26, 398-402 (1987)
CASE REPORT Sebaceous Gland Tumor of the Ovary TSUNEHISA KAKU, M.D.,* SATOSHI TOYOSHIMA, M.D.,* TOORU HACHISUGA, M.D.,* MUNETOMO ENJOJI, M.D.,* AND MASAHISA TANAKA, M.D.t *Second Department of Pathology, Faculty of Medicine, Kyushu University, and fDepartment Gynecology and Obstetrics, Fukuoka Red Cross Hospital, Fukuoka, Japan Received
July 13, 1985
A sebaceous gland tumor of the ovary was detected in a 60-year-old woman who underwent right salpingo-oophorectomy for a right ovarian cyst. The cyst was unilocular, weighed 820 g, and was filled with sebaceous material containing a few hair shafts. There was a protruded mass composed of lobules of mature or immature sebaceous cells over the inner surface of the cyst wall. She has been well for 4 years and 2 months after the surgery. This is the second well-documented case of this extremely rare type of tumor. This lesion is teratogenic with unilateral development of the sebaceous glands and malignant characteristics are nil. 0 1987 Academic Press, Inc.
A sebaceous gland tumor of the ovary consists almost entirely of sebaceous glands and was first described by Strauss and Gates in 1964 [l]. Only one similar case has since been briefly described . This uncommon ovarian tumor, a particular form of teratoma, was detected in a 60-year-old woman admitted to our clinic. CASE REPORT
A 60-year-old Japanese woman, G2, P2, had been followed for a probable uterine myoma for 6 months. Her past history was noncontributory except for a left nephrectomy for nephrolithiasis at age of 55. Ultrasonic tomography done at the end of 6 months revealed a right ovarian neoplasm. Laboratory data on admission were either normal or negative and there were no cutaneous lesions. At operation, the right ovary was enlarged and partly adhered to the pelvic peritoneum and to the serosa of the uterus. The left ovary and the uterus appeared to be atrophic and there was no ascites. A right salpingo-oophorectomy was done and no further therapy given. She is doing well 4 years and 2 months after surgery with no evidence of disease.
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The excised ovarian mass was a unilocular cyst weighing 820 g and was filled with sebaceous material containing a few hairs. The smooth outer surface was glistening white with focal fibrous adhesion, probably due to inflammation. Masses of papillary-like tissue (3.5 x 2.5 x 2.0 cm) protruded over the otherwise smooth inner surface of the cyst (Fig. 1). Microscopically, the cyst was lined with one to three layers of cuboidal epithelial cells with some squamous differentiation. The protruded areas were sharply demarcated from the adjacent wall, and were composed of irregular lobules of sebaceous glands made up of two different types of cells. Cells of the first type were identical to those present at the periphery of the normal sebaceous glands and represented undifferentiated germinative cells. These cells had uniform round or oval nuclei with less chromatin which was distributed in fine granules and threads. Each nucleus contained one or two nucleoli. Mitotic figures were present in a few of these cells, but atypical mitotic figures were absent. The cytoplasm varied and was sometimes small in amount and eosinophilic. The cell boundaries were indistinct. The second type cells were lipid-laden mature sebaceous cells occupying the center of the glands, were large, and had uniformly round nuclei. The cytoplasm was abundant and vacuolated with clear boundaries. In addition, transitional cells were often present between the foregoing two. Ratio ofgerminative to sebaceous cells varied. Some lobules mostly contained germinative cells, while others were made up mainly of mature sebaceous cells and, therefore, had mature sebaceous lobules (Fig. 2). Oil red-0 stain showed lipid material in the cytoplasm
FIG. 1. Gross internal appearance of the right ovarian tumor forming an unilocular cyst. Note protruded masses of papillary appearance.
KAKU ET AL.
