International Journal of Clinical Medicine, 2013, 4, 21-24
Published Online December 2013 (http://www.scirp.org/journal/ijcm)
http://dx.doi.org/10.4236/ijcm.2013.412A1005
Open Access IJCM
21
Basosquamous Cell Carcinoma of the Lower Lip Arising
from Actinic Cheilitis: Case Report and up Date
Ana Maria de Oliveria Miranda, Thiago de Miranda Ferrari, Daniel Cohen Goldemberg,
Luciana Pantaleão, Andrea Pires, Eliane Pedra Dias
Department of Pathology, Medicine school, Federal Fluminense University, Rio de Janeiro, Brazil.
Received September 2nd, 2013; revised October 3rd, 2013; accepted November 1st, 2013
Copyright © 2013 Ana Maria de Oliveria Miranda et al. This is an open access article distributed under the Creative Commons At-
tribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is prop-
erly cited. In accordance of the Creative Commons Attribution License all Copyrights © 2013 are reserved for SCIRP and the owner
of the intellectual property Ana Maria de Oliveria Miranda et al. All Copyright © 2013 are guarded by law and by SCIRP as a
guardian.
ABSTRACT
Background: Basosquamous carcinoma (BSC) is a rare non-melanoma skin cancer, considered to be a subtype of basal
cell carcinoma (BCC). BSC often produces distant metastases with a higher risk of recurren ce than that of BCC which
is not commonly fo und in the lip. Ca se Report: A 57-year-old white female patient presented an ulcer on her lower lip
that had an ongoing development for over six months. Physical examination, photo documentation, videoroscopy,
scraped cytology, toluidine blue test, and biopsy of the ulcer were carried out. Results: Upon physical examination we
observed an actinic cheilitis associated with the ulcer. Videoroscopy revealed the presence of fissures and erosion that
had not been seen by oroscopy. Toluidine blue test was only positive for the region of the ulcer. Cytological analysis
revealed rare nests compatible with carcinoma. Histopathology of the biopsy revealed a carcinoma with nests lined by
basal cells associated with areas of squamous differentiation. The patient was then referred to surgery for the removal of
the BCC. Analysis of the specimen showed free surgical margins and the immunohistochemical panel did not confirm
the initial diagnosis of BCC, indicating a subtype of BSC. After surgery, the patient has been followed by periodic
consultations. She is well and without further complications . Coments: BSC is considered to be an aggressive and rare
tumor affecting mainly upper face and primarily affects men over 60 years of age. Since our patient is a woman pre-
senting the lesion in the lower lip, this highlights the unusual and interesting presentation of this case report.
Keywords: Basosquamous Carcinoma; Lip; Actinic Cheilitis; Basal Cell Carcinoma
1. Introduction
Malignant labial neoplasms account for approximately
30% of all tumours occurring in the oral cavity including
the lips. Of these 95% are classified as squamous cell
carcinomas (SCC). The factors that influence the devel-
opment of lip SCC include the use of tobacco and alco-
hol, chronic exposure to sunlight and individual suscep-
tibility such as skin and eye color [1].
Basal cell carcinoma (BCC) is the most common ma-
lignant skin tumour worldwide, affecting mainly the head
and neck region, most frequently occurring in upper two
thirds of face. Like SCC, it presents many variant forms,
which need to be correctly diagnosed [2]. Basosquamous
carcinoma (BSC) or metatypical carcinoma [2,5-8] is one
of the subtypes of BCC. It is a rare non-melanoma skin
cancer, which often produces distant metastasis and pre-
sents a high recurrence risk [3,4].
Although the World Health Organization (WHO) and
most researchers consider BSC as an aggressive subtype
of BCC [2,5,9], other authors classify BSC as a separate
entity, causing controversy in the literature. Those that
classify BSC as a different entity propose that th is tumor
contains areas characteristic of both BCC and SCC with
no transition zone between them and with a propensity
for distant metastases thus making it different from BCC
or SCC once these tumors rarely metastasize [3,6,7,10].
Despite classification controversy, it is generally ac-
cepted that BSC is a rare skin tumor (1% to 2% of all
skin cancers), with higher local aggressiveness and a
tendency to generate distant metastasis, highlighting the
importance of a correct diagnosis [11]. Its location is

Basosquamous Cell Carcinoma of the Lower Lip Arising from Actinic Cheilitis: Case Report and up Date 22
mainly in head and neck, with only 1% appearing as la-
bial lesions [12]. In view of its clinical similarities with
BCC, it is only possible to diagnose BSC by performing
an incisional biopsy associated with histopathological ex-
amination. Care should be given to the size of the biopsy
once the histological features of BSC may be absent if
tissue biopsy is too small [13]. With regards to histopa-
thological features, this tumor presents areas typical of
both BCC and SCC with the presence of basaloid and
squamoid cells respectively. A transition zone with in-
termediate cells is evident between the area of BCC and
SCC tumor cells [3]. The most commonly used antibody
to differentiate basaloid SCC from BSC, is BerEP4 as the
latter presents BerEP4 immunopositivity in contrast to
the former [14]. Brantsch et al. [6] accept the use of BSC
term for tumors which are similar to BCCs that have ar-
eas of squamous differentiation presenting positivity for
Ber-EP4 associated with a negative result of epithelial
membrane antigen (EMA).
