ARTICLE
Auteur(s) :, Yuichiro Tsunemi*,
Kiyo Shimazu, Hidehisa Saeki, Hironobu Ihn, Kunihiko Tamaki
Department of Dermatology, Faculty of Medicine, University of
Tokyo, 7-3-1 Hongo, Bunkyo-ku, Tokyo 113-8655, Japan
accepté le 19 Août 2004
Eccrine angiomatous hamartoma (EAH) is a rare condition
characterized histologically by an increased number of eccrine
structures and numerous vascular elements [1-10]. EAH may also
contain fatty tissue and pilar structures, and sometimes shows
mucinous stroma [1-10]. The mucin is not usually abundant [3, 6].
In this report, we describe a rare variant of EAH with massive
mucin deposition, especially around the eccrine and pilar
structures.
Case report
A 23-year-old Japanese man presented with a nodule on the right
ring finger, which had been present since birth and had enlarged
with the growth of the patient. Physical examination revealed a
brown, 16 mm-diameter, elastic soft nodule on the proximal dorsal
aspect of the right ring finger. The patient felt slight
tenderness. There was mild hypertrichosis but no hyperhidrosis on
the surface of the nodule ( (figure 1) ). Mucinous
material oozed out during the biopsy. Histological examination
showed an increased number of eccrine glands and ducts, and
numerous vascular cavities adjacent to the eccrine structures at
the interface between the deep dermis and subcutaneous tissue (
(figure 2A) ).
Vascular cavities consisted of dilated small capillaries and
irregular thick-walled vessels (( figure 2B) ), and there
were also hair follicles among them. In addition, this lesion had
abundant myxoid stroma around the eccrine glands and ducts ( (figure 2C) ), and
some mucinous stroma around vessels and pilar structures, which was
stained for acid mucin by alcian blue stain (pH 2.5) ( (figure 2D) ). This patient
was diagnosed as having EAH.
Discussion
EAH is a rare benign condition with characteristic histological
findings; an increased number of eccrine secretory and ductal
elements, in close association with angiomatous channels [1-10].
Clinically it usually presents as one or several nodules or a
solitary large plaque on the extremities and sometimes on the trunk
[1-10]. Most cases are present at birth or develop before puberty
[1-10]. Its color may be red, violaceous, blue, yellow, brown, or
skin-color [2, 4, 6-9]. Hyperhidrosis, pain, or hypertrichosis may
be apparent, but not in all cases [2-10]. It may also contain fatty
tissue and pilar structures [1, 2, 4-9]; in these respects, our
case was a typical one. Sometimes it shows mucinous stroma [2-4, 6,
7, 9], but the mucin is not usually abundant [3, 6]. Our case was a
rare one in that markedly increased mucin deposition was found.
There has been only one case of EAH with a large pool of mucin and
that was reported by Seraly et al. [10]. They termed it
eccrine-pilar angiomatous mucinous nevus. These two cases indicate
that EAH includes a rare variant that has a large amount of mucin
as one of its hamartoma components.
References
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