Remitting Seronegative Symmetrical Synovitis with Pitting Edema Syndrome as a Manifestation of Recurrent Ovarian Cancer.

We herein report a case of ovarian cancer recurrence detected every time with symptoms of remitting seronegative symmetrical synovitis with pitting edema (RS3PE) syndrome. A 46-year-old woman who had a history of ovarian cancer 9 months earlier developed joint pain along with pitting edema in both hands and was diagnosed with RS3PE syndrome. Two and four years after initial surgery for ovarian cancer, symptoms of RS3PE syndrome appeared, and a recurrent site was detected. With resection of the relapsed sites and increased maintenance dose of methylprednisolone, these symptoms improved within a month.

Manual removal of placenta in women having unpredictable adherent placenta.

AIM: Our aim is to provide expected outcomes for undergoing manual removal of placenta (MROP) following vaginal delivery in women having an unpredictable adherent placenta (AP). METHODS: The data were obtained from four hospitals in Miyazaki Prefecture, Japan. We used propensity score-matched (1:1) analysis to match women who underwent MROP with women who did not undergo MROP (control). Total blood loss and hemorrhagic rate used as a ratio of women who reached a certain amount of blood loss were compared. Subgroup analysis was undertaken and was dependent on the presence of AP. We found the cut-off value of blood loss for detecting AP. RESULTS: Thirty-seven MROP cases were identified. Total blood loss and hemorrhagic rate differed significantly between MROP cases and controls; 95% of controls had blood loss of 1000 mL or less, whereas for the MROP cases, it was 14%. Fourteen MROP cases were diagnosed with AP. The hemorrhagic rate differed significantly between MROP cases with and without AP (n = 19); 79% of MROP cases without AP had blood loss of 2000 mL or less, whereas for the MROP cases with AP, it was 7%. There were seven incidents of hysterectomy and two of arterial embolization in MROP cases with AP. Through receiver operating characteristic curve analysis, 2035 mL of blood loss was determined to be the optimal cut-off value for detecting AP. CONCLUSION: The incidence of unpredictable AP in MROP cases was as high as 38%. The morbidity of MROP cases with unpredictable AP was severe. MROP should be prohibited in the absence of appropriate hemostatic preparations.

Prenatal diagnosis of premature chromatid separation/mosaic variegated aneuploidy (PCS/MVA) syndrome.

Premature chromatid separation/mosaic variegated aneuploidy (PCS/MVA) syndrome is a rare genetic disorder. In this case report, we describe the prenatal diagnosis of PCS/MVA syndrome in a 24-year-old, gravida 1, para 1, woman who was referred to us in her second trimester due to fetal growth restriction and extreme microcephaly (-5.0 standard deviations). Amniocentesis and chromosomal analysis confirmed PCS in 80% of cultured fetal cells. PCS findings were positive in 9% of paternal cells and 11% of maternal cells, indicative that both were PCS carriers. Genetic analysis confirmed that the fetus carried a combined heterozygote of maternal G > A point mutation of the promoter area of the BUB1B gene and a paternal Alu sequence insertion between intron 8 and exon 9 of the BUB1B gene. As PCS/MVA syndrome is associated with the development of various malignancies in early life, prenatal diagnosis is important for effective planning of post-natal care.

Candida Chorioamnionitis Leads to Preterm Birth and Adverse Fetal-Neonatal Outcome.

