Drainage by Ovarian Incision for the Treatment of Massive Ovarian Edema Torsion During Pregnancy.

Massive ovarian edema (MOE) is a rare benign condition presenting as unilateral ovarian enlargement with stromal edema, and only a limited number of MOE cases during pregnancy have been reported. MOE is often complicated by ovarian torsion, which requires detorsion. Although the diagnosis of MOE can be made using ultrasound and magnetic resonance imaging, its rarity makes diagnosis difficult, usually leading to overtreatment. Preserving the ovary in the treatment of MOE torsion is essential, and consideration of oophoropexy after detorsion is often reported. However, fixing an enlarged ovary to the pelvic wall in the limited space of the pelvis is challenging. Herein, we present a case of MOE of the right ovary diagnosed at the fifth week of gestation after ovulation induced by clomiphene citrate. Torsion of the ovary occurred in the seventh week. We achieved preservation of the ovary through laparoscopic surgery with detorsion and drainage by making a small incision to the enlarged ovary, resulting in an immediate size reduction. There was no recurrence of torsion or MOE throughout the pregnancy, and the patient gave birth in the 39th week of gestation. This is the third reported case of MOE after ovulation using clomiphene citrate, and it highlights the effectiveness of treatment with detorsion and a small incision of the ovary via laparoscopic surgery in patients with MOE torsion during pregnancy.

Ureter Injury in Laparoscopic Para-Aortic Lymphadenectomy for Endometrial Cancer by the Transperitoneal Approach.

The patient was 66 years old, had three pregnancies and two deliveries, and was menopausal at the age of 51. She had irregular bleeding and was found to have a chicken-egg-sized uterus and a thickened endometrium (23 mm). She underwent laparoscopic surgery for uterine endometrial cancer (endometrioid carcinoma G1, stage IB). Laparoscopic simple hysterectomy, bilateral adnexectomy, pelvic lymph node dissection, para-aortic lymph node dissection, and partial omentectomy were performed using the transperitoneal approach (TPA). The patient was obese, with a height of 148 cm, a weight of 68 kg, and a body mass index of 31 kg/m2. She had a large amount of visceral fat, which made it difficult to expand the surgical field during para-aortic lymph node dissection. A laparoscopic fan retractor (EndoRetract II, Medtronic) was used to lift the intestinal tracts and expand the field of view. It broke the fat around the left kidney, and the exposed left ureter was heat-damaged using a vessel sealing device (LigaSure, Medtronic). Postoperatively, a left ureteral stent was placed, and continuous urine draining into the retroperitoneum was performed. To prevent injury to the left ureter, the left ovarian vein branching from the left renal vein should be exposed as a landmark before the left ureter running parallel to it is isolated. It is essential that the fat around the left kidney is not broken during this operation. The left iliopsoas muscle should be exposed, and using this as a base, the left ovarian vein, left ureter, and left perirenal fat should be compressed and moved to the left side using a fan retractor to ensure a safe operation.

Laparoscopic intervention for solid pelvic tumours in Mayer-Rokitansky-Küster-Hauser syndrome: a case of bilateral uterine adenomyomas of the rudimentary uterus.

Mayer-Rokitansky-Küster-Hauser (MRKH) syndrome, a condition caused by Müllerian anomalies, is characterised by congenital vaginal aplasia and a rudimentary uterus. Case reports concerning uterine fibroids associated with MRKH syndrome are limited, and differentiating between uterine fibroids and ovarian solid tumours prior to surgical intervention is often challenging. Here, we present the case of a patient with MRKH syndrome and asymptomatic bilateral pelvic solid tumours located close to both ovaries. Based on intraoperative and histopathological findings, the tumours were diagnosed as adenomyomas of the rudimentary uterus. This is the first reported case of a uterine adenomyoma associated with MRKH syndrome. Moreover, our report highlights the fact that diagnostic laparoscopy is a valuable method to evaluate pelvic tumours in MRKH syndrome.

A randomized, double-blind, placebo-controlled, dose-escalating phase I trial to evaluate safety and immunogenicity of a plant-produced, bivalent, recombinant norovirus-like particle vaccine.

