J. Nihon
Case
Univ.
Sch.
Dent.,
Vol.
34. 214-223,
1992
Report
A Case
Minoru
of Adenocarcinoma the Upper Lip
Arising
in
HORI 1, Hiromitsu YAMANOI 1, Tadanobu SEKIWA 1, Mitsuhiko MATSUMOTO 1, Hiroshi TANAKA 1 and Kazuo KOMIYAMA 2 (Received12 December1991and accepted10 February 1992)
Key words:
adenocarcinoma, minor salivary gland tumor, lip tumor Abstract
A case of adenocarcinoma arising in the lip of a 20-year-old Japanese man is reported. After electro-irradiation therapy with a dose of 30 Gy, one third of the upper lip, including part of the cheek, was excised. Immediate reconstruction using transpositional flaps of the buccal skin and mucosa, and a functional neck dissection were performed. Nineteen months after surgery, there was no local recurrence or distant metastasis, and desirable results were obtained both functionally and cosmetically. Introduction Adenocarcinoma is a rare tumor of the minor salivary glands, occurring mainly in the parotid gland. Its incidence in the lip is extremely rare[1-5], and a review of the literature revealed that no previous case of adenocarcinoma at this site had been reported in Japan previously. This paper reports a case of primary adenocarcinoma in a minor salivary gland located in the lip. This was treated surgically by radical resection of about one third of the upper lip, together with a functional neck dissection. Case Report A 20-year-old Japanese man was referred to the Department of Oral Surgery for evaluation of a tumor on the right side of the upper lip. The patient had no history of serious medical problems. He claimed that the tumor had first been noticed in December 1988, when an acquaintance pointed out slight swelling of the patient's lip skin. However, as the tumor was not painful when first detected, the patient did not seek treatment. When the patient visited a dentist for treatment of dental caries, he was advised to have the mass on his lip examined, and was therefore referred to the Department of Oral Surgery on April 27 1989. Extra-oral examination revealed slight swelling of the upper lip skin in the 堀
稔1、 山 野 井 弘 充1、 関 和 忠 信1、 松 本 光 彦1、 田 中 博1、 小 宮 山 一 雄2
1 Department of Oral Surgery, Nihon University School of Dentistry 2 Department of Pathology, Nihon University School of Dentistry To whom all correspondence should be addressed: Dr. Hiroshi TANAKA, Department of Oral Surgery, Nihon University School of Dentistry, 1-8-13, Kanda-Surugadai, Chiyoda-ku, Tokyo 101, JAPAN.
215
vicinity
of
face.
the
Apart
had
no
right
from
one
tip.
nodes
in
the
lip
size
of
the
Chest
X-ray
a
revealed
side
of
no
patient
was
first
(Fig. of
the
tumor
by
had
tumor,
need
upper skin
flap
from
by
performed, the
a
size
palpable
of
a
lymph
the
with
to
of
modification
of
5).
was
obtained.
of
30
the
it
distal
were
obviating
defect
flap
of
mucous was
dissection
and
7),
and
the
defect
neck 6
of a vertical
secondary
nerve
lip
removal
flaps:
a functional (Figs.
lesion
tissues
full-thickness
the
after
upper
thus
mucosal
method
the after
and
to
days the
of
present,
and
mucous
decided 97
side
lip
However, the
was
Immediately
buccal
accessory
Gy. of
transposition
Finally,
upper
anesthesia,
were
sutured,
Bocca's
right
performed
medial
cells of
muscle,
dose
right
4).
the
horizontal
of
the
(Fig.
tumor
and
sternocleidomastoid
and
showed
biopsy
appearance
was
full-thickness
transposed (Fig.
elastic and
the
general
third
edges
two
a
Surgery
lip
no
a
was
was
Therefore,
Under
the
and
it
on
estimated
incisional
of
the flat.
Reconstruction
were
An
area
although
the
that
using
and
mm
The
induration,
2).
attempted
one of
from
skin.
adenocarcinoma
values.
about
approximation
using
conserve
in
tumor,
layers
cheek,
flaps
nodes
the
18•~15
lip
mm with
abnormal
department.
confirmed
the
direct
all
achieved
Both
no
the
patient
abnormalities.
excision.
our
excision.
then
of
crater-like
with
sections was
further
was
membrane. closed
it
for
lip
visited
frozen
the
surgical
through
and
of
the
20•~15
(Fig.
no
from
together
incising
examined, the
first
removed
of
The
other
measuring
of
tissue
pressure
was
size
wide
was
observed
revealed
the
changed
1).
lymph
the were
mass
swelling
diagnosis no
was
irradiation on
tumor
3), the
effect
the
remove the
data
electron
was
and
There
surrounding to
67Ga
laboratory
asymmetry (Fig.
submandibular tip,
granular the
palpation
the
subjected
of
Initially, there
to
by to
examination
However,
a red
histopathological
Accumulation
right
non-tender.
corresponding
when
and
minor
normal
region.
adhered
tenderness
performed,
Two
and
determined
tumor
only
appeared
a little-finger
showed
a site
causing
skin
of
mobile
cervical
tumor
The
slight
size
examination at
mouth, lip
tenderness.
the
were
right
mucosa
hard.
or
was
Both
Intra-oral
the the
pain
palpable:
forefinger
of
swelling,
spontaneous
were
the
corner
the
was
in
order
internal
to
jugular
vein. The
patient
surgery.
