Large uterine arteriovenous malformation successfully treated with combined endovascular treatment and supracervical hysterectomy: A case report.

Uterine arteriovenous malformations (AVMs) are rare conditions that cause life-threatening bleeding. Endovascular treatment or total hysterectomy is performed to safely treat most AVMs. This case report describes a 54-year-old female patient with a large uterine AVM, uterine bleeding, and cardiac overload that was difficult to manage but successfully treated. Total hysterectomy poses a high risk of hemorrhage due to significant uterine and internal iliac vein dilation; thus, embolization of feeding arteries was performed with N-butyl cyanoacrylate. However, a postembolization computed tomography scan detected paradoxical embolization of the liver, kidneys, and spleen. Therefore, supracervical hysterectomy was performed with preoperative coil embolization and intraoperative balloon occlusion of the feeding arteries. In this case, supracervical, not total, hysterectomy needed to be performed as the shunts were determined to be in the uterine corpus.

A case of solitary lymph node recurrence 9 years after initial treatment for advanced premenopausal endometrioid endometrial cancer; clinical usefulness of hormonal replacement therapy.

There is no consensus on the use of hormone replacement therapy (HRT) after treatment of advanced corpus cancer. We report a case of advanced corpus cancer at a young age, in which HRT was initiated 7 years after surgery, and regional lymph node recurrence was later detected. The patient was 35 years old at the time of initial treatment in X year, when she was diagnosed with stageIIIC2 corpus cancer and underwent a hysterectomy with bilateral salpingo-oophorectomy and a retroperitoneal lymphadenectomy. HRT was started at X + 7 years, and at X + 9 years, a 25 × 12-mm-sized mass was found in the hilum of the right kidney. A laparoscopic resection revealed regional lymph node recurrence of the corpus cancer. A retrospective study further revealed that a tumor measuring 12 × 3 mm was found at X + 3 years and grew to 18 × 7 mm in X + 6 years, just before the start of the HRT. We hypothesize that HRT did not induce tumor recurrence; instead, it allowed for long-term follow-up and early diagnosis.

A case of paraovarian tumor of borderline malignancy with decrease of apparent diffusion coefficient value and marked 18F-fluorodeoxyglucose accumulation.

Para-ovarian cysts are occasionally encountered in clinical practice; however, malignant tumors derived from them are rare. Due to its rarity, the characteristic imaging findings of para-ovarian tumors with borderline malignancy (PTBM) are largely unknown. Herein, we report a case of PTBM, along with imaging findings. A 37-year-old woman came to our department with a suspected malignant adnexal tumor. Pelvic contrast-enhanced magnetic resonance imaging (MRI) revealed a solid part within the cystic tumor with a decrease in the apparent diffusion coefficient (ADC) value (1.16 × 10-3 mm2/s). We also performed Positron Emission Tomography-MRI and showed a strong accumulation of 18F-fluorodeoxyglucose (FDG) in the solid part (SUVmax = 14.8). In addition, the tumor appeared to develop independently of the ovary. Because tumor was derived from para-ovarian cyst, we suspected PTBM preoperatively and planned fertility sparing treatment. Pathological examination revealed a serous borderline tumor and PTBM was confirmed. PTBM can have unique imaging characteristics, including a low ADC value and high FDG accumulation. When a tumor appears to develop from para-ovarian cysts, borderline malignancy can be suspected, even if imaging findings suggest malignant potential.

Versican provides the provisional matrix for uterine spiral artery dilation and fetal growth.

The extracellular matrix (ECM) in the endometrium plays a crucial role in mammalian pregnancy. We have shown that versican secreted from the endometrial epithelium promotes embryo implantation. Versican is a proteoglycan, a major player in the provisional matrix, and versikine, its N-terminal fragment cleaved by ADAMTS proteinases, serves as a bioactive molecule. Here, since versican expression in the placenta was dynamically altered in humans and mice, we investigated the role of versican in pregnancy using uterine-specific Vcan deletion mice (uKO mice) and ADAMTS-resistant versican expressing mice (V1R mice). uKO mice exhibited insufficient spiral artery dilation, followed by fetal growth restriction and maternal hypertension. Further analysis revealed impaired proliferation of tissue-resident natural killer cells required for spiral artery dilation. V1R mice showed the same results as the control, eliminating the involvement of versikine. Our results provide a new concept that versican, one factor of ECM, contributes to placentation and following fetal growth.

