Back to cited text no. 2  [PUBMED]    
3.Morrell DS, Ghali FE, Stahr BJ, McCauliffe DP. Eccrine angiomatous hamartoma: A report of symmetric and painful lesions of the wrists. Pediatr Dermatol 2001;18:117-9.  Back to cited text no. 3  [PUBMED]  [FULLTEXT]  
4.Silver SG, Ho VC. Benign epithelial tumors. In: Freedberg IM, Eisen AZ, Wolff K, Austen KF, Goldsmith LA, Katz SI, editors. Fitzpatrick's Dermatology in general medicine. 6 th ed. McGraw-Hill: New York; 2003. p. 767-85.  Back to cited text no. 4      
5.Gadroy A, Belhadjali H, Bayle P, Albes B, Lamant L, Bazex J. Eccrine angiomatous hamartoma: An atypical case. Ann Dermatol Venereol 2003;130:337-9.  Back to cited text no. 5      
6.Laeng RH, Heilbrunner J, Itin PH. Late-onset eccrine angiomatous hamartoma: clinical, histological and imaging findings. Dermatology 2001;203:70-4.  Back to cited text no. 6  [PUBMED]  [FULLTEXT]  
7.Challa VR, Jona J. Eccrine angiomatous hamartoma: A rare skin lesion with diverse histological features. Dermatologica 1977;155:206-9.  Back to cited text no. 7  [PUBMED]    
8.Lee HW, Han SS, Kang J, Lee MW, Choi JH, Moon KC, et al . Multiple mucinous and lipomatous variant of eccrine angiomatous hamartoma associated with spindle cell hemangioma: A novel collision tumor? J Cutan Pathol 2006;33:323-6.  Back to cited text no. 8  [PUBMED]  [FULLTEXT]  
9.Donati P, Amantea A, Balus L. Eccrine angiomatous hamartoma: A lipomatous variant. J Cutan Pathol 1989;16:227-9.  Back to cited text no. 9  [PUBMED]    
10.Srinivas CR, Rao PL. Sudoriparous angioma--regression following intravascular aethoxysclerol, a sclerosing agent. Br J Dermatol 1988;119:111-3.  Back to cited text no. 10  [PUBMED]    
11.Shwartz RA. Cosmeceuticals. [Last updated on 2006 Feb 28]. Available from: http://www.emedicine.com/derm/topic509.htm. [Last accessed on 2006 Jun 9].  Back to cited text no. 11      
12.Marrocco-Trischitta MM, Guerrini P, Abeni D, Stillo F. Reversible cardiac arrest after polidocanol sclerotherapy of peripheral venous malformation. Dermatol Surg 2002;28:153-5.  Back to cited text no. 12  [PUBMED]  [FULLTEXT]  


    Figures

  [Figure - 1], [Figure - 2], [Figure - 3], [Figure - 4], [Figure - 5], [Figure - 6], [Figure - 7], [Figure - 8]

This article has been cited by
1 Eccrine angiomatous hamartoma: A rare multifocal variant with features suggesting trauma
Naik, V., Arsenovic, N., Reed, M.
Dermatology Online Journal. 2009; 15(9): Art 6
[Pubmed]



 

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