Noroviruses (NoV) are the leading cause of epidemic acute gastroenteritis in humans worldwide and a safe and effective vaccine is needed. Here, a phase I, double-blind, placebo-controlled clinical trial was performed in 60 healthy adults, 18 to 40 years old. Safety (primary objective) and immunogenicity (secondary and exploratory objectives) of a bivalent (GI.4 and GII.4), plant-produced, virus-like particle (VLP), NoV vaccine candidate formulation were investigated at two dose levels (50 µg + 50 µg and 150 µg + 150 µg) without adjuvant. Overall, 13 subjects (65.0%) in the 50 µg group, 16 subjects (80.0%) in the 150 µg group, and 14 subjects (70.0%) in the placebo group reported at least 1 solicited local or general symptom during the 7-day post-vaccination periods following each dose. Severe solicited adverse events (AEs) were rare (2 events in the 50 µg group). A total of 8 subjects (40.0%) in each group reported at least one unsolicited AE during the 28-day post-vaccination periods. Immunogenicity was assessed on days 1, 8, 29, 57, 183 and 365. All subjects were pre-exposed to norovirus as indicated by baseline levels of the different immunological parameters examined. Vaccine-specific humoral and cellular immune responses increased after the first dose but did not rise further after the second vaccination. Increased GI.4- and GII.4-specific IgG titers persisted until day 365. The vaccine elicited cross-reactive IgG antibodies against non-vaccine NoV VLPs, which was more pronounced for NoV strains of the same genotype as the GII.4 vaccine strain than for non-vaccine genotypes. Significant blocking anti-GI.4 and anti-GII.4 VLP titers were triggered in both dose groups. Lymphoproliferation assays revealed strong cell-mediated immune responses that persisted until day 365. In conclusion, both dose levels were safe and well-tolerated, and no higher incidence of AEs was observed in the higher dose group. The data show that a single dose of the vaccine formulated at 50 µg of each VLP is sufficient to reach a peak immune response after 8 to 28 days. The results of this Phase I study warrant further evaluation of the non-adjuvanted vaccine candidate. Clinical trial registration: https://clinicaltrials.gov/ct2/show/record/NCT05508178, identifier (NCT05508178).

Gastrointestinal Stromal Tumor with Repeated Multiple Cerebral Infarction Mimicking Ovarian Cancer with Trousseau's Syndrome.

We report a case of gastrointestinal stromal tumor (GIST) with repeated multiple cerebral infarctions mimicking ovarian cancer. A 79-year-old postmenopausal woman had multiple cerebral infarctions with a giant pelvic tumor detected by computed tomography. Ovarian cancer with Trousseau's syndrome was suspected. Through laparoscopic biopsy on the tumor surface, she was diagnosed with left ovarian fibrosarcoma; although, the abdominal cavity could not be observed appropriately. Ovarian fibrosarcoma is an extremely rare tumor and still has no adequate treatment strategy. Complete resection was planned. The tumor was extremely fragile, and gelatinous that it easily bled. Meanwhile, the uterus and bilateral ovaries and fallopian tubes were all normal. The tumor invaded only the peritoneum near the left sacral uterine ligament and sigmoid colon, with no peritoneal dissemination. To completely remove the tumor, we performed total hysterectomy with bilateral salpingo-oophorectomy and omentectomy and sigmoidal and rectal resection with colostomy. Despite resuming her anticoagulant therapy on postoperative day 4, she had recurrent multiple strokes. On histopathological examination, tumor showed spindle cell proliferation with severe atypia, increased mitotic activity, and widespread necrosis. Immunohistochemical studies showed positive staining for c-kit, CD34, and DOG1. Thus, she was diagnosed with GIST. This case was rare and highly malignant, with a high risk of recurrence of GIST because of a giant ruptured tumor that had a mitotic activity of 36/10 high-power fields from the sigmoid colon. Multiple cerebral infarctions mimicking ovarian cancer recurred. Therefore, preoperative diagnosis of an atypical GIST was extremely difficult.

Placental polyp with arteriovenous malformation treated with a gonadotoropin-releasing hormone antagonist.

