Alpha-fetoprotein-producing serous carcinoma of the uterus metastasizing to the ovaries, mimicking primary ovarian yolk sac tumor: a case report and review of the literature.

In the uterus, most alpha-fetoprotein (AFP) producing neoplasms belong to the categories of malignant mixed muellerian tumor, hepatoid carcinoma, and yolk sac tumor. We describe the case of a 44-year-old woman who presented with vaginal bleeding, pelvic mass, and preoperative elevated AFP serum level, clinically suggestive of a primary ovarian yolk sac tumor. However, histological examination revealed a uterine AFP-producing papillary serous carcinoma, which has metastasized to the ovaries. Upon review of the literature on primary endometrial neoplasms with AFP production, 2 categories with possibly different histogenesis and biological behavior become evident: the primary yolk sac tumor of the uterus in young patients (range, 24-49 years; mean, 34 years) and the common high grade endometrial carcinoma with yolk sac dedifferentiation or aberrant AFP production in elderly patients (range, 55-69 years; mean, 63.7 years). In addition to being only the second case of uterine AFP-producing papillary serous carcinoma, this case is unusual for its clinical and radiological presentation as well as the relatively young age of the patient.     

Malignant mixed Mullerian tumor of the uterine corpus with alpha-fetoprotein-producing hepatoid adenocarcinoma component

OBJECTIVES: Hepatoid adenocarcinoma is a rare tumor and has the histological coexistence of well-differentiated adenocarcinoma and nests of hepatoid cells with immunoreactivity for alpha-fetoprotein (AFP). A case of hepatoid adenocarcinoma in malignant mixed Mullerian tumor of the uterus is presented with a review of the literature. CASE: The patient is a 63-year-old woman who complained of abnormal genital bleeding. Serum AFP concentration was 5060 ng/ml. Histologically, the tumor was composed of endometrioid adenocarcinoma, neoplastic hepatoid cells, and sarcoma component including leiomyosarcoma and rhabdomyosarcoma. After operation followed by six courses of platinum-based chemotherapy, serum levels of AFP dropped into normal range. CONCLUSIONS: This is, to our knowledge, the first report of malignant mixed Mullerian tumor of the uterus with an AFP-producing hepatoid adenocarcinoma component.     

Alpha-fetoprotein producing endometrial adenocarcinoma: report of a case

A case of endometrial adenocarcinoma which is producing alpha-fetoprotein (AFP) is described. AFP has been reported to be a tumor marker of gynecologic tumors, such as endodermal sinus tumor and embryonal carcinoma of the ovary. However, to our knowledge, there has been no report of AFP producing endometrial adenocarcinoma. The histologic study showed poorly differentiated adenocarcinoma of the endometrium and AFP was detected in the tumor cells by an immunoperoxidase-peroxidase-antiperoxidase technique.     

Verrucous carcinoma of the endometrium: case history, pathologic findings, brief review of literature and discussion

BACKGROUND: Verrucous carcinoma is a rare condition. A defined disease of the oral cavity, larynx, esophagus, skin, vulva, vagina and cervix. But a verrucous carcinoma arising from the endometrium without evidence of cervical malignancy or endometrial adenocarcinoma is extremely rare. CASE: A 67-year-old G2P2 menopausal patient that was referred for consultation 1 year after presenting with vaginal bleeding to her gynecologist who subsequently underwent several endometrial biopsies where the pathological findings were repetitively similar: papillary squamous proliferation, cytologically bland with low mitotic activity but extensive proliferation. A hysterectomy with bilateral salpingo-oophorectomy was performed. The final histologic examination revealed a squamous cell carcinoma of endometrium, verrucous and well differentiated, and there was no cervical invasion identified. CONCLUSION: This is a rare form of endometrial cancer with apparent favorable prognosis that must be considered when squamous cells are identified on endometrial samplings.     

Assessment of reliability endometrial brush cytology in detection etiology of late postmenopausal bleedings

