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

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[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

A case of adenocarcinoma arising in the upper lip.

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 thi...
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