Japanese Journal of Clinical Oncology 31:39-42 (2001)
© 2001 Foundation for Promotion of Cancer Research
Uterine Inversion Caused by Uterine Sarcoma: A Case Report
Department of Obstetrics and Gynecology, Institute of Clinical Medicine, University of Tsukuba, Tsukuba, Japan
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ABSTRACT |
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 TOP ABSTRACT INTRODUCTIONCASE REPORTSDISCUSSIONREFERENCES |
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Uterine inversion caused by uterine sarcoma is a rare condition with 12 reported cases to date according to a MEDLINE search. We report two cases of this rare condition. A 71- and a 72-year-old woman presented with uterine sarcomas rapidly extruded into the vagina. In both cases, magnetic resonance imaging (MRI) scans showed U-shaped uterine cavities and the pedicles of these tumors were attached to the uterine fundi. Pathological examination confirmed a leiomyosarcoma and a heterologous carcinosarcoma. Uterine inversion can occur when uterine sarcoma rapidly increases in size and extrudes into the vagina. MRI should be performed in the diagnosis of this rare combination.
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INTRODUCTION |
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TOPABSTRACTINTRODUCTIONCASE REPORTSDISCUSSIONREFERENCES |
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Most cases of uterine inversion are puerperal inversions and non-puerperal inversions are rare (1,2). Non-puerperal uterine inversions are often caused by leiomyomas, but rarely by uterine sarcomas (1). A search of the English literature in MEDLINE revealed only 12 reports on uterine inversions caused by uterine sarcomas from 1940 to October 2000. This paper describes two cases of this rare condition.
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CASE REPORTS |
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TOPABSTRACTINTRODUCTIONCASE REPORTSDISCUSSIONREFERENCES |
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Case 1
A 71-year-old woman, gravida 0, para 0, began to notice a tumor in the lower abdomen in September 1995 and the tumor was subsequently diagnosed as being a uterine leiomyoma by another physician. In August 1996, magnetic resonance imaging (MRI) confirmed that the tumor had increased in size, up to 20 cm in diameter, and the patient experienced anuresis. At laparotomy, the tumor could not be excised since it was easily hemorrhagic. After the surgery, gonadotropin-releasing hormone agonist (Gn-RHa) was used for 6 months, but the tumor did not diminish in size. In January 1997, the patient suffered massive vaginal bleeding caused by myoma delivery and required frequent blood transfusions. She also exhibited symptoms associated with ileus. She was referred to Tsukuba University Hospital in March 1997. Laboratory tests revealed a serum CA125 level of 76 U/ml, CA19-9 of 45 U/ml and LDH of 288 U/l. A T1-weighted dynamic MRI enhanced by gadolinium revealed an extruded tumor in the vagina and a high-intensity U-shaped uterine cavity (Fig.1a and b). We performed laparotomy with a preoperative diagnosis of uterine inversion caused by submucosal myoma. The inverted and concave uterine fundus was adhered with the sigmoid colon, which was considered to have been the cause of the ileus. The pedicle of the tumor was attached to the uterine fundus and the tumor extruded into the vagina was larger than the body of the uterus. A total abdominal hysterectomy and bilateral salpingo-oophorectomy were performed. The tumor weighed 580 g and was microscopically diagnosed as a leiomyosarcoma with cytological atypia and four mitotic figures per 10 high power fields (Fig. 2). She experienced anuresis and recurrent tumor in the vaginal cuff in January 1999. Total expiration of the tumor, 420 g in total, was performed. Pathological diagnosis was the same leiomyosarcoma as the previous one. Because of diabetes mellitus, she became blind. Therefore, she did not receive systemic chemotherapy postoperatively and has been treated orally with etoposide (25 mg/day for 3 weeks every 4 weeks) on an outpatient basis. As of October 2000, she remains disease free.
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Case 2
A 72-year-old woman, gravida 3, para 3, began to have vaginal bleeding in April 1998 and was diagnosed as having a leiomyosarcoma by a histological analysis of the endometrium by another physician. She was referred to Tsukuba University Hospital in August 1998. On admission, she presented a tumor extruded into the vagina. Laboratory tests revealed a serum CA125 level of 29 U/ml and LDH of 388 U/l. A T2-weighted MRI image showed a high-intensity U-shaped uterine cavity and an inverted uterus. Since the vaginal bleeding and anemia exacerbated, we performed laparotomy. The uterine fundus was concave (Fig. 3) and a total abdominal hysterectomy and bilateral salpingo-oophorectomy were performed. The tumor resembled myoma delivery, the uterine cervix was extended and thin and the uterine body was cylindrical. The pedicle of the tumor was attached to the uterine fundus and the tumor extruded into the vagina weighed 310 g and had a maximum diameter of 11 cm. Microscopically, the tumor had both carcinoma and sarcoma components, some of which had cross striations in the cytoplasm showing immunoreactivity to phosphotungstic acid–hematoxylin (Fig. 4), so that the final pathological diagnosis was a heterologous carcinosarcoma. Postoperatively, she received five courses of chemotherapy with etoposide (100 mg/day for 5 days) and cisplatin (20 mg/day for 5 days), followed by etoposide (25 mg/day for 3 weeks every 4 weeks) on an outpatient basis. In April 1999, metastatic tumors appeared in the right lung and gradually increased in size, resulting in accumulation of pleural effusion and respiratory distress in November 1999. Her condition deteriorated rapidly and she died in December 1999.
