AKGENTAFFIN 'T'UMOR OCCURRING IN A BENIGN CYSTIC TERATOMA OF THE OVARY

NATHAN MITCHELL, M.D., and BERNARD DIAMOND, M.D.

A R G E N T A F F I N tumors have been re- ported with increasing frequency during the past two decades in association with various portions of the gastrointestinal tract, par- ticularly of the appendix and ileum. Tumors of identical histology have also been described in connection with the lower respiratory tract and have been designated as the carcinoid type of bronchial adenoma.6 It is conceded that these neoplasms may show low-grade malignant characteristics and that numerous instances of metastases to regional and distant organs have been reported, but the tumor rarely is fatal in outcome unless some com- plication, such as intestinal obstruction, super- venes.3

In 1939, the first instances of argentaffin tumor occurring in the wall of an ovarian teratoma were reported by Stewart, Willis, and de Saram. They reported two cases of this lesion, in only one of which were they able to demonstrate intestinal mucous mem- brane. In both cases, hypertrophied smooth muscle elements were noted; and in both, respiratory mucosa, mucus glands, and frag- meats of hyaline cartilage were demonstrated. In their first case, one of the photomicro- graphs illustrates the juxtaposition of the argentaffin cells and the hyaline cartilage. In the second case, the tumor nodules were in more intimate association with gastrointes- tinal mucosa and smooth muscle. Gabrilove reported a case in which the argentaffin tumor was found in a fragment of gastric wall occurring in an ovarian teratoma. Hartz described an unusual case of a 20-year-old colored girl who exhibited a masculinizing syndrome associated with a large 12.1 Kg. ovarian tumor. Complete refeminization was observed after the removal of the tumor.

From the Departments of Pathology and Obstetrics and Gynecology, the Beth-El Hospital, Brooklyn, N. Y.

Received for publication, May 5, 1949.

Histologically, this tumor fulfilled all the cri- teria for a diagnosis of arrhenoblastoma. In several blocks, small cysts lined by typical intestinal epithelial cells were found. These cells, as well as small nodules of unmistakable argentaffin tumor cells, contained silver-posi- tive granules. Hartz concluded that in his case the arrhenoblastoma was a one-sided development of an ovarian teratoma. An argentaffin tumor occurring in the wall of an ovarian teratoma was also described by Black- well and Dockerty. These authors also demon- strated argentophilic granules in the tumor cells. They commented that, despite a careful search, no gastrointestinal mucosa was found. Microscopically, however, bronchial epithe- lium and peribronchial mucous glands were encountered.

I t is the purpose of this communication to report a case of an ovarian teratoma in which multiple argentaffin tumors were found in close association with respiratory-tract deriva- tives.

CASE REPORT A 63-year-old woman complained of a gen-

eralized eruption of the skin. During the course of a complete investigation for the cause of the rash, a flat plate of the abdomen revealed a large pelvic mass with irregular calcific densities within it. At operation, a large tumor of the left ovary was seen. The pedicle of the tumor was twisted, but no vascular changes were evident in the tumor at operation. The right ovary and tube, as well as the uterus were not abnormal. A left salpingo-oophorectomy was performed and the patient had an uneventful postoperative convalescence. She is well one year after operation.

Gross Description. The specimen consisted of a left fallopian tube and ovary. The fal- lopian tube was grossly negative. The ovary had been replaced by a large cystic mass, 12 cm. in diameter. The external surface of the cyst was smooth and glistening and elevated by several nodular masses, which were firm

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FIG. 1. T h e external aspect of the large ovarian cyst showing several well-demarcated nodules ele- vating the external lining of the cyst.

FIG. 2. The inner aspect of the cyst demonstrates the teeth and the promontory formed by the minia- ture bronchial structure.

and rubbery in consistency and which aver- aged 1 cm. in diameter (Fig. 1). On section, these nodules were well circumscribed but unencapsulated. The cut surface was granular and light yellow in color. The lumen of the cyst contained approximately 100 cc. of thin, yellow, oily fluid mixed with sebaceous mate- rial and hairs. Several teeth were noted in the inner lining of the cyst. Above the teeth, a small promontory was formed by a mass, 0.5 cm. in diameter, elevating the inner lining of the cyst (Fig. 2). On dissection, this mass was translucent gray in color and possessed a lumen that was filled with thin mucoid material.

