Case report of Basal cell adenocarcinoma of

the parotid gland: clinicopathological and

immunohistochemical study

García Pedro Emilio1, Avila Rodolfo Esteban2, Samar María Elena3

DOI: 10.22592/ode2018n31a8

Abstract
Basal cell adenocarcinoma is an epithelial neoplasm with the cytological characteristics of basal
cell adenoma but with a morphological pattern of infiltrative growth indicative of malignancy.
Due to its low incidence it is often difficult to diagnose a basal cell adenocarcinoma. The
objective of the present study was to identify morphological and immunohistochemical
characteristics that contribute to its diagnosis. A parotid tumor was resected in a 52-year-old
patient; postoperative biopsy and immunostaining with Ki-67, CK19, p63 and alpha- smooth
muscle actin were performed. It was diagnosed basal cell adenocarcinoma that invades the
tumor capsule, periglandular fat and lymph nodes. Immunostaining with Ki-67, CK19, p63
and alpha- smooth muscle actin was positive. Subsequently, a maxillary sinus metastasis
was diagnosed. The morphological characteristics, Ki-67 expression strongly positive and
metastasis give the malignant character to this tumor, which differentiates it from the basal
cell adenoma.

Keywords: parotid, basal cell adenocarcinoma, diagnosis.

1 Medical Doctor - Clinical Oncology Specialist. Assistant Professor. School of Medical Sciences. Universidad Nacional de Córdoba.
Argentina. ORCID: 0000-0001-5268-2339
2 Doctor of Medicine and Surgery. Associate Professor. School of Medical Sciences. Universidad Nacional de Córdoba. Argentina.
ORCID: 0000-0001-8857-2406
3 Doctor of Medicine and Surgery. Full Professor. School of Dentistry. Universidad Nacional de Córdoba. Argentina. ORCID: 0000-
0002-6093-3297

Odontoestomatología. Vol. XX - Nº 31 - Junio 2018

71
Adenocarcinoma de celulas basales de
parotida
Introduction Clinical case
The World Health Organization as well as El- In November 2002, a 52-year-old female pa-
lis and Auclair described basal cell adenocar- tient visited the dentist and an otorhinolar-
cinoma (ICD-O Code 8147/3, International yngology service since she felt pain in the left
Classification of Diseases for Oncology) as a region of the mandible; a diagnosis was not
cytological epithelial neoplasm, and in terms of reached. In December 2002 she went to a new
histomorphology it was found to be very sim- appointment and a nodular mass was found in
ilar to basal cell adenoma, but with infiltrative the left ascending ramus of the mandible, with
growth and a low incidence of metastasis(1‑3). increased pain on palpation. She was diagnosed
with trigeminal neuralgia, ruling out the im-
portance of the nodular lesion; physiotherapy
Background was indicated for the cervical region. However,
Some authors consider that this tumor stems the pain continued to increase, so in Decem-
from a basal cell adenoma while others argue ber 2003 she underwent a biopsy puncture of
that it originates de novo. Of all the cases, 80% an adenopathy, where an inflammatory process
are located in the parotid glands, 9% in the was identified.
submandibular gland and 11% in minor sali- In January 2004, a medical interconsultation
vary glands. It is a rare tumor, which is more was made with a head and neck surgeon who
frequent in adults between their 4th and 9th de- requested a CT scan of the neck which showed
cade of life; it is very rare in children. Swelling an image compatible with a tumor lesion. In
is usually the only symptom but sometimes it February of the same year, a left parotid deep
also causes pain and occasionally remains un- lobe tumor was excised with good postopera-
diagnosed for ten years. It has been categorized tive evolution.
into four types based on its histological pattern Anatomic pathology report: The macroscopy
of growth: solid, membranous, trabecular and showed a slightly lobed, encapsulated mass,
tubular(4-6). Given its low incidence and scarce 5  cm  x  1.7  cm  x  1.2  cm of violet gray color
bibliographic information, it is often difficult with firm areas when cut. In the microscopic
to diagnose it(7). examination of the colored sections with H/E
The aim of this study was to present a case of there was a fibrous capsule that surrounded the
basal cell adenocarcinoma in the parotid deep basaloid cells with pale eosinophilic cytoplasm
lobe in a 52‑year‑old woman and to identi- and rounded or oval nuclei organized in sheets
fy morphological and immunohistochemical and nests of variable shapes and sizes, separated
characteristics that contribute to its diagnosis. by bands of connective stroma. No areas of tu-
This work is part of the project called “The ex- mor necrosis, cellular atypia or mitotic figures
tracellular matrix and the myoepithelial com- were found.
ponent of epithelial tumors of human salivary The histological growth pattern was solid and
glands: structural, histochemical, immuno- tubular. The solid pattern presented nests and
histochemical and lectinhistochemical study”, strands of contiguous basaloid cells that in the
authorized by the Ethics Committee of the periphery of these structures formed a cellular
National Clinical Hospital. School of Medical layer organized in palisade cells.
Sciences. Universidad Nacional de Córdoba. In the tubular pattern there were basaloid cell
Argentina (File 188/14). islets that contained pseudocysts or prominent
lumen lined with cuboid cells (Fig. 1 A and B).