We evaluated the possibility of discovering bleeding causes in late postmenopausal period with cytological examination of material received by endometrial brush in comparison with Pap test and fractionated curettage. Sixty-two women in late postmenopausal period with cervical canal bleeding, treated in gynecological department of clinical hospital in Osijek, were cytological and histological processed. Final diagnosis in 29 from 62 (46.8%) women with late postmenopausal bleeding was cancer. 25 (40.3%) women had endometrial adenocarcinoma and 4 (6.5%) of them had squamous endocervical carcinoma. Two women had endometrial precancerous (3.2%). With Pap test accurate diagnosis was set up in 13 from 25 (52.0%) women with endometrial adenocarcinoma and in all of them with squamous endocervical carcinoma. With endometrial brush accurate diagnosis was set up in 14 from 25 (56.0%) women with endometrial adenocarcinoma and in 3 from 4 (75.0%) women with squamous endocervical carcinoma. With fractional curettage the diagnosis of endometrial adenocarcinoma was accurately correct in 21 from 25 (84.0%) women and in all of them with squamous endocervical carcinoma. Cytological examination of material derived with endometrial brush, alike vaginal cytology, is not enough reliable method in our conditions for discovering bleeding causes in late postmenopausal period. Diagnostic exactness of procedure could be increased by histopathological examination of material from endometrial brush procedure and with ultrasound evaluation of endometrium thickness.An erratum to this article can be found at     

The relationship of age and endometrial histology to blood loss and morbidity following vaginal hysterectomy

500 cases of vaginal hysterectomies were studied. Indications for vaginal hysterectomy were cervical carcinoma in situ (211), sterilization (210) and abnormal uterine bleeding (79). Blood loss was increased in those patients 35-44 years of age (mean of 331 ml) and was lowest in the 20-24 year age group (276 ml). Mean blood loss for all patients was 291 ml. Blood loss was significantly (p.01) increased in those patients with a secretory endometrium. Pelvic infection (cellulitis or abscess) was unrelated to absolute blood loss but was significantly increased (p.001) in those patients with pelvic hematomas. These finding suggest that vaginal hysterectomy performed on the perimenopausal patient with a secretory endometrium is most likely to be associated with increased blood loss. However, this increased blood loss does not cause a higher incidence of pelvic infection or prolonged postoperative hospital stay.     

Small cell carcinoma of the endometrium with concomitant pelvic inflammatory disease

Background: Small cell carcinoma of the endometrium is a rare disease entity characterized by bulkiness and predisposition to necrosis. Clinical presentations include postmenopausal bleeding, lower abdominal mass, chronic abdominal pain and menorrhagia. We present a case of small cell carcinoma of the endometrium with concomitant pelvic inflammatory disease. The literature is also reviewed. Case: A 64 year old female presented was admitted with the principal complaints of fever, lower abdominal pain and malodorous vaginal discharge. Bimanual examination revealed cervical motion tenderness with a WBC of 9400 cells/μL and increased levels of neutrophils, band cells and C-reactive protein. Sonography revealed an adnexal echocomplex compatible with tubo-ovarian abscess. Culture of the vaginal discharge revealed the presence of E. coli. Symptoms persisted despite three days of antibiotics administration so a laparotomy was performed with a friable hemorrhagic uterus revealed and an area of necrosis evident in the left adnexa. Malignancy was confirmed from frozen section. Total abdominal hysterectomy, with bilateral salpingo-oophorectomy and optimal debulking, was performed. The final pathology report confirmed small cell carcinoma of the endometrium. Conclusion: Malignancy and pelvic inflammatory disease have overlapping clinical characteristics. Once pelvic inflammatory disease is suspected in a postmenopausal patient, malignancy should also be suspected, and a thorough examination and a tumor-marker analysis performed. Received: 25 June 2001 / Accepted: 25 September 2001     

Yolk sac tumor of endometrium: A case report and literature review

ObjectiveTo report a rare case of endometrial yolk sac tumor (YST) and review published cases of YST of the endometrium.Case reportA 68-year-old female presented with intermittent vaginal spotting for nine months. An endometrial biopsy showed adenocarcinoma. Complete surgical staging operation was performed and the final pathology revealed stage II endometrial yolk sac tumor. The post-operative α-fetoprotein (AFP) level was 133.4 ng/mL. Post-operative adjuvant chemotherapy with bleomycin, etoposide, and cisplatin (BEP) regimen was prescribed for 6 cycles. AFP levels were normal before the fourth cycle of chemotherapy. She is disease free 6 months after completion of therapy.ConclusionPrimary YSTs arising in the endometrium is an extremely rare disease especially in postmenopausal women. Complete surgical staging operation with adjuvant chemotherapy will lead to good outcome in this disease.     

