ARTICLE Pyogenic
granuloma (PG) is a common, usually solitary, benign, angiomatous proliferation
of skin and mucous membrane [1]. This lesion, on rare occasions, develops
within a pre-existing vascular malformation. We report two cases of pyogenic
granuloma arising within port-wine stain.
Case reports
Case 1: A 31-year-old white woman was seen in April 1994 for an exophytic
tumour on the left heel. Since her childhood, she had noticed a "blue
stain" on the left foot, that she used to rub. This lesion appeared progressively
and developed within six months. Physical examination revealed a pedunculated
and ulcerative lesion with 3.5 cm diameter, looking like an achromic melanoma
(Fig. 1). It was secondary
infected, partially covered with scabs and very painful. Histological
examination showed a vascular proliferation arranged in lobules throughout
the dermis and hypodermis (Fig.
2). The epidermis was hyperplastic with collarettes partially present
at the edges. The center was eroded. The diagnosis of PG which had developed
within a port-wine stain was made. This tumour was surgically resected
without any complication or recurrence.
Case 2: A 67-year-old white man, presented in July 1998 with a pedunculated
tumour of the right wrist. Since his childhood, he had noticed port-wine
stains located on the arm, the hand and the right half of the chest. In
1991, a lesion on the right hand was excised, which led to the diagnosis
of PG. He presented in July 1998 with a pedunculated, partially necrotic
lesion that had developed over 6 months within the port-wine stain of
the right wrist (Fig. 3),
first evocative of a malignant vascular proliferation. It measured 4 cm
in diameter, and the center was ulcerated. A second smaller lesion developed
on the site of the prior excision. The two lesions were resected without
recurrence. Histological examination showed a vascular proliferation with
numerous capillaries and venules radially disposed to the skin surface,
located in the mid to upper dermis. It was surrounded by an oedematous
stroma containing an inflammatory cellular infiltrate predominantly composed
of neutrophils. In the lower dermis and hypodermis the stroma was fibrous
and infiltrated by mononuclear cells.
Discussion
PG is a very common solitary lesion of the skin and mucous membrane,
that usually occurs in children and young adults [2]. Subcutaneous [3]
and intravenous [4] localisations have also been described. It is predominantly
located on the trunk, face, and the extremities, including acral areas
[5]. Numerous satellite lesions have also been reported [5-8]. They occur
most of the time after extirpation of a primary single lesion, either
grouped in a localised area [2] (adjacent the original tumor site), or
disseminated.
PG usually develops at the site of a pre-existing
injury or chronic irritation [2, 5]. The involvement of an hormonal factor
is suggested by its development during pregnancy, in women using oral
contraceptives and its regression following delivery [1]. PG also appears
in patients receiving etretinate therapy, which induces skin fragility
[9, 10]. Although PG is a very common lesion, only five cases arising
within a port wine stain have so far been described. They occur in two
young boys [7, 11], a pregnant woman [12, 13] and a woman in the age of
pregnancy [11], as our first case. The last reported case was recently
described in a 65-year-old cirrhotic patient who developed a PG within
a solitary spider angioma [14]. Three of them presented with recurrences
adjacent to the site of the original tumour after a few months, one year
and for the third, during a new pregnancy, respectively [7, 12, 13]. Our
second patient had two recurrences seven years after the first excision,
one on the scar, the other within the port-wine stain, distant from the
initial lesion. There have been a number of theories put forward to explain
the pathogenesis of PG. Some authors suggest that a local traumatism could
trigger an inflammatory response with release of angiogenic factors [5].
Others have observed that port-wine stain and nevus flameus are composed
of arteriovenous (AV) fistulae. They explain the occurrence of PG by the
formation of abnormal AV communications in these highly vascularized area
[8, 15]. Shimizu has recently demonstrated the involvement of NOs Synthase
effector mechanism in promoting angiogenis and rapid growth of PG [16].
Our two observations are rare instances of PG arising within port-wine
stains, which were clinically evocative of malignant tumour. It is important
to know this particular presentation of PG to avoid misdiagnosis and large
unnecessary surgical resection.
Article accepted on 29/4/02
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