Microscopic Descriplion. Sections through the cyst wall revealed the typical structure of a dermoid cyst. The nodules elevating the external cyst lining were composed of well- differentiated tubular and glandular struc- tures embedded in a fibromuscular stroma (Fig. 3). In addition to the glandular struc- tures, compact masses of cells were found, the individual cells of which were remarkably uniform. The cells were composed of a promi- nent, vesicular, ovoid nucleus and light eosinophilic cytoplasm, the borders of which were ill-defined. Silver impregnation stains revealed numerous argentophilic granules located in the cytoplasm (Fig. 4). Section through the small mass containing the mucoid material revealed an organoid structure con- taining a muscular wall and lined by a mucous membrane containing pseudostrati- fied columnar to flattened cuboidal epithe- lium (Fig. 5). An occasional goblet cell was seen in the mucosal epithelium. Small lymph- oid accumulations were seen in the mucosa beneath the epithelium. In another section through one of the subserosal nodules, the nodule was covered by epithelium that was pseudostratified and contained several goblet cells. Definite cilia were noted covering one portion of this epithelial lining.

COMMENT

This case is apparently the sixth of its kind to be recorded. In three of the other five reported cases, the argentaffin tumor was found in intimate association with the gastro- intestinal wall.'. 4 9 'I In the two remaining cases, although the muscular tissue was thought to be suggestive of gastrointestinal muscle, no distinctive epithelium was demon- strated. In both cases respiratory-tract tissue was found. I t is significant in this connection

For captions see page 802.

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to refer to the findings of Blackwell ct al. on the microscopic findings in a series of 100 cystic ovarian teratomas. They found that respiratory-tract epithelium was present in 53 per cent of their cases, whereas gastro- intestinal epithelium was present in only 12

per cent. 111 thc case herein reported, there is no question about the origin of the argen- taffin nodules from cells within the respiratory- tract epithelium. One of the nodules was covered by ciliated, mucus-containing epi- thelium.

REFERENCES

1. BLACKWELL, W. J., and DOCKERTY, M. B.: Argentaffin carcinoma (carcinoid tumor) arising in an ovarian dermoid cyst. Am. J . Obst. @ Gynec. 51: 575-577, 1946.

2. BLACKWELL, W. J.; DOCKERTY, M. B.; MASSON,

their clinical and pathological significance. Am. 3. Obst. C3 Gynec. 51: 151-172, 1946.

3. DOCKERTY, M- B.9 and ASHBURN, F. S.: Carcinoid tumors (socalled) of ileum; report Of 13 cases in which there was metastasis. Arch. Surg. 47: 221-246, 1943.

4. GABRILOVE, J. L.: Carcinoid in stomach tissue within an ovarian dermoid. Arch. Path. 31 : 508-509, 1941.

5. HARTZ, P. H.: Giant cystic arrhenoblastoma of Ovary containing entodermal epithelium and car-

6. S O m E M , c. R., and h a S L E Y , J. w.9 JR.: Bronchial adenoma. New England J. Mcd. 239: 459- 466, 1948.

7, STEWART, M. .J.; WILLIS, R. A., and SARA^, G. S. W. DE: Argentfine carcinoma (carcinoid tumour) arising in ovarian teratomas: a report of two cases. 3. Path. & B a t . 49: 207-212, 1939.

J. C., and M U ~ E Y , R. D.: Demoid cysts of ovary: cinoid. Am. 3- Path. 21: 1167-1191, 1945*

FIG. 3 . The tubular and alveolar structure of the argenta8n tumor cells embedded in the j- bromuscular stroma. FIG. 4. Oil-immersion photomicrograph with the phase microscope, illus- trating black silver-positive Gytoplasmic granules in tumor cells. FIG. 5. The organoid structure is composed of a thick muscular layer lined by respiratory mucosa containing a few lymphoid nodules and occasional goblet cells.