72 García Pedro Emilio, Avila Rodolfo Esteban, Samar María Elena

Fig. 1: HISTOLOGICAL PATTERNS OF BASAL Fig. 2: BASAL CELL ADENOCARCINOMA
CELL ADENOCARCINOMA WITH INFILTRATING GROWTH AND METAS-
Tubular pattern. The arrow indicates the cells that TASIS
contain lights and pseudocysts lined by cells with The arrows show the invasion of the tumoral con-
dense chromatin nuclei. H/E 400x. nective capsule. H/E 100x.
Solid pattern. Nest of basaloid cells (asterisk). H/E The arrow shows the metastasis in a regional lym-
400x. phoid node (H/E). 100x.
The asterisk shows the infiltration of periparotid
adipose tissue. H/E 400x.
The diagnosis of mixed type basal cell adenocar-
cinoma was made on the basis of its histological
patterns (solid and tubular) and its malignant
character, with an aggressive growth with in- Immunohistochemical labeling was used to find
filtration of the tumor capsule and periparotid the differentiation of basaloid cells in epithe-
fat, perineural invasion and metastasis of a re- lial (CK 19) and myoepithelial cells (p63 and
gional lymphoid node (Fig. 2A, B and C). α-smooth muscle actin) (Fig. 3A). The prolif-
We then observed maxillary sinus involvement erating capacity of the tumor was studied with
with bone metastasis. the marker for Ki-67. A positive score was given
only if there were over 10% of marked cells(8):
the result was 25% of positive cells. (Fig. 3B).
The results are shown in Table 1.

Table 1: Immunohistochemical analysis of basal cell adenocarcinoma

Marker Marked cell Reaction *
Ki- 67 Epithelial cells (nuclei) Strong
(prognostic marker of cell proliferation)
p63 (myogenic differentiation) Myoepithelial cells (nuclei) Strong
Alpha-AML (myogenic differentiation) Myoepithelial cells (cytoplasm) Moderate
CK19 (epithelial differentiation) Epithelial cells (cytoplasm) Strong

Alpha-AML: α-smooth muscle actin. CK19: cytokeratin 19

* The qualitative assessment of the labeling was performed according to the intensity of coloration.

Case report of Basal cell adenocarcinoma of the parotid gland: clinicopathological and immunohistochemical study
73
 The radiation therapy scheme could not be
completed; palliative care was continued until
her death two months later.

Discussion
Primary basaloid tumors of the salivary glands
are lesions characterized by a predominance of
“basaloid” epithelial cells with round or ovoid
nuclei surrounded by a thin cytoplasm. The
term “basaloid epithelial cells” arose from ob-
Fig. 3: IMMUNOHISTOCHEMICAL LABELING
servations with standard histological techniques
OF BASAL CELL ADENOCARCINOMA
The arrow shows the nuclei of myoepithelial cells that do not make it possible to distinguish if
positive for p63. 400x. there are myoepithelial or ductal or basal epi-
The arrow indicates the nuclei of the epithelial cells thelial cells. In these cases, immunohistochem-
positively labeled for ical labeling is a useful tool to detect both cell
Ki-67. 400x. types, since it improves diagnostic accuracy.
The immunohistochemistry panel used in the
Given the results of the histopathological study, case described (p63, α-smooth muscle actin
the patient was referred for oncological treat- and CK19) was important for the diagnosis of
ment, with indication of postoperative radio- the “basaloid” cells of this carcinoma(9,10).
therapy for six weeks, which began in March The heterogeneous tumoral group of basaloid
2004. The treatment plan included the irradia- epithelial cells includes benign tumors such as
tion of the parotid plus the cervical region left pleomorphic adenoma, and basal cell adenoma,
at a total dose of 60  Gy, with a daily dose of and malignant tumors such as adenoid cystic
2  Gy. The complications were mucositis, dys-
carcinoma, undifferentiated small cell carcino-
phagia and mycosis, which, upon receiving
ma, and basal cell adenocarcinoma(10,11).
specific treatment, improved at the end of the
Basal cell adenocarcinoma mainly affects the
radiotherapy sessions.
parotid gland (more than 90%) and is histolog-
In September 2004, an orthopantomography
ically similar to basal cell adenoma. However, it
showed a cyst in the left horizontal ramus of
presents an infiltrating growth and a low inci-
the mandible and a vegetative image in the ho-
molateral piriform sinus. The surgery was per- dence of metastasis(1,2,12).
formed and the biopsy was negative for malig- It is generally considered a carcinoma of low
nancy. degree of malignancy, locally destructive and
Subsequently, in March 2005, the pain in the with frequent recurrences. The recurrence peri-
hard palate and the left region of the maxilla od varies from six months to two years and only
began. A CAT scan was indicated and a lesion occasionally produces distant metastases(12,13).
was identified at the maxillary floor level. The In this case the evolution was atypical, present-
surgery was performed and metastasis was di- ed a bone metastasis in an early form, less than
agnosed in the floor of the maxillary sinus. A a year after the end of its treatment. This led to
new radiotherapy plan was indicated, which the deterioration of the general condition of the
had mucositis and dysphagia as complications. patient and her death. This can be explained by
The general condition of the patient worsened the late diagnosis of the lesion, with lymphoid
and she presented dehydration and drowsiness. node metastasis; and histological factors of

