Cases reported "Hydrops Fetalis"

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1/83. Homozygous alpha-thalassaemia and hypospadias--common aetiology or incidental association? Long-term survival of Hb Bart's hydrops syndrome leads to new aspects for counselling of alpha-thalassaemic traits.

    Fetuses with homozygous alpha-thalassaemia develop Hb Bart's hydrops fetalis syndrome, which usually leads either to abortion or fetal/neonatal death. We report diagnosis, intrauterine transfusion therapy, neonatal intensive care management and long-term follow-up of a Vietnamese infant who survived Hb Bart's hydrops fetalis syndrome. During the first 2 years the child had normal development. In addition, the patient exhibited penoscrotal hypospadias. Despite a thorough endocrinological work-up the aetiology of genital ambiguity could not be elucidated. A review of the literature showed an association of homozygous alpha-thalassaemia and hypospadias in all surviving male children, suggesting a common aetiology for both entities. CONCLUSION: On the basis of our findings, we speculate that an unknown gene on chromosome 16 responsible for genital formation is altered in homozygous alpha-thalassaemia.
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ranking = 1
keywords = transfusion
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2/83. Severe nonimmune hydrops fetalis and congenital corneal opacification secondary to human parvovirus B19 infection. A case report.

    BACKGROUND: In parvovirus infections in animals, congenital anomalies are seen, but the teratogenic potential in humans seems fairly low. CASE: A fetus with hydrops, ascites and pleural effusion was seen at a prenatal ultrasound examination. Fetal cordocentesis was performed, and fetal blood was positive for parvovirus antibodies. Intravascular fetal blood transfusion was given at 21 and 23 weeks of gestation. At 39 weeks labor started spontaneously, and a 2,960-g, female infant was delivered. The newborn had bilateral opacification of the cornea. CONCLUSION: In this case a combination of fetal parvovirus B19 infection and congenital corneal opacification was seen. This case also demonstrates that blood transfusions in hydropic fetuses may reverse the hydrops and prevent intrauterine death.
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ranking = 2
keywords = transfusion
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3/83. Rapid development of hydrops fetalis in the donor twin following death of the recipient twin in twin-twin transfusion syndrome.

    Intrauterine death of one fetus in monochorionic twinning is associated with high rates of perinatal morbidity and mortality in the surviving fetus. Subsequent development of hydrops fetalis in the donor twin after fetal demise of the recipient twin has been described in only two case reports and pathophysiology remains unclear. We report on a monochorionic-diamniotic twin pregnancy complicated by severe twin-twin transfusion syndrome. Ultrasound examination at 20 weeks of gestation showed discrepant twins with oligohydramnios in the smaller twins' sac and polyhydramnios in that of the larger twin. Repeated amniocenteses permitted prolongation of the pregnancy. However, the recipient twin developed deteriorating hydrops fetalis and died at 28 weeks of gestation. After this event, subsequent development of hydrops fetalis in the surviving donor twin could be observed, as well as an increase of amniotic fluid. An elective cesarean section was performed at 29 weeks of gestation. Initial hypoxemia could be effectively treated by high frequency oscillatory ventilation, surfactant therapy and inotropic support. The infant was discharged in good condition at the age of 2 months. Although rare, antenatal demise of the recipient twin in a monochorionic pregnancy can be associated with the subsequent development of hydrops fetalis in the surviving donor twin. We speculate that this phenomenon is due to ischemia-reperfusion injury of the previously poorly perfused twin.
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ranking = 5
keywords = transfusion
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4/83. prenatal diagnosis and management of nonimmune hydrops fetalis.

    We examined the incidence, aetiological factors and outcome in 40 cases of nonimmune hydrops fetalis (NIH) and suggest a rational approach to management. The incidence of NIH was 1 in 830 deliveries during the last 10-year period. In spite of extensive antenatal and postnatal investigation no cause could be established in 14 (35%) cases. A probable aetiological factor was found in 65% of cases. These included viral infection (7), cardiovascular (6), twin-to-twin transfusion (3), chromosomal abnormality (3), other malformation syndromes (4), renal dysplasia (1), laryngeal atresia (1) and severe fetomaternal haemorrhage (1). Five of the 40 fetuses survived, 2 treated antenatally for tachyarrhythmia, 2 had spontaneous resolution and the fifth fetus had repeated intrauterine transfusions because of human parvovirus B19-induced anaemia. After diagnosis of nonimmune hydrops fetalis, early referral to a tertiary centre is to be encouraged for investigation and provision of intensive perinatal care. Investigation allows parents to be counselled appropriately that the mortality is no longer 100% and a steadily growing number may be amenable to some form of fetal therapy.
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ranking = 59.770029317171
keywords = fetomaternal, transfusion, haemorrhage
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5/83. Rapid determination of zygosity and common aneuploidies from amniotic fluid cells using quantitative fluorescent polymerase chain reaction following genetic amniocentesis in multiple pregnancies.

