Int. J. Oral Maxillofac. Surg.

2002; 31: 257261

doi:10.1054/ijom.2002.0223, available online at http://www.idealibrary.com on

Clinical Paper
Salivary Gland Pathology

Intraoral minor salivary gland

neoplasm: a single institution
experience of 80 cases

P. Jansisyanont1,
R. H. Blanchaert Jr2,
Robert A. Ord2
1

Formerly, Department of Oral and

Maxillofacial Surgery, University of Maryland,
Baltimore, MD, USA; Currently, Department of
Oral and Maxillofacial Surgery, Chulalongkorn
University, Bangkok, Thailand; 2Department of
Oral and Maxillofacial Surgery, University of
Maryland, Baltimore, MD, USA

P. Jansisyanont, R. H. Blanchaert Jr, R. A. Ord: Intraoral minor salivary gland

neoplasm: a single institution experience of 80 cases. Int. J. Oral Maxillofac. Surg.
2002; 31: 257261.  2002 Published by Elsevier Science Ltd on behalf of the
International Association of Oral and Maxillofacial Surgeons.
Abstract. From March 1991 to February 2001, 80 cases of minor salivary gland
tumours were diagnosed in the Department of Oral and Maxillofacial Surgery at
the University of Maryland at Baltimore (Baltimore, MD, USA). Data extracted
from a retrospective chart review included age, sex, symptoms, site, histological
diagnosis, treatment and outcome. Pleomorphic adenoma was the most common
benign tumour and makes up 89.5% of all benign tumours. The percentage of
malignancy (76.3%) was much higher than that found in other studies. Of the
malignant tumours, 54.1% were mucoepidermoid carcinomas. This study diers
from many previous reviews that were published by pathologists rather than a
surgical unit.

Introduction
Salivary gland tumours are uncommon.
The reported incidence is approximately
3% of all head and neck neoplasms13.
Tumours of minor salivary gland origin
account for only 1015% of all salivary
gland neoplasms10. The percentage of
minor salivary gland tumours is higher
in the African series6,8, although one of
these papers is from a maxillofacial
unit6, which may see minor salivary
gland tumour more than major salivary
gland tumour. Most studies include
major and minor salivary gland tumours
together and there are few articles that
study minor salivary gland tumours separately. The papers that have reported
minor and major salivary gland tumours
separately were done so from outside of
the USA.
The purpose of this study is to report
our experience with minor salivary gland
0901-5027/02/030257+05 $35.00/0

tumours at the University of Maryland

at Baltimore (UMAB), Department of
Oral and Maxillofacial Surgery.
Materials and methods
The records of all patients with minor
salivary gland tumours treated in the
Department of Oral and Maxillofacial
Surgery, UMAB during March 1991 to
February 2001 were reviewed. A total of
80 patients with intraoral minor salivary
gland tumours were identified.
Criteria for inclusion were biopsyproven minor salivary gland tumours
that occurred in the oral cavity.
Most patients with intraoral minor
salivary gland tumours received initially
incisional biopsy from the private oral
and maxillofacial surgeons. All patients
had incisional biopsy performed before
receiving the definitive treatment.

Key words: minor salivary gland tumour;

intraoral.
Accepted for publication 8 January 2002

All patients were investigated in a

retrospective manner by an extensive
chart review. Age, sex, symptoms,
duration of symptoms, location, race,
histology, and modalities of treatment
were examined and compared to those in
the existing literature.
Results
A total of 80 minor salivary gland
tumours were identified in 49 female
patients and 31 male patients. The
female to male ratio was 1.6:1, with a
female to male ratio of 1.9:1 with the
malignant tumours and 1:1.1 with the
benign tumours.
The patients range in age from 984
years with malignant and 3390 years
with benign tumours. The median age of
males and females with benign minor
salivary gland tumours was 64.5 and 58,

 2002 Published by Elsevier Science Ltd on behalf of the International Association of Oral and Maxillofacial Surgeons.

258

Jansisyanont et al.