At
tigraphy the
were
lip
time
was to
was
performed.
restore
the
Histological
cytoplasm
lumen groups
angle
had
appeared of
small
of
the
large
of
the of
the
lobules small
glands
mouth
resected
the
covering
showed was
cells showed
pale
identified
was
in
the
showed
lined
pleomorphism. connective (Figs.11,
on
the 9
that
The
Ga
the
cuboidal nuclei
of
section
tissue 12A
that
cheek. and
A 10).
nest
was
cells.
The
the
tumor
from between
and
in at
adenocar-
tumor
A
scin-
carcinoma
(Figs.
The by
after
67
performed
scar
obtained
vacuolated.
stroma
the
was
epithelium.
Fibrous
and
of
the
thus
glands
marked
staining.
remove
intervals
X-ray
recurrence
tissue
closely-packed
three-week
chest
operation
was
the
duct-forming and
and
result
under and
nucleolae gland
of
just
or
a secondary
functional
to
a
metastasis
However, of
and
two-
observation,
distant
examination
had
composed
8).
approximately
clinical No
(Fig.
cosmetic
cinoma
at
months,
found
satisfactory
cells
examined
thirteen
12B).
the the No
.
216
Fig. 1
Initial clinical appearance, showing slight swelling of the upper lip skin (arrow)
Fig.
Fig.
3
Operative
2
view
Mass
visible
showing
on the
delineated
lip mucosa
area
for
excision
of lesion
217
Fig.
4
Operative
view
the cheek.
Fig.
5
Postoperative
showing
Location
view
the resected
of a vertical
showing
skin
area flap
reconstruction
of one
third
of the upper
lip,
including
part
is outlined
with
two
full-thickness
transposition
flaps
of
218
Fig.
6
Operative
Fig.
view
7
showing
Gross
functional
surgical
specimen
neck
dissection
219
Fig.
Fig.
Fig.
10
9
8
Intraoral
External
view
13 months
appearance
When the patient constricts his lips, evidence of orbicularis function.
13 months
the
vermilion
after
surgery
after
border
surgery
wrinkles,
providing
visible
220
Fig.
12A
Fig.
11
H-E
stain •~10
Adenocarcinoma
Adenocarcinoma mucin
PAS
is
showing
composed
of
proliferation
just
mucus-secreting
cells,
stain •~20
Fig.
12B
Mucicarmine
stain •~20
under
the
epithelium
characterized
by
intercellular
221
metastasis could be detected in the cervical or submandibular lymph nodes. Discussion The morphological diversity of adenocarcinoma has posed problems for classification. Recently, however, the classification system of the World Health Organization (WHO) has been widely adopted, bringing about a better understanding of the biological and clinical course of these neoplasms. According to the WHO classification of salivary gland tumors, those with papillary, mucusproducing and trabecular patterns fall into the group of adenocarcinoma. The present case was classified as a combination of mucus-producing and trabecular types according to the WHO system. Adenocarcinoma accounts for about 5% of all salivary gland tumors[6]. It occurs mainly in the parotid gland, and rarely in the submandibular gland or minor salivary glands. EPKER et al. [31reported 90 cases of minor salivary gland tumors. Adenocarcinoma accounted for 16 cases, and occurred most often in the palate (7/16, 44%), the buccal mucosa (5/16, 31%), the floor of the mouth (3/16, 19%), and the lip (1/16, 6%). ISACSSON et al.111also reported that these lesions accounted for 15 of 201 minor salivary gland tumors, and only 2 occurred in the lip. A review of the Japanese literature showed that YOKOUet al. [7]reported only 6 cases of adenocarcinoma out of 107 malignant tumors of the salivary gland, of which none occurred in the lip. YOSHIDAet al.181reported 3 cases of adenocarcinoma out of 34 malignant salivary gland tumors, also with none occurring in the lip. ISHIKAWA[6] has described two types of adenocarcinoma: high-grade and low-grade malignancy. High-grade malignant lesions grow quickly with marked infiltration into the surrounding tissues, and develop distant metastasis. They tend to be larger and more infiltrative than low-grade malignant tumors. Clinically, low-grade malignant lesions may resemble benign tumors. OKABE[9] has suggested that most cases of adenocarcinoma are high-grade malignant lesions showing rapid infiltration into surrounding tissues, and causing pain and facial paralysis. On first examination, some of his patients with this type of tumor had already developed metastasis to the cervical lymph nodes, or distant metastasis, most often to the lung. CHAUDHRYet al. [4] considered that adenocarcinoma was the most serious malignant tumor arising in the intraoral minor salivary glands, and showed