Michinori Mayama

Hemophagocytic Lymphohistiocytosis Associated With a Parvovirus B19 Infection During Pregnancy

BACKGROUND: Hemophagocytic lymphohistiocytosis is potentially fatal. Prompt diagnosis and initiation of treatment are critical for ensuring the best possible prognosis. CASE: We present a case of parvovirus B19 infection related to hemophagocytic lymphohistiocytosis during pregnancy. The patient experienced fever and pancytopenia. A bone marrow biopsy demonstrated hemophagocytosis and a giant proerythroblasts, which is characteristic of a parvovirus B19 infection. Viral serology for parvovirus B19 was positive. Prompt treatment was started because of the high level of certainty of viral-associated hemophagocytic lymphohistiocytosis, and the patient was successfully treated with prednisolone administration. She delivered a healthy newborn without any complications. CONCLUSION: Hemophagocytic lymphohistiocytosis should be considered when encountering unexplained cytopenia and fever. Prednisolone was an effective treatment.

Factors influencing brain natriuretic peptide levels in healthy pregnant women

BACKGROUND The normal range of plasma brain natriuretic peptide (BNP) in pregnant women is still unclear. Moreover, pregnant women experience dynamic body weight changes and suffer from anemia, but effects on maternal BNP have not been investigated. This study aimed to reveal the normal plasma BNP range and examine the effects of physiological changes on BNP among pregnant women. METHODS AND RESULTS Plasma BNP, hemoglobin, plasma creatinine and BMI were measured in 58 non-pregnant control women and in 773 normal pregnant women at late pregnancy, early postpartum and 1-month postpartum. Mean plasma BNP (in pg/mL) was 11.8 (95% confidence interval: 0-27.5) in non-pregnant women, 17.9 (0-44.7, p<0.001) at late pregnancy, 42.5 (0-112.6, p<0.001) early postpartum and 16.1 (0-43.9, p=0.001) 1-month postpartum. Multiple regression analysis revealed that pre-delivery BNP levels were negatively correlated with BMI (p<0.001) and hemoglobin (p=0.002) and positively correlated with creatinine (p<0.001). Post-delivery BNP was positively associated with body weight change during pregnancy (p=0.001) and post-delivery creatinine (p=0.010) but negatively associated with body weight loss at delivery (p<0.001) and post-delivery hemoglobin (p=0.004). CONCLUSION Even normal pregnancy affects plasma BNP, particularly in the early postpartum period, indicative of cardiac stress. Plasma BNP levels are affected by BMI, body weight changes, creatinine and hemoglobin levels; therefore, these factors should be considered when analysing cardiac function and the physiological implications of BNP levels in pregnant women.

The efficacy of recombinant human soluble thrombomodulin for obstetric disseminated intravascular coagulation: a retrospective study

IntroductionRecombinant human soluble thrombomodulin (rhTM) is a novel anti-coagulant agent that regulates the imbalanced coagulation system by reducing the excessive activation of thrombin. rhTM potentially reduces the morbidity and mortality in patients with sepsis-induced disseminated intravascular coagulation (DIC). However, the efficacy of rhTM in obstetric DIC has not yet been established. We performed this study to examine whether the administration of rhTM was a potentially effective treatment for DIC induced by one or more underlying obstetric disorders.MethodsThis is a single-center, retrospective cohort study conducted between January 2007 and February 2015 using the records of the Department of Obstetrics at the Perinatal Medical Center of TOYOTA Memorial Hospital, Aichi, Japan. The eligibility criteria were known or suspected obstetric DIC documented on the basis of clinical and laboratory data and association with one or more major underlying obstetric disorders. Baseline imbalance between patients with and without treatment of rhTM was adjusted using an inverse probability of treatment weighting using propensity scores composed of the following independent variables: severe postpartum hemorrhage, placental abruption, and preeclampsia/eclampsia, including hemolysis, elevated liver enzymes, and low platelet syndrome, initial platelet counts, D-dimer levels, fibrinogen levels, and prothrombin time–international normalized ratio (PT–INR). We evaluated laboratory changes and clinical outcomes in the early phase of obstetric DIC.ResultsIn total, 66 of 4627 patients admitted to our department during the study period fulfilled the required criteria; of these, 37 and 29 patients were included in the rhTM and control group, respectively. After adjustment, treatment with rhTM was associated with significant improvements in platelet counts, D-dimer levels, fibrinogen levels, and PT–INR compared with the control group. The platelet concentrate transfusion volume was significantly lower in the rhTM treatment group (3.02 vs 6.03 units, P = 0.016). None of the adjusted group differences were statistically significant for all types of organ damage and failure.ConclusionrhTM administration was associated with clinical and laboratory improvement in patients with DIC caused by underlying obstetric conditions. Further clinical research is needed to clarify the optimal application of rhTM in each of the causative obstetric disorders.

