Adenoid cystic carcinoma of the
Bartholin gland and pregnancy
JOSEPHINE MURPHY, M.D.
JAMES M. WILSON, M.D.
DAVID A. BICKEL, M.D.
South Bend, Indiana
P R 1 M A R Y carcinoma of Bartholin's gland
is one of the rarest gynecologic tumors, and
its coincidental association with pregnancy
is extremely uncommon. Too, adenoid cystic
carcinoma or cylindroma is a rare finding in
general pathology; it has been occasionally
found in the salivary, lacrimal, and peri-
urethral glands, and the breasts. 1 - 6 Sayre 7
reported such a tumor originating in Bartho-
lin's gland.
Case report
A 27-year-old para i, gravida ii, whose last
menstrual period was Nov. 11, 1959, entered the
hospital Feb. 15, 1960, for diagnosis and re-
moval of a tumor of the left labium of the
vulva. This patient was a married white woman
whose past history revealed no serious illness,
and she had had no operations. There were no
previous symptoms of pelvic disease; menstrua-
tion had been normal before the present preg-
nancy and there was no history of abnormal
bleeding or vaginal discharge. There was no
family history of malignancy. For the preceding
year the patient had noticed a painless enlarge-
ment of the left side of the vulva. This tumor
mass was observed at her first delivery one year
before and thought to be a cyst of the Bartholin
gland. The tumor was never acutely inflamed
and had never opened and drained.
The general physical examination was nega-
tive. The uterus was enlarged to a size con-
sistent with a 3 months' pregnancy. The cervix
appeared normal and the cytologic smear was
negative; the urine, blood counts, and blood
From St. joseph Hospital.
612
cells were normal. The serologic test for syphilis
was negative and x-ray of the chest showed no
evidence of pulmonary disease. In the region of
the left Bartholin gland was an enlargement 3
to 4 em. in diameter which was firm, somewhat
movable, and not tender. The mucous membrane
covering the tumor was intact. There was no
palpable enlargement of the inguinal lymph
nodes. Except for a moderate rectocele, rectal
examination was negative. The obvious portion
of the tumor was removed Feb. 16, 1960, for
histologic study. It appeared to be loosely at-
tached to the surrounding structures but ex-
tended to the underlying muscle. The wound
healed by primary union.
Pathologic findings. The primary tumor,
which was incompletely encapsulated, consisted
of a firm, yellowish gray nodule about 2.5 em. in
diameter. The cut section presented a grayish
white homogeneous appearance, but the gross
specimen presented no identifying characteristics.
Microscopic specimens of this tumor were ex-
amined by several pathologists, whose interpre-
tations were almost identical and all concurred
in the diagnosis of adenoid cystic carcinoma or
cylindroma. The entire nodule was characterized
histologically (Figs. 1 and 2) by strands and
clumps of deeply staining basophilic cells; the
margins of these cell clumps showed peripheral
palisading. In many areas of the cell masses
there were acellular spaces filled with pale-
staining homogeneous material. Mitotic figures
were infrequent, and permeation of blood ves-
sels by tumor cells was not observed. Normal
gland structures were closely associated with the
tumor pattern (Fig. 1). The perineural lym-
phatic vessels were found to be filled with cords
of tumor cells (Fig. 2). In a separate fragment
Volume 83 Carcinoma of Bartholin gland and pregnancy 613
Number 5

of tissue, superficial infiltration of muscle by

the tumor was observed.
On March 1, 1960, a vulvectomy was per-
formed which included the entire perineum and
perirectal structures. Because of excessive blood
loss, the patient's condition did not warrant
inguinal dissection at this time. Tissue examina-
tion of the specimen removed by vulvectomy
disclosed minimal residual tumor without ex-
tensive invasion of muscle and other surrounding
tissue.
A left (homolateral) inguinal lymphadenec-
tomy was performed March 24, 1960. The his-
tologic examination of the nodes did not reveal
tumor involvement. There was some sloughing
of the inguinal wound which opened and healed
by secondary union.
The pregnancy progressed normally and the
patient went into active labor Aug. 11, 1~l60,
with a transverse presentation. A normal full-
term infant was delivered by cesarean section
and the puerperium was uncomplicated.
Chronic lymphedema of the left leg which
developed following the inguinal dissection has

Fig. 2. Perineural lymphatic invasion by tumor

cells; the essential method of spread of this tumor.
(Hematoxylin and eosin. x400.)

persisted, but the patient has had no fecal or

urinary incontinence.

Fig. 1. The characteristic structure of adenoid
cystic carcinoma, displaying anastomosing cords
of cells surrounding acellular spaces; normal
gland structures are associated with the tumor.
(Hematoxylin and eosin. xtOO.)
Comment
Adenoid cystic carcinoma is a rare type
of adenocarcinoma formerly designated as
cylindroma because of its distinctive histo-
logic architecture. It was first described by
Billroth 1 100 years ago. The term adenoid
cystic carcinoma was first employed by
Ewing3 and is now generally used by pa-
thologists who have a curious interest in
this uncommon tumor. The general his-
tologic picture presented in this case is
characteristic of reported adenoid cystic
carcinomas of salivary and other gland
structures. Although the precise histogenesis
is undetermined it is assumed that these
tumors are derived from glandular or ductal
epithelium. Adenoid cystic carcinoma of the
salivary glands, and the few reported tumors
arising from Bartholin's glands, have been
characterized by slow development and a

614 Murphy, Wilson, and Bickel
long duration of symptoms. Patients have
been known to live for years after local
extension and the development of distal
metastases.
Summary
A case of adenoid cystic carcinoma or
cylindroma of Bartholin's gland and preg-
nancy is presented.
REFERENCES
1. Billroth, Theodor: Virchows Arch. path. Anat.
17: 357, 1859.
2. Dockerty, M. D., and Mayo, C. W.: Surgery
13: 416, 1943.
3. Ewing, J.: Neoplastic Diseases, ed. 4, Phila-
delphia, 1940, W. B. Saunders Company, pp.
787-790.
4. Foote, F. W., and Frazell, E. L.: Atlas of
Tumor Pathology, Washington, D. C., 1954,
March 1, 1962
Am. J. Obst. & Gynec.
The clinical features of reported cases of
this tumor indicate that the immediate prog-
nosis is relatively good but the ultimate
prognosis poor.
Every solid tumor of the Bartholin gland
should be viewed with the same concern as a
tumor of the breast, and considered to be
cancer until proved otherwise.
Armed Forces Institute of Pathology, sec. IV,
fasc. 11, p. 103.
5. Newman, B., and Gray, D. B.: Am. J. Surg.
92: 400, 1956.
6. Quattlebaum, F. W., Dockerty, M. B., and
Mayo, C. W.: Surg. Gynec. & Obst. 82: 432,
1946.
7. Sayre, P.: Proc. Staff Meet. Mayo Clin. 24:
224, 1949.