ARTICLE
Auteur(s) : A
Shimizu, A Tamura, S Tanaka, T Syuto, Y Nagai, O Ishikawa
Department of Dermatology, Gunma University Graduate School of
Medicine, 3-39-22 Showa-machi, Maebashi, Gunma 371-8511, Japan
accepté le 5 Octobre 2004
Verrucous carcinoma is a distinct variant of cutaneous squamous
cell carcinoma, which may affect the skin or the oral mucosa. The
clinicopathological concept was first established by Ackerman, when
he reported 31 patients with “verrucous carcinoma” of the oral
cavity [1]. Here, we report a patient with pedunculated verrucous
carcinoma on the right thigh. Moreover, we reviewed 17 previously
reported cases, highlighting the clinical features.
Case report
A 73-year-old Japanese man presented with a 1-year history of a
constantly growing tumor on the right thigh. Physical examination
revealed a pedunculated, cauliflower-like tumor measuring 80 × 70 ×
13 mm. The size of the pedicle was 18 × 13 mm. The tumor
surface was covered by greasy yellow-to-brownish crusts with a
foul-smelling discharge ( (figure 1) ). Coarse
reticular pigmentation was observed around the tumor. There was no
lymphadenopathy. He had experienced neither a burn nor radiation
therapy on the thigh. Pseodomonas Aeruginosa was cultured from the
surface discharge. The computed tomography of the abdomen and
pelvis and 67Ga scintigraphy showed no abnormality.
Surgical resection and split-thickness skin grafting was performed
under lumbar anesthesia.
On histological examination, the excised tumor showed exophytic
growth through the sliced pedicle. The tumor branched out to form
several lobules connected to each other by thin stroma ( (figure 2A) ). The
epidermis was massively hyperplastic and papillomatously-folded
with marked hyperkeratosis and parakeratosis. The epithelial
strands contained many keratin-filled cysts and formed crypts.
There were large, bulbous, downward proliferations of
well-differentiated squamous cells ( (figure 2B) ). At deeper
levels, the tumor was sharply demarcated by an intact basement
membrane. In some parts, tumor cells displayed a loss of polarity
and slight nuclear atypia ( (figure 2C) ). There were
dense mononuclear cell infiltrations in the stroma. The biopsy
specimen taken from a pigmented lesion around the tumor revealed
slight infiltration of mononuclear cells in the upper dermis.
Flattening of the epidermis, fibrosis and hyalinization of collagen
bundles, which are suggestive findings of a burn scar, were not
seen. We speculated that the pigmentation might result from
repeated lymphangitis caused by the secondary infection of the
tumor.
For analyzing the human papillomavirus (HPV) status, DNA was
extracted from paraffin-embedded sections and subjected to
polymerase chain reaction (PCR) amplification. PCR using GP5(+)/GP
6(+) primers targeting specific sequences within the L1 region of
papillomavirus could not detect any HPV-sequence [2]. After a
follow-up period of 1 year, there has been no recurrence.
Discussion
According to the anatomical localization of verrucous carcinoma,
four types can be distinguished: (a) verrucous carcinoma of the
oral cavity, referred to as oral florid papillomatosis; (b)
verrucous carcnoma of the genitoanal region known as giant
condyloma of Buschke and Löwenstein; (c) verrucous carcinoma of the
plantar region, also known as epithelioma cuniculatum; and (d)
verrucous carcinoma occurring in other areas of the skin described
as cutaneous verrucous carcinoma.
Compared to oral, genitoanal and plantar verrucous carcinomas,
cutaneous verrucous carcinomas arising elsewhere in the skin are
exceedingly rare [3]. In our review of the literature published
between 1981 and 2003, we found 17 cases (summarized in table 1(
Table 1 )) of verrucous carcinoma not
localized in the classical regions [4-16]. There was a male
predisposition. The ages of the patients ranged from 47 to 83
years, with the mean age of 64 years. As for the tumor location, 6
cases were on the buttocks and 4 cases were on the head or leg,
followed by 3 cases on the trunk, excluding the buttock. Although
some reports were unclear on the shape, no case of pedunculated
verrucous carcinoma has been reported previously. The differential
diagnoses of the pedunculated cauliflower-like tumor of our case
include eccrine porocarcinoma and trichilemmal carcinoma. Tamura et
al. reported that 3 of 8 Japanese cases of trichilemmal carcinomas
were pedunculated [17]. In spite of the similarity of the clinical
findings, it is possible to distinguish a verrucous carcinoma from
these two entities by histological findings. As for the preceding
lesions, 4 cases were chronic ulcer and 2 cases were
syringocystadenoma papilliferum, followed by 1 case of hidradenitis
suppurativa or inflamed cyst. In half of the cases, obvious
preceding lesions were noted.
The pathogenesis of verrucous carcinoma is not yet fully
elucidated. The majority of reports deal with the mucosal type of
verrucous carcinoma and etiological factors discussed include HPV
infection and chemical carcinogenesis, e.g. due to tobacco and
betel quid chewing [3]. Indeed, several HPV types have been
detected in verrucous carcinomas including low-risk HPV types 6 or
11 and high-risk HPV types 16 and 18. HPV types 6 and 11 have been
very frequently demonstrated in Buschke-Löwenstein tumors [3].
Although HPV-DNA detection was performed in only 4 cases of
cutaneous verrucous carcinoma, two lesions were positive for HPV
DNA [8, 9]. One had type 1 and the other had type 11 and 18 HPV
genotypes, respectively. Alternatively, verrucous carcinoma
develops typically in moist areas and is frequently complicated
with chronic inflammation. Although not found in our case, about
half of the patients with cutaneous verrucous carcinomas had
preceding lesions. Chronic ulcer was a main preceding lesion, and
syringocystadenoma papilliferum, hidradenitis suppurativa and
inflamed cyst were also reported. Chronic inflammation may be
partly involved in the development of verrucous carcinomas.
Verrucous carcinoma defined as a low-grade malignant variant of
squamous cell carcinoma with slow invasive growth and low incidence
of metastasis is worthy of recognition. In fact, we sometimes
observe minute invasive foci within the verrucous carcinoma. Thus,
it should be kept in our mind that some verrucous carcinomas have
an invasive nature with metastatic potential [3, 18].
Table 1 Summary of clinical findings of 17 cutaneous
verrucous carcinomas reported between 1981 and 2004
|
The location of cutaneous verrucous carcinoma
|
|
Buttock
|
Head
|
Leg
|
Trunk (Excluding buttock)
|
Total
|
|
6
|
4
|
4
|
3
|
17
|
|
The shape of the tumor
|
|
Nodule
|
Ulcer
|
Subcutaneous massa
|
Others
|
Total
|
|
6
|
3
|
2
|
6
|
17
|
|
HPV DNA
|
|
Type 1
|
Type 11,18
|
Negative
|
Not done
|
Total
|
|
1
|
1
|
3
|
12
|
17
|
asuspected of the lesion developed from epidermal cyst.
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