Candida chorioamnionitis is rare but can lead to neonatal infection, high mortality, and neurodevelopmental impairment. We aimed to investigate maternal clinical features and perinatal outcomes and discuss future management strategies. We reviewed the medical records of women with Candida chorioamnionitis at our hospital over a 10-year period (n = 9) and previous published case reports and case series. The most prevalent Candida species was C. albicans (71.3% of the all cases). The most prevalent predisposing condition was preterm premature rupture of membranes (31/123, 25.2%), followed by pregnancy with a retained intrauterine contraceptive device (26/123, 21.1%) and pregnancy after in vitro fertilization (25/123, 20.3%). Preterm labor was the most common symptom (52/123, 42.3%), and only 13% of cases involved fever. Of the infants, 27% of the singletons and 23.8% of the twins were born before 22 gestational weeks, while 60% of the singletons and 76.2% of the twins were born at 22-36 weeks. The median birth weight of the babies born after 22 weeks was 1230 g. The mortality rates of the singletons and twins born after 22 weeks of gestation in the year 2000 or later were 28.6% and 52.4%, respectively. Antenatal treatment for Candida chorioamnionitis has not been established.

PCS/MVA syndrome caused by an Alu insertion in the BUB1B gene.

We report a case of premature chromatid separation/mosaic variegated aneuploidy syndrome identified by microcephaly on fetal ultrasound and confirmed by cytogenetic analysis of amniotic fluid. Initial mutational analysis of the entire coding region of the BUB1B gene failed to identify any causative mutations. However, further analysis revealed a known compound heterozygous mutation in the upstream region of this gene and a novel Alu insertion mutation in the intron.

Maternal Morbidity in Women with Placenta Previa Managed with Prediction of Morbidly Adherent Placenta by Ultrasonography.

Objective. To determine maternal morbidity in women with placenta previa managed with prediction of morbidly adherent placenta (MAP) by ultrasonography. Methods. A retrospective cohort study was undertaken comprising forty-one women who had placenta previa with or without risk factors for MAP. Women who had all three findings (bladder line interruption, placental lacunae, and absence of the retroplacental clear zone) were regarded as high suspicion for MAP and underwent cesarean section followed by hysterectomy. We attempted placental removal for women having two findings or less. Results. Among 28 women with risk, nine with high suspicion underwent hysterectomy and were diagnosed with MAP. Three of 19 women with two findings or less eventually underwent hysterectomy and were diagnosed with MAP. The sensitivity and positive predictive value for the detection of MAP were 64% and 100%. The pathological severity of MAP was significantly correlated with the cumulative number of findings. There were no cases of MAP among 13 women without risk. There was no difference of blood loss between women with high suspicion and those without risk (2186 ± 1438 ml versus 1656 ± 848 ml, resp.; p = 0.34). Conclusion. Management with prediction of MAP by ultrasonography is useful for obtaining permissible morbidity.

Antithrombin improves the maternal and neonatal outcomes but not the angiogenic factors in extremely growth-restricted fetuses at <28 weeks of gestation.

AIMS: Severe preterm fetal growth restriction (FGR) remote from term is problematic. We aimed to investigate the effect of maternally-administered antithrombin on maternal and neonatal outcomes. A prospective, one-arm, pilot study was performed in 14 women with severe FGR (≤5th centile) at <28 weeks of gestation, without hypertensive disorders. Maternal plasma concentrations of soluble Feline McDonough Sarcoma (FMS)-like trypsin kinase-1 (sFlt-1) and placental growth factor (PlGF) were measured and categorized into three groups: group 1; low sFlt-1 and high PlGF, group 2; moderate sFlt-1 and low PlGF, and group 3; high sFlt-1 and low PlGF. Antithrombin was administered for 3 days. The incidence of perinatal mortality, infant morbidity, and the period of pregnancy prolongation were compared. RESULTS: In group 1 (n=4), their pregnancies were extended for longer periods and the maternal and infant outcomes were good. The prolongation periods were shorter in groups 2 (n=3) and 3 (n=7), which resulted in poor maternal [severe preeclampsia or hemolysis, elevated liver enzymes, and low platelet count (HELLP) syndrome] and infant outcomes. CONCLUSIONS: The evaluation of the maternal sFlt-1 and PlGF at 21-27 weeks of gestation is useful in the managements of severe FGR. Antithrombin treatment could prolong the pregnancies with low sFlt-1 and high PlGF without negatively affecting maternal or fetal health.