    Following second-trimester twin amniocentesis, we used quantitative fluorescent polymerase chain reaction (QF-PCR) assays and polymorphic small tandem repeats (STR) for rapid determination of zygosity and common aneuploidies from amniotic fluid (AF) cells in four pregnancies with like-sex twins, fused placentae and inconclusive chorionicity. The first and the second cases were suspected to have inadvertent sampling of the same amniotic cavity twice. The first case showed a dizygotic (DZ) pattern and repeat amniocentesis was thus avoided. The second case was monozygotic (MZ) and was complicated by discordant fetal growth and twin-twin transfusion syndrome. The third case was associated with a co-twin malformation, occipital encephalocele. dna studies revealed MZ twinning with a discordant structural defect. The fourth case was associated with co-twin abnormalities of cystic hygroma and hydrops fetalis. dna studies showed DZ twinning with discordant structural and chromosomal defects. The QF-PCR assay with STR has the advantages of rapid determination of zygosity and common aneuploidies in AF cells. This simple test appears to be useful in the instances of possible inadvertent puncture of the same amniotic cavity twice during amniocentesis and of discordant fetal structural and/or chromosomal abnormalities following genetic amniocentesis in multiple pregnancies with uncertain chorionicity.
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ranking = 1
keywords = transfusion
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6/83. B19 parvovirus-induced fetal hydrops:good outcome after intrauterine blood transfusion at 18 weeks of gestation.

    We report a successful treatment of a B19 parvovirus-induced fetal hydrops diagnosed at 16 weeks of gestation. This disease could be corrected by means of a unique intraperitoneal blood transfusion performed at 18 weeks, once diagnosis was established. The delivery occurred at 36 weeks, leading to the birth of a healthy baby. This case suggests that transfusion should be attempted, as the spontaneous fetal recovery remains uncertain and shows that intraperitoneal blood transfusion is an effective therapeutic option of the B19 parvovirus-induced anemia, in the absence of a viral myocarditis.
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ranking = 7
keywords = transfusion
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7/83. Successful fetal sacrococcygeal teratoma resection in a hydropic fetus.

    BACKGROUND/PURPOSE: The development of hydrops in a fetus with a sacroccocygeal teratoma (SCT) usually is a predictor of fetal demise; in utero resection may offer the only chance of survival. Although the authors had performed this procedure in 3 previous cases, they had no long-term patient survival. The authors report a successful case of in utero resection of a fetal sacrococcygeal teratoma. methods: The authors resected a fetal SCT from a 23-weeks-gestation hydropic fetus, using gradually tightening umbilical tapes at the tumor base, electrocautery, and careful sharp dissection. After a blood transfusion, the fetus suffered cardiac arrest but was resuscitated and returned to the uterus. RESULTS: Postoperatively, residual SCT growth ceased, and hydrops rapidly resolved. Five weeks after the procedure, the infant was delivered because of preterm labor, and, after resection of residual SCT, was discharged home at 3 months of age. She is now a healthy 3 year old. CONCLUSION: This case shows that successful fetal SCT resection and long-term patient survival is possible.
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ranking = 1
keywords = transfusion
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8/83. Endoscopic documentation of unintentional perforation of the dividing membrane during amnioreduction for twin-twin transfusion syndrome.

    OBJECTIVE: The purpose of this report is to document endoscopically the occurrence of unintentional piercing of the dividing membrane in a monochorionic twin pregnancy with twin-twin transfusion syndrome (TTTS). MATERIALS AND methods: The dividing membrane was visualized endoscopically during laser therapy in a patient with TTTS who had previously undergone therapeutic amniocentesis. The donor twin had no visible bladder, but the amniotic fluid volume had normalized after amniocentesis. RESULTS: The suspected membrane perforation was identified endoscopically during laser surgery. Five areas of vascular communications were identified and photocoagulated. CONCLUSIONS: Sonographic evidence of normalization of the amniotic fluid volumes after therapeutic amniocentesis with persistent nonvisualization of the bladder of the donor twin should not be interpreted as a sign of improvement of TTTS, but rather as indirect evidence of unintentional perforation of the dividing membrane. This complication can be confirmed endoscopically.
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ranking = 5
keywords = transfusion
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9/83. prenatal diagnosis of twin-twin transfusion syndrome complicated with hydrops fetalis at 14 weeks of gestation.

    Twin-twin transfusion syndrome (TTTS) may complicate multiple pregnancy. Monochorionic discordant twins with oligohydramnios and polyhydramnios may be diagnostic. hydrops fetalis is particularly ominous. All the signs can appear independently at any stage of gestation. However, TTTS with hydrops fetalis in early pregnancy is rare. We report here a case of TTTS complicated with hydrops fetalis diagnosed at 14 weeks of gestation. Our case may be one of the earliest cases of the prenatal diagnosis of TTTS complicated with hydrops fetalis in the literature.
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ranking = 5
keywords = transfusion
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10/83. In utero erythrocyte transfusion for fetal xerocytosis associated with severe anemia and non-immune hydrops fetalis.

    Hereditary xerocytosis is a rare hemolytic anemia occurring secondary to a defect in cell membrane potassium flux. We report a case of severe fetal anemia and non-immune hydrops secondary to hereditary xerocytosis that was managed successfully with in utero erythrocyte and albumin transfusion.
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ranking = 5
keywords = transfusion
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