A 35-year-old woman (gravida 1, para 0) underwent termination of pregnancy (ToP) at 12 weeks of gestation. One month after ToP, she experienced significant vaginal bleeding and the mass with blood flow was identified on imaging. The presence of a placental polyp with arteriovenous malformation (AVM) was suspected on transvaginal sonography and MRI. Since the bleeding had ceased when she visited our hospital, we decided to treat the placental polyp with AVM with gonadotropin-releasing hormone (GnRH) antagonist therapy instead of surgery. Two months after GnRH antagonist treatment, the mass and blood flow in the uterus disappeared. Menstruation resumed 1 month after the completion of treatment. In our case, we were able to successfully treat placental polyps with AVM using GnRH antagonist therapy.

Characteristics and outcomes of women with adenocarcinoma versus squamous cell carcinoma of the vulva: A Japanese Gynecologic Oncology Group study.

OBJECTIVE: Studies on vulvar adenocarcinoma are lacking. Thus, we aimed to compare the characteristics and survival outcomes between vulvar adenocarcinoma and squamous cell carcinoma (SCC). METHODS: This was a preplanned sub-analysis of a previously organized nationwide retrospective observational study in Japan conducted between 2001 and 2010 (JGOG-1075S). Surgically treated women with stage I-IV vulvar invasive adenocarcinoma were compared to those with SCC. Multivariable analysis was performed to identify patient and tumor characteristics related to adenocarcinoma. Inverse probability of treatment weighting was used to balance the background differences, and a Cox proportional hazards regression model was fitted to estimate the effect of the histological type on survival. RESULTS: Forty-eight women with adenocarcinoma were compared with 537 women with SCC. On multivariable analysis, women with adenocarcinoma were younger (median age, 64.5 vs. 70 years, adjusted odds ratio [OR] per age 0.975, 95% confidence interval [CI] 0.955-0.995, P = 0.016) and had higher positive surgical margin rates (31.2% vs. 18.4%, adjusted OR 2.376, 95% CI 1.188-4.754, P = 0.014) than those with SCC. However, according to the weighted model, the survival outcomes were comparable (hazard ratio for progression-free survival, 1.088, 95% CI 0.740-1.601, P = 0.667 and hazard ratio for overall survival, 1.008, 95% CI 0.646-1.573, P = 0.973). Similar associations were observed when the cohort was stratified by age (≤70 or >70 years), stage (I-II or III-IV), and surgical margin (negative or positive) (all, P > 0.05). CONCLUSION: Vulvar adenocarcinoma is characterized by a younger age at diagnosis and higher positive surgical margin rates than SCC, but the survival outcomes are comparable.

Robot-assisted laparoscopic hysterectomy for endometrial cancer in a patient with Herlyn-Werner-Wunderlich syndrome.

Herlyn-Werner-Wunderlich syndrome, a rare Mullerian duct anomaly, includes a triad of uterine didelphys, obstructed haemivagina and ipsilateral renal agenesis. A 58-year-old woman with Herlyn-Werner-Wunderlich syndrome, reported of recurrent genital bleeding for 9 years, was finally diagnosed with endometrial cancer. She had a history of vaginal septum resection and nephrectomy of atrophic right kidney. MRI demonstrated uterine didelphys, a tumour filling the left uterus and a cyst on the right lateral side of the uterus. Robot-assisted hysterectomy, including bilateral salpingo-oophorectomy and pelvic lymphadenectomy, was performed. As the cyst communicated with the right cervix, but not with the urinary tract, a Gartner duct cyst was diagnosed. Uncertain diagnosis and delay of treatment in endometrial cancer may occur in patients with Herlyn-Werner-Wunderlich syndrome. We should preoperatively fully evaluate the anatomy of the uterus and surrounding tissues and plan surgical procedures, especially in patients with urogenital malformations.

Management of pulmonary contusions during the COVID-19 pandemic.

