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CORRESPONDENCE
Year : 2017  |  Volume : 62  |  Issue : 1  |  Page : 99-100
Eccrine angiomatous hamartoma in a patient with nevus depigmentosus and nevus spilus


1 Department of Dermatology, Fukushima Medical University, Fukushima, Japan
2 Department of Plastic Surgery, Fukushima Medical University, Fukushima, Japan

Date of Web Publication10-Jan-2017

Correspondence Address:
Toshiyuki Yamamoto
Department of Dermatology, Fukushima Medical University, Fukushima
Japan
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0019-5154.198034

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How to cite this article:
Yamamoto T, Hirano M, Ueda K. Eccrine angiomatous hamartoma in a patient with nevus depigmentosus and nevus spilus. Indian J Dermatol 2017;62:99-100

How to cite this URL:
Yamamoto T, Hirano M, Ueda K. Eccrine angiomatous hamartoma in a patient with nevus depigmentosus and nevus spilus. Indian J Dermatol [serial online] 2017 [cited 2020 Nov 24];62:99-100. Available from: https://www.e-ijd.org/text.asp?2017/62/1/99/198034


Sir,

Eccrine angiomatous hamartoma (EAH) is a relatively rare hamartoma of eccrine glands and blood vessels in infancy. Clinically, EAH usually presents with a solitary nodule or plaque, involving the distal extremities.

A 3-year-old boy visited the Department of Plastic Surgery of our hospital, complaining of a painful nodule on the hand which had existed since birth. Physical examination revealed a skin-colored, smooth, small nodule on the left thumb [Figure 1]a. Neither palmar hyperhidrosis nor finger hyperplasia was observed. In addition, the patient had a depigmented macule on the left abdomen [Figure 1]b and a pigmented freckle on the right upper extremity [Figure 1]c, both of which were developed soon after birth. All the skin lesions were considered benign; however, according to the parents' wish, the digital nodule was completely resected and biopsy was performed from the depigmented macule under general anesthesia. Histological examination showed nodular clusters composed of eccrine glands and dilated vessels in the lower dermis [Figure 2]a. Immunohistochemistry showed a number of CD31-positive vessels within the foci of eccrine sweat glands [Figure 2]b. A biopsy specimen from the depigmented macule showed no specific features. Unfortunately, histological examination was not carried out on the nevus spilus on the arm. The patient has been followed up without local recurrence.
Figure 1: (a) A skin-colored, smooth nodule on the left thumb (arrow). (b) A depigmented macule on the left abdomen. (c) A brownish macule on the right upper extremity

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Figure 2: (a) Nests of proliferative eccrine glands embedded within the lipomatous tissues in the lower dermis (H and E, ×40). (b) A number of CD31-positive blood vessels within the eccrine sweat glands (×200)

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EAH usually presents with a papule, nodule, plaque, patch, or macule.[1],[2],[3],[4] Verrucous lesions, hemangioma-like lesions, and verrucous hemangioma-like lesions are rarely reported.[5],[6],[7] The diagnostic criteria are hyperplasia of normal or dilated eccrine glands, close association of the eccrine structures with capillary angiomatous foci, and the presence of pilar, lipomatous, mucinous, or lymphatic structures.[4] EAH is often accompanied by local pain, hypertrichosis, and local hyperhidrosis. Pelle et al. [8] reviewed 37 cases of EAH and described that pain/tenderness was observed in 42.4% and sweating in 34.3%.

EAH is a hamartoma and has been reported to occur in a patient with neurofibromatosis Type I [9] and in one with Cowden syndrome.[10] Our case developed other congenital nevi such as nevus depigmentosus and nevus spilus. We examined throughout the body, but did not detect congenital hamartomas or nevoid lesions of any other origins. Unfortunately, because the patient was an infant, detail examinations such as sweating test, echography, or magnetic resonance imaging were not carried out. Although association of nevus depigmentosus and nevus spilus has rarely been reported,[11] cooccurrence of EAH with either nevus depigmentosus or nevus spilus has not been reported to date. Therefore, the etiopathogenesis of the rare cooccurrence of the three conditions is still unknown. Although the coexistence may be only incidental, accumulation of similar cases is needed in the future. The prognosis of EAH is good, and thus, surgical excision is occasionally chosen, especially in cases accompanying local pain.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Pelle MT, Pride HB, Tyler WB. Eccrine angiomatous hamartoma. J Am Acad Dermatol 2002;47:429-35.  Back to cited text no. 1
    
2.
Holcomb M, Sun G, Eldin K, Brandon K. Verrucous hyperpigmented plaque in a 15-month-old boy. Eccrine angiomatous hamartoma (EAH) associated with verrucous hemangioma (VH). Int J Dermatol 2013;52:25-6.  Back to cited text no. 2
    
3.
Tsuji T, Sawada H. Eccrine angiomatous hamartoma with verrucous features. Br J Dermatol 1999;141:167-9.  Back to cited text no. 3
    
4.
Jorge-Finnigan C, Conejero C, Hernández-Martín A, Sánchez-Gómez J, Noguera-Morel L. Congenital erythematous plaques and papules on the right arm. Eccrine angiomatous hamartoma. Pediatr Dermatol 2015;32:285-6.  Back to cited text no. 4
    
5.
Tennant LB, Mulliken JB, Perez-Atayde AR, Kozakewich HP. Verrucous hemangioma revisited. Pediatr Dermatol 2006;23:208-15.  Back to cited text no. 5
    
6.
Cheong SH, Lim JY, Kim SY, Choi YW, Choi HY, Myung KB. A case of eccrine angiomatous hamartoma associated with verrucous hemangioma. Ann Dermatol 2009;21:304-7.  Back to cited text no. 6
    
7.
Galan A, McNiff JM. Eccrine angiomatous hamartoma with features resembling verrucous hemangioma. J Cutan Pathol 2007;34 Suppl 1:68-70.  Back to cited text no. 7
    
8.
Pelle MT, Pride HB, Tyler WB. Eccrine angiomatous hamartoma. J Am Acad Dermatol 2002;47:429-35.  Back to cited text no. 8
    
9.
Castilla EA, Schwimer CJ, Bergfeld WF, Skacel M, Ormsby A. Eccrine angiomatous hamartoma in a neurofibromatosis Type-1 patient. Pathology 2002;34:378-80.  Back to cited text no. 9
    
10.
Oh JG, Yoon CH, Lee CW. Case of Cowden syndrome associated with eccrine angiomatous hamartoma. J Dermatol 2007;34:135-7.  Back to cited text no. 10
    
11.
Chokoeva A, Wollina U, Lotti T, Tana C, Tchernev G. Nevus depigmentosus associated with nevus spilus:First report in the world literature. Georgian Med News 2015;248:73-6.  Back to cited text no. 11
    


    Figures

  [Figure 1], [Figure 2]



 

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