ARTICLE
Auteur(s) :, Yoshihiro Handa1,2,*,
Yoichi Kato1, Hirohiko Ishikawa3, Yasushi
Tomita2
1Division of Dermatology, Anjo kosei Hospital, 28
Higashi-hirokute, Anjo-cho, Anjo, 446-8602, Japan
2Department of Dermatology, Nagoya University Graduate
School of Medicine, 65 Tsurumai-cho, Showa-ku, Nagoya, 466-8550,
Japan
3Department of Plastic and Reconstructive Surgery,
Nagoya University Graduate School of Medicine, 65 Tsurumai-cho,
Showa-ku, Nagoya, 466-8550, Japan
accepté le 1 Septembre 2004
Basal cell carcinoma (BCC) is the most common type of malignant
tumor of the skin in Caucasians, but is less common in orientals
[1]. BCC may occur anywhere on the skin, but more than 80% are
located on the sun-exposed skin of the face and neck [2-7], with
less than 1% of the cases located on unexposed areas of the
genitalia [2, 3, 8-10]. About 200 cases have been described on the
vulva [9], but only 41 cases of scrotal BCC have been reported in
the English language literature [2-4, 6, 8, 9, 11-14]. In 51 cases
of perianal and genital BCCs, most frequent histopathologic
subtypes is nodular (66%), and the second is superficial (18%) [3],
but giant BCC of the superficial subtype is rare [7]. We report a
very rare case of a superficial basal cell carcinoma over 5 cm
in diameter at the large end on the scrotum in a Japanese patient.
Case report
A 74-year-old man presented with an itchy lesion on his scrotum
which had been slowly enlarging over the previous 10 years. The
lesion had been diagnosed as chronic eczema and treated with
corticosteroid ointment by a practitioner for 3 years without
improvement. We suspected that the lesion was extramammary Paget’s
disease and performed an incisional biopsy. The histopathological
diagnosis was compatible with BCC. Surgery was performed 5 years
after our first observation because of an initial refusal of the
patient. The patient had no history of BCC, any other skin tumors,
intake of arsenic, nor treatment with irradiation.
On examination he had a light reddish plague 5.5 × 2.0 cm
in size with erosions in spots and a grayish black periphery
located on the right upper side of his scrotum ( (figure 1) ).
Histopathologic examination of the total resected specimens
showed buds and irregular proliferations of tumor tissue attached
to the undersurface of the epidermis. The tumors had small
hyperchromatic nuclei with peripheral palisading. Large amounts of
melanin pigment were present within the tumor nests and in the
upper dermis. Tumor nests were separated from the surrounding
stroma by cleft formations, which are typical features for
superficial BCC ( (figure 2) ).
Neither recurrence nor metastasis has been observed during 59
months of follow-up examinations.
Discussion
Although BCC is more common in males than females [1, 5, 7, 14], in
the genital area, however, vulvar lesions are more frequently
reported [3, 8, 9]. Although the precise reason for this female
predominance is yet unknown, one possible explanation is the
presence of chronic skin irritation such as chronic vulvovaginitis,
because most patients with vulvar BCC tend to be post menopausal
[8].
Scrotal carcinoma is rare, with an average annual incidence of
0.1 per 100,000 [6, 11, 12]. The majority of these are squamous
cell carcinoma [9], with 5-15% being BCCs [4, 11-13]. A recent
report described that, of all non-nevoid BCC syndrome cases, only
0.27% (51/18,910 BCCs) were located within the perianal and genital
regions, and 0.03% (6 cases) on the scrotum [3]. In other reports,
0.05% (1/2072 BCCs) were located on the scrotum [14], and none was
located on the scrotum or male genitalia in the first episodes of
688 BCCs [15].
Exposure to ultraviolet (UV) radiation is the most likely cause
and accounts for the preferential distribution of these lesions
over heavily sun-exposed areas of the body [3, 4, 6, 13]. Other
etiologic factors must be considered whenever BCCs are located in
non-sun-exposed areas including the genital area. Early reports
have suggested that scrotal carcinoma may be a result of
nonspecific factors such as poor hygiene and chronic irritation [4,
6]. Another possible explanation is that a generalized depression
in immunosurveillance from advancing age or UV light at distinct
sites predisposes their development [3, 4, 6, 11-13]. However,
other factors must be involved, or BCC would be more frequent in
patients on immunosuppressive medication [4, 13]. Chronic
inflammation, dermatitis, exposure to arsenic and previous
radiation therapy have also been reported [1-4, 9, 12, 13]. The
patient of the present case however had no history of arsenic
intake and irradiation. No other common risk factors were
identified in terms of occupation, coexisting illness or exposure
to potential carcinogens [4, 13].
As the most common clinical presentation of BCC is nodules or
ulcers, the differential diagnosis of the scrotal BCC included
extramammary Paget’s disease, Bowen disease, melanoma [4, 9, 11,
12], angiokeratoma, pigmented seborrheic keratosis and melanocytic
nevus. Paget’s disease of the scrotum usually manifests as red,
raised, indurated and occasionally ulcerative lesions [11, 12]. We
highly suspected the lesion of the present case to be extramammary
Paget’s disease because of the location and erythematous plaque.
The absence of raised induration together with the grayish black
periphery might have differentiated superficial BCC from
extramammary Paget’s disease.
Most BCC are less than 1 cm in diameter. Giant BCC was
defined as a tumor more than 5 cm in the greatest dimension
according to the classification of the American Joint Committee on
Cancer [5, 7, 10]. Less than 1% of all BCCs reach this size [5].
The reasons for the enlarged size in the present case were that the
patient neglected trivial itching for 7 years, the lesion was
treated with corticosteroid ointment for 3 years having been
diagnosed as chronic eczema, and then excision of the lesion was
delayed because of the patient’s refusal.
Histopathological subtypes of BCC can be grouped as
nonaggressive; nodular and superficial subtypes, and aggressive;
morpheaform, micronodular and metatypical subtypes [5, 7]. In 50
cases of giant BCC, 36 cases (72%) showed aggressive
histopathological subtypes, 13 cases (26%) showed nodular, and
superficial subtype was only 1 case (2%) [7]. BCCs are locally
invasive and slowly enlarging. They rarely metastasize and result
in the death of the patients [5]. Metastases occurred in 9.5% (4/42
cases) of scrotal BCCs reported. However, due to the small number
of case reports and the higher likelihood of metastasis than
reported, it is difficult to determine whether the biologic
behavior of scrotal BCCs is more aggressive than BCCs located in
other areas [2, 4, 6]. Giant BCCs greater than 10 cm, with
long duration and aggressive histopathological subtypes often have
a high mortality representing fatal and metastatic BCCs [10]. On
the other hand, the superficial subtype BCC that do not invade
deeply into the subcutaneous tissue and muscle as in our case are
considered not to have a high mortality rate.
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