Table 1. Race and histological distribution

Tumour type

Caucasian

Black

Other

Pleomorphic adenoma
Mucoepidermoid carcinoma
Polymorphous low-grade adenocarcinoma
Adenoid cystic carcinoma

10 (58.8%)
25 (75.8%)
8 (88.9%)
3 (42.9%)

7 (41.2%)
4 (12.1%)
1 (11.1%)
3 (42.9%)

0
4 (12.1%)
0
1 (14.2%)

respectively. For the malignant neoplasms, the median age for males and
females was 52 and 54, respectively. The
median age for males and females with
minor salivary gland tumour was
approximately the same: 54 and 55,
respectively. The median age for those
with malignant minor salivary gland
tumours was 6 years younger than for
those with benign tumours (54 years for
malignant and 60 years for benign). By
histological type, the median age for
those with mucoepidermoid carcinoma
was younger than for those with
other histological types (48.5 years for
mucoepidermoid carcinoma, 58 years
for pleomorphic adenoma, 63 years for
adenoid cystic carcinoma and 56 years
for polymorphous low-grade adenocarcinoma). Six of the malignant tumours
(6 out of 61) (10%) occurred in children
aged 916 years and four of these were
mucoepidermoid carcinomas; all of the
benign tumours occurred in adults.
Race was reported in 77 out of 80
patients71.3% were Caucasian and
22.5% were black. The distribution
of minor salivary gland tumours by
race indicated that the majority of
patients who had pleomorphic adenoma,
mucoepidermoid carcinoma, and polymorphous low-grade adenocarcinoma
were Caucasian, while there was an
equal Caucasian to black predilection
with adenoid cystic carcinoma (Table 1).
The most common complaint that
brought patients to see either the dentist
or oral and maxillofacial surgeon was a
painless swelling in the mouth (61.25%).
Of these patients, 20% were without
symptoms and the tumour was found
on routine dental examination. The
majority of patients with benign minor
salivary gland tumours (68.4%) had
symptoms for more than 1 year before
seeking treatment (Fig. 1). Of patients
with malignant minor salivary gland
tumours, 50.8% had symptoms less than
1 year before receiving treatment. Of the
patients with malignant tumours, 27.9%
had experienced symptoms for longer
than 1 year and 13.1% experienced no
symptoms (found on routine dental
examination), respectively (Fig. 2).
The majority of the patients with
minor salivary gland tumours who

received treatment at UMAB presented

with malignant minor salivary gland
tumours. Of these (76.3%) (61 out of 80)
were classified as malignant and (23.7%)
(19 out of 80) were classified as benign.
The commonest sites for minor salivary gland tumours were the palate, buccal mucosa and upper lip, which
accounted for 77.5% of cases. The palate
was the most common site for all minor
salivary gland tumours (53.8%) and
(60.5%) (26 out of 43) of palatal tumours
were malignant. The buccal mucosa was
the second most common site for malignant salivary gland tumours (Table 2).
The most common sites for malignant
minor salivary gland tumours were the

palate, buccal mucosa, maxilla, and

retromolar fossa at 42.6%, 21.3%, 8.2%,
and 8.2%, respectively.
There were 19 patients who had
benign minor salivary gland tumours of
which two (10.5%) were recurrent
tumours that were initially treated elsewhere. Pleomorphic adenoma was the
most common minor salivary gland
tumour (17 out of 19 cases, 89.5% of
benign minor salivary gland tumour)
and accounting 21.3% of all minor salivary gland tumours. The palate was the
most commonly involved site of pleomorphic adenoma (88.2%, 15 out of 17
cases of pleomorphic adenoma). The
other site of presentation for pleomorphic adenoma was the upper lip. There
was one patient who had basal cell
adenoma of the palate and one with
canalicular adenoma of the upper lip.
Mucoepidermoid carcinoma was the
most common malignant lesion noted in
this study. There were 33 tumours diagnosed as mucoepidermoid carcinoma,

Fig. 1. Duration of symptom in benign minor salivary gland tumours.

Fig. 2. Duration of symptom in malignant minor salivary gland tumours.