Spontaneous regression of stage IV clear cell carcinoma of the endometrium in a patient with essential thrombocytosis

OBJECTIVE: The aim of this study was to document a case of advanced stage clear cell carcinoma of the endometrium which underwent spontaneous regression (SR) and comment on the possible contribution of the patient's thrombocytosis. CASE REPORT: A 73-year-old woman with essential thrombocytosis presented with vaginal bleeding. Imaging demonstrated a complex uterine mass, a 4-cm infrarenal mass, and a 5-cm subumbilical mass. Biopsy of the subumbilical mass revealed adenocarcinoma, and endometrial curettage revealed extensively necrotic clear cell endometrial carcinoma. At hysterectomy 5 weeks later, the infrarenal and subumbilical masses were not identified. The endometrial tumor was almost completely necrotic. She received no adjuvant therapy and remains disease-free 6 years later. Interestingly, her platelet-lowering agent (hydroxyurea) was discontinued shortly before, and her platelet count was significantly elevated at the time of her presentation with endometrial carcinoma. CONCLUSION: This report documents a rare case of SR of advanced endometrial carcinoma, and we speculate that increased circulating platelets were a major contributing factor.     

Association of low-grade endometrioid carcinoma of the uterus and ovary with undifferentiated carcinoma: a new type of dedifferentiated carcinoma?

Low-grade endometrioid carcinomas, either of the endometrium or the ovaries, usually have an excellent prognosis. The association of this type of tumor with undifferentiated carcinoma is rare. In this study, we present the clinicopathologic features of 25 such cases. The age of the patients ranged from 30 to 82 years (median, 51 years). At presentation, the patients had either vaginal bleeding or pelvic pain. The endometrioid carcinoma involved the endometrium in 14 cases, the endometrium and 1 or both ovaries in 9 cases, and the ovaries in 2 cases. Undifferentiated carcinoma associated with low-grade endometrioid carcinoma was found at presentation in 19 grade 1 or 2 endometrioid carcinomas: 15 in the endometrium and 5 in the ovary. In one of these cases, undifferentiated carcinoma was found in the endometrium and the ovary. Undifferentiated carcinoma was found after resection of low-grade endometrioid carcinoma in six cases, involving the retroperitoneum, pelvis, vagina, or liver. The undifferentiated carcinoma was composed exclusively of diffuse sheets and solid nests of epithelial cells in l0 cases. Epithelial cells with isolated foci of keratinization were seen in nine cases and rhabdoid cells in a myxoid background in six cases. Twenty-four patients were treated with total abdominal hysterectomy and with bilateral salpingo-oophorectomy. Twenty-two patients received additional therapy as follows: chemotherapy (), radiotherapy (), and tamoxifen (). Follow-up showed that 15 patients died of disease in 1 to 60 months (median, 6 months), and 5 patients are alive with progressive disease with a follow-up between 6 and 8 months; 1 patient is alive with no evidence of disease at 104 months. In four cases, the diagnosis was made recently, with short follow-ups of 3 and 4 months. Foci of undifferentiated carcinoma may be confused with solid endometrioid adenocarcinoma erroneously leading to the diagnosis of a grade 3 or a significantly less aggressive grade 2 endometrioid carcinoma. The recognition of undifferentiated carcinoma in an otherwise low-grade endometrioid adenocarcinoma is extremely important because it indicates aggressive behavior. In asynchronous cases, being aware of this association can explain the absence of a second primary.     

Percutaneous embolization in the management of intractable vaginal bleeding

AIM: To assess the effectiveness of arterial embolization in gynecological malignancies by Polyvinyl Alcohol particles. MATERIAL AND METHODS: Six patients, four with cervix carcinoma, one endometrium carcinoma, and one vaginal metastasis of ovarian carcinoma underwent percutaneous embolization due to intractable vaginal bleeding. As an embolic agent PVA particles were used. RESULTS: Cessation of the bleeding was observed immediately after the embolization. Complete embolization has been achieved in all the patients Recurrent bleeding did not occur in any of the cases. There were no complications related to the embolization procedure. CONCLUSION: Transarterial embolization is a lifesaving procedure in treating intractable vaginal bleeding. PVA particles are effective and it is a simple way in ceasing the hemorrhage due to pelvic malignancies.     

Mixed papillary transitional cell carcinoma and adenocarcinoma of the uterine cervix: a clinicopathologic study of three cases.

