bluuebunny/crossref_metadata_2025 huggingface dataset:
ACE Inhibition Preserves Heparan Sulfate Proteoglycans in the Glomerular Basement Membrane of Rats with Established Adriamycin Nephropathy
Authors: Wapstra F.H. , Navis G.J. , van Goor H. , van den Born J. , Berden J.H.M. , de Jong P.E. , de Zeeuw D.
Published: April 2003

The gradual onset of the antiproteinuric effects of ACE inhibition suggests that structural effects on the glomerular basement membrane (GBM) may be involved in their renoprotective action. To test this hypothesis, we studied the effects of lisinopril (5 mg/kg/24 h) on proteinuria, focal glomerulosclerosis (FGS) and glomerular heparan sulfate (HS) proteoglycan (HSPG) GBM staining in rats with established Adriamycin nephrosis. Treatment was started 6 weeks after disease induction. As expected, lisinopril reduced blood pressure, proteinuria and the FGS score. In control rats, Adriamycin nephrosis was associated with significantly impaired GBM staining for both HSPG core protein (assessed from BL-31 staining) and HS staining (assessed from JM-403 staining) 12 weeks after disease induction. In rats treated with lisinopril (5 mg/kg/24 h) GBM stianing was significantly better preserved for HS as well as for HSPG core protein. These data suggest that structural effects on the GBM, improving glomerular permselectivity, may be involved in the renoprotective effects of ACE inhibition in proteinuria-induced renal damage.
Oxidant Stress Activates AP-1 and Heparin-Binding Epidermal Growth Factor-Like Growth Factor Transcription in Renal Epithelial Cells
Authors: Sakai Masahiro , Tsukada Toshiaki , Harris Raymond C.
Published: April 2003

Ischemia/reperfusion injury increases the expression of bioactive heparin-binding epidermal growth factor-like growth factor (HB-EGF) in the rat kidney, suggesting that oxidant stress or cell injury related to oxidant stress might affect HB-EGF expression in the injured renal parenchyma. We utilized a nontransformed rat renal epithelial cell line (NRK-52E cells) to investigate whether reactive oxygen species induced transcriptional activation of HB-EGF mRNA. Hypoxia/reoxygenation increased HB-EGF expression in NRK-52E cells, and at concentrations that induced sublethal cell injury, hydrogen peroxide (H2O2) increased HB-EGF mRNA expression 4.7-fold. The free radical scavengers, dimethylthiourea and N-acetylcysteine inhibited HB-EGF mRNA induction. In contrast, another free radical scavenger, pyrrolidine thiocarbamate (PDTC), augmented H2O2-mediated HB-EGF expression. Since PDTC has been reported to augment AP-1-mediated transcriptional activation, we utilized an electrophoretic mobility shift assay to confirm that H2O2 administration to NRK-52E cells did increase nuclear extract DNA-binding activity to a consensus AP-1 sequence. Using a CAT reporter assay coupled to the proximal 2,000 bp of the human HB-EGF 5′-untranslated region, we determined that H2O2 administration increased CAT activity 5.5-fold. Truncation or deletion mutations of a putative AP-1-binding site reduced the H2O2-stimulated activity by >60%, and there was increased DNA binding of nuclear extracts from H2O2-treated cells to a 24-bp oligonucleotide containing this putative AP-1 site. Anti-fos and jun antibodies inhibited this binding, and there was no binding to an oligonucleotide in which the putative AP-1 site was mutated.The site of the residual activation was found to exist in the most proximal 5′-untranslated region (–121 to +60), which contains two putative SP1 sites. Timing and localization of AP-1-binding activity from nuclear extracts from the post-ischemic tissue correlated with HB-EGF mRNA expression. Therefore, in renal epithelial cells, oxidant stress increases HB-EGF expression, which appears to be mediated in part by an increase in AP-1 binding. This activation may play an important role in the induction of HB-EGF mRNA in response to tissue injury and may regulate early stages of recovery following ischemic damage.
Renal and Urinary Glycosaminoglycans in an Experimental Model of Chronic Renal Failure in Rats
Authors: Michelacci Yara M. , Cadaval Ricardo A.M. , Rovigatti Roberto M. , Kohlman Oswaldo
Published: April 2003

The present paper reports the glomerular and renal individual glycosaminoglycan levels in an experimental model of chronic renal failure (CRF) that was induced in Wistar rats by five-sixths mass ablation. Glycemia, body weight, blood systolic pressure and urinary excretions of creatinine, albumin and glycosaminoglycans were measured for 12 weeks. At the end of the experiment, the weight and the glycosaminoglycan composition of the kidneys were determined. In control rats, heparan sulfate was the main glycosaminoglycan found both in whole kidney and isolated glomeruli, with trace amounts of dermatan sulfate. Isolated glomeruli presented higher heparan sulfate concentrations than whole kidney (expressed as mg/g dry weight). In CRF rats, albuminuria appeared from the 2 week on, and dermatan sulfate and chondroitin sulfate contents of the kidney increased, whereas heparan sulfate levels remained unaltered. Changes in urine glycosaminoglycans (heparan sulfate, chondroitin sulfate and dermatan sulfate) were not statistically significant. The increase in glomerular dermatan sulfate and chondroitin sulfate observed in this experimental model could be related to the mechanisms involved in the glomerulosclerosis and proteinuria that occur in CRF.
Fetal Parvovirus B19 Infection and Meconium Peritonitis
Authors: Schild R.L. , Plath H. , Thomas P. , Schulte-Wissermann H. , Eis-Hübinger A.M. , Hansmann M.
Published: April 2003