Intraoral minor salivary gland tumour

Table 2. Anatomical distribution of benign and malignant minor salivary gland tumours (n=80)
Malignant
Site
Palate
Buccal
Upper lip
RMF*
Maxilla
Mandible
Lower lip
Total

Benign

No. of
cases

% by
location

No. of
cases

% by
location

26
13
4
5
5
4
4
61

60.5
100
66.7
100
100
100
100

17
0
2
0
0
0
0
19

39.5
0
33.3
0
0
0
0

Total

% of all
tumours

43
13
6
5
5
4
4
80

53.8
16.3
7.5
6.2
6.2
5
5
100

*RMF: retromolar fossa.

accounting for 41.3% of all tumours and

54.1% of all malignant tumours. There
were 13 males and 20 females in this
mucoepidermoid carcinoma group. The
palate and buccal mucosa were the most
common sites of mucoepidermoid carcinoma (15 and six cases, respectively),
Furthermore, there were four cases
located intraosseously (three cases in the
mandibular retromolar region and one
case in the maxillary premolar region).
The mucoepidermoid carcinomas were
further classified into low grade (16
cases), intermediate grade (eight cases),
high grade (seven cases) and an
unknown grade (two cases).
Polymorphous low-grade adenocarcinoma was the second most common
malignancy. The incidence of polymorphous low-grade adenocarcinoma was
14.8% (9 out of 61 cases) of the malignant minor salivary gland tumours and
11.3% (9 out of 80 cases) of all tumours.
The buccal mucosa and palate were the
most common sites for polymorphous
low-grade adenocarcinoma and the
remaining locations were the upper lip
and maxilla.
Adenoid cystic carcinoma was the
third most common site of malignant
minor salivary gland tumours. There
were seven patients who had adenoid
cystic carcinoma, accounting for 11.5%
of malignancies and 8.8% of all minor
salivary gland tumours. The palate was
the most common site. The other sites
where adenoid cystic carcinoma presented were the maxilla and upper lip.
All benign minor salivary gland
tumours were treated surgically by wide
local excision, except for two patients
who did not receive any treatment due to
their advanced age and poor general
health. The commonest treatment
modality for malignant minor salivary
gland tumours (70.8%) was surgery
alone. Surgery consisted of wide local
excision and composite resection with
or without neck dissection. The other

treatment modalities for malignant salivary gland tumours were combined

treatment (Table 3). There were five
patients who either received treatment at
other institutions or refused treatment.
There were no recurrences in benign
minor salivary gland tumours with a
follow-up from 3 months to 5 years.
There were 61 patients with malignant
tumours, of which 48 had low- or
intermediate-grade
cancers.
These
included low- and intermediate-grade
mucoepidermoid carcinoma, polymorphous low-grade adenocarcinoma, clear
cell carcinoma, low-grade adenocarcinoma, basal cell adenocarcinoma,
mucous-producing cyst adenocarcinoma
and acinic cell carcinoma. Three patients
were lost to follow-up; of the remaining
45 patients, 42 remain cancer-free
(93.3%) with a variable follow-up ranging from 3 months to 9 years. One
woman with persistent intermediategrade
mucoepidermoid
carcinoma,
which recurred following four surgeries,
radiation therapy and injections with
p53 adenovirus, died of other causes.
Two patients remain alive with persistent disease. One male patient had a
massive, neglected low-grade adenocarcinoma with persistent tumour at the
skull base. One woman had polymorphous low-grade adenocarcinoma that
recurred locally and in the neck. Following neck dissection and maxillectomy,
the tumour again recurred in the maxilla

