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Ovarian cysts develop rarely in fetuses during pregnancy and usually disappear after birth. However, during pregnancy, torsion and rupture of the cyst can occur and it is necessary to manage such cases. At present, there is no standardized prenatal or neonatal treatment. However, the preservation of ovarian function is an important consideration. Here, we present a case involving a 35-year-old woman who gave birth to an infant with a complicated ovarian cyst, which was resolved through laparoscopic surgery on the third day after birth. The prenatal diagnosis of the fetal ovarian cyst was performed prenatally using ultrasound and magnetic resonance imaging (MRI). However, torsion of the ovarian pedicle was suspected because of dorsal thickening of the cyst wall and fluid formation indicated by high intensity on T1- and T2-weighted MRI. Surgery was complicated due to involvement in the torqued pedicle of the left fallopian tubal fimbria, which was released. The cyst was drained and partially resected, and then the pedicle torsion was released. By four years and five months of follow-up, there had been no ovarian cyst findings on ultrasound or MRI. Larger cysts are more likely to involve torsion of the ovarian pedicle and potential bleeding. Considering that, in this case, the cyst was large and proved to be ischemic–hemorrhagic, the decision to operate seemed justified.

Sigmoid volvulus requires urgent treatment, and it is particularly rare among pregnant women without a history of laparotomy. A delay in diagnosis may lead to serious consequences for the mother and fetus, and a rapid diagnosis and treatment in this setting is essential. The patient was a 19-year-old primiparous woman. She complained of repeated exacerbations and remissions of abrupt lower abdominal pain for the past 2 days and was transported to our hospital at 33 weeks of gestation. Ultrasonography revealed no placental thickening, and maternal bowel dilation was difficult to identify. Plain abdominal X-ray showed a dilated colon on the left side of the abdomen. Contrast-enhanced CT scan of the abdomen revealed a volvulus on the dorsal side of the uterus. The proximal end of the transverse to sigmoid colon was markedly dilated, and the distal end was collapsed. The elevated lactate level on blood gas analysis suggested intestinal ischemia. She was suspected of having a sigmoid volvulus at 33 weeks and 3 days of gestation. We decided to perform a cesarean section to secure the operative field for an intestinal resection following delivery. A male weighing 1840 g with Apgar scores 8/8 was delivered. The sigmoid colon was approximately 80 cm in length. A 360-degree clockwise rotation of was observed with a very distended but viable sigmoid loop. Following reduction of the volvulus, the sigmoid colon was fixed to the left side of the peritoneum. The mother had an uneventful postoperative course, and the infant was discharged without any sequelae. This case demonstrates two important lessons. First, sigmoid volvulus can occur in pregnant women even if they never had a laparotomy. Second, abdominal contrast-enhanced CT is useful for rapid diagnostic and treatment decisions relative to this pathology.

Pseudoaneurysms generally develop when an arterial puncture site is inadequately sealed. We encountered a case of vaginal pseudoaneurysm that developed 3 years after cesarean section in a 35-year-old gravida 7 para 4 woman who was prescribed with anticoagulant and antiplatelet drugs after surgeries for ventricular septal defect and aortic valve replacement. Pelvic computed tomography scan revealed a large mass, which showed a dappled contrast filling on the arterial phase, located in the posterior vaginal wall. The vaginal pseudoaneurysm was completely occluded by embolization of the left vaginal artery. Anticoagulation and antiplatelet therapies can be potential causes of spontaneous pseudoaneurysm rupture. Extrauterine pseudoaneurysm has a long period of time between cesarean section and pseudoaneurysm discovery. Considering that pseudoaneurysm shows different clinical features for each patient, we should always consider pseudoaneurysm when we assess a patient with postpartum hemorrhage.

Cefoselis is a widely used ß-lactam antibiotic, but occasionally induces seizures and convulsion in elder and renal failure patients. However, ß-lactams are known not to pass through the blood-brain barrier (BBB). In this study, we examined the BBB penetration of cefoselis in normal and renal failure rats by means of brain microdialysis. Cefoselis was dose-dependently appeared in brain extracellular fluid in proportion to its blood level. The elimination constant from brain extracellular fluid (apparent) was slightly lower than that from blood. These results indicated that cefoselis might penetrate the BBB or be discharged by a certain transport system. In contrast to the result of cefoselis, cefazolin, a leading drug of cephalosporins, could not be detected in the brain extracellular fluid after an intravenous injection. In renal dysfunction rats, the elimination half-lives of cefoselis from both blood and brain were extensively prolonged. This would be one of responsible factors inducing seizures seen in patients. However, the additional factor, such as decrease in brain function related to aging, would be involved in seizures in patient received cefoselis, because an extremely high dose was required to induce seizures even in renal failure rats. A local administration of cefoselis into the hippocampus through the microdialysis probe caused a striking elevation of extracellular glutamate, with a minimum increase in ?-aminobutyric acid (GABA). However, a systematic cefoselis administration via the tail vein did not elevate extracellular glutamate and GABA concentrations in the hippocampus of renal failure rats that exhibited marked seizures. These results suggested that not the stimulation of glutamate release, but the blockade of GABA receptors might be responsible for the seizure induced by cefoselis. [ABSTRACT FROM AUTHOR]

