Minimal Deviation Adenocarcinoma (Adenoma Malignum) of the Uterine Cervix Associated with Mucinous Ovarian Carcinoma

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1 THE KURUME MEDICAL JOURNAL Vol. 38, p, , 1991 Mimal Deviation Adenocarcoma (Adenoma Malignum) of Utere Cervix Associated with Mucous Ovarian Carcoma TAKASHI NISHIDA, KIMIO USHIJIMA, TAKAAKI ODA, TOR SUGIYAMA, YOSHIMASA TSUJI, KIYOHISA IMAISHI AND MICHIAKI YAKUSHIJI Department of Obstetrics and Gynecology, National Kokura Hospital, 10-1 Harugaoka, Kokuramami-ku, Kitakyusyu, 802, **Department of Obstetrics and Gynecology, National Kurume Hospital, Kokubu-machi, Kurume, 830 and tdepartment of Obstetrics and Gynecology, Kurume University School of Medice, Kurume, 830 Japan Received publication July 18, 1991 Summary: A case of mimal deviation adenocarcoma (MDA : adenoma malignum) of utere cervix associated with ovarian mucous carcoma is documented. Diagnosis was possible only retrospectively after surgery by histological examations cludg immunohistochemistry. Three courses of chemorapy, consistg of cisplat, doxorubic and ifosfamide, could eradicate residual diseases of ovarian cancer from peritoneal cavity, but was sufficiently effective agast lymph node metastases of cervical MDA. Subsequently, disease flared-up retroperitoneal ly durg sixth course of treatment course, suggestg chemoresistance developed, and furr chemorapy usg different regimens were not effective. Therapeutic tractability of MDA as well as diagnostic difficulty was aga emphasized. Key words : cervical adenoma malignum-ovarian mucous tumor-s - chronous cancer-immunohistochemistry combation chemorapy. Introduction Adenoma malignum, now designated as mimal deviation adenocarcoma : MDA (Silverberg and Hurt, 1975) is an uncommon variant of cervical gland type adnocarcoma. Sce first description of Gusserow 1870, much has been described concerng its diagnostic difficulty and rapeutic tractability. Moreover, cases of coexistence of cervical and ovarian tumors with similar histology, it will become more difficult to distguish wher cervical tumor is an dependent primary tumor or a metastasis from ovarian cancer. In this paper, we document a case of cervical MDA associated with ovarian mucous carcoma, and discuss prognostic Case Report significance. A 52 year old woman with uneventful past history came to National Kurume Hospital her back pa and mucous vagal discharge. Ultrasonography revealed a large multicystic tumor with thick septum lower abdomen. Although a cytological screeng test cervical cancer was negative, cervical biopsy was permed because of firm consistence of cervix. The biopsy spec- 265

266 NISHIDA, ET AL. imen revealed branchg glands deep part of cervix, but evidence of malignancy was not conclusive. Repeated cytologic examations were aga nega - tive.

2 266 NISHIDA, ET AL. imen revealed branchg glands deep part of cervix, but evidence of malignancy was not conclusive. Repeated cytologic examations were aga nega - tive. At laparotomy, which cluded total hysterectomy, bilateral salpgo-ophorectomy, retroperitoneal lymph nodes samplg and peritoneal washg cytologic examation, unresectable tumors remaed abdomal cavity and pelvic retroperitoneal space. Fostsurgical diagnosis was ovarian cancer stage llc (FIGO: International Federation of Gynecology and Obstetrics) associated with stage llb cervical cancer. At second laparotomy followg 3 courses of postoperative chemorapy consistg of 50 mg/m2 cisplat, 50 mg/m2 doxorubic and 5 g/m2 ifosfamide, traperitoneal implants of ovarian cancer had been completely eradicated, and furr attempts to retroperitoneal lymph nodes dissection were unsuccessful at that time. Three additional courses of combation chemorapy were permed after second surgery. At end of 6th course, however, it was found that disease was spreadg i pelvis, suggestg that drug resistance had developed. Despite changes of chemorapy, patient took a down-hill course and fally died of disease at 12th month followg first diagnostic operation. Histological Fdgs In addition to route hematoxyleos stag, an immunohistochemical stag carcoembryonic antigen () was done by a avid-biot complex method on f ormal -fixed, paraff-embedded tissue usg polyclonal anti - antiserum (Dako Corp., Santa Barbara, CA. U. S. A.). Dilated and branchg glands proliferated deep stroma of utere cervix. The glands were irregular size and shape, and led by columnar epilial cells with basal nuclei resemblg cervical gland cells ( 1). Immunohistochemical stag revealed positive reaction cytoplasm of neoplastic gland cells cervix ( 2). None of cancer cells ovarian tumor was -positive ( 3). -positive reaction was also observed cytoplasm of cancer cells retroperitoneal lymph nodes obtaed at second surgery ( 4). Discussion The cervical tumor showed sidious presentation, and conventional cytological screeng procedures were persistently negative. The cervical biopsy, which mucous glands extended deeply to cervix, was diagnostically suggestive but not conclusive. Diagnosis was confirmed only retrospectively after surgery ovarian cancer, and immunohisto - chemically positive stag of cervical cancer dicated that cervical and ovarian tumors were dependent primary cancers. The diagnostic difficulty of adenoma malignum (MDA) have been recognized previously. Although some studies supported diagnostic importance of cytology (Kaku and Enjoyi, 1983; Szyfelbe et al. 1984), ors (Kamski and Norris, 1983; Michael et al, 1984) questioned usefulness of this diagnostic technique. Cytological diagnosis of MDA requires subtle observation as well as adequate samplg, as expected from its histologic appearance. The results of repeated cytologic examations were false-negative case reported here. On histologic examation, relation between tumor cells and depth of stroma is only suggestive feature of MDA,