259

and retropharyngeal nodes; following

further excision, it recurred in the orbit
and she has refused further treatment.
There were 13 patients with highgrade tumours, either high-grade
mucoepidermoid carcinoma or adenoid
cystic carcinoma. Of the seven patients
with high-grade mucoepidermoid carcinoma, four (57%) had lymph node metastases at presentation. Two patients
(15.3%) died because of disease: one
male with adenoid cystic carcinoma with
lung, brain, and bone metastases, and
one female with high-grade mucoepidermoid carcinoma of locoregional recurrence after surgery plus radiation and
further salvage surgery. One patient with
adenoid cystic carcinoma died 1-week
postoperatively of a presumed pulmonary embolism. One patient with highgrade mucoepidermoid carcinoma is
alive with pulmonary metastases unresponsive to chemotherapy. Thus nine
out of 13 (69%) of individuals with
high-grade malignancies are alive without cancer but the follow up period
is short7 years 6 months is the
longestthis is very inadequate for
adenoid cystic carcinoma.
Discussion
Most studies including this one have
shown that minor salivary gland
tumours are more common in females
than males, with a ratio range from 1.2:1
to 1.9:117,21.
The age range of the patients was 990
years. There was no statistically significant dierence in the ages of females and
males in the benign and malignant minor
salivary gland tumour groups. The
present study showed that the median
age of the patients with malignant minor
salivary gland tumours is 6 years
younger than those with benign
tumours. This may have been due to the
large proportion of mucoepidermoid
carcinomas. On the other hand, the
study by W et al. revealed that
the patients with benign tumours were

Table 3. Treatment of malignant minor salivary gland tumours

Treatment
Wide local excision
Composite resection
Composite resection+neck dissection
Surgery+radiation+chemotherapy
Chemotherapy and/or radiation
Other
Total

No. of
cases

% of malignant
minor salivary
gland tumours

32
8
3
11
2
5
61

53.0
13.0
4.8
18.0
3.2
8.0
100

260

Jansisyanont et al.

5 years younger than those with malignant tumours21. However, there was no
statistically significant dierence in the
age of patients with the benign and
malignant salivary gland tumours.
There was a Caucasian predilection
for minor salivary gland tumours that
corresponds to the study by W
et al. (71.3% from the present study and
84.3% from W et al.)21. However
this may be due to our racial population
mix as our percentage for oral squamous
cell carcinoma is similar.
The percentage of malignant minor
salivary gland tumours from most
studies was quite similar, representing
34.8% to 65%11,16,17,20,21. However, a
high percentage of benign tumours were
reported in the study by I &
S12, which they attribute to an
increased proportion of black patients in
their study group. They stated that black
people are aected by pleomorphic
adenoma about 3.5 times more often
than Caucasian people.
In this study, the percentage of malignant minor salivary gland tumours was
76.3%, which corresponds well to the
studies of S & P14,18. The high
percentage of malignant minor salivary
gland tumours may result from a harvesting eect, with the majority of our
referrals from oral and maxillofacial surgeons who treat the benign minor salivary gland tumours and refer the
malignant minor salivary gland tumours
to our institution.
The frequency of pleomorphic
adenoma in this study was 21.3%, which
was lower than that found in other
studies. The incidence of pleomorphic
adenoma has been reported to range
from between 40.850% up to 70%
(Table 4)4,12.
The incidence of mucoepidermoid
carcinoma in this study (41.3%)
was higher than in most published
studies,
accounting
for
8.6
33.9%5,11,12,17,18,20,21. Mucoepidermoid
carcinoma was the most common malignant salivary gland tumour reported in
this study, although a number of previous surveys have reported that adenoid
cystic carcinoma is the most common
malignant
minor
salivary
gland
tumour1,11,1719. This dierence in the
histological distribution may be related
to the histological criteria that was used
for the diagnosis of adenoid cystic carcinoma. Series prior to 1984 did not
recognize polymorphous low-grade
adenocarcinoma as an entity and it was
frequently diagnosed as adenoid cystic
carcinoma. Furthermore, the study by

Table 4. Distribution and percentage (shown in parentheses) of minor salivary gland tumours
in the present study and previously reported series
Variable

Present
study

Rivera Van heerden & Isacsson & Eveson &

Bastides Raubenheimer
Shear
Cawson

Total no. cases

80
62
Total no. benign
19
34
PA
17 (21.3) 24 (38.7)
CA
4 (6.5)
SP
3 (4.8)
IDP
1 (1.6)
Total no. malignant
61
28
MC
33 (41.3) 18 (29.0)
ACC
7 (8.8)
6 (9.7)
AC
3 (3.8)
2 (3.2)
AceC
3 (3.8)
UC
2 (3.2)
PLGA
9 (11.3)

70
34
34 (48.6)

36
6 (8.6)
9 (12.8)
3 (4.3)
1 (1.4)
11 (15.7)

201
145
140 (70)

56
13 (6.5)
21 (10.4)
15 (7.5)

11 (10.5)

Waldron
et al.

