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ABSTRACT: Endometrioid adenocarcinoma of the uterine cervix is rare and difficult to distinguish from endometrial adenocarcinoma, particularly in large bulky cases. Herein, we report a rare case of a large endometrioid adenocarcinoma in a 58-year-old Japanese woman in which the tumor origin was difficult to identify. The patient presented with abdominal pain and vaginal bleeding and was referred to a gynecologist for suspected uterine cancer based on an evaluation performed at another institution. Imaging studies revealed an 11-cm cervical tumor without parametrial invasion, lymph node, distant metastases, or hydronephrosis, whereas cervical biopsy revealed an endometrioid adenocarcinoma. The patient was subsequently referred to our department for the determination of the tissue origin. The results of immunohistochemical staining were equivocal for cervical and endometrial origin. However, rectal examination confirmed a barrel-shaped cervix extending from the lower uterine segment to the hymenal ring. Human papillomavirus and microsatellite instability tests were negative. Finally, the patient was diagnosed with stage III endometrioid adenocarcinoma of the uterine cervix and achieved complete remission after concurrent chemoradiotherapy, followed by platinum-based systemic chemotherapy.

Demons syndrome is defined by hydrothorax and ascites associated with a benign genital tumor that resolves after resection of the tumor. However, Demons syndrome with pericardial effusion has never been reported. Intensive care unit–acquired weakness is a neurological sequela to sepsis/systemic inflammatory response syndrome, or multi-organ failure. A 47-year-old, nulligravid, Japanese woman, was transferred to our hospital for refractory heart failure and a ruptured ovarian tumor. She had an 11-cm left ovarian tumor with ascites, hydrothorax, and pericardial effusion; she was intubated for pulmonary hypertension and admitted to the intensive care unit for septic shock. Four days later, a left salpingo-oophorectomy was performed for Demons syndrome with pericardial effusion. The histological diagnosis indicated a serous cystadenoma with fibrotic changes. Following surgery, ventilator weaning was delayed due to intensive care unit–acquired weakness. The association between Demons syndrome and pericardial effusion should be recognized to ensure early treatment and for preventing sequalae from the disease.

A 65-year-old, gravida 3, para 2 Japanese woman was referred to our hospital for symptomatic thickening of the endometrial lining. Endocervical and endometrial cytology revealed an adenocarcinoma. The endometrial biopsy specimen was mixed, with a glandular part diagnosed as endometrioid carcinoma and a solid part diagnosed as high-grade mixed large and small cell neuroendocrine carcinoma (L/SCNEC). She underwent extra-fascial hysterectomy with bilateral salpingo-oophorectomy, complete pelvic and para-aortic lymphadenectomy, and omentectomy (FIGO IIIB, pT3b pN0 M0). She currently has no deleterious germline mutation, but high tumor mutation burden and high microsatellite instability (MSI) were identified. She underwent six cycles of platinum-based frontline chemotherapy and achieved complete remission. Immune checkpoint blockade therapy is a promising second-line therapy for MSI-high solid tumors. However, the MSI or mismatch repair (MMR) status of endometrial L/SCNEC remains unclear in the literature. Universal screening for MSI/MMR status is needed, particularly for a rare and aggressive disease.

Although parasitic leiomyoma could be spontaneous or iatrogenic in origin, port-site implantation of parasitic leiomyoma is an iatrogenic benign sequela of laparoscopic surgery. A 30-year-old, primigravida Japanese woman was referred after unresponsiveness to preoperative gonadotropin-releasing hormone for intramural fibroids. Magnetic resonance imaging showed multiple intramural fibroids and left ovarian endometrioma with no malignant features. Laparoscopic myomectomy with power morcellation and ovarian cystectomy were performed, followed by treatment with a combined oral contraceptive. Seven years after the primary surgery, she underwent abdominal myomectomy for a port-site, and peritoneal recurrence of the leiomyoma and intramural leiomyomas was detected. Microscopic examination revealed that resected specimens from the port-site demonstrated leiomyoma with lesser cell density and more prominent hyalinization than those from the uterus. Therefore, clinicians should counsel patients regarding the risks and benefits of laparoscopy with morcellation versus laparotomy. Further development of techniques for uterine tissues extraction is warranted.