The recommended treatment for BSC is the total re-
moval of the tumour with a safety margin and a long
term follow-up. For high-risk lesions (greater than 2 cm
in diameter) and those with lymphatic and perineural
invasion, radiotherapy and sentinel node biopsy should
be considered [10]. A recent publication claiming to be
the first European survey on different epidermal tumours
treated by Mohs Micrographic Surgery (MMS) has
pointed out the dangers that BSC poses if not properly
managed. BSC seems to present a higher recurrence rate
when compared to BCC, with an aggressive growth pat-
tern, even when compared to SCC and treated by MMS
[15], reinforcing the need for a wide surgical margin of
tumors and close follow-up of these patients.
Actinic cheilitis (AC) is a potentially malignant dis-
order that gives rise to 95% of carcinomas of the lip and
is associated with sun exposure, affecting mostly white
men over 50 years of age [16,17]. Histologically, the
most frequent findings of AC are solar elastosis asso-
ciated with various degrees of dysplasia and inflamma-
tion [18]. The objective of th is report is to present an un-
usual case of basosquamous carcinoma on the lower lip
associated with actinic cheilitis.
2. Case Report
A 57-year-old, alcoholic, non-smoker, white female pa-
tient, came July 2010, to the Oral Medicine Outpatient
Facility at the Antonio Pedro University Hospital, Scho ol
of Medicine, UFF, Niterói, RJ, due to the development of
an ulcer on her lower lip. The ulcer evolved over six
months at the site of a recurrent herpes labialis (Figure
1(A)).
During medical history, although the patient reported
that she had not work ed in activities that involved exces-
sive sun exposure; it was common for her to be
Figure 1. Basosquamous cell carcinoma: (A) clinical aspects;
(B) videoroscopy of ulcer; (C), (D) Histopathological aspects
by hematoxilin-eosin; (E) immunostaining with CK-17 an-
tibody, positive; (F) EMA antibody, negative; (G), (H) CK-
16 antibody, negative; Ber-EP4 antibody, positive.
exposed to sunlight during leisure moments. She was
born and lives in a town by the seaside, and when she
was younger, she used to go to the beach, every weekend
in summer, being exposed to the sun approximately 4
hours per day for more than 15 years with no protection
of any kind of sunscreen. The patient underwent physical
examination, photo documentation, videoroscopy (Fig-
ure 1(B)), scraped cytology, toluidine blue test, and bi-
opsy.
During physical examination the lower lip presented
an ulcer with spontaneous bleeding measuring 0.8 × 0.8
cm located partially on the lip and partially on the skin.
The surrounding vermilion zone of the lip was dry, har-
dened and flaky presenting oedema and the blurring of
the vermilion border leading to the initial clinical diag-
nosis of a severe actinic cheilitis, with th e possibility of a
SCC at the ulceration site. Crust, erythema, pigmented
blemish and white plaques, were also observed during
physical examination. No intra-oral lesions were ob-
served.
Videoroscopy was performed with MAXX digital mi-
croscope (AVANTSCOPE MAXX™), with a magnifica-
tion factor of 50 times (Figure 1(B)) revealing lesions
with the presence of fissures and erosion with a positive
toluidine blue test. Cytological analysis revealed rare
nests of compatible with carcinoma. Histopathology of
the directed biopsy fragment revealed presence of solar
elastosis and a carcinoma with nests lined with basal
cells, with areas of squamous differentiation (Figures
1(C) and (D)). Immunohistochemistry showed immuno-
reactivity to anti-CK17 and anti-CK 16 (Figures 1(E),
(G) and (H)).
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Basosquamous Cell Carcinoma of the Lower Lip Arising from Actinic Cheilitis: Case Report and up Date 23
After being submitted to all tests the patient was re-
ferred for removal of the BCC in October 2010. Analy-
sis of the specimen from surgical fragment showed free
surgical margins and the same histological aspects as was
described for the incisional biopsy. We expanded the
immunohistochemical panel, using epithelial membrane
antigen (EMA) and BerEP4, which confirmed the diag-
nosis of BSC subtype and not BCC (Figures 1(F) and
(I)). After surgery the lesion site showed good healing.
During the last two years the patient has been followed
up by periodic consultations every 6 months and until
now she is well with no further changes, interestingly
without showing new herpes labialis episodes.
3. Comments
BSC is a controversial rare aggressive skin tumour. It is
characterized in some studies as a variant of BCC with
squamous differentiation while in others it is considered
as an independent tumour. Although there is a high risk
of recurrence and one of its characteristics is its capacity
to metastasize, giving rise both to local and distant me-
tastasis, this is not a rule [2,6,19]. Some authors believe
that sentinel lymph node biopsy could be useful for sug-
gested high-risk BCC, althoug h the routine use of SLNB
for BCC is not recommended [20].
