Outcome of Monochorionic Diamniotic Twins with Twin Reversed Arterial Perfusion Sequence: Interstitial Laser versus Endoscopic Cord Occlusion.

Twin reversed arterial perfusion sequence (TRAP) is a rare complication of monochorionic twins (MC). This study aimed to describe and compare the short- and long-term outcomes of MC pregnancies with the TRAP sequence treated with two different techniques: interstitial fetal laser (IFL) (n = 22) versus endoscopic cord occlusion (CO) (n = 24). The study population included 46 MC pregnancies with TRAP. Pregnancy loss within 2 weeks after the procedure occurred in 27% of cases (6/22) in the group treated with IFL and in 8% of cases (2/24) in the group treated with CO. The survival rate of the pump twin was 73% (16/22) in the IFL group and 83% (20/24) in the group treated with CO. The median gestational age at birth was 38 weeks in the group treated with IFL and 35 weeks in the group treated with CO. The rate of preterm birth before 34 weeks was 12.5% (2/16) in the group treated with IFL and 32% (7/22) in the group treated with CO. In the group treated with IFL, there were no cases of neurological disabilities reported by the parents compared to three cases in the CO group. IFL is associated with a higher risk of early pregnancy loss; however, if the pregnancy progresses, it is associated with lower risks of preterm birth and neurological disabilities in the survivors.

Management Options for Fetal Bronchopulmonary Sequestration.

To evaluate the prenatal course and perinatal outcome of fetuses with bronchopulmonary sequestration (BPS) managed expectantly or using minimally invasive methods. This was a retrospective study of 29 fetuses with suspected BPS managed between 2010 and 2021 in three fetal medicine centers in Poland. Medline was searched to identify cases of BPS managed expectantly or through minimally-invasive methods. In 16 fetuses with BPS, there was no evidence of cardiac compromise. These fetuses were managed expectantly. Thirteen hydropic fetuses with BPS qualified for intrauterine intervention: a thoraco-amniotic shunt (TAS) was inserted in five fetuses, laser coagulation of the feeding vessel was performed in seven cases, and one fetus had combined treatment. In the combined data from the previous and the current study of various percutaneous interventions for BPS associated with hydrops, the survival rate was 91.2% (31/34) for TAS, 98.1% (53/54) for laser coagulation, and 75% (3/4) for intratumor injection of sclerosant. After taking into account cases with available data, the rate of preterm birth before 37 weeks in the group treated with laser coagulation was 14.3% (7/49) compared to 84.6% (22/26) in the group treated with TAS. The need for postnatal sequestrectomy was lower in the group of fetuses treated with laser coagulation 23.5% (12/51) in comparison to fetuses treated with TAS 84% (21/26). In fetuses with BPS without hydrops, progression of the lesion's volume, leading to cardiac compromise, is unlikely. In hydropic fetuses with BPS, intrauterine therapy using minimally invasive methods prevents fetal demise. Both, the rate of preterm birth and the need for postnatal surgery is significantly lower in the group treated with laser coagulation compared to the group treated with TAS.

Long-Term Amnioinfusion through an Intrauterine Catheter in Preterm Premature Rupture of Membranes before 26 Weeks of Gestation: A Retrospective Multicenter Study.

INTRODUCTION: The objective of this study was to elucidate the efficacy of long-term amnioinfusion on perinatal outcomes in patients with preterm premature rupture of membranes (PPROM) before 26 weeks' gestation. MATERIAL: A total of 31 patients with PPROM at a periviable gestational age (21 + 0-25 + 0 weeks) were enrolled. Long-term amnioinfusion was performed in 22 patients, and 9 patients did not receive amnioinfusion. Data were collected retrospectively from 2 clinical sites between January 2017 and March 2019. RESULTS: In the medical management group, there was a significantly higher rate of chorioamnionitis compared to the long-term amnioinfusion group (89 vs. 15%, p = 0.001). The latency period between PPROM and delivery was higher in the amnioinfusion group (median, 5.5 vs. 3 weeks, p = 0.04). The frequency of bronchopulmonary dysplasia was higher in the control group compared to the amnioinfusion group (89 vs. 40%, p = 0.03). The rates of other neonatal complications were similar in both groups. CONCLUSIONS: Long-term amnioinfusion through an intrauterine catheter in PPROM before 26 weeks' gestation may improve pregnancy and newborn outcomes.