A 33-year-old primigravida at 26 weeks gestation presented with fetal hydrops and fetal anemia following prior parvovirus B19 infection. The fetus required two intrauterine transfusions of packed red cells. At 35 weeks gestation, a cesarean section was performed for obstetric reasons. As a consequence of a prenatal bowel perforation, the neonate developed meconium peritonitis, for which she needed laparotomy. This case demonstrates that there may be an association between intrauterine parvovirus infection and meconium peritonitis, the latter possibly caused by vascular injury in fetal life.
Renal Hemodialysis and Maternal Serum Triple Analyte Screening
Authors: Shulman Lee P. , Briggs Rodney , Phillips Owen P. , Friedman Steven A. , Sibai Baha
Published: April 2003

The obstetrical management of women with renal disease is complicated and associated with increased fetal and maternal morbidity. However, maternal serum screening is an integral part of obstetrical care and should be offered to all women. We found that maternal serum levels of α-fetoprotein and human chorionic gonadotropin did not significantly change as a result of hemodialysis, whereas levels of unconjugated estriol were markedly decreased following hemodialysis. Maternal serum screening should be limited to α-fetoprotein analysis in women undergoing hemodialysis until the effects of hemodialysis on all serum analytes are better delineated.
Enlarged Cisterna Magna in the Third Trimester as a Clue to Fetal Trisomy 18
Authors: Chen Chih-Ping , Hung Tai-Ho , Jan Sheau-Wen , Jeng Cherng-Jye
Published: April 2003

Six of 19 fetuses with trisomy 18 confirmed by late karyotyping in the third trimester were found to have an enlarged cisterna magna (10 mm or more). For trisomy 18 conceptuses, there is a significant excess of females in both fetuses and livebirths; however, we found this ratio to be reversed in the third-trimester trisomy 18 fetuses with an enlarged cisterna magna (5 males vs. 1 female), indicating males are more likely to have this associated defect. Each of the 6 cases was associated with other trisomy markers including intrauterine growth retardation and polyhydramnios. Since a significant proportion of pregnancies may escape early prenatal care and some potentially detectable fetal abnormalities may be missed on early ultrasound and/or by maternal serum screenings, prenatal detection of an enlarged cisterna magna associated with intrauterine growth retardation and/or polyhydramnios in late gestation should prompt complete genetic counseling, rapid karyotyping and a careful search for other congenital anomalies.
A Case with Duodenal Duplication Cyst: Prenatal Diagnosis and Surgical Management
Authors: Yamataka Atsuyuki , Pringle Kevin C.
Published: April 2003

Objective: We report the first case of a subhepatic cystic mass diagnosed in utero that subsequently proved to be a duodenal duplication cyst. Method: Conventional prenatal abdominal ultrasound. Results: On prenatal ultrasonography, the differential diagnosis was choledochal cyst, bowel duplication cyst, or omental cyst. Conclusion: The prenatal detection of a cystic mass allowed close neonatal surveillance and timely surgical intervention prior to the onset of potentially serious complications.
Analysis of the Origin of the Extra Chromosome in Trisomy 8 in 4 Cases of Spontaneous Abortions
Authors: Nicolaidis Peter , von Beust Gabriela , Bugge Merete , Karadima Georgia , Vassilopoulos Dimitris , Brøndum-Nielsen Karen , Petersen Michael B.
Published: April 2003

Objective: To determine the origin of the extra chromosome in trisomy 8 in spontaneous abortions. Methods: We analyzed 4 cases of nonmosaic trisomy 8 in 1st-trimester spontaneous abortions and their parents with DNA polymorphism analysis using microsatellite DNA markers. Results: In 3 cases the extra chromosome was maternal in origin and in 1 case paternal in origin. In 2 of the cases the nondisjunction had occurred in maternal meiosis, while the other 2 cases were consistent with a postzygotic (mitotic) origin of the additional chromosome. Conclusion: Although a small number of cases studied, these results suggest differences from the common autosomal trisomies 21, 18, 16, and 13 where the vast majority of cases are due to errors in maternal meiosis.
The Apparently Isolated Choroid Plexus Cyst: Importance of Minor Abnormalities in Predicting the Risk for Aneuploidy
Authors: Leonardi Michael R. , Wolfe Honor M. , Lanouette Jan M. , Landwehr Joseph B. , Johnson Mark P. , Evans Mark I.
Published: April 2003

Objectives: To assess the risk of aneuploidy in cases of isolated choroid plexus cysts (CPCs) and to compare the risk when associated with minor or major anomalies. Methods: All fetuses with CPCs and known karyotype were identified. CPCs were categorized as ‘isolated’ or associated with minor or major sonographic anomalies. Preexisting risk factors for aneuploidy were compared between groups. The frequency of aneuploidy was compared between fetuses with isolated CPCs and those with CPCs associated with minor or major anomalies. Continuous and categorical variables were analyzed using one-way analysis of variance or chi-square as appropriate with p < 0.05 considered significant. Results: One hundred and forty-nine fetuses with CPCs diagnosed at a mean gestational age of 19 weeks were identified. No significant differences in the frequency of preexisting risk factors for aneuploidy were identified between groups. Eighteen of 149 (12%) fetuses with CPCs had other sonographic anomalies; in 10 they were minor, and 2 of the 10 had abnormal karyotypes. Four of 8 fetuses with major anomalies were aneuploid. All 131 fetuses with isolated CPCs had normal karyotypes, and all aneuploid fetuses had additional anomalies. Conclusions: The overall rate of aneuploidy in patients with CPCs was 4% with no abnormal karyotypes among isolated CPCs. The presence of even minor sonographic abnormalities substantially increased the risk of aneuploidy. Isolated CPCs identified sonographically may not place the patient at risk of aneuploidy, but should prompt a diligent search for other minor or major anomalies. The finding of any other anomaly warrants consideration for karyotypic evaluation.
Management of a Case of Twin-to-Twin Transfusion Syndrome by a Combined Surgical Approach
Authors: van Peborgh P. , Pelletier G. , Suarez B. , Ville Y.
Published: April 2003