Fulminant liver failure resulting from massive hepatic infarction associated with hemolysis, elevated liver enzymes, and low platelets syndrome

Hepatic infarction is an extremely rare and fatal complication associated with hemolysis, elevated liver enzymes, and low platelets (HELLP) syndrome. It can develop into fulminant liver failure, which increases both maternal and neonatal mortality rates. A 34‐year‐old woman with no remarkable past medical history developed eclampsia after delivery at 40 weeks of gestation. Imaging indicated massive hepatic infarction and rupture followed by cardiac arrest and fulminant liver failure. Despite liver replacement therapy with plasma exchange and continuous hemodiafiltration, the patient gradually deteriorated with persistent bacterial infection until death at 98 days after delivery. The management of fulminant liver failure complicated with HELLP syndrome should be multidisciplinary. Liver transplantation, the only radical treatment for fulminant liver failure, is worth attempting, if applicable.

A Case Report and Literature Review of Spontaneous Perforation of Pyometra.

BACKGROUND Pyometra is defined as an accumulation of purulent material in the uterine cavity. Spontaneous perforation is a very rare complication of pyometra. The clinical findings of perforated pyometra are similar to perforation of the gastrointestinal tract and other causes of acute abdomen. CASE REPORT We report a rare and difficult case of peritonitis in an elderly female that was caused by a spontaneous perforation of pyometra. A 90-year-old postmenopausal woman was referred to our hospital with complaints of vomiting, fever, and abdominal pain. Computed tomography revealed a large amount of ascites, cystic mass in the uterus, and intraperitoneal and intrauterine air. Transvaginal ultrasound demonstrated a thin area around the fundus. An emergency laparotomy was performed for the suspected gastrointestinal perforation or perforation of pyometra. At laparotomy, copious purulent fluid was present in the peritoneal cavity; however, no perforation of the gastrointestinal tract was observed. We identified a perforation site over the uterine fundus and purulent material exuding from the cavity. Subsequently, hysterectomy and bilateral salpingo-oophorectomy were performed. The patient was discharged on postoperative day 13 with no complications. Histopathologic studies revealed endometritis and myometritis with no evidence of malignancy. WHY SHOULD AN EMERGENCY PHYSICIAN BE AWARE OF THIS?: With diffuse peritonitis, ruptured pyometra should be considered, even in elderly female patients. This case illustrates the importance of clinical knowledge of acute gynecologic diseases. Here we also review the perforation of pyometra with no evidence of malignancy.

Pseudo Gitelman Syndrome Associated With Pregnancy.

BACKGROUND: Gitelman syndrome is a rare inherited renal tubulopathy associated with metabolic alkalosis and electrolyte disorders. Pseudo Gitelman syndrome presents with the same clinical characteristics as Gitelman syndrome, yet without genetic mutations in SLC12A3. CASE: A 32-year-old woman with no remarkable medical and family history developed hypokalemia at 32 weeks of gestation. Laboratory findings were consistent with Gitelman syndrome and potassium supplementation was initiated. The patient delivered a healthy neonate at 40 weeks of gestation and the electrolyte disorders drastically improved. After delivery, genomic analysis revealed no evidence of mutations in SLC12A3, and pseudo Gitelman syndrome was finally diagnosed. CONCLUSION: Pseudo Gitelman syndrome, presenting with Gitelman syndrome-like renal tubulopathy without mutations in SLC12A3, can cause a temporary electrolyte imbalance based on the physiologic changes of pregnancy. Although pregnant women with isolated hypokalemia need not be evaluated for Gitelman or pseudo Gitelman syndrome, if it is accompanied by metabolic alkalosis, hypocalciuria, hypomagnesia, and activation of the renin–angiotensin–aldosterone system without hypertension, this evaluation should be considered.