Summary. The role of glutamate in the N-methyl-4-phenyl-dihydropyridinium (MPP+) toxicity has been argued in the past decade. However, the effects of glutamate efflux and NMDA antagonist on MPP+-induced dopamine overflow have not been documented. To clarify this, we perfused MPP+ through a microdialysis probe in the striatum of freely moving mature C57BL/6 mice. The 60-min perfusion of 10 and 100 μM MPP+ strikingly increased dopamine levels to 28- and 93-fold of the basal values, respectively. In contrast, an administration of MPP+ did not induce marked glutamate release: the MPP+-perfusion slightly increased the glutamate level at 100 μM, but not at 10 μM. The addition of 100 μM (+)-MK-801 or 200 μM (±)-AP-7 to the perfusate did not attenuate MPP+-induced dopamine overflow. The extent of dopamine release only depended on the amount of MPP+ accumulation into the cells. These results indicated that, at least in the striatum, neither glutamate release nor the NMDA antagonist, including (+)-MK-801, could regulate MPP+-evoked dopamine overflow. [ABSTRACT FROM AUTHOR]

We report a case of Cornelia de Lange syndrome (CdLS) where prenatal diagnosis was not made even with major anomaly. A 33-year-old Japanese woman was referred to our institution at 23 weeks of gestation because of fetal forearm defect. Ultrasound examination revealed short forearms and short humeri and femurs (–2.1 SD). The fetal estimated body weight was 450 g (–1.3 SD). Fetal MRI at 26 weeks of gestation revealed short forearms and hypoplasty of hand fingers. Fetal growth restriction became evident thereafter, leading to intrauterine fetal death occurring at 29 weeks of gestation. A stillbirth baby was of 798 g in body weight and 33.0 cm in length. External examination showed a low hairline, synophrys, low-set ear, hypertrichosis, and smooth long philtrum with thin lips. The neck appeared short and broad. Finally, CdLS was diagnosed. The prenatal diagnosis might be possible as the arm findings were totally characteristic in a small fetus, regardless of whether an overhanging upper lip was identified. Because CdLS is a rare condition, it is important to consider its possibility as a part of differential diagnosis.

Massive subchorionic thrombohematoma (MST), termed Breus’ mole, is a rare condition in which a large maternal blood clot separates the chorionic plate from the villous chorion. Common complications of MST include fetal growth restriction, preeclampsia, and intrauterine fetal death. Here, we present a case of a 17-year-old Japanese woman referred to our institution at 21 weeks of gestation. Ultrasound examination revealed a large placental mass with mixed high and low echogenicity measuring approximately 7.6 cm in thickness. Doppler examination showed absence of end-diastolic velocity of the umbilical artery. At 22 weeks of gestation, the patient had a stillbirth weighing 138g. The placenta weighed 502 g and was 8 cm thick, and the total blood loss was 270 g. Macroscopic examination revealed that a subchorionic blood clot measuring 12 cm × 5 cm covered a large portion of the placenta with well-defined margins on the fetal surface. Microscopic examination revealed an intervillous hematoma and fibrinous deposits directly beneath the chorionic plate with adjacent compressive effects. Based on these findings, MST was diagnosed. Because MST is rare, it must be considered in the differential diagnosis of parental conditions. Magnetic resonance imaging can be optimal for diagnosing MST when ultrasound diagnosis is difficult.