Objective: The management of a case of severe twin-to-twin transfusion syndrome at 24 weeks of gestation is presented, using a combined surgical and medical approach. Methods: Operative fetoscopy was used to coagulate placental vessels crossing the intertwin membranes. Repeated intrauterine transfusions were used to correct recurrent anemia in the donor twin. Selective three-dimensional placental angiography and directed histological analysis were used to study the placenta after delivery. Results: Resolution of twin-to-twin transfusion syndrome was obtained by coagulation of placental anastomoses, but was followed by recurrent anemia in the donor twin. This was successfully treated by serial intrauterine blood transfusions, and 2 healthy twins were delivered at 31 weeks of gestation. One artery-to-vein anastomosis was demonstrated to be the only communication left between the two circulations. Conclusions: This case illustrates the limitations of placental surgery in twin-to-twin transfusion syndrome and highlights the need for ultrasound and Doppler follow-up of these high-risk fetuses to indicate further treatment. Placental anastomoses can be overlooked by macroscopic examination and injection technique, but can be demonstrated by placental angiography.
Surgical Approach of an Early Mammalian Embryo: The Rabbit Model
Authors: Beaudoin S. , Simon L. , Simeoni J. , Sacquin P. , Bargy F.
Published: April 2003

We describe an experimental surgical model in early rabbit embryo (12.5 days of gestation). Twenty-one embryos were operated, of which 9 survived the first postoperative week and 5 had normal further growth until term. As far as we can ascertain, this is the first successful report of a surgical approach in early mammalian embryos. We think that this model may be useful for workers involved in the study of early congenital malformations.
Amniotic Septostomy for the Treatment of Twin Oligohydramnios-Polyhydramnios Sequence
Authors: Saade George R. , Belfort Michael A. , Berry David L. , Bui The-Hung , Montgomery Lynn D. , Johnson Anthony , O’Day Mary , Olson Gayle L. , Lindholm Henry , Garoff Leena , Moise Jr. Kenneth J.
Published: April 2003

Objective: To report our experience with intentional puncture of the intervening membrane (‘septostomy’) for the treatment of the twin oligohydramnios-polyhydramnios sequence (TOPS). Methods: 12 patients were diagnosed with TOPS based on ultrasonographic findings. A 20- to 22-gauge spinal needle was used to puncture the membrane between the twins without any attempt at amnioreduction in 9 patients, while the procedure was combined with amnioreductions in 3 patients. Results: Gestational age was 23.1 ± 3.3 weeks at the time of septostomy and 31.1 ± 4.4 weeks at delivery. Rapid accumulation of fluid around the ‘stuck’ fetus occurred in all cases following a single procedure. Three of the 24 fetuses died in utero and 1 died on the fifth day of life, for a combined survival of 83.3%. In the survivors, the septostomy to delivery interval ranged between 0.6 and 13 weeks (mean ± SD 8.3 ± 4.8). Conclusion: Amniotic septostomy is a promising new method for the management of TOPS and is associated with survival rates that are better than, or comparable to, more invasive modalities. A multicenter trial comparing septostomy to other modalities is warranted.
‘Dual Positivity’ for Neural Tube Defects and Down Syndrome at Maternal Serum Screening: Gestational Outcome
Authors: Zanini Roberto , Tarantini Michele , Cerri Valeria , Jacobello Carmelo , Bellotti Daniela , Lancetti Sara , Scalchi Sabrina , Groli Caterina , Bianchi Umberto A.
Published: April 2003

Objective: To evaluate the gestational outcome of pregnancies screen-positive for both neural tube defects (NTD) and Down syndrome (DS) (‘dual positivity’). Methods: Among 10,667 mid-trimester women screened for DS and NTD with α-fetoprotein (AFP), unconjugated estriol (uE3), and human chorionic gonadotropin (hCG), delivered up to July 1996, we have selected cases with both an unexplained AFP value ≥2.5 multiples of median (MoM) and a DS risk ≥1:250. All these pregnant women were managed with amniocentesis and/or CVS, ultrasound scans, and Doppler velocimetry. We have collected all data about the gestations with ‘dual positivity’ and no obvious explanation for these findings (cases with fetal malformations related to raised AFP). Results: Twelve women (1.1:1,000) showed unexplained ‘dual positivity’. Abnormal karyotypes were found in 3 fetuses, and pregnancies were terminated: there were 2 triploidies with partial hydatiform mola, and 1 DS. In 9 cases the fetal karyotype was normal, but a confined placental trisomy 16 was found in 4. Of the 9 continuing gestations, 8 displayed fetal growth retardation (FGR). One gestation ended with fetal death at 27 weeks. All 9 fetuses were morphologically normal, and 8 were small for gestational age. Conclusions: ‘Dual positivity’ at NTD/DS screening may anticipate pregnancy complications. The finding of trisomy 16 confined to the placenta and FGR in 4 cases suggests that at least some fetuses with growth restriction may suffer from a distinct placental disease. Maternal serum screening may have implications different from DS and NTD, as demonstrated by the 2 cases with triploidy and incomplete hydatiform mola, the 4 cases with placental trisomy 16, and the 4 cases of FGR of the 5 fetuses without chromosome abnormalities. As the pathologic outcome of these pregnancies is more important than the mere serum screening results, we feel that these cases need a strict work-up, including CVS, amniocentesis and ultrasound studies to better address the obstetrical management.
Early Antenatal Diagnosis of Cardiac Defects Using Transvaginal Doppler Ultrasound: New Perspectives?
Authors: Areias José Carlos , Matias Alexandra , Montenegro Nuno , Brandão Otília
Published: April 2003