OC07.05: Maternal cardiac function in patients with mild and severe pre‐eclampsia compared with normal pregnant women

demographics and pregnancy outcome were obtained from obstetric records. Descriptive statistics was performed. Mean gestational age at delivery and fetal birthweight with z-scores were compared between groups. Prevalence of small for gestational age fetuses (SGA, <5th percentile) and preterm birth (<37 weeks) was calculated and compared. Mann-Whitney U, independent samples t-tests and chi-square test were used. Results: A total of 35,194 women were included at a mean gestational age of 21.5±1.1 weeks. UtAD impedance was normal in 30,915/35,194 (87.8%) women and elevated in 4,279/35,194 (12.2%). In the group with normal UtAD 30,570/30,915 (98.9%) had a live birth. Intrauterine demise or neonatal death was reported in 345/30915 (1.1%). In the group with raised UtAD, 4,159/4,279 (97.2%) women had a live birth, whereas intrauterine demise or neonatal death was reported in 120/4,279 (2.8%). Mean gestational age at delivery was 39.1±2.4 and 38.1±3.3 weeks in the normal and raised UtAD group, respectively (p=0.001). Mean birthweight z scores were 0.06 and -0.33 in the normal and abnormal UtAD groups, respectively (p=0.001). The prevalence of SGA was 1,221/30,915 (3.9%) in the normal and 600/4,279 (14%) in the raised UtAD group (p=0.001). The prevalence of preterm birth was 2,534/3,0915 (8.2%) and 756/4,279 (17.7%) in the two groups (p=0.001). Conclusions: Raised UtAD at the anomaly scan is associated with an increased rate of intrauterine demise or neonatal death. Fetal birthweight is lower in women with raised UtAD.

EP29.08: The efficacy of Superb Micro-vascular Imaging on diagnosing endometrial cancer

Objectives: Metabolic syndrome (MetS) has been recognised as a risk factor for malignancies. The aim of this study was to evaluate the association of MetS and risk of endometrial carcinoma (EC), by measuring endometrial thickness (ET). Methods: The Ragama Health Study (RHS) recruited 35-64-year-old female cohort by age-stratified random sampling in 2007 and re-evaluated them in 2014, using a structured interview, anthropometric measurements and biochemical tests. Liver ultrasound to detect fatty liver was performed in 2007. Pelvic ultrasound to detect ET was performed in 2014 among consenting participants. MetS was diagnosed on established International Diabetes Federation (IDF 2012) criteria. Increased ET was defined as >5mm. Simple logistic regression was used to screen variables and multiple logistic regression was used to obtain adjusted effects of risk factors for increased ET. Results: 813/1636(49.7%) of the original female cohort attended follow-up; ET was measured in 567(69.7%). Median (IQR) age of females was 61 (56-66) years. 323 fulfilled criteria for MetS (prevalence 57.1%) in 2007. 57(10.1%) had increased ET in 2014. Increasing plasma triglycerides [OR=1.004 per mg/dl, 95%CI:1.001-1.007, p<0.05] and being hypertensive [OR=2.16, 95%CI:1.11–4.08, p<0.05] were associated with increased ET, while advancing age [OR=0.93 per year, 95%CI:0.89–0.98, p<0.01] and being diabetic [OR= 0.34, 95%CI:0.10–0.89, p<0.05] were protective. Conclusions: Hypertension and increased plasma triglyceride levels, in the pre-menopausal period, were risk factors for future asymptomatic increased ET.

Sarcoid-like reaction mimicking vaginal cancer recurrence

A sarcoid‐like reaction is a development of non‐caseating granuloma in patients with underlying malignancy and represents a false positive finding on positron emission tomography/computed tomography (PET/CT). A sarcoid‐like reaction is a benign condition; therefore, differentiating a sarcoid‐like reaction from cancer recurrence is necessary. Only uterine and ovarian cancer related cases have been reported in the gynecological field and to the best of our knowledge, this is the first case of a sarcoid‐like reaction in vaginal cancer. A 59‐year‐old vaginal cancer patient received concurrent chemoradiotherapy and achieved complete remission. Recurrence of vaginal cancer was suspected because of the elevation of serum squamous cell carcinoma antigen level. PET/CT revealed abnormal uptake at the bilateral mediastinal and hilar lymph nodes. A non‐caseating granuloma was detected from the biopsy of the swollen lymph nodes. No evidence of cancer recurrence was observed. A sarcoid‐like reaction should be considered when evaluating PET/CT in cancer patients to prevent unnecessary treatments.