Although tumors consisting of a combination of transitional cell carcinoma (TCC) and adenocarcinoma have been described in the endometrium, they have not been documented in the uterine cervix to our knowledge. Three such cervical cases are reported in this article. Three patients, whose ages ranged from 40 to 61 years, presented with vaginal bleeding and malignant cells on routine Papanicolaou smears. The initial diagnoses based on a biopsy specimen were poorly differentiated squamous cell carcinoma in two patients and adenocarcinoma with a solid component in the third patient. All patients underwent radical hysterectomy. The hysterectomy specimens each contained a polypoid endocervical mass with minimal invasion of the cervical stroma. On microscopic examination, each tumor consisted of a component of papillary TCC admixed with an adenocarcinoma of endometrioid type. Both carcinomatous components were immunoreactive for cytokeratin (CK) 7 but not CK20. The three patients were alive and disease-free from 10 months to 4 years postoperatively. Recognition of this unusual variant of cervical carcinoma is important to delineate its clinical and pathologic features and establish prognostic differences, if any, from other histologic subtypes of cervical carcinoma. Papillary TCC mixed with adenocarcinoma broadens the morphologic spectrum of transitional cell neoplasms of the uterine cervix.     

The development of adenocarcinoma of the endometrium in young women receiving long-term sequential oral contraception.

4 cases of adenocarcinoma of the endometrium were reported in young women. All were using and had used for periods of 4-12 years a sequential oral contraceptive, Oracon, manufactured by Mead Johnson Laboratories. Ethinyl estradiol and mestranol, the 3 methyl ether of ethinyl estradiol, are 25 times more potent than diethylstilbestrol, which has recently been implicated in the development of vaginal adenocarcinoma in female offspring of women treated during pregnancy with it. A study is now in progress to determine whether long-term use of Oracon produces premalignant changes in the endometrium. Doctors with patients on similar sequential contraceptives should watch for abnormal vaginal bleeding.     

Endometrial carcinoma in septate uterus detected 6 months after full-term delivery: case report and review of the literature

BACKGROUND: Endometrial carcinoma associated with pregnancy is rare. We present the case of a 39-year-old woman with endometrial carcinoma in the septate uterus detected 6 months after normal delivery. CASE: The patient complained of vaginal bleeding from 32 weeks' gestation, and had a spontaneous vaginal delivery at 37 weeks. The bleeding continued after the delivery, but repeated endometrial cytology and biopsy did not reveal evidence of malignancy. An exploratory laparotomy was performed 6 months postpartum with suspicion of uterine myoma. A histological diagnosis of endometrial carcinoma was made based on examination of frozen sections from the hysterectomy specimen, and bilateral salpingo-oophorectomy and pelvic/para-aortic lymphadenectomy were also performed. Histological examination revealed G3 endometrioid adenocarcinoma with squamous differentiation, which arose in the septum of the uterus and deeply invaded the myometrium. The patient received postoperative chemotherapy and is healthy with no evidence of disease 3 years after the treatment. CONCLUSIONS: Although pregnancy-associated endometrial carcinoma is rare, careful examinations are needed when unexplained vaginal bleeding continued.     

Localized amyloidosis in endometrioid carcinoma of the uterus: a rare association

Background Amyloidosis is associated with infections, chronic disorders, hematological malignancies and endocrine neoplasms. Only rarely has it been associated with other tumors. Amyloidosis of the endometrium is rare and has only been reported as part of a systemic disease. Case report A 65-year-old postmenopausal female presented with irregular vaginal bleeding. Ultrasonography revealed a growth in the uterine cavity and enlarged internal and external iliac lymph nodes. Total hysterectomy with Bilateral salpingo-ophorectomy and dissection of pelvic fascia with external and internal iliac lymph nodes was done. The histologic diagnosis was that of a well-differentiated endometrioid adenocarcinoma: villoglandular type. Additionally, there were intratumoral nodular deposits of amyloid which were positive with Congo red showing apple green birefringence on polarization. Conclusion This is an unusual case of amyloidosis associated with endometrioid carcinoma of the uterus.     

A case of triple primary gynecological malignancy

A 71-year-old woman was admitted due to postmenopausal vaginal bleeding. During a routine physical examination, a tumor in the left breast was discovered. A fractionated curettage was performed. Endometrial adenocarcinoma and squamous cell carcinoma of the cervix was found on histological examination. A total hysterectomy was performed. Subsequently, a breast biopsy was performed which revealed an infiltrating duct carcinoma of the breast and a mastectomy was performed. Triple gynecological cancer is discussed.     

Endometrioid adenocarcinoma after insertion of a levonorgestrel-releasing intrauterine system

The levonorgestrel-releasing intrauterine system (IUS) is now widely used and recommended as a reliable contraceptive and treatment for menorrhagia. It also potentially provides a simple alternative to treatment for women with stage-1, grade-1 endometrial cancers who are at high risk for surgery. This case describes a 39-year-old multiparous patient who visited the gynecology outpatient department at a district general hospital with a history of heavy, prolonged periods for 1 year. Hysteroscopy and biopsy specimen revealed benign nonsecretory endometrium and a levonorgestrel-releasing IUS was inserted. The patient visited again 4 years later with an 8-month history of prolonged bleeding. Endometrial biopsy specimen confirmed well-differentiated endometrioid adenocarcinoma. This is the first case report, to our knowledge, where hysteroscopy was performed before levonorgestrel-releasing IUS insertion with conclusive evidence of initial negative malignancy followed by development of endometrial cancer 4 years after insertion. This case raises the question of the safety of levonorgestrel-releasing IUS for the prevention and treatment of endometrial hyperplasia and carcinoma.     