Objective: Cardiac defects are the most prevalent congenital anomalies. Screening policies have adopted an 18- to 22-week ultrasound scan to detect such anomalies. However, diagnosis may be feasible early in pregnancy using transvaginal Doppler ultrasound. Methods: Transvaginal ultrasound, including nuchal translucency (NT) measurement, is routinely performed at 10–13 weeks of gestation at our department. Complementary arterial and venous blood flow Doppler evaluation is also performed in cases of increased NT as a part of an ongoing research project. Results: Ultrasound revealed complete atrioventricular septal defects in 2 trisomic fetuses (trisomy 18 and 21) at 12 and 13 weeks of gestation, respectively, with increased NT and abnormal venous blood flow velocity. Conclusions: Incrased NT has been associated with major chromosomal anomalies and is being increasingly related to cardiac defects. Considering that venous blood flow patterns may provide additional clues to the cardiac function, it may be useful as a complementary tool for the earlier diagnosis of structural cardiac anomalies.
Blood Gas and pH in the Human Fetus with Severe Anemia
Authors: Vandenbussche Frank P.H.A. , van Kamp Inge L. , Oepkes Dick , Hermans Jo , Bennebroek Gravenhorst Jack , Kanhai Humphrey H.H.
Published: April 2003

Leiden University Hospital is the national referral center for the management of fetal isoimmunization in The Netherlands. In this observational study, blood gas and acid-base measurements from 286 pretransfusion samples and 214 paired posttransfusion samples of 113 fetuses were analyzed. In umbilical arterial blood, we found a significant positive correlation between the degree of anemia and pH, as well as a significant negative correlation between degree of anemia and pO2. However, umbilical venous blood gas and pH remained virtually unchanged even in severe anemia. During intrauterine transfusion with unbuffered adult red cells, there was a small but statistically significant decrease of pH and pO2 in fetal blood. We conclude that severe fetal anemia is associated with decreased umbilical arterial pH, but that umbilical venous pH remains normal until shortly before death.
Pastoral Care Utilization among Women Electing Pregnancy Termination for Fetal Anomalies
Authors: Brady Teresa B. , Lidums Sue , Yaron Yuval , Evans Mark I. , Johnson Mark P. , Kramer Ralph L.
Published: April 2003

Objective: To identify determinants of requesting pastoral care (PC) at the time of pregnancy termination for fetal anomalies. Methods: PC was provided by a hospital-based chaplain. Data were reviewed for 88 women who elected pregnancy termination. Nominal logistic regression and Kruskal-Wallis and Student t tests were used as appropriate. Results: None of 37 women who underwent dilatation and curettage (D&C) or dilatation and evacuation (D&E) requested PC as compared with 40 of 51 patients who underwent prostaglandin induction of labor (p < 0.001). The decision to seek PC was related to gestational age (p < 0.001), but not to maternal age, prior termination of pregnancy, gravidity, parity, racial background, or insurance status. The gestational age was also a significant predictor of the procedure performed (p < 0.001). D&C/D&E were significantly more likely to be performed at earlier gestational ages. Women seeking PC were less likely to have experienced previous pregnancy loss or the death of a child. Among women who sought PC, 20% had experienced previous pregnancy loss or the death of a child, as opposed to 54% of women in the prostaglandin group who did not seek PC (p = 0.03). In the D&C/D&E group, the figure was 46%. Conclusions:Utilization of PC is much more frequent among women undergoing prostaglandin induction of labor than D&C/D&E. Determinants of seeking PC are related to issues congruent with choices of the termination procedure. Patients who experienced a previous pregnancy loss or the death of a child are also less likely to feel the need, or do not want, chaplaincy involvement.
Cytogenetic Studies in Fetal Blood
Authors: Costa D. , Borrell A. , Soler A. , Carrió A. , Margarit E. , Ballesta F. , Puerto B. , Caballín M.R. , Fortuny A.
Published: April 2003

In order to assess the effectiveness and reliability of cytogenetic diagnosis provided by fetal blood, we report the first 186 cases of fetal blood sampling performed for rapid karyotype between 19–37 weeks of pregnancy in our Prenatal Diagnosis Unit. The overall diagnostic success rate was 98%, achieving 100% in the last period of the study. Chromosomal anomalies were detected in 16% (29/182) of the fetuses. In malformed fetuses this rate increased from 8–9% in isolated malformation or markers of aneuploidy to 50% in multiple malformations. In pregnancies in which a previous cytogenetic study in amniotic fluid was inconclusive, fetal blood made it possible to obtain a definitive result, with no discrepancies found at phenotypic follow-up examination. Interestingly enough, one of the four previously defined as pseudomosaicisms was found to be a non-mosaic in fetal blood, and only 1 of 4 mosaicisms was confirmed in fetal blood. In conclusion, cytogenetic analysis of fetal blood samples appears to be effective, rapid and reliable to establish the fetal karyotype in selected cases.
Insulinlike Growth Factor 1 in Controls and Growth-Retarded Fetuses
Authors: Bocconi Luisa , Mauro Fabio , Maddalena Silvia E. , De Iulio Cinzia , Tirelli Amedea S. , Pace Elisabetta , Nicolini Umberto
Published: April 2003