3 CERVICAL ADENOMA MALIGNUM WITH OVARIAN CANCER Mimal deviation adenocarcoma (MDA) of cervix obtaed at first surgery. Dilated and branchg glands proliferat deep cervical stroma. (HE X100) MDA. 2. Immunohistochemical (Immunoperoxydase reaction sta ~200) cytoplasm of

4 268 NISHIDA, cancer cancer peroxydase 3. Negative tissue. 4. stag (Immunoperoxydase Positive sta node obtaed ~200) AL. immunoreactive sta stag pelvic ET at ovarian ~100) was also second found operation. metastatic (Immuno-

5 CERVICAL ADENOMA MALIGNUM WITH OVARIAN CANCER 269 and benign lesions cludg deep nabothian cysts should be excluded (Clement and Young, 1989). Intracytoplasmic stag by immunohistochemical method has a diagnostic value (Michael et al. 1984), and positive stag can support a diagnosis of MDA, although a negative stag dose not exclude that diagnosis (Steeper and Wick, 1986). The prognosis of patients with cervical MDA recent literature is clearly worse than or types of cervical adenocarcoma (Kaku and En jyo ji, 1983; Gilks et al. 1989). Recurrence can develop regardless of postsurgicai treatment. After failure of secondary tumor reduction followg 3 courses of chemorapy, patient this study took a down-hill course and fally died of progression of cervical disease at 12th month followg diagnostic surgery. The success of surgery agast a recurrent tumor after failure of cisplat combed with radiation rapy (Fusi and Gordon, 1988), suggests that despite current advances treatment of gynecological malignancies, surgical resection still remas best method treatment of patients with cervical MDA. References CLEMENT, P.B. and YOUNG, R. H. (1989). Deep nabothian cysts of utere cervix. A possible source of confusion with mimal-deviation adenocarcoma (adenoma malignum). Int. J. Gynecol. Pathol. 8, FUSI, L. and GORDON, H. (1988). Insidious presentation of adenocarcoma of cervix (adenoma malignum). J. Obstet. Gynecol. 8, 363. GILKS, C. B., YOUNG, R. H., AGUIRE, P., DeLELLIS, R. A. and SCULLY, R. E. (1989). Adenoma malignum (mimal deviation adenocarcoma) of utere cervix. A clicopathological and immunohistochemical analysis of 26 cases. Am. J. Surg. Pathol. 13, GUSSEROW, A. L. S. (1870). Ueber Sarcome des Uterus. Arch Gynakol. 1, KAKU, T. and ENJOJI, M. (1983). Extremely well-differentiated adenocarcoma ("adenoma malignum") of cervix. Int. J. Gynecol. Pathol. 2, KAMINSKI, P. F. and NORRIS, H. J. (1983). Mimal deviation carcoma (adenoma malignum) of cervix. Int. J. Gynecol. Pathol. 2, MICHAEL, H., GRAWE, L. and KRAUS, F. T. (1984). Mimal deviation endocervical adenocarcoma : clica; and histologic features, immunohistochemical stag carcoembryonic antigen, and differentiation from confusg benign lesions. Int. J. Gynecol. Pathol. 3, SILVERBERG, S. G. and HURT, W. G. (1975). Mimal deviation adenocarcoma ("adenoma malignum") of cervix. Am. J. Obstet. Gynecol. 123, STEEPER, T. A. and WICK, M. R. (1986). Mimal deviation adenocarcoma of utere cervix ("Adenoma Malignum"). Cancer 58, SZYFELBEIN, W. M., YOUNG, R. C. and SCULLY, R. E. (1984). Adenoma malignum of cervix : cytologic fdgs. Acta Cytol. 28,

Chapter 8 Adenocarcinoma

Page 80 Chapter 8 Adenocarcinoma Overview In Japan, the proportion of squamous cell carcinoma among all cervical cancers has been declining every year. In a recent survey, non-squamous cell carcinoma accounted