the poorest prognosis, irrespective of the mode of treatment. As to the patient described in this paper, it was first thought that the tumor might be benign because it produced no pain upon pressure, and had grown slowly over a period of 3 months. However, the tumor was also immobile, suggesting a possibility of malignancy. Therefore a biopsy was performed. The tumor appeared to be a combination of the mucinous and trabecular types, and had infiltrated just under the covering epithelium. Various cytological abnormalities were also identified in the tumor cells, including irregular duct formation and mucus production. These histological findings confirmed that the tumor should be classified as an adenocarcinoma. Because of the relative rarity of all types of lip carcinoma in Japan, there are
222
few reports of surgical procedures or treatment for such patients. In this case, there was a conflict between the need to make a wide surgical excision, and the need to conserve tissue in order to achieve a satisfactory reconstruction. Removal of the full thickness of the lip means that direct closure becomes impossible, because mouth-opening becomes restricted and the oral aperture is asymmetrical. In such cases, use of an Abbe flap is usually recommended for upper and lower lip defects of one third of the lip width. BURGETet al. [10]described using a combination of rotation and Abbe flaps. In choosing the method of reconstruction in the present case, three major factors had to be considered. First, the size of the excision, second, the preservation of adequate function, and third, the patient's young age (20 years). Upon excision of a tumor, there should usually be a peripheral safety margin of at least 1 cm. However in this case, in order to achieve a satisfactory functional result, the mouth commissure and philtrum column needed to be retained. Thus the surgical margin was reduced to 5 mm. Examination of frozen sections from the edges of the excision revealed no evidence of carcinoma, and thus the reduced safety margin was confirmed to be adequate. The reconstruction method in this case involved the use of rectangular transpositional flaps of buccal skin and mucosa to obtain a good color match with the vermilion border. Conclusion A case of adenocarcinoma arising in the lip of a 20-year-old Japanese man is reported. After electro-irradiation therapy with a dose of 30 Gy, one third of the patient's upper lip, including part of the cheek, was excised. Immediate reconstruction using transpositional flaps of the buccal skin and mucosa, and a functional neck dissection were performed. Nineteen months after surgery, there was no local recurrence or distant metastasis, and desirable results were obtained, both functionally and cosmetically. References [1] ISACSSON, G. and SHEAR, M.: Intraoral salivarygland tumors: a retrospectivestudy of 201 cases,J. Oral Pathol., 12, 57-62,1983 [2] SPIRO,R. H., HUVOS, A. G. and STRONG, E. W.: Adenocarcinomaof salivary origin: clinicopathologicstudy of 204patients, Am. J. Surg., 144,423-431,1982 [3] EPKER, B. N. and HENNY, F. A.: Clinical,histopathologic,and surgicalaspectsof intraoral minor salivary gland tumors: reviewof 90 cases, J. Oral Surg., 27, 792-804,1969 [4] CHAUDHRY, A. P., VICKERS, R. A. and GORLIN, R. J.: Intraoral minor salivarygland tumors: an analysisof 1414cases, Oral Surg., Oral Med. Oral Pathol., 14., 1194-1226,1961 [5] CHAU, M. N. and RADDEN, B. G: Intra-oralsalivarygland neoplasms:a retrospectivestudy of 98 cases, J. Oral Pathol., 15, 339-342,1986 [6] IsHIKAwA, G.: Oral Pathology,Vol. 2, 757-758,Nagasue Books, Kyoto, Japan, 1982(in Japanese) [7] YOKOU, E., AMAGASA, T., IWAKI, T., TACHIBANA, T., FUJII,E., ISHII, J., SHIMIZU, M. and SHIODA, T.: Clinical studieson malignant tumorsof the salivaryglands, Jpn. J. Oral Maxillofac. Surg., 28, 75-85,1982
223
[8] YOSHIDA, H., CHEN,L., YOKOE,Y., SAGAMI, Y., SEGAMI, N., HYO, Y. and IIZUKA,T.: Clinicostatistical study of malignant salivary gland tumors in our clinic for past 16 years, Jpn. J. Oral Maxillofac. Surg., 36, 1445-1450,1990 [9] OKABE,S.: Treatment of Malignant Salivary Gland Tumors. In Oral Cancer-Diagnosis and Treatment, 193-202, SHIMIZU, M. and KOHAMA, G., Eds., Dental Diamand, Tokyo, Japan, 1989 (in Japanese) [10] BURGET, G. C. and MENICK.F. J.: Aesthetic restoration of one-half the upper lip, Plast. Reconstr. Surg., 78, 583-593, 1986