FIG. 2. General microscopic view of the tumor composed of irregular lobules of sebaceous glands. Note two types of cells, mature sebaceous cells and undifferentiated germinative cells (H&E, X 84).
of both sebaceous and transitional cells (Fig. 3). There were, in addition, foci of squamous epithelium with keratinization around a hair shaft. The squamous cells, however, showed neither cellular atypism nor mitotic figures. These sebaceous components were partly surrounded by xanthogranulomatous elements such as foamy histiocytes, foreign body giant cells, and polymorphonuclear leukocytes (Fig. 4). The wall of the cyst was partially thickened and such areas were composed of dense fibrous connective tissue, with calcification. DISCUSSION
In 1964,Strauss and Gates [ 11reported a huge ovarian tumor which weighed 3345g and consisted of mature and immature sebaceous cells. Hair shafts, lobules of fat tissue, and globules of keratin were found in a small portion of the tumor. They regarded this tumor as a variant of dermoid cyst characterized by an unilateral development of sebaceous glands. Although the tumor ruptured in the course of oophorectomy, the patient was well 4 years and 7 months after the surgery, when the report was made. This tumor was not considered to be malignant. Another example of such a tumor was shown by Janovski as “sebaceous adenoma,” developing in teratoma of the ovary, but details were not given . Scully categorized this rare tumor as one of the monodermal and highly specialized
FIG. 3. High-power view revealing lipid material in the sebaceous and transitional cells (oil red0, x288).
FIG. 4. Area of keratinized squamous epithelium surrounded by xanthogranulomatous elements (H&E, x 84).
teratomas, regarding it to be comparable with struma ovarii, carcinoid, and pure squamous cell carcinoma . The lesion in the present case was sharply demarcated and composed of mature and immature sebaceous gland cells, arranged in an organoid pattern. Slight pleomorphism of the nuclei and occasional mitotic figures are recognized. The histologic features of this ovarian tumor are consistent with that of a sebaceous adenoma usually seen in the skin of the head and so forth, except for the eyelids. In the dermatopathologic field, there are lesions of sebaceous differentiation, such as sebaceous cyst, nevus sebaceous, sebaceous hyperplasia, sebaceous adenoma, sebaceous epithelioma, and sebaceous carcinoma [4-81. Based on the degree of differentiation, sebaceous adenoma is categorized between sebaceous hyperplasia, in which the sebaceous lobules appear fully or early matured around a central duct, and sebaceous epithelioma, in which the tumor is not made up of lobules but rather is composed of irregular cell nests and the percentage of the sebaceous cell component is much less than 50% . The presence of certain pleomorphism and mitotic activity in sebaceous adenoma sometimes makes it difficult to distinguish from sebaceous carcinoma, but the former lacks severe nuclear atypia and is not invasive of adjacent structures. In sebaceous adenoma of the skin, in extraocular locations, recurrence is unusual, that is only 3 of 36 cases reported by Rulon and Helwig . In these 3 cases, the tumor never recurred after reexcision or radiation . In the ovarian tumor of our patient, there are foci of keratinizing squamous epithelium, probably representing areas with differentiation toward the sebaceous duct structure . In the previously reported case and ours, both patients had been treated only with unilateral salpingo-oophorectomy. Therefore, this sebaceous gland tumor of the ovary histologically resembles a sebaceousadenoma of the skin in extraocular locations and may show behavior of a benign lesion. ACKNOWLEDGMENT We thank M. Ohara for critical
readings of the manuscript.
REFERENCES 1. Strauss, A. F., and Gates, H. S., Giant sebaceous gland tumor of the ovary, Amer. J. C/in. Puthol. 41, 78-83 (1964). 2. Janovski, N. A., and Dubrauszky, V. Atlas ofgynecologic and obstetric diagnostic histoputhology, McGraw-Hill, New York, pp. 394-395 (1967). 3. Scully, R. E., Tumors of the ovary and maldeveloped gonads, in Atlas of tumor pathology, (W. H. Hartmann, Ed.), Vol. 16, 2nd series, Armed Forces Institute of Pathology, Washington, D.C. (1980). 4. Lever, W. F., and Lever, G. S. Histopathology of the skin, ed. 6, Lippincott, Philadelphia (1983). 5. Pinkus, H., and Mehregan, A. H. A guide to dermatohistopathology, ed. 3, Appleton-CenturyCrofts, New York (1981). 6. Lever, W. F., Sebaceous adenoma: Review of the literature and report of a case, Arch. Dermatol. 57, 102-111 (1948). 7. Essenhigh, D. M., Jones, D., and Rack, J. H., A sebaceous adenoma: Histological and chemical studies, Brit. J. Derrnatol. 76, 330-340 (1964). 8. Rulon, D. B., and Helwig, E. B., Cutaneous sebaceous neoplasms, Cancer 33, 82-102 (1974).