A new procedure for the determination of 21 macro- and trace elements in human fetal urine using an inductively coupled plasma mass spectrometry with dynamic reaction cell (ICP-DRC-MS) equipped with a micro-flow nebulizer.

The procedure for determination of 21 macro- and trace elements - Li, Na, Mg, Al, K, Ca, V, Cr, Mn, Fe, Co, Cu, Zn, Se, Sr, As, Cd, Sb, Ba, Pb and U - in human fetal urine by inductively coupled plasma mass spectrometry (ICP-MS) was developed and validated. The application of a micronebulizer and a dynamic reaction cell (DRC) allowed to perform a full analysis of small volumes (200 µL) of urine collected from human fetuses without the need for sample digestion with closed microwave systems. The procedure and ICP-MS instrument was thoroughly optimized in order to reliably determine both macroelements and ultra-trace concentrations of elements. The internal standard method (Ge, Rh and Tb) was applied in order to encompass signal drift and non-spectral interferences. The rules of metrology were used in order to ensure the quality of the results: (1) the procedure was validated, (2) the uncertainty of the measurement results was estimated and (3) the traceability of the measurement result was established by using the certified reference material with matching matrix (Seronorm Trace Elements Urine L-1). Also, the analyte addition method to the artificial urine was employed for additional confirmation of trueness of the procedure. The selected parameters of the procedure were as follows: (a) limits of detection - (0.00023-53 µg L-1) for U and Ca, respectively, (b) recoveries of the reference value - 81%-136% for Mn and Cd, respectively (c) linearity expressed as R - greater than 0.999, and (d) expanded relative uncertainties (k = 2) - 13%-66% for Sr and Cd, respectively. The developed and validated procedure was applied to 58 samples of urine collected from human fetuses. The samples were diluted with nitric acid and analyzed without further treatment. The procedure allowed to reliably determine both macro- and trace elements in very low volume of sample in a single analytical run.

Percutaneous Intratumor Laser Ablation for Fetal Sacrococcygeal Teratoma.

OBJECTIVE: To evaluate the efficiency of percutaneous intratumor laser ablation for fetal solid sacrococcygeal teratoma (SCT). SUBJECTS AND METHODS: We carried out percutaneous ultrasound-guided intratumor laser ablation through a 17-gauge needle using an output of 40 W in 7 fetuses with large solid SCT and reviewed the literature for minimally invasive therapy for this condition. RESULTS: Laser ablation was carried out at a median gestational age of 20 (range 19-23) weeks, and in all cases there was elimination of obvious vascularization within the tumor and improvement in cardiac function. Three (43%) babies survived and had surgical excision of the tumor within 2 days of birth, 3 liveborn babies died within 5 days of birth and before surgery, and 1 fetus died within 2 weeks after the procedure. In previous series of various percutaneous interventions for predominantly solid SCT the survival rate was 33% (2/6) (95% CI 9.7-70%) for endoscopic laser to superficial vessels, 57% (4/7) (95% CI 25-84%) for intratumor laser, 67% (8/12) (95% CI 39-86%) for intratumor radiofrequency ablation, and 20% (1/5) (95% CI 3.6-62%) for intratumor injection of alcohol. CONCLUSIONS: In solid SCT, the reported survival from intratumor laser or radiofrequency ablation is about 50%, but survival does not mean success, and it remains uncertain whether such interventions are beneficial or not because the number of fetuses is small and there were no controls that were managed expectantly.

A single-center study to evaluate the efficacy of a fetal urine peptide signature predicting postnatal renal outcome in fetuses with posterior urethral valves.