Two types of congenital bronchial atresia (proximal and peripheral) have been classified. We report two cases of peripheral bronchial atresia diagnosed by prenatal ultrasonography (US) and magnetic resonance imaging (MRI). Evaluating an enlarged lung mass that is homogeneously hyperechoic on US and hyperintense on T2-weighted MRI can help in determining whether bronchial atresia is present. Proximal type is suggested when a dilated main bronchus is observed as a tubule structure of an involved lung hilum. In our cases, T2-weighted MRI revealed homogeneously hyperintense lung lesion with decreased signal intensity of adjacent lobe, flattening diaphragm, and mediastinal shift. Dilatation of the main bronchus was not observed and the opposite lung was normal in appearance. These findings were explained by secondary compression due to enlargement of the involved lung. The preservation of vascular structure and the retained normal shape, though enlarged, in the affected lobe were observed, which demonstrated undisrupted pulmonary architecture of the lobe. Thus, congenital cystic adenomatoid malformation was excluded because pulmonary architecture was relatively preserved. Finally, presumed diagnoses of the peripheral bronchial atresia were made and confirmed by postnatal chest computed tomography.

A 37-year-old (G4P3) woman was referred to our hospital at 32 weeks of gestation for the evaluation of a fetus with an intrathoracic cystic lesion. Ultrasonography and magnetic resonance imaging revealed that a fetal cystic lesion without a mucosal layer was located in the posterior mediastinum. These findings were consistent with a bronchogenic cyst. At 38 3/7 weeks of gestation, an elective cesarean section was performed because of her previous cesarean section. A female neonate without any external anomalies, weighing 2,442 g, with Apgar scores of 8 and 9, and requiring no resuscitation was born. Four weeks after delivery, the neonate was admitted because of respiratory distress due to mass effect. At right lateral thoracotomy, a 105 × 65 mm of solitary smooth-walled cyst containing serosanguineous fluid was found in the posterior mediastinum, which was excised completely. Histologic examination revealed the diagnosis of the mediastinal gastric duplication cyst. The neonate made an uneventful recovery. Accurate diagnosis is not necessary, but detection and continuous observation are logical. Although gastric duplication, particularly intrathoracic, is a rare pathology, it should be considered in the differential diagnosis of any intrathoracic cyst.

A 40-year-old pregnant woman presented with a fetal abdominal cyst and oligohydramnios. Color Doppler scan revealed a single blood vessel from the fetal aorta into a single umbilical artery. Severe oligohydramnios limited ultrasonographic evaluation of the fetal lower limbs, kidneys, or bladder. The pregnancy was terminated; the fetus showed fused lower limbs, bulging abdomen, and absent external genitalia and was diagnosed with type III sirenomelia. On autopsy, no normal bladder was observed, but duodenal atresia, anorectal atresia, and right renal agenesis were found. An intra-abdominal cyst, diagnosed histologically as a saccular cloaca, occupied the abdominal cavity. Ultrasonographic diagnosis of fetal sirenomelia is difficult due to poor depiction of the lower limbs. A vitelline artery leading to a single umbilical artery and a fetal abdominal cyst occupying most of the abdominal cavity are considered fetal sirenomelia associated with large defects of the gastrointestinal and genitourinary tracts.

Abstract Aim We report a case of congenital pulmonary airway malformation (CPAM) with hydrops in which the fetus underwent thoracoamniotic shunting. Case Report A 40-year-old (G1P1) woman was diagnosed with a macrocystic CPAM. Thoracoamniotic shunting was performed at 19 weeks of gestation but not well drained and was successfully performed again at 23 weeks. However, the CPAM volume ratio, abdominal circumference, and amniotic fluid index started increasing from 28 weeks and hydrops worsened. The insufficient shunting and the fetal cardiac failure had to be considered. At 32 weeks, a male infant with general edema and massive ascites was born weighing 3,362 g (+4.79 SD) with Apgar scores of 2 and 4. The infant was intubated and high-frequency oscillation and nitric oxide therapies were instituted. The resection of CPAM was performed on day 2. Nasal continuous positive airway pressure was instituted on day 16. The infant was discharged and prescribed with home oxygen therapy (HOT) on day 65. The infant was able to leave the HOT at 30 months and is currently 34 months of age in good condition. Conclusion Fetal thoracoamniotic shunting may be life-saving in CPAM complicated by hydrops and that this treatment might be sufficient to cure the child.

Background. We experienced a rare case of a pregnant woman with Klippel-Trénaunay syndrome complicated with diffuse venous malformation of the uterus. This is the first report on the usefulness of dynamic contrast-enhanced-MRI for the diagnosis of diffuse venous malformation of the uterus. Case Presentation. A 23-year-old woman presented with convulsions and talipes equinus position of both lower limbs at 11 weeks of gestation. At 27 weeks, ultrasonography demonstrated tubular echolucent spaces throughout the myometrium. Dynamic MRI at 37 weeks revealed that the myometrial lesion was enhanced slowly and showed homogeneous enhancement even on a 10 min delayed image. Taken together with unilateral foot hypertrophy, varices, and port-wine stain, the patient was diagnosed as having Klippel-Trénaunay syndrome complicated with diffuse venous malformation of the pregnant uterus. The patient underwent elective cesarean section because of severe dystonia. The lower uterine segment was thickened and heavy venous blood flow was observed at the incision. Histological diagnosis of the myometrial biopsy specimen was venous malformation. Conclusions. Both diffuse venous malformation and Klippel-Trénaunay syndrome during pregnancy can involve considerable complications, in particular, massive bleeding during labor. Women who suffer from this syndrome should be advised about the risk of complications of pregnancy.

