Carcinoma primary in Bartholin's gland

Carcinoma primary in Bartholin's gland

CARCINOMA PRIMARY IN BARTHOLIN’S GLAND* Case Report NELSON B. SACKETT, (From the Woman’s Hospital, Division of St. Luke’s M.D., NEW YORK, N...

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CARCINOMA

PRIMARY

IN BARTHOLIN’S

GLAND*

Case Report NELSON B. SACKETT, (From

the Woman’s

Hospital,

Division

of St. Luke’s

M.D., NEW YORK,

N. Y.

Hospital)

HILE pelvic malignancy occurred in 3.5 per cent of all gynecological adW missions to-the Woman’s Hospital, only 1.8 per cent of these cancers were primary in the vulva, compared to the 3 to 4 per cent reported in the literature. Carcinoma primary in the gland of Bartholin accounting for 9, or 6.5 per cent, of Tam&g’s” per cent, of 940 vulvar malignancies collected by primary ease was operated upon at the Woman’s to 1956.

or its duct is even more rare, 138 cases, and only 17, or 1.8 Parrott and Miller.4 No such Hospital in the 27 years prior

L. T., No. 127565, aged 55, divorced, gravida iii, para iii, was complaining of intermittent vaginal bleeding of 4 months’ duration. itching and burning inside the introitus onthe left side at infrequent years. current

Menses were spotting.

normal until the menopause in 1952, three There was no venereal or specific infection.

The

The obstetrical second and third

history included a traumatic confinements were without

breech incident

first seen Jan. 25, 1956, Other symptoms were intervals for several

years

onset

of

the

extraction with perineal repair. and all three children survive.

Although examination following the last confinement in 1931 was since then the patient had noted soreness or itching in the left vaginal tervals, not requiring treatment. There was no dyspareunia. Of interest in connection with physical findings the patient struck the right groin against her sewing

before

on admission machine.

Examination showed a well-nourished woman, 5 feet, 1 inch In the right groin was a tender, partly mobile lymph node about wise, cervical, axillary, and inguinal nodes were not unusual.

was

said to be normal, region at long ina fall

in 1951

when

tall, weighing 130 pounds. 4 cm. in diameter. Other-

Pelvic examination showed a deep laceration of the perineum with a thin mucomucous bridge, and a small rectocele. Just inside the hymeneal ring was a dull red, partly ulcerated lesion on the left posterolateral vaginal wall corresponding to the site of the orifice of the duct of Bartholin’s gland, with an irregular border and a diameter of 2.5 cm. This was continuous with a wedge of induration extending some 6 cm. into the left ischiorectal fossa and nearly to the periphery of the external sphincter ani, but nowhere near the ischiopubic ramus. The rest of the vaginal walls were not involved. The cervix presented a tiny cyst, and the corpus was in second-degree adherent retroversion. Otherwise no palpable abnormality was noted in it or the adnexa. Rectal examination disclosed no enlarged sacral nodes or intrinsic lesion. *Presented

at

a meeting

of

the

New

York

183

Obstetrical

Society,

March

12,

1957.

184

SACKETT

On admission the long bones, and pelvis spine. The Wassermann Examination

under

temperature, blood, were normal, while test was negative. anesthesia

on Jan.

and only

Am. J. Oh. & Gyner. Jnnuary. 19%? urine were normal. arthrit,ic changes

31, 1956,

confirmed

the

X-rays appeared previous

of the lungs, in the lower findings.

To rule out a tlowngrowth from the uterus or cervix a fractional curettage preceded the biopsy and coagulation of the lesion in the left vaginal sulcus. The curettings revealed atrophic endometrium and chronic endoeervicitis with scarring. Sections from the vaginal lesion showetl partly epitlermoi,l and partly transitional-cell carcinoma with some plexiform and some adenoid patterns. The pathologist, Dr. Motylofl’, clrew attention to a possible origin of the carcinoma from the left Bartholin tluct. Radical rulvectomy and l$mphadenecton~y were planned and the first stage was formetl on February 1. The inner vulrectomy incision included au inverted V-shaped ment of the left vaginal wall extending up to the cervix and incorporating a 1.5 to 2 The outer incision was wider on the left side uninvolvell margin around the growth. includell the entire perineal bridge and a portion of the sphincter ani muscle in order excise deeply the tonguelike prolongation of growth into the left ischiorectal fossa. Recovery was complicated and the patient was discharged wounds filled in slowly, delaying

persegcm. and to

by fecal incontinence which steadily improved in 2 weeks, The deep perineal and ischiorectal ou the twenty-first day. the second stage for about two months.