74 García Pedro Emilio, Avila Rodolfo Esteban, Samar María Elena

poor prognosis such as capsular, perineural and cutaneous basal cell carcinoma with deep inva-
periglandular fat invasion. sion(12,15).
There is no predilection for gender, and 80% of Wilson and Robins claim that local invasion of
tumors occur after 50 years of age with an aver- the surrounding soft tissues and the gland are
age of 60 years of age(14). This case corresponds the best markers for differentiating the basal
to a 52‑year‑old woman. cell adenoma from basal cell adenocarcinoma.
Its histomorphological patterns are described as They also consider it important to complement
solid, membranous, tubular or trabecular. The the recognition of these tumors on the bases of
solid pattern is the most frequent, characterized invasion with proliferation markers such as Ki-
by solid cellular nests of different shapes and 67 and apoptosis markers(18).
sizes. A collagen stroma, consisting of bands of In a previous study we obtained a negative Ki-
different thickness, separates these nests that 67 labeling in the basal cell adenoma, unlike
correspond to the most frequent histological the basal cell adenocarcinoma, where we ob-
pattern and with the highest risk of metastasis. served a proliferation index greater than 25%,
In the membranous pattern there are abundant which also explains the unfavorable evolution
deposits of a strongly eosinophilic hyalinized of this patient(10).
basal membrane. The growth pattern is tubu- Saluja et al. also claim that the definitive diag-
lar when luminal spaces are formed between nosis of malignancy is established by the inva-
basaloid epithelial cells, whereas in trabecular sive nature of the neoplasm in the surrounding
growth, basaloid epithelial cells are arranged in tissues(19).
interconnected cell bands(15). On the other hand, Jung et al. report that basal
In our study, the tumor was located in the pa- cell adenoma with capsular invasion can also be
rotid gland with a mixed growth pattern since considered an infiltrating tumor, which makes
the basaloid epithelial cells were placed in solid us question the category of basal cell adenocar-
and tubular areas. cinoma. These authors say that the tumors do
Regarding the prevalence of this neoplasia in not have morphological differences, frequently
Latin America, in the consulted databases we develop a cribriform or solid growth pattern
only found data in two studies by Ruiz-Godoy and are larger than the basal cell adenoma with-
and Rivera et al. and Ito et al.(16,17). out capsular invasion. However, they consider
In 1996 Ruiz-Godoy Rivera et al. conducted that the cribriform structure of basal cell ade-
a clinicopathological study of six cases of basal nocarcinoma may correspond to a misdiagnosis
cell adenocarcinoma analyzed at the Instituto of adenoid cystic carcinoma. In addition, the
Nacional de Cancerología, México(16). solid variant of adenoid cystic carcinoma pres-
Ito et al. examined the archives of the Depart- ents cells with marked nuclear atypia and fre-
ment of Pathology of the Instituto del Cáncer quent mitosis(7).
de Londrina (Paraná State, Brazil) for the years Although regional and distant metastases are
1972-2001, and analyzed a total of 496 cases of not common, in this case there was metastasis
major and minor salivary gland tumors. These in a regional lymphoid node and also in the
researchers found only three cases of basal cell maxillary sinus floor, which led to the patient’s
adenocarcinoma(17). death(12).
Histologically, the main differential diagnosis is In general, there is no consensus regarding its
made with basal cell adenoma; but it also in- treatment, since some recommend local exci-
cludes the solid variant of cystic adenoid carci- sion and others total parotidectomy, even in
noma, basaloid squamous cell carcinoma and cases of membranous basal cell adenoma, as
mentioned by Zhan et al.(12).

Case report of Basal cell adenocarcinoma of the parotid gland: clinicopathological and immunohistochemical study
75
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María Elena Samar: [email protected]

Received on: 09.01.2017 – Accepted on: 05.10.2017

Case report of Basal cell adenocarcinoma of the parotid gland: clinicopathological and immunohistochemical study
77