BACKGROUND: Coronavirus disease (COVID-19) is an emerging infectious disease with human-to-human transmission. Early identification of patients with COVID-19 is important in preventing its spread. CASE PRESENTATION: A 36-year-old man with a fever for 3 days fell from the 5th floor and was transported to our emergency department. Pan-scan computed tomography (CT) revealed multiple ground-glass opacities just below the pleura, pelvic fractures, thoracic vertebral fracture (Th11), and right femoral fracture. COVID-19 was ruled out based on negative real-time reverse transcription-polymerase chain reaction results on days 2 and 3, and a reduction in the multiple ground-glass opacities on CT carried out on day 5. Until the suspicion of COVID-19 was cleared, strict and standardized processing procedures were implemented in the same way as for a patient with COVID-19. CONCLUSIONS: It is very difficult to rapidly differentiate between pulmonary contusions and COVID-19, as these conditions have many similarities on CT.

A Case of Trousseau's Syndrome Accompanying Ovarian Cancer with Widespread Thromboembolisms.

The patient was a 41-year-old woman, gravida 0. She had no notable medical history. Laparoscopic right salpingo-oophorectomy and left cystectomy were performed for bilateral ovarian endometriomas, which were both pathologically diagnosed as benign. Six months later, she presented with left lower abdominal pain and expressive aphasia. Examination revealed multiple cerebral infarctions and pulmonary embolism. The patient was diagnosed with Trousseau's syndrome secondary to ovarian cancer, and anticoagulant therapy was initiated. Despite treatment, she developed visual field loss due to occlusion of the left retinal artery; dizziness due to cerebellar infarction and myocardial infarction; and right hemiplegia due to new cerebral infarction. She received chemotherapy (two courses of paclitaxel and carboplatin), which did not improve her condition, and died two months after onset. An autopsy revealed that her left ovary was enlarged to a size of 12 cm and an endometrioid carcinoma G2 was identified. Ovarian cancer had spread throughout the abdominal cavity, and a large amount of pleural and ascites fluid was present. Multiple thrombi were found in bilateral pulmonary arteries and bilateral common iliac veins. There was a 2.5 cm thrombus in the left ventricle apex, and the anterior descending branch was obstructed by thrombus with recanalization.

Correction to: Sudden fatal bleeding from a uretero-arterial fistula combined with pre-existing uretero-colic and uretero-vaginal fistulas 7 years after a cervical cancer surgery: a case report.

In the original publication of this article [1], the family name of an author is wrong.

Sudden fatal bleeding from a uretero-arterial fistula combined with pre-existing uretero-colic and uretero-vaginal fistulas 7 years after a cervical cancer surgery: a case report.

BACKGROUND: Uretero-arterial fistulas (UAFs) are a rare cause of hematuria and could be fatal often due to a diagnostic delay despite recent advances in the treatment modalities. CASE PRESENTATION: A 52-year-old woman with a history of advanced cervical cancer developed a fever and was diagnosed with a left uretero-colic fistula. She also had a uretero-vaginal fistula and suffered from repeated urinary tract infections over 6 years. While waiting for an elective colostomy, she developed sudden perineal bleeding and died 14 h after the onset. The autopsy findings revealed that bleeding from a newly developed UAF spreads out to the extracorporeal space through the pre-existing fistulas. CONCLUSIONS: Bleeding from a UAF complicated by other uretero-genital and/or uretero-enteric fistulas could proceed rapidly resulting in a fatal outcome because of a lack of a tamponade effect. Early recognition of a UAF in high-risk patients is crucial for a prompt diagnosis, which might lead to a treatment success.

Management of Airway Obstruction due to Diffuse Idiopathic Skeletal Hyperostosis in the Cervical Spine: A Case Report and Literature Review.

Diffuse idiopathic skeletal hyperostosis (DISH) is a relatively common progressive noninflammatory entheses disease. Patients are often asymptomatic or are undiagnosed due to minor chronic symptoms. We herein report a rare case in which the primary symptom was sudden-onset upper airway obstruction due to exuberant osteophytosis in the cervical spine. Treatment was successful with careful airway management and surgical osteophyectomy. Most DISH cases in the literature with airway obstruction have been managed with tracheotomy. However, the safety and necessity of this approach remain questionable. We herein discuss the possibility of conservative management as a choice of airway control. Airway obstruction due to DISH may be underrecognized. This highlights the importance of including DISH in the differential diagnosis of airway obstruction. In addition, a detailed evaluation and personalized care for each individual case is essential.

