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CASE REPORT
Year : 2016  |  Volume : 7  |  Issue : 6  |  Page : 533-535  

Great saphenous vein dilatation with reflux at the saphenofemoral junction: A rare underlying association of eccrine angiomatous hamartoma


Department of Dermatology, Venereology and Leprosy, Jawaharlal Nehru Medical College (DMIMSU-DU), Sawangi, Wardha, Maharashtra, India

Date of Web Publication11-Nov-2016

Correspondence Address:
Sanjiv Choudhary
28 Modern Nagpur society, Chhatrapati Nagar, Ring road, Nagpur – 440 015, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2229-5178.193912

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   Abstract 

Eccrine angiomatous hamartoma (EAH) is an exceedingly rare benign tumor-like lesion prevalent in childhood, which can produce pain and marked sweating. Histological features include proliferation of eccrine sweat glands and angiomatous capillary channels. It may be rarely associated with underlying pathological conditions. A 15-year-old female patient presented with multiple tender reddish papules and nodules coalescing to form plaques of 10 × 8 cm over the anterior aspect of the right lower thigh since birth. It was associated with hypertrichosis, hyperhidrosis, pain, and occasional bleeding on trauma. Histopathological examination of the lesion showed increased proliferation of both eccrine and angiomatous channels. Ultrasonography and Color Doppler of the right thigh showed dilatation of the great saphenous vein (GSV) above the right knee, with evidence of grade 3 reflux at saphenofemoral junction. Magnetic resonance imaging revealed large dilated GSV with slow flow and venous malformation in the anterior part of the right knee

Keywords: Dilated great saphenous vein, eccrine angiomatous hamartoma, saphenofemoral junctional reflux


How to cite this article:
Choudhary S, Sabiha Q, Singh AL. Great saphenous vein dilatation with reflux at the saphenofemoral junction: A rare underlying association of eccrine angiomatous hamartoma. Indian Dermatol Online J 2016;7:533-5

How to cite this URL:
Choudhary S, Sabiha Q, Singh AL. Great saphenous vein dilatation with reflux at the saphenofemoral junction: A rare underlying association of eccrine angiomatous hamartoma. Indian Dermatol Online J [serial online] 2016 [cited 2019 May 21];7:533-5. Available from: http://www.idoj.in/text.asp?2016/7/6/533/193912


   Introduction Top


Eccrine angiomatous hamartoma (EAH) is a rare nevoid proliferation of eccrine glands, small blood vessels, and occasionally other elements such as fatty tissue and pilar structures in the middle and deep dermis. It is associated with variable clinical manifestations such as pain, hyperhidrosis and hypertrichosis. It is one of the differential diagnoses of painful tender nodular lesions. It may be rarely associated with underlying pathological conditions.


   Case Report Top


A 15-year-old female patient presented with multiple painful red to dark-coloured raised lesions with associated sweating over the right thigh just above the knee since birth. It was initially asymptomatic and smaller in size, but as the age of the patient advanced, it gradually increased in size. Cutaneous examination revealed multiple reddish to blue-brown hyperpigmented papules and nodules at some places coalescing to form plaques of 10 × 8 cm size associated with hypertrichosis over the anterior aspect of the right lower thigh extending upto the knee joint. The circumference of the right lower thigh was greater than that of the left one. Engorgement of veins surrounding the lesion was seen in standing position [Figure 1]. Histopathology showed dermal benign proliferation of capillaries and eccrine glands [Figure 2]: H and E, ×40]. X-ray findings were suggestive of soft tissue involvement without involving bone. Ultrasonography and color doppler revealed dilated great saphenous vein (GSV), dilated draining veins with grade III reflux, and reflux on valsalva at the saphenofemoral junction (SFJ) [Figure 3]. Venogram showed dilated GSV with no reflux in the deep veins. Magnetic resonance imaging revealed vascular malformation with subcutaneous multiple tortuous channels and its draining vein, which was further draining into the large dilated GSV [Figure 3]. For vascular malformation sclerotherapy was done by using sclerosing agent- sodium tetradecyl sulphate, and for reflux at SFJ, endovascular laser ablation was done. However, the patient did not turn up for follow-up.
Figure 1: Multiple reddish to blue-brown hyperpigmented papules, nodules, and plaques with hypertrichosis over the anterior aspect of the right lower thigh