Objectives: To establish a reference range of insulinlike growth factor 1 (IGF-1) values in normal fetuses and to assess whether intrauterine growth retardation is associated with increased or decreased IGF-1 levels. Methods: Retrospective analysis of blood samples collected from 64 fetuses who underwent blood sampling at 18–38 weeks’ gestation was performed: 40 fetuses, who were considered controls, were appropriately grown for gestational age and were found unaffected by the condition for which they were tested; the remainder (n = 24) underwent fetal blood sampling to assess fetal karyotype and acid-base balance following ultrasonic diagnosis of intrauterine growth retardation. (In this group, 8 survived, and 16 died during the perinatal period). IGF-1 was measured using a radioimmunoassay after acid-ethanol extraction in order to avoid interference by the binding proteins. All samples from controls and growth-retarded fetuses were measured using the same batch, and the intra-assay coefficient of variation of the test ranged from 4.1 to 6.1%. Results: In control fetuses, IGF-1 serum levels increased linearly with gestational age. In growth-retarded fetuses, IGF-1 levels were not significantly different from the reference range (median Z-score –0.3; range –4.4 to 291) and did not correlate with fetal size, hematocrit, and acid-base balance values. There was a significant difference in IGF-1 and pH values when the fetuses were divided into two groups based on the perinatal outcome: those who survived had values of IGF-1 mostly within the normal range, whereas the fetuses who died in utero or postnatally had significantly decreased pH and elevated IGF-1 values (median Z-score 2.1; 95% confidence interval 0.4–13.9; p = 0.04). Conclusions: This study confirms previous observations that IGF-1 levels parallel the increase in fetal size which occurs with advancing gestation. Increased levels of IGF-1 may indicate a terminal process in the fetal adaptation to placental failure.
Ultrasound-Guided Gene Transfer to Hepatocytes in utero
Authors: Wang Guoshun , Williamson Roger , Mueller Glenda , Thomas Patricia , Davidson Beverly L. , McCray Jr Paul B.
Published: April 2003

Objectives: Several inherited liver diseases are associated with a progressive course that begins early in life. Such disorders may be amenable to treatment with gene transfer in the fetal or neonatal period. Methods: We used ultrasound guidance to deliver an adenoviral vector to the liver of 28-day gestation fetal rabbits by cardiocentesis. β-Galactosidase reporter gene expression in hepatocytes was analyzed 3, 7, and 21 days after vector delivery. Using this nonsurgical approach, the viral vector was efficiently delivered into the fetal circulation. Results: The liver was the main organ targeted by this route of administration with up to 40% of the hepatocytes β-galactosidase positive in some animals. The β-galactosidase expression in hepatocytes gradually declined between 3 and 21 days following gene transfer. Associated with the decline in gene expression, an increased number of inflammatory cells were noted in the livers of adenoviral vector treated animals. This suggests that an immune response limits the duration of gene expression in the fetal rabbit, similar to the findings in postnatal animals. Conclusions: This animal model and vector delivery method may be useful for evaluating gene transfer to the fetus with viral and nonviral vectors. Further modifications of the adenoviral vector to reduce immunogenicity may enhance the duration of expression.
Fetal and Neonatal Hypertension in Twin-Twin Transfusion Syndrome: A Case Report
Authors: Baud O. , Lebidois J. , van Peborgh P. , Ville Y.
Published: April 2003

We report a case of fetal systemic hypertension. This occurred in an ex-donor twin soon after coagulation of chorionic vessels and amniodrainage performed for severe twin-twin transfusion syndrome during the 2nd trimester of pregnancy. Systemic hypertension was suspected because of a high systolic velocity through the tricuspid valve, and Bernoulli’s equation was used to estimate the right intraventricular pressure. As both pulmonary arteries and ductus arteriosus were normal, the pressure in the aorta was considered to be equal to that in the right ventricle (60 mm Hg). Fetal systemic hypertension could have happened either because of a dramatic increase in placental resistances in the territory of the ex-donor twin or by reversal of the fetofetal transfusion pathological process.
Detection of an Obstructive Membrane in the Ductus arteriosus of a Fetus Using High Frame Rate Echocardiography
Authors: Rein A.J.J.T. , Nadjari Michel , Bromiker Ruben , Omokhodion Samuel I. , Nir Amiram
Published: April 2003

We examined a fetus at 38 weeks of gestation because of marked disproportion in size of the left and right ventricles. A membrane was detected at the distal end of the ductus arteriosus with significant flow gradient through it. New echocardiographic technologies such as high frame rate imaging and dynamic beam focusing increase spatial and temporal resolution and enhance more precise anatomical diagnosis in the fetus. This ductal membrane might have been related to the transient tachypnea from which the baby suffered after birth.
Prenatally Diagnosed Fetal Neck Teratoma
Authors: Gagnon Alain L. , Bebbington Michael W. , Kamani Ali , Solimano Alfonso
Published: April 2003

A case of fetal neck teratoma diagnosed at 32 weeks’ gestation is reported. Planning before delivery included consultations with the neonatologist, the anesthesiologist, the otolaryngologist and the social worker. Delivery by cesarean section under epidural anesthesia was carried out at 34 weeks because of preterm labor. The head was delivered and the fetus was intubated within 4½ min using a rigid bronchoscope. Magnesium sulfate and nitroglycerin provided uterine relaxation allowing fetal oxygenation via the uteroplacental circulation. Umbilical artery blood gases were satisfactory. The early neonatal course was complicated by hemorrhage from the teratoma externally as well as intratracheal bleeding and ventilatory problems. The baby died at 40 min of age in his mother’s arms.
Unexpected Inherited Chromosomal Translocation during Prenatal Diagnosis for Maternal Age: Risk for a Nondetectable Karyotype Imbalance in Offspring
Authors: Siffroi Jean-Pierre , Heim Nazbanou , Benzacken Brigitte , Franco Jean-Claude , Le Bourhis Corine
Published: April 2003