Acute Abdomen due to Mutual Tangle of Two Small Paratubal Cysts.

A 30-year-old woman (gravida 0) visited our hospital with a complaint of right lower abdominal pain. Transvaginal ultrasonography revealed a 5-cm swollen right ovary, which was suspected to be a mature cystic teratoma. Pelvic examination revealed moderate pain. Contrast-enhanced computed tomography showed a 44-mm cystic mass containing fat and calcified material in the right pelvis. Since torsion was suspected, emergent laparoscopic surgery was performed. Intraoperative findings were a swollen right ovary without torsion or congestion. Two small pedunculated 1- and 2-cm diameter paratubal cysts that grew from almost the same place of the ampulla of the right fallopian tube were observed. The thin stalk of the 1-cm paratubal cyst was entangled around the stalk of the 2-cm paratubal cyst, with its head congested. Through a small abdominal laparoscopic incision, the tumor of the right ovary and the two paratubal cysts were excised. Histopathological examination revealed that the right ovarian tumor was a mature cystic teratoma, and the two paratubal cysts had no malignancy. This case showed that only a 2-cm tumor with congestion caused the acute abdomen.

The Cytoreductive Effect of Radiotherapy for Small Cell Ovarian Carcinoma of the Pulmonary Type: A Case Report and Review of the Literature.

Small cell ovarian carcinoma of the pulmonary type is a rare and highly aggressive tumor for which a suitable treatment strategy has not been established. A 45-year-old woman presented with abdominal swelling, and primary ovarian carcinoma was suspected. The postoperative pathological diagnosis was small cell ovarian carcinoma of the pulmonary type. She also had complicated grade 1 endometrioid carcinoma of the uterine corpus. Three courses of cisplatin and etoposide therapy were administered as adjuvant chemotherapy. Because the tumor was chemotherapy resistant, she underwent palliative abdominal irradiation at a dose of 26 Gy in 13 fractions, which induced cytoreduction and provided symptomatic relief. She died 4 months after surgery. Lactate dehydrogenase was a useful tumor marker during treatment. Here, we present an extremely rare case of a patient with small cell ovarian carcinoma of the pulmonary type treated with radiotherapy after surgery and chemotherapy.

Adult-onset Krabbe disease presenting with an isolated form of peripheral neuropathy.

INTRODUCTION: Adult-onset Krabbe disease is clinically rare and usually affects the pyramidal tracts in the central nervous system. Patients develop a spastic gait, and peripheral neuropathy sometimes occurs simultaneously. METHODS: A 55-year-old woman with consanguineous parents developed slowly progressive, asymmetric muscle weakness and atrophy in her forearms, while her ability to walk remained unaffected without pyramidal tract signs after onset at age 51 years. RESULTS: Nerve conduction studies demonstrated an asymmetric demyelinating-type peripheral neuropathy, and sural nerve biopsy documented reduced myelinated nerve fiber density with uniformly thin myelin sheaths, suggesting hypomyelination. Brain MRI demonstrated minor white-matter injury along the optic radiations, which was associated with asymptomatic, mild, prolonged latency on visual evoked potentials. Laboratory analysis documented low enzyme activity of galactocerebrosidase (GALC) and a known mutation of the GALC gene. CONCLUSION: Isolated peripheral neuropathy occurs very rarely in adult-onset Krabbe disease. Muscle Nerve 54: 152-157, 2016.

Trichoepithelioma Arising in an Ovarian Mature Cystic Teratoma.

Here, we report an extremely rare case of trichoepithelioma (TE)-a benign epithelial tumor originating from the outer root sheath of a hair follicle-arising in an ovarian mature cystic teratoma (MCT) with fluorodeoxyglucose-positron emission tomography (FDG-PET) findings. A 48-year-old Japanese woman presented to our hospital for her annual follow-up of adenomyosis. Ultrasonography and magnetic resonance imaging revealed a left ovarian tumor with irregular-shaped septum, which was suspicious of malignancy. However, tumor marker levels were within normal range. On FDG-PET, the maximum standardized uptake value (SUVmax) of the tumor was 2.9. Laparotomy with left salpingooophorectomy was performed. Pathologic examination revealed the probability of TE, rather than basal cell carcinoma (BCC), arising in an ovarian MCT. After five years of follow-up, the patient had no sign of recurrence. The FDG-PET SUVmax was low in TE, as with other benign tumor. However, future investigation is needed to evaluate the findings of FDG-PET imaging in TE cases.