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Figure 2: Benign proliferation of eccrine glands with dilated capillaries filled with red blood cells [H and E, ×40]

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Figure 3: (a) Magnetic resonance imaging - large dilated great saphenous vein. (b) Ultrasonography - reflux on valsalva at the saphenofemoral junction

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   Discussion Top


EAH is a rare condition. It often occurs at birth [1],[2],[3] or during early childhood.[4],[5],[6] It has been reported to occur in both the sexes. The exact pathogenesis is not known but various theories such as abnormal induction of heterotypic dependency with resultant malformation of adnexal as well as mesenchymal elements have been proposed.[7] It is usually asymptomatic but occasionally presents with pain and focal hyperhidrosis (accounting for the designation of sudoriferous angioma adopted by some authors).[8] The hyperhidrosis is presumably an expression of its eccrine component.EAH usually occurs as a solitary lesion, however, cases with multiple lesions have been reported and account for approximately 26% of all cases in the literature.[9] Many underlying associated conditions have been reported in the literature. Lin et al. in their retrospective study of 15 cases of EAH found that, in most of the cases, EAH arose as a single lesion on a lower extremity. The symptoms and signs most commonly associated with EAH were pain (60%), hypertrichosis (13.3%), itching (13.3%), and hyperhidrosis (6.7%). Additional pathological findings associated with EAH were hemangioma (13.3%), verrucous hemangioma (6.7%), arteriovenous malformation (6.7%), and angiokeratoma (6.7%).[10]

Till date approximately 50 cases of EAH are published. However, EAH with dilated GSV along with reflux at saphenofemoral junction has not been reported in the literature.

Acknowledgement

We are thankful to Dr. Pankaj Banode, Professor of Radiology Department, JNMC, Sawangi, Wardha for carring out all the necessary radiological investigations and interpreting the radiological findings of this case.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Hyman AB, Harris H, Brownstein MH. Eccrine angiomatous hamartoma. N Y State J Med 1968;68:2803-6.  Back to cited text no. 1
    
2.
Domonkos AN, Suarez LS. Sudoriparousangioma. Arch Dermatol 1967;96:552-3.  Back to cited text no. 2
    
3.
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. 3
    
4.
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. 4
    
5.
Sulica RL, Kao GF, Sulica VI, Penneys NS. Eccrine angiomatous hamartoma (nevus): Immuno-histochemical findings and review of the literature. J Cutan Pathol 1994; 21:71-5.  Back to cited text no. 5
    
6.
Smith VC, Montesinos E, Revert A, Ramon D, Molina I, Jorda E. Eccrine angiomatous hamartoma: Report of three patients. Pediatr Dermatol 1996;13:139-42.  Back to cited text no. 6
    
7.
Zeller DJ, Goldman RL. Eccrine-pilar angiomatous hamartoma: A report of a unique case. Dermatologica 1971;143:100-4.  Back to cited text no. 7
    
8.
Sanmartin O, Botella R, Alegre V, Martinez A, Aliaga A. Congenital eccrine angiomatous hamartoma. Am J Dermatopathol 1992;14:161-4.  Back to cited text no. 8
    
9.
Lee SY, Chang SE, Choi JH, Sung KJ, Moon KM, Koh JK. Congenital eccrine angiomatous hamartoma: Report of two patients. J Dermatol 2001;28:338-40.  Back to cited text no. 9
    
10.
Lin YT, Chen CM, Yang CH, Chuang YH. Eccrine angiomatous hamartoma: A retrospective study of 15 cases. Chang Gung Med J 2012;35:167-76.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]


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