An unexpected t(1;19) translocation is described in a fetus. Inherited from the mother, this translocation was found during the course of a normal prenatal diagnosis made for maternal age. The very short length of chromosomal translocated segments and their labelling pattern made high-resolution cytogenetic methods and fluorescence in situ hybridization techniques necessary for the correct identification of this karyotype rearrangement, both in mother and fetus. Different modes of meiotic segregation, leading to potential erroneous prenatal diagnoses, are discussed.
Maternal Serum Placental Growth Hormone and Insulinlike Growth Factor Binding Proteins 1 and 3 in Pregnancies Affected by Fetal Aneuploidy and Other Abnormalities: Implications for Prenatal Diagnosis of Trisomy 21
Authors: Moghadam S. , Engel W. , Bougoussa M. , Hennen G. , Igout A. , Sancken U.
Published: April 2003

In the present study, we determined circulating serum levels of human placental growth hormone (hPGH) and insulinlike growth factor binding proteins 1 and 3 (IGFBP-1 and IGFBP-3) using two-site radioimmunoassays during the gestational midtrimester of pregnancies affected by chromosomal disorders with the aim of identifying potential marker substances that might have a significant discriminative and predictive value for prenatal diagnosis of fetal chromosomal aberrations and of organ malformations such as neural-tube defect. Our results show that the maternal serum levels of hPGH were significantly elevated in pregnancies affected by chromosomal anomalies or organ malformations as compared with controls. The distribution of IGFBP-1 concentrations for all experimental groups except trisomy 21 were closely similar to the normal population. IGFBP-3 decreased slightly in pregnancies affected by Down syndrome. These findings suggest that hPGH may be useful as an additional marker in prenatal screening for Down syndrome.
Peripartal Temporal Course of Endothelin 1, Angiotensin II, and Atrial Natriuretic Peptide in Pre-Eclampsia and Normotensive Pregnancy
Authors: Zunker P. , Happe S. , Louwen F. , Evers S. , Ringelstein E.B.
Published: April 2003

Objective: To investigate the temporal peripartal course of plasma endothelin 1 (ET-1), angiotensin II (ANG II), and atrial natriuretic peptide (ANP) in patients with pre-eclampsia as compared to women with normotensive pregnancy. Methods: Levels of ET-1, ANG II, and ANP were measured by means of radio-immunoassay twice a week in 17 patients with pre-eclampsia and in 17 women with normotensive uncomplicated pregnancy during four different periods: (a) from admission to 1 week before delivery, (b) 1st week before delivery, (c) 1st week after delivery, and (d) 2nd week after delivery. Results: ET-1 levels were not statistically different between the two groups. ANG II concentrations were lower in the pre-eclampsia group than in the control group with statistical significance in period c (2.8 ± 3.0 vs. 7.1 ± 5.9 pg/ml, mean ± SD). ANP levels were higher in pre-eclamptic women than in the control group, the difference being statistically significant in period a (54 ± 46 vs. 11 ± 16 pg/ml) and in period c (122 ± 134 vs. 39 ± 22 pg/ml). Conclusions: There are considerable differences in ANP and ANG II concentrations during the peripartal period between patients with pre-eclampsia and normotensive pregnant women. Decreased ANG II and elevated ANP levels in the 1st week postpartum may reflect the clinical observation that some women deteriorate in this period.
Massive Intracranial Immature Teratoma with Extracranial Extension into Oral Cavity, Nose, and Neck
Authors: Alagappan A. , Shattuck K.E. , Rowe T. , Hawkins H.
Published: April 2003

We present a case of congenital massive intracranial teratoma with extracranial extension into oral cavity, nose, and neck diagnosed by antenatal ultrasonography at 25 weeks of gestation. The fetus was delivered by cesarean section because of massive fetal head size and severe maternal pregnancy-induced hypertension. At necropsy the tumor was found to be an immature teratoma, with no recognizable normal brain tissue. An additional finding was hepatomegaly secondary to extensive extramedullary hematopoiesis. Karyotyping of both the fetus and the teratoma revealed a normal female chromosomal composition: 46,XX.
Fetal Glioblastoma: Rapid Growth during the Third Trimester
Authors: Kamitomo Masato , Sameshima Hiroshi , Uetsuhara Kouichi , Tomosugi Tetsuzou , Matsuda Yoshio , Ibara Satoshi , Sueyoshi Kazunobu
Published: April 2003

We present a case of fetal glioblastoma which appeared after 28 weeks’ gestation. The first ultrasonographic finding was an enlarged fetal head with right shifted falx cerebri at 31 weeks’ gestation. At 33 weeks, a large and high echogenic mass in the left hemisphere and right enlarged ventricle was identified. Magnetic resonance imaging showed that the tumor was localized in the left hemisphere and a margin of the tumor was defined. Because fetal well-being judging from biophysical parameters was good and we considered that the tumor was resectable, a male fetus weighing 2,670 g was delivered at 34 weeks’ gestation by cesarean section. However, he was inoperable due to consumptive coagulopathy and rapid growth of the tumor, and died on the 41st day of life.
Frequencies of Fetal Nucleated Red Blood Cells in Maternal Blood during Different Stages of Gestation
Authors: Kuo Pao-Lin
Published: April 2003

Nucleated red blood cells (NRBCs) isolated by a triple density gradient from 100 ml peripheral blood samples of 100 pregnant women and 30 women postpartum were subjected to morphological analysis and PCR quantitation. The number of NRBCs steadily increased from 5.3 (frequency: 2.4 × 10–7) in early gestation to 98.2 (frequency: 4.2 × 10–6) near term. The number of male cells increased from 6 (frequency: 2.7 × 10–7) in early gestation to a peak of 31 (frequency: 1.48 × 10–6) in the second trimester, and slightly decreased to 27 (frequency: 1.31 × 10–6) near term. Both NRBCs and male fetal cells rapidly disappeared after delivery. The result implies that a significant proportion of NRBCs in maternal blood are of fetal origin before 24 weeks of gestation while in late gestation the majority of NRBCs may be of maternal origin.
Lethal Congenital Dyserythropoietic Anaemia Type I in Siblings Presenting as Pericardial Effusions in the Second Trimester
Authors: Stone Peter , Zuccollo Jane
Published: April 2003