On April 18, 1956, bilateral lymphadenectomy was performed, including the superficial and deep femoral and inguinal, external and common iliac groups. Both obturator and hyIt is noteworthy that, while the pogastric regions were explored but only partially cleared. ipsilateral nodes showed no cancer, a metastasis was demonstrated in the contralateral enlarged inguinal node. pital repair patient

After profuse drainage both wounds May 26. Bilateral sinuses persisted sloughed out.

slowly while

Interim

26, 1957, 13 months after the first and had no evidence of recurrence.

Result.-When last seen, Feb. was well, back at work as a librarian,

granulated, several silk

and the patient left the hossutures from the Bassini-type operation,

the

Pathologic& Ez:cLmination (from the report by Dr. Leon Motyloff):-The vulva, removed en bloc with perineum, ischiorectal fat, and part of the vagina, showed the lesion of the left vaginal wall and fourchette bearing a crater 1.5 cm. in diameter and 0.5 cm. deep. Subjacent, induration of t,he vaginal wall for I.5 cm. was continuous with a wedge of thickened ischiorectal tissue. Microscopic sections showed partly transitional epidermoid plexiform carcinoma associated with a solid tubular glandular growth closely related to adjacent lobules of Bartholin’s gland. The latter showed chronic inflammatory infiltration, involutional changes, and occasional cystic distention of the lobular ducts near the carcinoma, which apparently involved the main duct of the gland. The solid tubular elements of the carcinoma imitated the structure of the tubules of the Bartholin glanrl (Figs. 1 and 2). The depth of infiltration of the malignant cells reached 7 mm. in some areas, but there was no evidence of invasion of the ischiorectal fat beyond the main lesion, which was apparently confined to the site of the left Bartholin gland. Inflammatory infiltration of the entire ischiorectal tissue extended down to the fragments of sphincter ani muscle. Study of tissues removed at bilateral lymphadenectomy showed on the left (ipsilateral) side 8 superficial and 3 deep nodes with chronic lymphadenitis but with no evidence of metastasis. On the right (contralateral) side a large superficial inguinal node measuring 4 by 2 cm. showed epidermoid partly transitional-cell carcinoma with glandular pattern in some areas, and with calcification and psammoma bodies. One smaller superficial node showed extensive hilar metastases. In summary, the metastatic carcinoma was found only in the superficial inguinal nodes of the side opposite the primary lesion.

CARCINOMA

PRIMARY

IN Fig.

Fig. Fig. Fig.

l.-Bartholin’s Z.--Partly

gland, epidermoid,

showing partly

carcinoma transitional-cell

Summary

BARTHOLIN’S

GLAND

185

1.

2. in

apposition carcinoma

to lobules with solid

of the tubular

gland. pattm ems.

and Conclusions

1. A case of carcinoma occupying the region of the left gland of Barth \olin an .d its duct is reported. Its origin from that gland is indicated by its local lion,

186

Am. .I. Obst. % Gynec. Jmcary. l5iP

SACKETT

by the presence of transitional-cell differentiation, solid tubular or adenoid patterns, all repeated in the regional lymph node metastasis, and by the absence of demonstrable malignancy elsewhere. 2. Possible etiological factors are obstetric trauma, chronic inflammation in the neighboring vulvar and ischiorectal tissues, and involutional and cystic changes in the lobules of Bartholin’s gland. In 3 of Taussig’s 6 cases there for prophylaxis was a history of previous Bartholin cyst. The implications are obvious. 3. As to prognosis, favorable factors are the absence of tissue permeation beyond the main lesion, the negative clinical and histological findings in the deep iliac and obturator regions, and the apparent lack of recurrence in the first 13 months. Nevertheless, the long history, insidious onset, and contralateral superficial lymph node metastases make the long-range outlook rather doubtful, in conformity with the poor results reported in t,he literature.