Calcium channel blocker attenuated opioid withdrawal syndrome.

Case: A 61-year-old woman was diagnosed with deep cervical abscess and enlarged mediastinal abscess. These required a protracted period of mechanical ventilation and neck and thoracic drainage surgery with daily wound lavage, necessitating the administration of large amounts of fentanyl and dexmedetomidine. After extubation, fentanyl was discontinued but dexmedetomidine was continued, and she developed hypertension, tachycardia, tachypnea, and hyperthermia within several hours; therefore, she was diagnosed with opioid withdrawal syndrome. Her symptoms failed to improve with either an increased dexmedetomidine dose or a diltiazem infusion for symptomatic management. Ultimately, 20 mg nifedipine was given through a nasogastric tube, which led to a resolution of withdrawal symptoms. Outcome: This is the first case of calcium channel blockers attenuating opioid withdrawal syndrome symptoms in a human. Conclusion: Calcium channel blockers might be alternative therapy to refractory opioid withdrawal syndrome. Case accumulation in the future is expected.

Inhibition of hepatocellular carcinoma by PegIFNα-2a in patients with chronic hepatitis C: a nationwide multicenter cooperative study.

BACKGROUND: We investigated whether the administration of maintenance doses of interferon prevented hepatocellular carcinoma (HCC) in patients with chronic hepatitis C. METHODS: Study 1: A multicenter, retrospective, cooperative study was carried out to determine whether long-term administration of low-dose peginterferon alpha-2a (PegIFNα-2a) prevented HCC development in patients with chronic hepatitis C. In total, 594 chronic hepatitis C patients without a history of HCC were enrolled and treated with 90 µg PegIFNα-2a administered weekly or bi-weekly for at least 1 year. Study 2: HCC developed in 16 of 99 additional patients without PegIFNα-2a treatment during 3.8 years of observation. A propensity-matched control study was then carried out to compare the incidence of HCC between the 59 patients who received low-dose PegIFNα-2a (PegIFNα-2a group) and 59 patients who did not receive PegIFNα-2a treatment (control group), matched for sex, age, platelet count, and total bilirubin levels. RESULTS: Study 1: HCC developed in 49 patients. The risk of HCC was lower in patients with undetectable hepatitis C virus RNA, ≤40 IU/L alanine aminotransferase (ALT), or ≤10 ng/L alpha-fetoprotein (AFP) 24 weeks after the start of therapy. Study 2: The incidence of HCC was significantly lower in the PegIFNα-2a group than in the control group. CONCLUSIONS: Low-dose and long-term maintenance administration of PegIFNα-2a decreased the incidence of HCC in patients with normalized ALT and AFP levels at 24 weeks compared with patients without normal ALT and AFP levels.

[Case of primary racemose hemangioma of a bronchial artery with life-threatening hemoptysis].

A 22-year-old woman was admitted to our hospital because of hemoptysis and respiratory insufficiency. The chest roentgenogram and the chest computed tomogram showed infiltrative shadows in the bilateral lower lobes and mediastinal emphysema. On the second day of hospitalization, we performed double lumen endotracheal tube intubations for the repeated life-threatening hemoptysis. Bronchoscope examination revealed normal bronchus with fibrin formation. Bronchial autobiography (BAG) showed a convoluted and enlarged right bronchial artery and bronchial-pulmonary artery shunt. We diagnosed primary racemose hemangioma of a bronchial artery and performed bronchial artery embolism (BAE) of the right upper bronchial artery using coil. There was no reccurence of hemoptysis after BAE procedure. BAE with coil seems to be effective for life-threatening hemoptysis due to racemose hemangioma.