Back issues No.4 - 2007 / Resident Forum  

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Eccrine Angiomatous Hamartoma with Features of Verrucous Hemangioma
Ching-Wen Huang , Ya-Ju Hsieh , Yu-Hung Wu , Yang-Chin Lin , Kuo-Ming Chuang
Dermatol Sinica 25: 282-284, 2007

A 43-year-old man presented with a solitary asymptomatic lump on his left lower leg present since early childhood. The lesion had enlarged slowly and become verrucous. There was no evidence of sweating in the lesion evoked by physical work or emotional stress. The patient was otherwise healthy. Physical examination disclosed a raised, 3 × 2-cm, purple-to-black hyperkeratotic plaque on the left shin (Fig. 1). Histopathologic examination of tissue from an excisional biopsy showed a hyperkeratotic epidermis with prominent papillomatosis and irregular acanthosis. Numerous thin-walled, dilated, blood-filled vessels filled the papillary dermis (Fig. 2). There was lobulated proliferation of intermingled mature eccrine ducts, glands, vascular structures, and adipose tissue in the mid- and deep dermis extending to the subcutis (Fig. 3A, 3B). The histopathologic diagnosis was EAH with features of verrucous hemangioma (VH). There was no recurrence of the lesion six months after the excision.
EAH was first described by Lotzbeck in 1859 and given its name by Hyman et al. in 1968.1 Clinically, it presents as single or multiple hemangiomata or nevus-like red, blue-brown, or flesh-colored nodules or plaques,2-4 The lesions are usually asymptomatic, but pain and hyperhidrosis have been reported in 32% to 42% of cases.2 Most EAHs are congenital but adult onset has been reported.5, 6 There is no gender predilection.3, 4 Most lesions are reported on the extremities but rarely involve the forehead, neck, chest, back, and sacral region.3, 6
Histologically, EAH usually lies in the deep dermis and contains a proliferation of eccrine coils and numerous capillary channels surrounding or intermingled with the eccrine structures. It may also contain fatty tissue and pilar structures. The various components and their ratios are the major features differentiating EAH from other hamartomas, such as blue rubber bleb nevi, fibrous hamartoma of infancy, smooth muscle hamartoma, angiolipoma, and eccrine nevus.7, 8
Other vascular components, such as spindle cell hemangiomas, cavernous channels, and arterio-venous malformation have been reported as part of EAH.5, 11-13, Epidermal change, however, has only been documented in a few cases. Such reports include mild hyperkeratosis, papillomatosis, and mild acanthosis.7, 9, 10 We have seen only one published case describing EAH with the prominent epidermal signs resembling VH as seen in our patient.14
VH is a rare, localized, vascular malformation that is usually congenital and rarely presents later in life. Most cases present as circumscribed, warty papules or plaques with a predilection for the lower extremities.15-18 VH has histopathologic features of capillary or cavernous hemangiomas in the dermis and subcutaneous tissue associated with reactive epidermal hyperkeratosis, acanthosis, and papillomatosis. The superficial component in VH resembles angiokeratoma but differs with respect to the deeper dermal components.16
The unusual presentation in our patient may be attributed to either verrucous hyperkeratotic epidermal change occurring in a congenital EAH or hamartomatous proliferation of sweat gland apparatus in a congenital VH. The development of VH has been hypothesized to depend on local factors such as trauma, secondary infection, or a physiological interaction between keratinocytes and endothelial cells.10, 13, 16 While there’s no way to be certain, we speculate that our patient most likely had VH as a reactive phenomenon in an already established EAH, rather than VH being the primary lesion.7, 9, 10
Both EAH and VH are benign lesions that do not regress spontaneously. Excision is usually curative, although VH reportedly recurs in up to 33%.16 For this reason, simple excision or electrocautery are suitable for small lesions less than 2 cm, while larger lesions require wide, deep excision and, sometimes, skin grafting.
In summary, we report an unusual case of EAH with overlying verrucous hemangiomas-like features. It is interesting to speculate on the pathophysiology and our findings expand the histological spectrum of this hamartoma.

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