References 1. Ackerman, L. V., and de1 Regato, J. A.: Cancer, ed. 2, St. Louis, 1954, The Mosby Company, pp. 953-965. 2. Cosbie, W. ct.: AM. J. OBST. & GYNEC. 63: 251, 1952. 3. McKelvey, J. L.: Obst. & Gynec. 5: 452, 1955. 4. Parrott, M. H., and Miller, N. F.: In Davis, C. H., and Carter, B.: Gynecology Obstetrics, Hagerstown, 1956, W. F. Prror Co., Inc., vol. 2, chap. 9. Aa6. J. OBST. & GYNEC. 40: 764, 1940. 5. Taussig, F. J.: of Cancer and 6. Tam&g,. F. J.: In Pack, G. T., and Livingston, E. M. : Treatment Diseases, New York, 1940, Paul B. Hoeber, Inc., vol. 2, pp. 1’783-1812. Brit. M. J. 2: 780, 1954. 7. Way, 8.:

C. V.

and Allied

Discussion DR. FRANK R. SMITH.-The incidence of vulva1 cancer is generally being about 4 per cent of all genital cancer. At Memorial Hospital where about 435 patients with cancer of the vulva, I think there have been no patients with cancer of Bartholin’s gland, a very low incidence.

regarded as we have had more than 8

In his last book, Dr. Emil Novak stated that 90 per cent of Bartholin’s gland cinomas are adenocarcinomas and the other 10 per cent are epidermoid carcinomas. epidermoid type causes some confusion because it may be hard to determine whether a case has its origin as a primary cancer of the anus or of Bartholin’s gland.

carThe such

At Memorial Hospital, we have thought that adenocarcinoma of the Bartholin’s gland did not behave exactly like the other carcinomas of the vulva, but behaved like other adenocarcinomas in that distant metastases resulted. This was based on the fact that one patient in 4 had distant metastases. Perhaps we often confuse primary cancer of Bartholin’s gland with a secondary cancer. there 1952

We have 2 patients with carcinoma of Bartholin’s are about 135 patients who had had cancer of who survive and are free of disease.

DR. GRAY case presentation lieve in the files

H. TWO’MBLY.-I is the rarity of this at Bellevue Hospital

think lesion. there

One case that I saw had been ceedingly radioresistant. So, judging that this is a surgical problem.

treated from

gland in the vulva

the thing that I have seen only are only 2.

our “alive’ during the

’ files years

should be emphasized one in my experience

with radiation therapy that very infinitesmal

whereas 1926 to in this and I be-

and proved to be exexperience, I would say

!J$;Ll;r:’ ”

CARCINOMA

PRIMARY

We have had 2 recent cases carcinoma of the Bartholin gland. a metastasis from a hydronephroma,

IN

BARTHOLIN’S

GLAND

that were thought by various They proved to be metastatic the second a metastasis from

These cases aroused considerable interest on the service, some of the literature on the subject. There is a good review Gynecological Swrvey of 1951 by Wharton and Everett, in which 109 cases in the entire world literature, which emphasized again

187

observers to be primary disease, in the first case a chorionepithelioma.

so we have been looking up article in the Obstetrical and they were able to collect only the rarity of this disease.

