“Lipoleiomyoma with Metaplastic Cartilage” (Benign Mesenchymoma) of the Uterine Cervix

“Lipoleiomyoma with Metaplastic Cartilage” (Benign Mesenchymoma) of the Uterine Cervix

Path. Res. Pract. 188, 799-801 (1992) "Lipoleiomyoma with Metaplastic Cartilage" (Benign Mesenchymoma) of the Uterine Cervix R. Volpe, V. Canzonieri,...

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Path. Res. Pract. 188, 799-801 (1992)

"Lipoleiomyoma with Metaplastic Cartilage" (Benign Mesenchymoma) of the Uterine Cervix R. Volpe, V. Canzonieri, A. Gloghini and A. Carbone Division of Pathology, Centro Regionale di Riferimento Oncologico, Istituto Nazionale di Ricovero e Cura a Carattere Scientifico, Aviano, Italy

SUMMARY Benign mesenchym011la (BM) is a11 ullcommoll tumor colltai11ing two or more differentiated mesenchymal elements in addition to fibrous tissue. A case of BM of the uterine cervix in a 51-year-old woman is reported. This tumor, wh ich we designated "lipoleiomyoma with metaplastic cartilage" because of its morphological features, apparently represents the first documented example of BM of the cervix.

Introduction The term mesenchymoma was first applied to a group of malignant mixed tumors in 1948 by 5toutlO • Itwas pointed out at that time that there were also benign mixed mesenchymal tumors that could be called mesenchymomas. Benign mesenchymoma (BM) is composed of at least two mesenchymal elements, in addition to fibrous tissue, not ordinarily found together in a tumor. All of these elements, of which fat, blood vessels and smooth muscle are most common, are differentiated; none is anaplastic or suggestive of cancer9 • We encountered an unusual tumor of the uterine cervix composed of three benign mesenchymal elements. This tumor, which we designated "lipoleiomyoma with metaplastic cartilage" apparently represents, to our knowledge, the first documented example of BM of the uterine cerVIX. Case Report A 51-year-old multiparous woman was evaluated for a ten-month history of abundant menstrual bleeding. Her past history was unremarkable. A pelvic examination showed enlargement of the uterus. No other physical abnormality or malformation was detected. The chest roentgenograms and results of barium enema examination were normal. Hysterectomy and bilateral salpingo oophorectomy were performed in December 1988. There was no evidence of disease ten months after the surgery. © 1992 by Gustav Fischer Verlag, Stuttgart

Results Pathologie Findings

The resected uterus weighed 1120 g. The posterior wall of the uterine cervix contained a well-circumscribed tumor measuring 15 X 10.2 x 9 cm. The tumor was separated from the mucosa by compressed fibrous and muscular tissue. It had a pale-grey whorled cut surface (Fig. 1). The ovaries and the fallopian tubes were normal. Histopathologically, the margin of the cervical tumor was sharply demarcated. The tumor was composed predominantly of interlacing bundles of smooth muscle cells with areas of myxoid change. There were haphazardly arranged islands of mature adipose tissue and several islands of mature cartilage (Fig. 2). Confirmatory immunohistochemical studies were performed on paraffin-embedded tissue sections by an avidinbiotin peroxidase complex method as previously described3, using a panel of antibodies including 5-100 protein (Dakopatts als, Glostrup, Denmark), vimentin (Boehringer, Mannheim, FRG) and desmin (Monosan, Nistelrode, The Netherlands). Their immunoreactivity has been specified in separate papers 2,3,5. Immunohistochemical analysis documented that cartilaginous and adipose islal)ds expressed positive immunostaining for 5-100 protein, whereas smooth muscle cells were positively immunostained with vimentin and desmin antibodies, as expected (Fig. 2). None of the mesenchymal elements showed mitotic activity or significant nuclear pleomorphism. In scattered 0344-0338/92/0188-0799$3.50/0

800 . R. Volpe et al.

Fig. 1. Grass photograph of the resected uterus showing the large tumor of the cervix. The tumor was separated from the mucosal surface by compressed tissue.

areas there were blood vessels that varied from simple capillaries, capillaries with thick walls, and larger vessels with smooth muscle in the walls. Skeletal muscle, lymphoid and hematopoietic tissue, hemangiopericytoma-like constituents, bone areas, and epithelial elements were not observed.

Discussion Non-epithelial benign neoplasms of the cervix are uncommon. Cervicalleiomyomas represent about 8% of all uterine myomas 7 . A less frequent variant of cervical as weIl as uterine body leiomyomas is the so-called vascular leiomyoma. Hemangiomas are rarely found in the cervix; a single instance of cervicallymphangioma was reported and several cases of lipoma of the cervix are on record 7 .