BACKGROUND: Posterior urethral valves (PUVs) account for 17% of pediatric renal failure. The management of pregnancies involving fetuses with PUV is hampered by the fact that current clinical parameters obtained from fetal ultrasound and/or fetal urine biochemistry are insufficient to predict postnatal renal function. We previously have developed a fetal urine peptide signature (12PUV) that predicted with high precision postnatal renal failure at 2 years of age in fetuses with PUV. Here, we evaluated the accuracy of this signature to predict postnatal renal outcome in fetuses with PUV in an independent single-center study. METHODS: Thirty-three women carrying fetuses with suspected PUV were included. Twenty-five fetuses received vesicoamniotic shunts during pregnancy. PUV was confirmed postnatally in 23 patients. Of those 23 fetuses, 2 were lost in follow-up. Four and 3 patients died in the pre- and perinatal periods, respectively. Follow-up renal function at 6 months of age was obtained for the remaining 14 patients. The primary outcome was early renal failure, defined by an eGFR < 60 mL/min/1.73 m2 before 6 months of age or pre- or perinatal death. RESULTS: The peptide signature predicted postnatal renal outcome in postnatally confirmed PUV fetuses with an AUC of 0.94 (95%CI 0.74-1.0) and an accuracy of 90% (95%CI 78-100). The signature predicted postnatal renal outcome for the suspected PUV cases with an AUC of 0.89 (95%CI 0.72-0.97) and an accuracy of 84% (95%CI 71-97). CONCLUSIONS: This single-center study confirms the predictive power of the previously identified 12PUV fetal urinary peptide signature.

Neonatal survival and kidney function after prenatal interventions for obstructive uropathies.

OBJECTIVES: Prenatal interventions in LUTO (lower urinary tract obstruction) usually are still question of a debate between gynaecologist and paediatric nephrologist. We aimed the study to assess the early survival rate and renal outcome in LUTO foetuses. MATERIAL AND METHODS: The study was a prospective data analysis of 39 foetuses from singleton pregnancies. All pregnant women with LUTO in the foetus were qualified for VAS based on a local practice. The mean time of first urine analysis ranged between 13-30 weeks of pregnancy. Primary end-point analysis included live birth, 28d-survival, pulmonary and renal function assessment in neonatal period. RESULTS: From initial number of 39, six patients miscarried before the procedure was performed. Overall, 33 VAS were performer at the mean 21 week of pregnancy (range 14-30 weeks). 25/39 foetuses survived until delivery. Three neonates died in first 3 days of life. In the first month 3 children required peritoneal dialysis, but at 28 day all children were dialysis-free. Overall survival rate at 28 day was 56%. Renal function preservation of the initial group (39) turned out to be low - 18% (7/39). CONCLUSIONS: Our study showed average survival curves and complications. LUTO in the foetus had mostly unfavourable outcome in the neonatal period. The prenatal intervention did not increase it significantly and did not guarantee the preservation of normal kidney function.

Ventriculo-amniotic shunting for severe fetal ventriculomegaly.

INTRODUCTION: Severe fetal cerebral ventriculomegaly, observed in about 1 in 1000 newborns, is associated with a high risk of perinatal death and neurodevelopmental delay in survivors. The objective of this study was to evaluate the efficiency of ventriculo-amniotic shunting for drainage of severe fetal cerebral ventriculomegaly and the neurodevelopment of survivors at the age of 2 years. MATERIAL AND METHODS: This was a retrospective study of 44 fetuses with severe bilateral ventriculomegaly treated with ventriculo-amniotic shunting in a tertiary fetal therapy center between 2010 and 2015. RESULTS: Shunt insertion was successfully carried out at a median gestational age of 25 weeks (range 20-33 weeks). There were three fetal deaths within 24 hours of the procedure and 41 live births at a median gestational age of 37 weeks (range 28-39 weeks). Neurodevelopment at 2 years of age was evaluated using the Bayley scale in the 38 survivors. In the 27 cases with isolated ventriculomegaly 19 (70.4%; 95% confidence interval [95% CI] 51.5%-84.2%) had normal or mild neurodevelopmental delay and 8 (29.6%; 95% CI 15.6%-48.5%) were moderately or severely delayed. In the 11 with non-isolated ventriculomegaly 2 (18.2%; 95% CI 5.1%-4.8%) had normal or mild neurodevelopmental delay and 9 (81.8%; 95% CI 52.3%-94.9%) babies were moderately or severely delayed. CONCLUSIONS: Ventriculo-amniotic shunting is an option for the management of severe ventriculomegaly and results in normalization of the ventricular diameter. However, a high proportion of survivors have neurodevelopmental delay and the possible beneficial effect of ventriculo-amniotic shunting needs to be assessed by randomized studies.