Endometrial metastasis from signet-ring breast carcinoma: case report

Breast cancer rarely metastasizes to the endometrium. The signet-ring variety of breast cancer is very rare and has been reported to metastasize to the endometrium in only three previous reports. We present a patient with signet-ring breast carcinoma with metastasis to the endometrium. The patient presented with vaginal bleeding and was found to have wide spread metastasis. Endometrial metastasis in patients with signet-ring breast cancer usually denotes a poor prognosis.     

Tamoxifen and endometrial pathologies: a prospective study.

OBJECTIVE:The purpose of this study was to prospectively follow a group of women with breast cancer, on tamoxifen, for the development of endometrial pathologies. MATERIALS AND METHODS: Eighty women with breast cancer, on tamoxifen, were prospectively followed every 6 months with pelvic examination, Pap smear, vaginal ultrasound, and endometrial biopsy. RESULTS: Nine women were lost to follow-up prior to initiation of treatment and 4 refused biopsies, leaving 67 patients for evaluation. Fifty (74.6%) of the 67 patients were already on tamoxifen for a mean duration of 15.8 +/- 16.6 months and had a baseline benign, unremarkable endometrium at the time of entry into the study. The total duration of treatment was 32.5 +/- 19.6 months (median 30 months). The mean age of the patients was 51.7 +/- 9.9 years (median 52 years). Of the patients, 56.7% were postmenopausal. Sixty-three patients had a benign endometrium (mean age 51.8 +/- 10.1 years, mean duration 33.1 +/- 19.6 months). Two patients had simple hyperplasia (mean age 43.5 years, duration 28.5 +/- 33.2 months), 1 patient had complex hyperplasia with atypia (age 57 years, duration 13 months), and another patient developed adenocarcinoma (grade 3) after 22 months. These 4 patients had abnormal vaginal bleeding. Seven patients developed endometrial polyps (mean age 54.0 +/- 8.5 years, duration 36 +/- 24.2 months). The mean endometrial thickness for patients with histologically unremarkable and abnormal endometrium was not significantly different (7.6 +/- 3.9 vs 8.8 +/- 5.0 mm, respectively) (median 7.0 mm for both groups). No endometrial thickness cutoff point reached statistical significance. The patient who developed endometrial cancer had a thickness of only 3 mm. CONCLUSION: All patients who developed an abnormal endometrium had abnormal vaginal bleeding. There was no correlation between endometrial thickness and endometrial pathology; thus the value of routine screening remains controversial.     

Secretory adenocarcinoma of the endometrium

Secretory adenocarcinomas of the endometrium are uncommon tumors distinct from clear cell carcinomas. We reviewed nine cases that included the original endometrial curettings and the specimens of uteri with both adnexa [total abdominal hysterectomy-bilateral salpingo-oophorectomy (TAH-BSO)]. The patients' ages ranged from 36 to 79 years (with an average of 55 years). Six women were postmenopausal, and most complained of vaginal bleeding. Two patients were nulliparous and the others had one to four children. Four patients were obese, of whom one was diabetic and hypertensive. Eight tumors were of grade I and one was grade II. The histologic pattern was comparable to that of secretory endometrium, days 17 to 22, and the glands were positive with periodic acid-Schiff (whether predigested or not); they were partly positive with alcian blue and negative with Best Carmine. The carcinoma in one case was positive for carcinoembryonic antigen; all cases were negative with alpha-fetoprotein. Six patients were staged as IA and three as IB. One 47-year-old patient had a concurrent endometrioid adenocarcinoma (grade II) of the right ovary with squamous, clear cell, and mucinous components. Three cases had no tumour penetration of the myometrium while in the others penetration varied from 5 to 70%. One patient received intracavitary radium prior to TAH-BSO and two had postoperative radiation. All patients are alive 11 to 113 months following surgery. Secretory adenocarcinoma of the endometrium should be separated from clear cell carcinoma, as it has the pattern of secretory endometrium day 17 to 22, is very well differentiated, and has a relatively good prognosis.