Nivolumab for malignant transformation of ovarian mature cystic teratoma.

Mature cystic teratoma of the ovary (MCT) occasionally undergoes malignant transformation (MT) that is resistant to chemotherapy and has a poor prognosis. We experienced a case of clinically aggressive MCT-MT that invades surrounding organs and tissues. Although tumor was resected entirely, a rapid tumor recurrence occurred during postoperative chemotherapy (paclitaxel + ifosfamide + cisplatin). The results of comprehensive genomic profiling test performed early in the postoperative period showed a high tumor mutational burden of 23 mutations/Mb. Treatment with nivolumab monotherapy has promptly been initiated and has been very successful for more than one year.

A Case of a Patient with Adhesive Small Bowel Obstruction in Pregnancy after Extensive Myomectomy for Diffuse Uterine Leiomyomatosis.

Background: Diffuse uterine leiomyomatosis is a rare disease in which countless, poorly defined, and small nodules are present in most parts of the uterine myometrium. It frequently occurs in fertile women and causes infertility. A deep, median, longitudinal incision of the uterine corpus with the opening of the endometrial cavity, "extensive myomectomy," is required to restore fertility. However, myomectomy may also be a risk factor for perinatal complications. We present a rare case of adhesive small bowel obstruction after extensive myomectomy for diffuse uterine leiomyomatosis. Case: A 37-year-old primigravida presented with sharp epigastric pain and vomiting at 21-week gestation. The patient had a history of extensive myomectomy for diffuse uterine leiomyomatosis. Abdominal radiography revealed moderate air fluid levels in the small intestine, and the patient was diagnosed with adhesive small bowel obstruction. The patient was also diagnosed with placenta previa. Bowel rest with intestinal tube was continued until delivery. Cesarean section was performed at 32-week gestation due to (i) prolonged fasting and total parenteral nutrition for conservative treatment and (ii) fear of sudden massive bleeding from placenta previa. Because the ileum was strongly adherent to the uterine scar from the previous myomectomy, adhesiolysis and enterectomy were performed. The placenta was uncomplicatedly delivered and the hemorrhage was well-controlled. Conclusions: Pregnancy with a history with extensive myomectomy for diffuse uterine leiomyomatosis should be carefully monitored because of the occasional occurrence of adhesive small intestine obstruction during pregnancy.

Laparoscopic hysterectomy for endometrial cancer after the Fontan procedure: The first case report.

The Fontan procedure is a palliative cardiac surgery performed for children with a single functional ventricle, and its aim is to divert central venous return to the pulmonary artery without passing through the right ventricle. We herein report the first case of endometrial cancer after the Fontan procedure that was successfully treated with laparoscopic hysterectomy. A 38-year-old female who underwent the Fontan procedure at the age of 23 presented with abnormal genital bleeding and was diagnosed with endometrial cancer. Computed tomography revealed distinctive venous vasculature in the pelvis: most of the venous return from the left lower extremity occurred via the extremely distended left ovarian vein. Laparoscopic total hysterectomy and bilateral salpingo-oophorectomy were performed. The pneumoperitoneum was maintained at 8-10 mmHg during the operation so that the high intra-abdominal pressure would not interfere with venous return. In addition, the left ovarian vein was first test clamped to ensure that circulation was maintained and was then resected. Although the volume of blood loss reached 358 ml due to high venous pressure, her postoperative course was favorable, and she had no signs of recurrence at the 12-month follow-up. Our case suggests that laparoscopic hysterectomy might be safe and feasible for patients with endometrial cancer after the Fontan procedure, as long as a preoperative study of pelvic vascularization and intraoperative monitoring of the circulation is carried out.

Recurrent vaginal intraepithelial neoplasia successfully treated with topical imiquimod: A case report.