Objective: Although uterine didelphys per se is not associated with an impaired ability to conceive, the association between uterine anomalies and gestational trophoblastic neoplasia (GTN) remains unclear. The management of chemotherapy-resistant GTN in women with uterine didelphys raises a new issue regarding whether to perform a hemi-hysterectomy. Case report: A 23-year-old, gravida 1, para 0 Japanese woman was referred with a failed intermittent cervical dilatation for hematometra. Four years previously, she developed a GTN Stage III, score 5. As two cycles of chemotherapy with methotrexate (MTX) and one cycle of EMA-CO (etoposide, MTX, actinomycin D, cyclophosphamide and vincristine) did not result in remission, we performed an abdominal hemi-hysterectomy. After a canalization procedure and cervicoplasty were performed, the patient conceived naturally and prematurely delivered by cesarean section twice. Conclusion: A hemi-hysterectomy should be considered for fertility preservation when GTN develops on either side of a didelphic uterus and adjuvant chemotherapy does not result in remission. Keywords: Adjuvant chemotherapy, Fertility preservation, Gestational trophoblastic disease, Hysterectomy, Anomalies

Small cell carcinoma of the uterine cervix is a rare histological entity that has a poor prognosis. We report the case of a patient with small cell carcinoma of the uterine cervix who underwent a radical hysterectomy during pregnancy. A 33-year-old Japanese woman with genital bleeding was referred at 15 weeks’ gestation. A speculum exam revealed a 5.4-cm-dia. mass in the cervix, and a cervical biopsy revealed small cell carcinoma of the uterine cervix. Imaging studies demonstrated a tumor confined to the cervix, swelling of intra-pelvic lymph nodes, and no distant spread of the tumor. She was diagnosed as having small cell carcinoma of the uterine cervix, stage IB2, and underwent a radical hysterectomy with pelvic lymphadenectomy. She refused any adjuvant therapies, had a systemic relapse 4 months after surgery, and died of the disease 5 months after surgery. Early-stage small cell carcinoma of the uterine cervix should be treated with a definitive therapy soon after diagnosis whether the patient is pregnant or not. Saving the mother’s life should be the top priority.

The dissemination of minimally invasive in utero surgery reduced the mortality of twin reversed arterial perfusion sequence, but the mortality of expectantly treated surgical candidates remains high. A 26-year-old, non-parous, Japanese woman at 13 weeks of gestation had been diagnosed with twin reversed arterial perfusion sequence and was judged as a surgical candidate for radiofrequency ablation. However, she did not undergo surgery because of the anatomical location of the acardiac twin. At 18 weeks of gestation, the blood flow to the acardiac twin disappeared spontaneously. The pump twin began to demonstrate fetal growth retardation during the third trimester. The patient delivered a 1891 g female at term. We macroscopically identified the cause of the fetal growth retardation as velamentous insertion of the umbilical cord and microscopically diagnosed the acardiac twin with acardiac acephalus. We should give the same attention to the management of post–twin reversed arterial perfusion sequence as twin reversed arterial perfusion sequence itself.

Objective: Pregnancy does not increase the risk of bleeding from a brain arteriovenous malformation (AVM), but once an AVM has bled during pregnancy, the rate of rebleeding during the same pregnancy is high. Therefore, termination of the pregnancy is an option for patients in whom the AVM is located in an eloquent area. We report a woman with an intracerebral hemorrhage from a brain AVM who underwent a second-trimester therapeutic abortion by vaginal cesarean section. Case report: A 30-year-old multiparous woman visited our emergency department at 17 weeks of gestation complaining of a sudden-onset headache with vomiting. She had no history of headaches or seizures. Based on the clinical presentation, computed tomography and magnetic resonance imaging, we made a clinical diagnosis of Spetzler–Martin Grade III AVM. Before undergoing stereotactic radiosurgery as a primary treatment, we advised her to terminate her pregnancy and performed a vaginal cesarean section at 19 weeks of gestation. Two months later, the patient underwent gamma knife surgery for the underlying lesion, without complications. Follow-up angiography and magnetic resonance imaging showed that the AVM had disappeared completely. Conclusion: Although its indications are limited, vaginal cesarean section is a useful option for terminating a pregnancy that compensates for the disadvantages of dilatation and curettage and systemic abortifacients.