Congenital cystic adenomatoid malformation - diagnostic and therapeutic procedure: 8-year experience of one medical centre.

INTRODUCTION: Congenital cystic adenomatoid malformation (CCAM) is a rare anomaly. The mechanisms and the time at which the abnormality develops are still unclear. The malformation is characterized by the presence of single large or multiple but smaller cysts. AIM: To present the experience of our medical centre, the Polish Mother's Memorial Hospital - Research Institute. MATERIAL AND METHODS: We analysed the medical records of 32 neonates hospitalized in 2008-2017 at the Department of Paediatric Surgery and Urology ICZMP due to pre- or postnatally diagnosed congenital cystic adenomatoid malformation. RESULTS: In 2008-2017 32 children were hospitalized at our department due to prenatally diagnosed CCAM. An intrauterine procedure of thoracoamniotic shunting was performed in 13 fetuses. All newborns had chest computed tomography. Surgery had been performed in 21 children. Histopathological analysis of specimens prepared from the resected lung tissue confirmed type 1 CCAM in 4 cases, type 2 CCAM in 11 cases and type 3 CCAM in 1 case. Four patients were diagnosed with a bronchogenic cyst and one with extralobar pulmonary sequestration. CONCLUSIONS: Postnatal management of CCAM depends on the clinical status of the newborn. Newborns without clinical symptoms require radiological diagnostics to confirm the presence of malformation. Surgical treatment is recommended before the age of 10 months. Resection of the malformed lung fragment in patients with CCAM is the first-line treatment. It should be emphasized that despite the tremendous advances in diagnostic imaging, the final diagnosis of CCAM is made based on histopathological findings.

Intake of folic acid by Polish women with higher education - a survey research: can we do more?

OBJECTIVES: The objective of the study is to determine efficacy of the Primary Prevention Program of Neural Tube Defects in Polish women with higher education in 5-year interval. MATERIAL AND METHODS: Survey research was conducted twice (in 2008 and 2013) in 630 female students of universities: 305 female medical students and 325 female non-medical students. The survey was also done among women aged 27- 35 who graduated from medical or non-medical universities and have at least one child. Questions concerned knowledge about prophylaxis and periconceptional folic acid intake. Chi square test was used to assess the significance. RESULTS: Knowledge concerning prophylaxis was significantly higher in female medical students comparing to non-medical ones, both in 2008 (p < 0,001), and in 2013 (p < 0,001). 92.9% in 2008 and 93.9% in 2013 of medical students knew about the necessity of periconceptional folic acid intake. Awareness of female non-medical students was lower (2008 - 35.3% and 2013 - 41.1%) and did not change in the 5-year long period (p = 0.3). There was no significant difference in preconceptional folic acid intake among mothers with medical and non-medical education (53.3% vs. 45% p = 0.4). However, the highest folic acid intake was among mothers -medical doctors who treat children with neural tube defect. CONCLUSIONS: Difference between medical and non-medical students shows that better educational programs may improve knowledge about prophylaxis. Aside from knowledge, compliance with recommendations of Primary Prevention Program of Neural Tube Defects is unsatisfactory.

Combined screening for early and late pre-eclampsia and intrauterine growth restriction by maternal history, uterine artery Doppler, mean arterial pressure and biochemical markers.