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DISCUSSION |
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TOPABSTRACTINTRODUCTIONCASE REPORTSDISCUSSIONREFERENCES |
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Non-puerperal uterine inversion is an extremely rare disease, accounting for only one sixth of all cases of inversion (2). In a MEDLINE English literature search from 1940 to October 2000, using the search terms ‘uterine sarcoma’ and ‘uterine inversion’, we found only 12 reports of uterine inversion caused by uterine sarcoma (1–5). Therefore, there have been only 14 cases including our cases. Table 1 shows the frequency of primary disease of non-puerperal uterine inversion in the published reports (1–11). Furthermore, it has been described that the symptoms associated with non-puerperal uterine inversion were vaginal bleeding, vaginal tumor, lower abdominal pain, menorrhagia and urinary disturbance. Including our cases, none of the reported cases were in shock, which is sometimes associated with puerperal uterine inversion.
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Uterine inversion is suspected when a tumor is palpable in the vagina but the uterine fundus is not palpable by a pelvic examination (5). In the present cases, extruded tumors were observed in the vagina, but a pelvic examination failed to identify uterine inversion. MRI has been shown to be a useful diagnostic tool since it can examine the characteristic image of uterine inversion. Lewin et al. reported that, in T2-weighted MRI scans, a U-shaped uterine cavity and a thickened and inverted uterine fundus on a sagittal image and a ‘bulls-eye’ configuration on an axial image are signs indicative of uterine inversion (12). In case 1, a T1-weighted dynamic image enhanced by gadolinium was effective in detecting an extruded tumor in the vagina and a U-shaped uterine cavity. One explanation for why both a T1- and a T2-weighted MRI scan can examine uterine inversion caused by sarcoma may be that uterine sarcoma has varieties of tumor components.
The etiological factors of this combination include (a) sudden extrusion of a tumor from the uterus, (b) thin uterine wall, (c) dilatation of the uterine cervix, (d) tumor size, (e) thickness of the tumor pedicle and (f) tumor attachment site (3). Including our cases, as the tumor with its pedicle attaching to the uterine fundus increased in size, the uterine wall became thinner and the uterine muscle became progressively relaxed at a point where the pedicle had attached. Then, as the cervix further dilated, the tumor extruded into the vagina, thus resulting in uterine inversion. When a rapidly growing tumor exhibits signs indicative of myoma delivery, patients should be treated as having not only uterine sarcoma, but also uterine inversion.
Ehrlich et al. reported that the abdominal approach is the best and least hazardous treatment for uterine inversion caused by uterine sarcoma (5). As mentioned above, patients with this disease are less likely to suffer shock condition. Consequently, there is enough time to perform tests such as MRI to deliver accurate preoperative diagnosis. As long as extrauterine infiltration is not confirmed, total abdominal hysterectomy and bilateral salpingo-oophorectomy are suitable. In the present cases, chemotherapy, mainly utilizing etoposide, was performed postoperatively on the grounds that oral and intravenous chemotherapy with etoposide had effectiveness in the management of uterine sarcoma (13,14). Whereas one patient is free of disease postoperatively, the other developed metastatic tumors in the lung 8 months postoperatively and ultimately died of sarcoma. At present, operation and/or chemotherapy have been recommended for the treatment of uterine inversion caused by sarcoma, but the standard therapy has not yet been established. Therefore, we should investigate the treatment for this combination in the future.
We have presented two cases with uterine inversion caused by uterine sarcoma. When a tumor extruded into the vagina rapidly increases in size, MRI is a useful tool in diagnosing not only uterine sarcoma, but also uterine inversion.
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FOOTNOTES |
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+ For reprints and all correspondence: Yoshihito Ichikawa, Department of Obstetrics and Gynecology, Institute of Clinical Medicine, University of Tsukuba, 1–1–1 Tennohdai, Tsukuba, Ibaraki 305-8575, Japan E-mail:yoichika@md.tsukuba.ac.jp
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REFERENCES |
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Received July 24, 2000; accepted October 18, 2000.
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