Congenital dyserythropoietic anaemias (CDA) are rare inherited disorders of erythropoiesis characterised by abnormal red cell morphology and haemolysis. The diagnosis of CDA should be considered in the fetus or patient presenting with a normocytic or macrocytic anaemia especially if red cell morphology is abnormal. Three types and other possible variants have been described. There are few reports of clinical presentation of CDA in utero. We present 2 cases of lethal CDA in siblings that presented with pericardial effusions in the second trimester.
Management and Outcome of Fetomaternal Rh Alloimmunization in Twin Pregnancies
Authors: Lepercq Jacques , Poissonnier Marie-Hélène , Coutanceau Marie José , Chavinie Jacques , Brossard Yves
Published: April 2003

Between 1987 and 1996, nine twin pregnancies with fetomaternal Rh alloimmunization were delivered at our institution. Eight pregnancies were dizygotic, and the fetal blood groups were different in 3 cases. The remaining pregnancy was monozygotic and monochorionic-diamniotic. Intravenous fetal exchange transfusion was performed in five pregnancies, up to five times in each twin in one pregnancy. No fetal death occurred. The average gestational age at birth was 35 (range 33–37) weeks. The hemoglobin level was 13.2 (range 9.2–16.5) g/dl. Fetomaternal Rh alloimmunization in twin pregnancy is according to zygosity; each fetus has to be treated separately, except in case of transplacental communication.
Extra Euchromatic Band in the qh Region of Chromosome 9
Authors: von Beust Gabriela , Bink Karin
Published: April 2003

Chromosomal analysis of amniotic cell culture revealed an extra euchromatic band in the variable heterochromatin region 9q12. Cytogenetic analysis of the fetus was compared with the chromosomes of the parents. Using different cytogenetic banding techniques and fluorescence in situ hybridization with specific DNA probes, the structural rearrangements involved were considered. The very rare variant proved to be familial. Demonstrating the inheritance of a normal individual supports the interpretation of the prenatal analysis of chromosome 9 as a variant without clinical relevance for the fetus.
Early Transvaginal Measurement of Cephalic Index for the Detection of Down Syndrome Fetuses
Authors: Rosati Paolo , Guariglia Lorenzo
Published: April 2003

Objective: The aim of this study was to produce normal cephalic index values by transvaginal scan early in pregnancy and to evaluate the screening utility of this measurement for the identification of fetuses at risk for Down syndrome. Method: In this prospective cross-sectional study, transvaginal high-resolution sonography was performed between 9 and 16 weeks of gestation in 1,087 euploid fetuses and 36 Down syndrome fetuses. Measurements of the cephalic index, calculated as the ratio biparietal diameter/occipito-frontal diameter, were plotted against gestational age. Results: The cephalic index was found to show fairly constant values throughout the period evaluated. The best correlation with gestational age was described by a linear correlation. All the measurements obtained in Down syndrome fetuses were within the normal ranges. Conclusion: Our data show that in early pregnancy the cephalic index cannot be considered a useful tool in the detection of fetuses at risk for Down syndrome.
Acardiac Twins: Pathophysiology, Diagnosis, Outcome and Treatment
Authors: Søgaard Kirsten , Skibsted Lillian , Brocks Vibeke
Published: April 2003

Twin gestation is often a hazardous pregnancy and especially the monochorionic twin pregnancy significantly contributes to fetal morbidity and mortality. Among the serious complications with twins, the twin-twin transfusion syndrome complicates 5–35% of monozygotic twin pregnancies with monochorionic placentation. Acardiac twinning, earlier known as chorioangiopagus parasiticus, is the most extreme manifestation of this condition. An acardiac twin is a rare complication of multifetal pregnancy, in the literature reported at an incidence of 1% of monochorionic twin pregnancies, i.e. 1 of 35,000 pregnancies. In the following paper we review the literature on the subject and report 6 cases, 5 twins and 1 triplet, that were diagnosed at our department during the period of 1993–1997 and treated conservatively. Only 1 child survived.
Risk Factors of Hypoglycemia in Premature Infants
Authors: Zanardo V. , Cagdas S. , Golin R. , Trevisanuto D. , Marzari F. , Rizzo L.
Published: April 2003

Hypoglycemia is a frequent complication of preterm birth and may lead to later CNS damage. The hypoglycemia incidence and the relative risk factors for the affected preterm infants were assessed. We examined 1,500 preterm infants (<37 weeks of gestational age) consecutively admitted between January 1994 and December 1996 at the Department of Pediatrics of Padua University, and screened for hypoglycemia by Dextrostix within the first hour of life. Hypoglycemia was defined as blood glucose levels <40 mg% at Dextrostix. Among study prematures, 35% had hypoglycemia; while the incidence was 9% at levels of Dextrostix <20 mg%. The relative risk for hypoglycemia (odds ratio, OR) was computed assuming a 99% confidence interval (CI). We found 5 risk factors for hypoglycemia: cesarean section (OR 2.24, CI 1.66–3.03), intrauterine malnutrition (SGA) (OR 1.65, CI 1.08–2.53), NICU hospitalization (OR 1.45, CI 1.09–1.93), gestational age between 30 and 33 weeks (OR 1.93, CI 1.34–2.78), and twinning (OR 2.49, CI 1.77–3.56). At levels of Dextrostix <20 mg%, 3 more risk factors were found: cardiopulmonary resuscitation at birth (OR 4.06, CI 2.52–6.54), neonatal respiratory distress syndrome (OR 2.21, CI 1.34–3.36) and gestational age between 26 and 29 weeks (OR 2.16, CI 1.02–4.25). The identification of relative risk factors could be useful in improving the hypoglycemia prophylaxis, and in reducing related later CNS abnormalities.
Antenatal Diagnosis of Homozygous Alpha Thalassemia
Authors: Doridot V. , Sibony O. , Luton D. , Reyal F. , Feraud O. , Multon O. , Oury J.F. , Blot P.
Published: April 2003