BACKGROUND: Pre-eclampsia is a systemic disease connected with high maternal and fetal morbidity and mortality. Despite significant progress achieved in perinatal medicine, pre-eclampsia is still one of the most significant current problems in obstetrics. OBJECTIVES: The aim of the study was to establish diagnostic algorithms for early and late pre-eclampsia (PE) and intrauterine growth restriction (IUGR). MATERIAL AND METHODS: A total of 320 pregnant women between 11 + 0 and 13 + 6 weeks of gestation were recruited for a case-control study. The study group consisted of 22 patients with early PE, 29 patients with late PE and 269 unaffected controls. The following parameters were recorded: maternal history, mean arterial pressure (MAP), mean uterine artery pulsatility index (UtA-PI), and the concentrations of placental growth factor (PlGF), pregnancy-associated plasma protein A (PAPP-A) and free beta-human chorionic gonadotropin (free ß-hCG). RESULTS: A multivariable stepwise logistic regression analysis indicated that the best screening model for the prediction of early PE is based on a combined analysis of maternal risk factors, UtA-PI and PlGF levels (sensitivity: 91%; specificity: 84%). The best screening model for the prediction of late PE is based on a combined analysis of maternal risk factors, UtA-PI and MAP (sensitivity: 85%; specificity: 83%). The most effective screening model for the prediction of IUGR is based on a combined analysis of maternal risk factors, UtA-PI and PlGF concentrations (sensitivity: 91%; specificity: 83%). CONCLUSIONS: The integrated model of screening established in this study can be a valuable method to identify patients at increased risk of developing pre-eclampsia and related complications. The ability to predict the occurrence of pre-eclampsia in early pregnancy would enable maternal and fetal morbidity to be reduced through the introduction of strict obstetric surveillance as well as planned delivery in a reference center.

Thoracoamniotic Shunts in Macrocystic Lung Lesions: Case Series and Review of the Literature.

OBJECTIVE: To evaluate the efficiency of thoracoamniotic shunts for drainage of macrocystic-type congenital cystic adenomatoid malformation (CCAM). SUBJECTS AND METHODS: This was a retrospective study of 12 fetuses with a large thoracic cyst treated with thoracoamniotic shunting between 2004 and 2014 in a tertiary fetal therapy center. Medline was searched to identify cases of CCAM treated with thoracoamniotic shunting. RESULTS: In all cases the thoracic cyst was associated with major mediastinal shift, the CCAM volume ratio (CVR) was >1.6, and in eight cases there was associated hydrops. Shunt insertion was successfully carried out in all cases at a median gestational age of 24 weeks (range 18-34). In 10 cases there was live birth at a median age of 38 weeks (range 35-41), but in two hydropic fetuses there was intrauterine death. A literature search identified a total of 98 fetuses with CCAM treated with thoracoamniotic shunting between 1987 and 2016. In the combined data from the previous and the current study, the survival rate was 77% (53 of 69) for hydropic and 90% (37 of 41) for nonhydropic fetuses. CONCLUSIONS: The role of thoracoamniotic shunting in macrocystic lung lesions associated with hydrops is well accepted. Intrauterine intervention is also likely to be beneficial in the subgroup of nonhydropic fetuses with a CVR >1.6.

Difficulties in prenatal diagnosis of tumour in the fetal sacrococcygeal area.

Prenatal ultrasound at the 20th week of gestation revealed a 3-cm tumour in the sacrococcygeal area. Initially, a sacrococcygeal teratoma was suspected on the basis of fetal ultrasonography, which revealed normal heart anatomy and an increasing tumour mass. The diagnosis was then changed to fetus in fetu or teratoma. Prenatal magnetic resonance imaging at the 34th week of pregnancy confirmed the ultrasound diagnosis. No other anomalies were found. Elective caesarean section was performed at term. The care team included a paediatric surgeon, obstetricians, neonatologists, midwives, and an anesthesiologist. A female newborn was delivered in good condition. The tumour was resected in the operating room and mature teratoma was established by histopathological evaluation. Surprisingly, agenesis of the right forearm was revealed which had not been detected prenatally, despite many examinations (both in our hospital and earlier at a primary care obstetrician office).

Wspólczesne metody monitorowania dobrostanu plodu w ciazy powiklanej wewnatrzmacicznym zahamowaniem wzrastania.

Intrauterine growth restriction (IUGR) is one of the most important problems in current perinatology. The number of complications such as intrauterine fetal hypoxia, preterm and operative labours, intrauterine demises and neonatal deaths are signifcantly higher among pregnant women with IUGR. The proper monitoring and assesement of the fetal well-being are crucial to make the right decision about optimal time and mode of delivery.