It was demonstrated that second- and third-trimester therapeutic termination of pregnancy (TOP) is feasible in cases with placenta previa. We report a 34-year-old woman with complex fetal malformations associated with placenta previa. An ultrasound examination at 21 weeks of gestation revealed fetal growth restriction (FGR) and complex fetal malformations associated with a placenta previa. After extensive information, the parents opted for careful observation. Thereafter, FGR gradually progressed and we observed arrest of end-diastolic velocity of the umbilical artery. Finally, intrauterine fetal death (IUFD) was confirmed at 33 weeks of gestation. Two days after IUFD, the patient experienced labor pain. The placenta and dead fetus weighing 961 g were vaginally delivered, and total bleeding was 270 mL. Although further studies to confirm the dynamic change of the uteroplacental blood flow are necessary to avoid the risk of maternal hemorrhage, vaginal TOP with placenta previa after feticide or IUFD would be feasible.

Dose-dependent local control is known for stereotactic radiotherapy (SRT) of brain metastases (BM). We summarize here our experience with patients of BM treated by the high-dose SRT of 38 Gy or 35 Gy in 3 fractions. This study is a retrospective review of all cases of BM treated by Linac-based SRT with dynamic conformal arcs technique at our institution from July 2014 to April 2023. The margin from gross tumor volume (GTV) to planning target volume (PTV) was 1-2 mm. The total dose of 38 Gy or 35 Gy was administered in 3 fractions, and the prescribed dose covered 95% of the PTV. We analyzed the impact of patient and treatment characteristics on overall survival (OS), local control (LC) and radionecrosis (RN) after SRT by Kaplan-Meier method and cox regression models. Statistical significance was set at P < 0.05. There were 236 patients. The total number of lesions was 331, and the median follow-up period was 9.5 months (range = 0.1-106.5). Patients had a median age of 69 years (range = 35-92), 149 (63%) were male. The most common presenting illness was non-small-cell lung (NSCLC) cancer (120 patients, 51%), followed by breast cancer (33 patients, 14%), SCLC (25 patients, 11%), colorectal cancer (12 patients, 5%), esophageal cancer (8 patients, 3%), renal cell carcinoma (8 patients, 3%), gastric cancer (6 patients, 3%) and others (24 patients, 10%). The median GTV was 2.9 cc (IQR = 1.5-5.3). The dose of 38 Gy in 3 fractions was prescribed for 251 lesions and 35 Gy in 3 fractions for 80 lesions. The median OS was 10.7 months, and the 6-months, 1-year, and 2-year OS rates were 65%, 47%, and 26%, respectively. In univariate analysis, the prognosis was significantly better in the group with good performance status, control of extracranial lesions, and chemotherapy after SRT, respectively. The LC rates at 6 months, 1 and 2 years were 97%, 93% and 91%, respectively, for all lesions. The total of 13 lesions (4%) had local recurrence, of which 7 lesions underwent surgery. There was no significant difference in LC rate between the 38 Gy and 35 Gy groups (P = 0.89). Symptomatic RN was observed in a total of 26 lesions (8%), of which 14 lesions underwent surgery. The cumulative incidence of symptomatic RN at 1 year, 2 years and 5 years was 4.7༅, 17.2༅ and 42.5༅, respectively. The cumulative incidence of RN requiring surgery at 1 year, 2 years and 5 years was 1.1%, 6.1%, and 32.8%, respectively. In univariate analysis, GTV ≥3 cc (vs. <3 cc) was associated with higher rates of RN, not with LC or OS. In 14 lesions with surgery for RN, the median surrounding brain volumes circumscribed with 18 Gy and 24 Gy were 28.8 cc (range = 13.9-57.4) and 19.2 cc (range = 8.3-36.3), respectively. While high-dose, three-fraction SRT for BM can be expected to have a high LC rate, it is necessary to be careful about RN in long-term survival cases with high exposure doses to the normal brain. [ABSTRACT FROM AUTHOR]