335
426
180
245
143 (42.7) 174 (40.8)
20 (4.7)
5 (1.2)
155
30 (8.9)
44 (13.1)
40 (11.9)
6 (1.8)
7 (2.1)

181
65 (15.3)
40 (9.4)
15 (3.5)
1 (1.4)

PA: pleomorphic adenoma; CA: cystadenoma; SA: sialadenoma papilliferum; IDP: inverted
ductal papilloma; MC: mucoepidermoid carcinoma; ACC: adenoid cystic carinoma; AC:
adenocarcinoma; AceC: acinic cell carcinoma; UC: undierentiated carcinoma; PLGA:
polymorphous low-grade adenocarcinoma.

E & C noted that the incidence of mucoepidermoid carcinoma in

British and western European studies
was considerably lower than that in
reports from the USA, which might
represent a true geographic variation11.
The frequency of polymorphous lowgrade adenocarcinoma in the present
study was 11.3% of all minor salivary
gland neoplasms which was the same
range reported in other studies (1.4
15.7%)3,12,20,21. However, in some
reports polymorphous low-grade adenocarcinoma was not found or reported as
a small number. This was probably
because there was overlap of histological
features between polymorphous lowgrade adenocarcinoma and adenoid
cystic carcinoma and some studies were
reported before the polymorphous lowgrade adenocarcinoma was first introduced as a distinctive neoplasm in
19849,11.
The anatomical distribution of minor
salivary gland tumours found in the
present study is slightly dierent from
that in other studies. In this study, the
palate, buccal mucosa, and maxilla were
the most predominant sites, while in
other major surveys the palate, upper
lip, and buccal mucosa were the predominant sites. The palate was reported
to be the predominant site of occurrence
and is the location of occurrence in
between 4055%4,15,21 of all cases; this
has been reported to be as high as 75%.
This study reports that 53.8% of all
minor salivary gland tumours occurred
in the palate and that 60.5% (26 out of
43) were malignant; other studies
reported that approximately 50% of all
minor salivary gland tumours occurred

in the palate and 50% were malignant2,7,11,21. It is not possible to dierentiate malignant from benign tumours
clinically. A total of 27.95 (one in four)
malignant tumours were present for
more than 1 year and 13.1% (one in
seven) were a symptomatic. Therefore all
suspected minor salivary gland tumours
require biopsy to avoid further delay in
diagnosis.
Although two of our 19 patients with
benign tumours had recurrent lesions,
we encountered no recurrence following
adequate local surgery. In our 61 malignant tumours, only five were found to
have clinically proven lymph node metastases (8.2%): four high-grade mucoepidermoid carcinomas and one polymorphous low-grade adenocarcinoma.
Two patients died of cancer, one of
distant metastases and one of locoregional recurrence. One patient died perioperatively; four patients have persistent
disease, three with local recurrence (one
died of other causes) and one with lung
metastases. A total of 54 patients (54 out
of 61; 88.5%) remain alive without obvious cancer, although the follow-up
period is short. Histological type, grade,
stage and site are all important determinants of prognosis and aggressive surgery with wide margins is the best
method of treatment.
The information from this report was
unique and dierent from other studies
because it came from a maxillofacial
oncology unit rather than a pathology
unit.
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Address:
Dr Pornchai Jansisyanont DDS, MS
Department of Oral and Maxillofacial
Surgery
Faculty of Dentistry
Chulalongkorn University
Henri-Durrant Road
Paturawon, Bangkok
10330 Thailand
Tel: +662 218 8582
Fax: +662 218 8581
E-mail: jpornchai@hotmail.com