Previous studies report that BSC is predominant in
fair-skinned males over sixty years of age, affecting areas
of higher sunlight exposure, such as the head and neck
[7]. Martin et al. [10] evaluated 31 cases of BSC in 28
patients with an average age of 68 years. Of these only
25% were female patients. Their results confirm the main
trend in the literature finding BSC most often (75%) in
head and neck region. Similar results were obtained by
Tarallo et al. [7] who found twice the number of men
with BSC when compared to female patients, with an
average age of 70 years. Leibovitch et al. [12] performed
a larger study involving 178 cases of BSC with a mean
age of 63 years. Of these approximately 2/3 (115 cases)
occur in men and 1/3 (63) in women. The patient re-
ported in this case is slightly below sixty years of age,
matching the suggested most prevalent age of onset by
most studies. Although, the patient is female, while most
BSCs occur in male patients, her fair skin is consistent
with the majority of cases reported in the literatu re.
Garcia et al. [20] performed a recent review of the
literature report that a variety of studies have revealed
that within the head and neck the most affected region is,
the central area of the face and in particular the perinasal
region. In accordance, after evaluating 35 patients with
BSC, Borel [21] showed that the head and neck, particu-
larly the central region of face, correspond to the area
that is subjected to higher sunlight exposure. Yet in an-
other study Leibovitch et al. [13] found the same results
showing that the main location of BSC was the nas-
al region (33%), followed by the ear (18.5%) and the
periocular area (11.2%), while the lip was affected in
only 1% of the cases.
The case reported here aroused interest in its unusual
localization—the patient’s lower lip—and its low fre-
quency especially in women given that lesions in this
particular region represent only 1% of BSC cases re-
ported in previous studies. Two reviews published in the
1970s report that a frequency of 1 to 2% of non mela-
noma skin cancers is BSC. Schuller et al. [11] investi-
gated 2565 cases of non-melanoma lesions of skin and
identified 33 cases of BSC (1.2%) while Borel [21] per-
formed a review of the literature showing that among
1706 cases of non-melanoma cancers reported, 35 (2%)
cases were BSC. In a more recent study (2011) per-
formed by Tarallo et al. [7], this incidence has grown to
approximately 5%.
BSC has considerable pathological relevance because
of its local aggressiveness, risk of distant metastasis
mainly affecting the lungs, and its increased risk of re-
currence. Bowman et al. [3] reported a prevalence of
metastasis of 7.4% for BSC, which was significantly
higher than the prevalence of metastasis 0.87% for SCC
and concluded that tissue invasion displayed by BSCs is
similar to that of BCC or SCC but with a higher frequen-
cy of pulmonary metastasis than SCC.
Borel’s [21] studies showed that 47% of the 35 pa-
tients with BSC presenting recurrent lesions even after
treatment with a wide surgical excision. Martin et al. [10]
evaluated 31 cases of BSC in 28 patients, observing the
presence of recurrent tumours in 9 (32%) patients after
treatment, lymph node metastasis in five (17%) patients,
and one (4%) patient developing lung metastasis. In their
study, they concluded that the prognostic factors for re-
currence were positive surgical margins, male gender,
lymphatic invasion, perineural invasion and although
they reported that tumor size was not statistically signifi-
cant, all cases of lymphatic metastasis had tumours larger
than 2 cm.
As there are no clinical aspects that can distinguish
BSC from BCC, diagnosis is based on its histopatho-
logical features. But Giacomel et al. [22] suggest that
dermoscopy can be used to provide additional morpho-
logical information, as it was used in this study. This
tumour has been poorly defined in the dermatological
literature with confusing clinical and histological results,
and in many studies not mentioning or recognizing the
lesion [2,7,20]. Brantsch et al. [6] claim that immuno-
histochemistry distinguishes BSC from BCC and SCC.
BSC is a similar to BCC in that it shows areas of squa-
mous differentiation with immunoreactivity to BerEP4
and immunonegativity to EMA. Histopathological ex-
amination of the patient reported in this paper revealed
carcinoma with tumor nests lined by basal cells, areas of
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Basosquamous Cell Carcinoma of the Lower Lip Arising from Actinic Cheilitis: Case Report and up Date
Open Access IJCM
24
squamous differentiation, positivity for BerEP4 and
negativity for EMA, exactly as described in papers con-
cerning BSC de scri pt ion [14].
Although rare in women and in the lower lip, this case
alerts us to the possibility of tumors other than SCC af-
fecting lips. This reinforces the need to investigate ulcers
associated or not associated with actinic cheilitis, by cy-
topathological and histopathological examination. BSC,
although rare and nonspecific clinical presentation, re-
quires an accurate diagnosis because of its frequency of
recurrence and greatly increased incidence of pulmonary
metastasis compared to BCC and SCC.
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