In Dr. Sackett’s ease there was a crossed metastasis, that is, the nodes on the same side as the tumor were not involved but those on the opposite side were. A few years ago we had occasion to put some radioactive gold underneath a carcinoma of the vulva and then do a radical vulvectomy and bilateral groin dissection in continuity. The whole specimen was fixed and cleared. This gave us a most interesting demonstration, because the lymphatics from the vulva were outlined by the radioactive gold and appeared as pale, dove-gray lines lying very deep in the tissue right on the deep fascia. The profusion of these lymphatics was really quite striking. There were hundreds of them coming up over the mons. The other thing that was enlightening was that, when the radioactive gold was put in on one side, radioactivity was found promptly not only in the nodes on the same side but also on the opposite side. In other words, there was a free anastomosis of lymphatics. Evidently anything passing up the lymphatic channels would go either to one side or the other quite freely. Dr. Sackett stated that the groin dissection drained for a long time. Recently we have been experimenting with the possibility of reducing this drainage time and the necrosis that SO often occurs in groin dissections by putting catheters with many holes cut in the ends of them under the skin flap of the groin dissections and connecting them with suction, an idea taken from radical breast surgery. The most recent case we have tried this out in has given a beautiful result. There was no necrosis, but prompt adhesion of skin flaps, so there was really no collection of serum at all under the skin flaps. 1 think it is a method that might well be taken into consideration by other members of this Society. DR. MICHAEL J. JORDAN.-1 would like to present a case of a patient with carcinoma of Bartholin’s gland who was operated on yesterday. She was seen for the first time 10 weeks ago and at that tme she presented a lesion involving the right Bartholin gland with ulceration. There were large flxed nodes in both groins, measuring anywhere from 2.5 to 4 cm. The nodes in the left groin were subjected to biopsy and showed essentially the same pattern as the primary lesion itself, which was a mixture of adenocarcinema and anaplastic epidermoid carcinoma, It was decided at that time to try this patient on some million volt rotating therapy and she was given a total of approximately 4,000 r units tumor dose to the primary lesion and approximately 2,000 to each groin. This patient returned to Memorial Hospital last Saturday. The primary lesion and the fixed nodes had completely disappeared. In view At the time of this operation, of this it was decided to do an exploratory operation. which was done yesterday, a total hysterectomy was performed because of the presence of fibroids, and at the same time a dissection of the pelvic lymph nodes bilaterally down to the external ring was carried out. A bilateral groin dissection, superficial and deep, was done. The pelvic nodes present were small and grossly negative, and the specimen in general showed no gross evidence of disease. There is a small amout of thickening in the right Bartholin gland which was not reIf one were making an examination for the first time, one would think moved at this time. perhaps that there had been a mild inflammation of the gland at some time. Some of these cases are therefore particularly radiation sensitive. This was not at first a case that would justify surgery and yet 10 weeks later the patient is without any apparent disease. If all specimens taken yesterday come back negative, I intend to remove the vulva, to see if there is any residual disease in the right Bartholin gland itself.

DR. JOHN G. MASTERSON.--I became interested in the problem of carcinoma of the Bartholin gland in 1953 as the result of a rase that we hall on the Gynecological Tumor Service at the State University of New York. Dr. Sidney Goss and I made a survey of the literature We found that there had been some 116 cases reported up to that time In conjunction with some of the comments that have been made by the previous discussants, I think that our survey, as published in 1955, would indicate that the extension of radical surgery to the disease has been associated with an improvement in the end results that parallels the extension of radical surgery to the more common varieties of carcinoma of the vulva. There are some further pertinent comments that might be made on the basis of our survey. The reported cases indicate that this is a disease found in younger age groups than is the usual carcinoma of the vulva. The latter is most frequently seen in the seventh and eighth decades. The peak incidence in our analysis was actually around the fifth decade. This fact might implicate inflammation as a predisposing factor. Although we commonly consider Rartholin gland carcinoma to be of an adenomatous variety, a rcviem of the literature shows that half of them are epidermoid and half of them are adenomatous. Some work in this connection by the German investigators, Sitzenfrey and Schweizcr, indicates that inflammation may produce a metaplasia in the Bartholin gland. Hence, infection may possibly be a predisposing factor in the appreciably increased incidence of epidermoid carcinoma. Since, however, the ducts of the Bartholin gland are lined by epidermoid cells, they coultl very well be the site of origin of many tumors. In conclusion, I certainly agree with Dr. Smith and Dr. Twombly that radical surgery appears to be the best treatment that one can offer these patients, and that, with the extension of this procedure to Bartholin gland carcinoma, one should anticipate a survival rate comparable to that seen in other types of carcinoma of the vulva.