Biochemical markers in screening for preeclampsia and intrauterine growth restriction.

OBJECTIVES: The aim of the study was to evaluate the relationship between the concentrations of substances released by the placenta: placental growth factor (PIGF), pregnancy-associated plasma protein A (PAPP-A) and free beta-human chorionic gonadotropin (beta-hCG) and the risk of early and late preeclampsia (PE) and intrauterine fetal growth restriction (IUGR). MATERIAL AND METHODS: A total of 180 pregnant women between 11+0 and 13+6 weeks gestation were recruited for a case-control study Twenty-two patients suffered from early PE, 29 patients from late PE. Data analyzed during the study included maternal histoty and concentrations of PAPP-A, PlGF beta-hCG. RESULTS: The multiple of the median (MoM) value of the PAPP-A concentrations was 1.01 in the control group (interquartile range (1QR), 0.65-1.55), 0.67 (1QR, 0.382-0.82) in the group of patients with early preeclampsia and 0.74 (IQ, 0.33-1.09) in the group of patients suffering from late preeclampsia. MoM value of the PIGF concentrations was 1.21 in the control group (IQR, 0.93-1.57), 0.62 (IQR, 0.51-0.96) in the group of patients with early preeclampsia and 0.92 (lQR, 0.63-1.09) in the group of patients suffering from late preeclampsia. MoM value of beta-hCG concentrations was 1.14 in the control group (IQR, 0.75-1.49), 1.08(IQR, 0.74-1.23) in the group of patients with early preeclampsia and 1,25(IQR, 1,05-1,49) in the group of patients suffering from late preeclampsia. The performance of screening was determined by the areas under the curve and detection rates, with a fixed false-positive rate of 10%. CONCLUSIONS: Decreased levels of PAPP-A and PIGF are related to an increased risk of preeclampsia and its complications.

[The diagnostic algorithm in twin pregnancy]. / Schemat diagnostyczny w ciazy blizniaczej.

This paper presents the diagnostic algorithm in twin pregnancy. The most important sonographic parameters in the assessment of twins have been discussed. Moreover, the most significant complications of twin pregnancy as well as diagnostic possibilities and management, have been also presented and defined.

[Fetal therapy--evaluation of intrauterine therapy in congenital cystic adenomatoid malformation of the lung (CCAM)]. / Terapia plodu--ocena skutecznosci leczenia wewnatrzmacicznego wrodzonego zwyrodnienia gruczolakowato-torbielowatego pluc (CCAM).

OBJECTIVES: The aim of the study was to evaluate the efficiency of intrauterine treatment of large cysts in fetal lungs using thoracoamniotic shunts. MATERIAL AND METHODS: Our observational retrospective study was carried out on a series of 8 fetuses who under went thoracoamniotic shunting after sonographic statement of large macrocystic lesions in the lungs at the Department of Gynecology Fertility and Therapy of the Fetus, Polish Mother's Research Institute, between 2009-2014. RESULTS: Mean gestational age at shunt insertion was 26.6 (range 18-33) weeks. Marked mediastinal shift in the echocardiographic examination was observed in all of the investigated cases. Five fetuses had polyhydramion, with 4 hydropic cases. Out of the remaining 4 fetuses without impaired cardiac function, 3 had very large lesions at initial presentation and 1 had a lesion that was rapidly increasing in size. Shunt insertion was successful in all cases. Only one patient went into premature labor (at 36 weeks of gestation). Mean gestational age at delivery was 38.2 weeks. Cesarean section was necessary in the half of the patients due to obstetric complications. All newborns underwent resection of the lesions. Three of them were operated in the first month after birth. The rest of the operations were postponed. Prenatal diagnosis of congenital cystic adenomatoid malformations was confirmed by pathologists in all cases. CONCLUSION: Intrauterine therapy of macrocystic lesions in fetal lungs enables to achieve good perinatal outcome It needs to be considered in every case of a fetus with developing impaired cardiac function.