Neoplasms of neurogenie derivation arising in the cervix are extremely rare and include neurofibroma and ganglioneuroma 7 . To the best of our knowledge, no case of BM of the cervix has been reported in the literature. The most striking examples of these tumors are found in the kidney and perirenal tissues 8• 9 ; however, BM has been observed in the head and neck 1,4,6,9, 11 and, though rarely, in the upper and lower extremities, trunk, retroperitoneum, spinal cord and other exceptional sites 9 • This is the first report of a BM situated in the cervix. The tumor was composed of a mixture of mesenchymal tissues in addition to fibrous tissue. Easily recognized adipose tissue and areas of smooth muscle were scattered haphazardly. Islands of cartilage and abnormal vessels were also observed, while other elements such as striated muscle, bone, lymphoid tissue, areas of extramedullary hematopoiesis and hemangiopericytoma-like foci were lacking. Immunohistochemical results confirmed the presence of combined mesenchymal elements within the tumor. Therefore the lesion fulfilled the histological definition of BM9. In particular, all tissues were weIl differentiated and there was no suggestion of malignancy. The benign nature of the lesion was also supported by the fact that the tumor was weil circumscribed with demarcated margins. It has been reported that BM may invade contiguous anatomie structures and infiltrate to some degree 1,9. Thus, like other benign neoplasms, this tumor may recur if incompletely excised 8 • Le Ber and Stout9 reported a recurrence rate of 20% and therefore recommended wide local excision as the treatment of choice. In the present case no further treatment was considered after hysterectomy. Even if a metastasizing malignant counterpart of BM may occur, it has been stated that BM apparently remains benign with no malignant potentiaI 8,9. This may support the concept that BM may be of hamartomatous origin. According to Bures and Bames 1 factors favouring its hamartomatous origin include the patient's age at the time of diagnosis, the majority of patients being under 25 years of age, and its association with congenital anomalies or with other lesions of hamartomatous nature and various phacomatoses. By contrast, in the present case BM was found in an adult woman without apparent association with other pathologie conditions. Moreover, this BM was composed of at least two mesenchymal tissues that, when viewed individually, could be regarded as true neoplasms. The mature cartilage was considered as a result of metaplastic change because of the absence of this tissue in the normal cervix. In conclusion, our unusual case confirms that BM, showing gross and microseopie features of a benign mesenchymal neoplasm, can occur in adult patients in the absence of other pathologie conditions.

Acknowledgement This work was supported in part by the Associazione Italiana per la Ricerca sul Cancra, Milan, Italy.

Benign Mesencbymoma . 801

Fig. 2. Tbis representative portion of tbe tumor was composed of adult far cells, smootb muscle cells, and mature cartilage. Hemaroxylin-eosin, x 220. lnset A, smootb muscle cells sbowing positive immunostaining for desmin. Inset B, Cartilage tissue with cells positively immunostained for S-100 protein. Avidin-biotin peroxidase complex metbod, hematoxylin counterstain, x 230.

References Bures C, Barnes L (1978) Benign mesencbymomas of the bead and neck. Arcb Patbol Lab Med 102 : 237-241 2 Carbone A, Glogbini A, Volpe R, Boiocchi M (1990) Anti-vimelltin antibody reactivity witb Reed-Sternberg cells of Hodgkin's disease. Virchows Arch (A) 417: 43-48 3 Carbone A, Manconi R, Poletti A, Colombatti A, Tirelli U, Volpe R (1986) S-100 protei n, fibronectin, and laminin immunostaining in lymphomas of follicular center eell origin. Cancer 58:2169-2176 4 Cracovaner AJ, Vieta JO (1969) Benign mesenchymoma of the tongue. Arch Otolaryng Head Neck Surg 89: 665-668 5 Debus E, Weber K, Osbo1'11 M (1983) Monoclonal antibodies to desmin, tbe muscle-specific intermediate filament protein. EMBO J 2: 2305-2312 1

6 Ferbo U, Di Blasi A (1988) Mesencbimoma benigno dell'ipofaringe. Pathologica 80: 371-377 7 Ferenczy A (1982) Benign lesions of the cervix. In: Blaustein A (Ed) Pathology of tbe Female Genital Tract, Second edition, 147-148. Springer Verlag, New York 8 Latres R(1982) Tumors of the soft tissues. In: Atlas ofTumor Pathology, Second series, fascicle 1: 101-104. Armed Forces Institute of Patbology, Wasbington D.C. 9 Le Ber MS, Stout AP (1962) Benign mesenchymomas in children. Cancer 15: 598-605 10 Stout AP (1948) Mesencbymoma, mixed tumor of mesenchymal derivates. Ann Surg 127: 278-290 11 Wholters OD, SteI1feId M (1987) Benign mesenehymoma in tb e tracbea of a patient with the nevoid basal eell eareinoma syndrome. J Laryngol Otol 101: 522-526

Received Oerober 10, 1990 . Accepted in revised form July 11, 1991

Key words: Benign Mesenchymoma - Cervix - Cervix tumor - Lipoleiomyoma Dr. Antonino Carbone, Division of Pathology, Centro Regionale di Riferimento Oneologieo, Via Pedemontana Oceidentale, Aviano 1-33081,ltaly