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Hemophagocytic syndrome (HPS) is a syndrome presenting with signs of persistent remittent fever, hepatosplenomegaly, pancytopenia, hepatic dysfunction, and disseminated intravascular coagulation (DIC) due to hypercytokinemia caused by activated T lymphocytes and macrophages. The mortality in adults is high and a small number of complicated cases during pregnancy have been reported. We report one HPS case that developed a remittent fever, leukocytopenia, and thrombocytopenia in the 2st week of pregnancy, and abnormal blood coagulation, hepatic dysfunction, and hypercytokinemia were found. Antibiotics and immunoglobulin were given but failed to improve clinical and laboratory findings. At the 24th week, the patient was diagnosed with DIC, and antithrombin (AT) concentrate was given. With the increase in plasma levels of AT, improvements were seen in both clinical signs and laboratory findings. Bone marrow biopsies were carried out, and a diagnosis of HPS was made. Preeclampsia developed in the 27th week and it became severe. Cesarean section was performed in the 29th week because of severe preeclampsia, intrauterine growth retardation (IUGR), and fetal distress. The courses of mother and newborn were uneventful. We discuss the mechanism of AT in the treatment of this syndrome and the association between this syndrome and severe preeclampsia. In conclusion, AT concentrate was very effective in suppressing cytokine production, and the possibility that severe preeclampsia developed because of hypercytokinemia, which may be one of the pathogeneses of severe preeclampsia and IUGR, was suggested.

ObjectiveThe liquid embolic agent n-butyl cyanoacrylate (NBCA) is a tissue adhesive used as an immediate and permanent embolic agent when mixed with oil-based contrast medium. In this study, the preservation of fertility with TAE using NBCA for massive haemorrhage during pregnancy or the peripartum period and the utility of this therapy were investigated.MethodsCases from January 2005 to October 2010 in which TAE was performed for massive haemorrhage in pregnant women, particularly during the peripartum period, were investigated.ResultsTAE was performed in 27 pregnant women. The embolic agent used was GS only in five cases, NBCA only in 19 cases, and additional embolization with NBCA when the effect with GS was insufficient in three cases, one each of abruptio placentae, cervical pregnancy, and uterine atony.A comparison of mean blood loss when each embolic agent was used for haemostasis showed a significant difference between cases in which GS only was used and cases in which NBCA only was used. In a comparison of mean transfusion volume, a significant difference was seen between cases in which both GS and NBCA were used and cases in which NBCA only was used. In a postoperative follow-up survey, menses resumed in eight patients, including four patients who later became pregnant and three who delivered.ConclusionsTAE with NBCA, which has an embolic effect unrelated to clotting dysfunction for massive haemorrhage during the peripartum period, is a minimally invasive and very effective treatment method for patients with severe DIC.

Conjoined twinning is a unique complication of monochorionic pregnancy. This report describes the clinical findings in two cases of conjoined twins, and discusses their management. One case involved thoracopagus complicating a triplet pregnancy, and the other involved cephalothoracopagus, in which the outcome was intrauterine fetal death due to abruptio placentae after amniocentesis. Recent improvements in ultrasound imaging have facilitated the diagnosis of conjoined twins as early as the first trimester. Although many mothers opt to terminate pregnancy when conjoined twins are diagnosed, a few do not, as in the cases described. In such cases, pregnancy management, including accurate determination of the degree of organ fusion and psychological follow up, are important. On the basis of the two present cases, we present a systematic flow diagram for management of conjoined twin pregnancy from the time of diagnosis until delivery.

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