Objective: Diagnosis of the α-thalassemia-2 trait. Method: Homozygous α-thalassemia was discovered by chance in the fetus of a female Chinese patient. Major intrauterine growth retardation, oligohydramnios, an immobile fetus, and cardiomegaly were the principal echographic signs. Cordocentesis showed fetal anemia, and electrophoresis of fetal hemoglobin revealed the presence of Bart’s hemoglobin. Result: As there is no known effective treatment, termination of pregnancy was proposed to the patient. Conclusions: An α-thalassemia-2 trait is a lethal condition. Early echographic signs (cardiothoracic index >0.50, placental thickening) can be screened during weeks 17–18 or even during weeks 13–14 of gestation. These signs would permit a reduction of invasive examinations in couples at risk.
Induction of Labor in Patients with Term Premature Rupture of Membranes
Authors: Hallak Mordechai , Bottoms Sidney F.
Published: April 2003

Objective: To determine whether delayed induction of labor in patients with premature rupture of membranes (PROM) at term has beneficial effects on the mother or the infant. Study Design: Retrospective analysis of our database revealed 576 patients >37 weeks of gestation with PROM, who delivered live-born infants without major congenital anomalies. We analyzed the frequencies of primary cesarean, neonatal intensive care unit (NICU) admissions, and oxytocin use by time since hospital admission and interval until onset of labor. Results: NICU admission increased from 1.9% in <3 h between admission to onset of labor to 13.3% after >18 h. Admission-onset of labor interval, birth weight of <2,500 or >4,000 g and meconium were all more important determinants of NICU admission than gestational age, duration of labor, PROM, and ROM. Prolonged admission-onset of labor interval was associated with an increased risk of variable decelerations (p < 0.001). Primary cesarean rates increased progressively with longer intervals between admission and onset of labor. Stepwise discriminant function analysis revealed that labor duration, admission-onset of labor interval, gestational age, and birth weight of <2,500 g were all more important determinants of primary cesarean delivery than the durations of PROM or ROM. Conclusions: The increased frequencies of NICU admission, variable decelerations, and primary cesarean suggest that delayed labor induction after hospital admission was linked to worsened perinatal outcomes. These results may have been influenced by usually performing a single digital examination as part of initial evaluation of term patients who present with PROM. Based on our data, we suggest immediate induction for PROM at term, especially if digital examination has been performed.
Amnioinfusion in the Evaluation of Fetal Obstructive Uropathy: The Effect of Antibiotic Prophylaxis on Complication Rates
Authors: Feldman Baruch , Hassan Sonia , Kramer Ralph L. , Kasperski Stefanie B. , Evans Mark I. , Johnson Mark P.
Published: April 2003

Objective: Amnioinfusion plays an important role in the intrauterine evaluation and treatment of fetal obstructive uropathy. However, it may significantly increase the risk for chorioamnionitis, premature rupture of membranes and premature labor. We evaluated the impact of prophylactic antibiotics on postamnioinfusion complications. Methods: Thirty pregnancies complicated by fetal obstructive uropathy, treated by amnioinfusion and with documentation of pregnancy outcome were identified from our database. Pregnancy outcomes were compared between patients who were treated with prophylactic antibiotics and those with no prophylaxis. Results: Chorioamnionitis was diagnosed in 3 out of 15 (20%) patients who did not receive prophylactic antibiotics as compared to 2 (13%) in the treated group. The overall rate of serious obstetrical complication was significantly higher in the untreated group (66 vs. 20%; p = 0.021). Patients receiving prophylactic antibiotics delivered at a significantly greater gestational age than those who did not receive antibiotics (34.0 ± 3.7 vs. 31.3 ± 1.9 weeks, respectively; p = 0.018). Conclusions: Our study supports the use of oral prophylactic antibiotics as being effective in reducing the previously observed significant risks associated with amnioinfusion in fetal obstructive uropathy.
Differential Effect of Advanced Maternal Age on Prenatal Diagnosis of Trisomies 13, 18 and 21
Authors: Drugan Arie , Yaron Yuval , Zamir Ronit , Ebrahim Salah A.D. , Johnson Mark P. , Evans Mark I.
Published: April 2003

Nondisjunction associated with advanced maternal age, a well-established factor in the etiology of autosomal trisomy, should equally affect all chromosomes. In this study we evaluate the association of advanced maternal age with the occurrence of potentially viable autosomal trisomies (13, 18 and 21). 275 aneuploid pregnancies were ascertained prenatally and were grouped according to chromosome anomaly diagnosed. Mean maternal age was significantly younger (p = 0.009) in pregnancies affected by trisomy 13 than in pregnancies with trisomy 21. An intermediate mean maternal age was observed in pregnancies affected by trisomy 18. Our study shows a trend of the more severe, but potentially viable, autosomal trisomies to be diagnosed at younger maternal age. This may substantiate the ‘relaxed selection hypothesis’ proposed to explain the association of aneuploid conceptions with advanced maternal age.