Background. Although radiation exposure is of great concern to expecting patients, little information is available on the fetal radiation dose associated with prophylactic internal iliac artery balloon occlusion (IIABO). Here we estimated the fetal radiation dose associated with prophylactic IIABO in Caesarean section (CS). Cases. We report our experience with the IIABO procedure in three consecutive patients with suspected placenta previa/accreta. Fetal radiation dose measurements were conducted prior to each CS by using an anthropomorphic phantom. Based on the simulated value, we calculated the fetal radiation dose as the absorbed dose. We found that the fetal radiation doses ranged from 12.88 to 31.6 mGy. The fetal radiation dose during the prophylactic IIABOs did not exceed 50 mGy. Conclusion. The IIABO procedure could result in a very small increase in the risk of harmful effects to the fetus.

Meningeal metastasis is rare in the clinical course of ovarian carcinoma and its prognosis is extremely poor. We experienced a case of carcinomatous meningitis from metastatic ovarian small cell carcinoma. A 33-year-old woman with atypical genital bleeding, was diagnosed with a right ovarian tumor and referred to our department. She underwent a total abdominal hysterectomy, bilateral salpingo-oophorectomy, omentectomy, and lymphadenectomy. It was an optimal debulking surgery. She was diagnosed with ovarian carcinoma classified as Stage IIIc according to the Féderation Internationale de Gynécologie et d’Obstétrique classification system. Histological findings showed small cell carcinoma of the pulmonary type. The tumor was bilateral with paraaortic lymph node involvement. The patient was treated with irinotecan and cisplatin (CPT-P therapy). After 4 courses of CPTP therapy, multiple liver metastases and Virchow’s lymph node metastases were found. She was treated with amrubicin as a secondline chemotherapy, but the treatment was ineffective. Five months after surgery, the patient complained of severe headache and nausea. Lumbar puncture was performed and cytology was positive. Magnetic resonance brain imaging indicated meningeal thickening. The patient was diagnosed with meningeal metastasis and received 19-Gy whole cranial irradiation. In spite of these treatments, her disease progressed rapidly and she was often drowsy. She died of aspiration pneumonia 6 months after surgery.

Minimally invasive surgery (MIS) is performed for various human cancers, among which robot-assisted surgery is widely used in the United States, Europe, and other countries. Presently, the total number of robot-assisted surgeries in gynecology exceeds that in urology and any other clinical department. Our group began using robot-assisted surgery for early-stage uterine corpus cancer as from November 2017 and has already performed 40 robot-assisted operations. For this study, we examined the cases of 35 patients, excluding 3 in whom lymphadenectomy was not performed and 2 who underwent additional vaginal wall plasty. Compared with the first 20 cases, the latter 15 had reduced bleeding and shorter operation times. No serious complications were observed in the whole cohort, but infections, including urinary tract infections and peritonitis, were common among the first 20 patients. Although robot-assisted surgery is considered more advantageous for obese patients than laparoscopic and open surgery, we found no significant differences in bleeding, surgical time, or the number of resected lymph nodes between obese patients (Body Mass Index (BMI) ≥30) and other patients in this present study. Robot-assisted surgery is expected to be more widely used, and further technical improvements are needed. [ABSTRACT FROM AUTHOR]