Vaginal intraepithelial neoplasia (VAIN) is a rare disease associated with human papillomavirus infection. High-grade VAIN is typically treated with either excisional or ablative therapy. However, recurrent VAIN lesions are common and these treatments cause vaginal scarring. Recent studies have indicated that 5% imiquimod is an effective treatment for VAIN. The present report describes a case of a woman diagnosed with recurrent VAIN 3 who was treated with a 5% topical imiquimod cream and achieved a complete response after excision and CO2 laser vaporization. A 53-year-old, gravida 5, para 2 postmenopausal woman who was diagnosed with papillary squamous cell carcinoma by biopsy underwent conization, total abdominal hysterectomy and bilateral salpingo-oophorectomy. A histological examination revealed grade 3 cervical intraepithelial neoplasia with free surgical margins. At 3 years after the hysterectomy, the vaginal smear revealed atypical squamous cells, leading to a pathological diagnosis of VAIN 3. Partial vaginectomy was performed, and VAIN 3 was detected in the lesion with positive margins. At 4 months into follow-up, the vaginal smear revealed a high-grade squamous intraepithelial lesion (HSIL), and subsequent biopsy during colposcopy revealed a pathological diagnosis of VAIN 3. At 3 months after CO2 laser vaporization, the vaginal smear revealed HSIL with suspected recurrence and imiquimod treatment was initiated. One sachet of 5% imiquimod cream (0.25 g) was placed in the entire vagina three times per week for 14 weeks with no apparent complications. At 3 years after the treatment, there has been no recurrence. This case demonstrated that topical imiquimod with careful follow-up is an effective treatment for VAIN and is well-tolerated. Further clinical evidence of the effectiveness and safety of imiquimod in patients diagnosed with VAIN is required.

A Case of Recurrent Hemorrhagic Corpus Luteum with Elevated Follicle-Stimulating Hormone, Controlled by Estrogen/Gestagen Therapy.

A high secretion of follicle-stimulating hormone (FSH) in reproductive-aged women is unusual. We report a case of recurrent corpus luteum hemorrhage and subsequent ovarian torsion with markedly elevated FSH levels in a reproductive-aged woman in the absence of functional gonadotroph adenoma (FGA) or premature ovarian failure (POF). A 22-year-old nulligravid woman with a history of bilateral hemorrhagic corpus luteum and subsequent ovarian torsion presented with acute abdominal pain. An emergency salpingo-oophorectomy of the right side was performed, and the right ovarian torsion due to hemorrhagic corpus luteum was diagnosed. Laboratory tests revealed markedly elevated FSH levels (77.6 mIU/mL). FGA was suspected, but no evidence of tumor was identified. The left ovary enlarged again at one-month follow-up. Estrogen/gestagen therapy (EGT) was started, which reduced the enlarged ovary to normal size. Two years later, her pituitary hormonal status was evaluated in detail. Besides markedly elevated FSH level, slightly elevated LH (31.2 mIU/mL), normal total inhibin B (35.3 pg/ml), abnormally low anti-Müllerian hormone (AMH) (<0.03 ng/mL), and poor FSH response to gonadotropin-releasing hormone stimulation test were found. In the absence of FGA, we conclude that certain disorders of inhibitory factors for FSH function, including inhibin and AMH may exist, which could attribute to the patient's symptoms. EGT was very effective in suppressing the ovarian hyperactivity.

Precocious Solitary Cervical Metastasis from Endometrial Cancer Presenting as Cervical Radicular Pain.

BACKGROUND: Bone metastases from endometrial cancer are rare. To our knowledge, only 2 cases of solitary vertebral metastases from endometrial cancer presenting with osseous and/or neurologic symptoms before the diagnosis of the primary endometrial cancer have been reported; however, in both cases, the metastases were thoracic. Thus, cervical vertebral metastases are extremely rare. CASE DESCRIPTION: We describe the case of a 55-year-old woman who presented with right C6 radicular and neck pain. Neurologic imaging showed destruction of the C6 vertebral body by an extradural mass with kyphotic changes in the cervical spine. Fluorodeoxyglucose positron emission tomography showed increased uptake in the enlarged body of the uterus as well as the C6 vertebral body. Additional pelvic magnetic resonance images and endometrial biopsy confirmed endometrial cancer. The patient underwent excision of the cervical tumor with anterior reconstruction and posterior fixation followed 1 month later by semiradical hysterectomy and bilateral salpingo-oophorectomy with pelvic and para-aortic lymph node dissection. The cervical and endometrial histopathologic findings were similar, and the final diagnosis made was cervical metastatic adenocarcinoma originating from an endometrial cancer. CONCLUSIONS: We describe an extremely rare case of a precocious solitary cervical metastasis from an endometrial cancer presenting as cervical radicular pain. In our review of published reports, we found that solitary spinal metastases are significantly associated with longer overall survival than are multiple lesions. Resection of the spinal lesion with rigid spinal reconstruction followed by radical hysterectomy may be beneficial in such patients.