Purpose of investigation: Endometrial stromal (ES) tumours may show different types of differentiation including smooth muscle and sexcord like elements. A common problem is the differential diagnosis of ES tumour variants from other tumours that have smooth muscle or sex cordlike elements. We report a case of collision tumour that was composed of a lowgrade endometrial stromal sarcoma (ESS) with sexcord like differentiation, a uterine tumour resembling ovarian sexcord tumour (UTROSCT), and a leiomyoma. Result: The patient was a 63yearold multiparous woman who was referred to us with the complaint of recurrent abnormal uterine bleeding. Magnetic resonance imaging demonstrated a 3.4cmdia. solid tumour in her uterine cavity. We performed an abdominal total hysterectomy and a bilateral salpingooophorectomy. On gross and microscopic examination, the tumour consisted of a CD10positive lowgrade ESS, a CD10negative UTROSCT, and a leiomyoma. Conclusion: ESS should be distinguished from UTROSCT and leiomyoma because of its malignant potential. [ABSTRACT FROM AUTHOR]

Objective: We conducted a retrospective, multi-institutional, collaborative study to accumulate cases of neuroendocrine carcinoma of the endometrium, to clarify its clinicopathologic features, treatment, prognosis and prognostic factors to collate findings to establish future individualized treatment regimens. To our knowledge, this is the largest case study and the first study to statistically analyze the prognosis of this disease. Methods: At medical institutions participating in the Kansai Clinical Oncology Group/ Intergroup, cases diagnosed at a central pathologic review as neuroendocrine carcinoma of the endometrium between 1995 and 2014 were enrolled. We retrospectively analyzed the clinicopathologic features, treatment, prognosis and prognostic factors of this disease. Results: A total of 65 cases were registered from 18 medical institutions in Japan. Of these, 42 (64.6%) cases were diagnosed as neuroendocrine carcinoma of the endometrium based on the central pathological review and thus included in the study. Advanced International Federation of Gynecology and Obstetrics stages (stage III and IV) and pure type small cell neuroendocrine carcinoma cases had a significantly worse prognosis. Upon multivariate analysis, only histologic subtypes and surgery were significant prognostic factors. Pure type cases had a significantly worse prognosis compared to mixed type cases and complete surgery cases had a significantly better prognosis compared to cases with no or incomplete surgery. Conclusion: Our findings suggest that complete surgery improves the prognosis of neuroendocrine carcinoma of the endometrium. Even among cases with advanced disease stages, if complete surgery is expected to be achieved, clinicians should consider curative surgery to improve the prognosis of neuroendocrine carcinoma of the endometrium. [ABSTRACT FROM AUTHOR]

Background: Glycosylation is one of the most common post-translational modifications of eukaryotic proteins and is known to undergo dynamic changes in a wide range of biological processes. To date, however, the glycan expression profiles in endometriosis are largely unknown. The objective of the study was to identify the panel of glycans that were aberrantly expressed in endometriosis, a hormone-dependent disease. Methods: The glycan expression profiles in primary cultured human endometriotic cyst stromal cells (ECSCs) and normal endometrial stromal cells (NESCs) were determined by lectin microarray analysis. Distribution of Wisteria floribunda agglutinin (WFA)-binding glycans in ovarian endometriotic cysts and eutopic proliferative phase endometrium were assessed by lectin histochemistry. The expressions of N-acetylgalactosaminyl transferases that synthesize WFA-binding glycans were evaluated in ECSCs and NESCs. Results: We found that the levels of WFA-binding glycans were decreased in ECSCs. Lectin histochemistry revealed that WFA-binding glycans were decreased only in the stromal components of the ovarian endometriotic cysts, but not in the epithelial components, compared to the eutopic proliferative phase endometrium. The expressions of N-acetylgalactosaminyl transferases that synthesize WFA-binding glycans were downregulated in ECSCs. Conclusions: Utilizing lectin microarray analysis and lectin histochemistry, we found that WFA-binding glycans were decreased in endometriosis. The synthetic enzymes of WFA-binding glycans were significantly downregulated in ECSCs. It is suggested that reduced expression of N-glycans with WFA-binding properties on ECSCs is a novel characteristics of endometriosis. [ABSTRACT FROM AUTHOR]