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To compare the outcomes of dichorionic triamniotic (DCTA) triplets with that of monochorionic diamniotic (MCDA) twin gestations undergoing fetoscopic laser surgery (FLS) for treatment of twin-to-twin transfusion syndrome (TTTS).Retrospective cohort study of prospectively collected data of consecutive DCTA triplet and MCDA twin pregnancies with TTTS that underwent FLS at two fetal treatment centers between 2012 and 2020. Preoperative, operative and, postoperative variables were collected. Perinatal outcomes were investigated. Primary outcome was survival to birth and to neonatal period. Secondary outcomes were gestational age (GA) at birth and procedure-to-delivery interval. Literature review was conducted in which PubMed, Web of Science, and Scopus were searched from inception to September, 2020.Twenty four sets of DCTA triplets were compared to MCDA twins during the study period. There were no significant differences in survival (no survivor, single, or double survivors) to birth and to the neonatal period of the MC twin pairs of the DCTA tripletsPerinatal survival including fetal and neonatal are comparable between DCTA triplets and MCDA twins. However, this might have resulted from the small sample size of the DCTA triplets. GA at delivery is earlier in triplets, which could be due to the nature of triplet gestation rather than to the laser procedure itself.

Fetal anemia secondary to incompatibility between maternal-fetal blood types can result in hydrops and demise. Intrauterine transfusions have improved survival in experience centers. Our objective was to determine the practice patterns amongst fetal centers.Thirteen fetal centers across the world were surveyed. Results from all participating centers were recorded, analyzed, and presented as ratios. Questions on the survey were related to experience of the physician, preferred methods of transfusion, fetal surveillance, and timing of delivery.Differences amongst centers were as follows: 54% of the centers performed transfusions in operating room, the remaining did them in a clinic room or close to the operating room; 31% did not use maternal anesthesia, 31% used oral or intravenous sedation and 38% used a combination of local with oral or intravenous sedation. The similarities include: 84% performed intravenous transfusions, while 2 centers reported intraperitoneal and intracardiac transfusions were performed for very early cases; 85% of centers performed the last transfusion at 34-35 weeks and 77% electively delivered their patients at 37 weeks.Method of transfusion and delivery timing was similar in most centers; however, differences were seen in location of procedure, anesthetic coverage, and surveillance. Further assessment is needed to determine if these differences in practice have any potential neonatal effects.

Introduction: To characterize the changes in fetal lung volume following endoluminal tracheal occlusion (FETO) that are associated with infant survival and need for extracorporeal membrane oxygenation (ECMO) in congenital diaphragmatic hernia (CDH). Methods: Fetuses with CDH who underwent FETO at a single institution were included. CDH cases were reclassified by MRI metrics, [observed-to-expected total lung volume (O/E TLV) and percent liver herniation]. The percent changes of MRI metrics after FETO were calculated. ROC-derived cutoffs of these changes were derived to predict infant survival to discharge. Regression analyses were done to determine the association between these cutoffs with infant survival and ECMO need, adjusted for site of CDH, gestational age at delivery, fetal sex, and CDH severity. Results: Thirty CDH cases were included. ROC analysis demonstrated that post-FETO increases in O/E TLV had an area under the curve of 0.74 (p=0.035) for the prediction of survival to hospital discharge; a cutoff of less than 10% was selected. Fetuses with a

To compare the perinatal outcomes of pregnancies complicated by chorioamniotic separation (CAS) vs septostomy following fetoscopic laser photocoagulation (FLP) for twin-twin transfusion syndrome (TTTS).This was a retrospective cohort analysis of monochorionic diamniotic twin pregnancies with TTTS that underwent FLP at one of two university-affiliated tertiary medical centers between January 2012 and December 2020. CAS and septostomy were diagnosed either during the procedure or by ultrasonography within 24-48 h after FLP. Data on procedure and postprocedure parameters, pregnancy outcomes and survival were collected from the patients' electronic medical records. Pregnancies were stratified according to the presence of CAS, septostomy or neither. Patients diagnosed with both CAS and septostomy were analyzed separately.Of the 522 women included in the cohort, 38 (7.3%) were diagnosed with CAS, 68 (13.0%) with septostomy and 23 (4.4%) with both CAS and septostomy. The remaining 393 (75.3%) women comprised the control group. Groups did not differ in demographic characteristics. The septostomy group had a lower rate of selective fetal growth restriction than did the CAS and control groups (24.2% vs 36.8% vs 42.7%, respectively; P = 0.017). Moreover, intertwin size discordance was lower in the septostomy group (15.1% vs 23.4% in the CAS group and 25.5% in the control group; P = 0.001). Median gestational age at FLP was significantly lower in the CAS group (19.3 weeks vs 20.4 weeks in controls and 20.9 weeks in the septostomy group; P = 0.049). The rate of delivery prior to 34 weeks was significantly higher in the CAS group (89.2%), followed by the septostomy group (80.9%), compared with the control group (69.0%) (P = 0.006). A secondary analysis demonstrated that patients with both CAS and septostomy presented the highest rates of delivery prior to 34 weeks (100%) and 32 weeks (68.2%).CAS and septostomy following laser surgery for TTTS are independently associated with higher rates of preterm delivery. The presence of these two findings in the same patient enhances the risk of prematurity. © 2021 International Society of Ultrasound in Obstetrics and Gynecology.

Introduction: Without fetal or perinatal intervention, congenital high airway obstruction syndrome (CHAOS) is a fatal anomaly. The ex utero intrapartum treatment (EXIT) procedure has been used to secure the fetal airway and minimize neonatal hypoxia but is associated with increased maternal morbidity. Case Presentation: A 16-year-old woman (gravida 1, para 0) was referred to our hospital at 31 weeks gestation with fetal anomalies, including echogenic lungs, tracheobronchial dilation, and flattened diaphragms. At 32 weeks, fetoscopic evaluation identified laryngeal stenosis, which was subsequently treated with balloon dilation and stent placement. The patient developed symptomatic and regular preterm contractions at postoperative day 7 with persistent sonographic signs of CHAOS, which prompted a repeat fetoscopy with confirmation of a patent fetal airway followed by Cesarean delivery under neuraxial anesthesia. Attempts to intubate through the tracheal stent were limited and resulted in removal of the stent. A neonatal airway was successfully established with rigid bronchoscopy. Direct laryngoscopy and bronchoscopy confirmed laryngeal stenosis with a small tracheoesophageal fistula immediately inferior to the laryngeal stenosis and significant tracheomalacia. A tracheostomy was then immediately performed for anticipated long-term airway and pulmonary management. The procedures were well tolerated by both mom and baby. The baby demonstrated spontaneous healing of the tracheoesophageal fistula by day of life 7 with discharge home with ventilator support at 3 months of life. Conclusion: Use of repeated fetoscopy in order to relieve fetal upper airway obstruction offers the potential to minimize neonatal hypoxia, while concurrently decreasing maternal morbidity by avoiding an EXIT procedure. Use of the tracheal stent in CHAOS requires further investigation. The long-term reconstruction and respiratory support of children with CHAOS remain challenging.

Objective To evaluate laparoscopy-assisted fetoscopic laser photocoagulation (FLPC) of placental anastomoses in the treatment of twin-to-twin-transfusion syndrome (TTTS). Study design We performed a retrospective cohort study analyzing pregnancies complicated by TTTS who underwent FLPC in a single university-affiliated tertiary medical-center. Outcomes were compared between patients who received laparoscopy-assisted FLPC (study group) and patients who underwent the conventional FLPC technique (control group). Baseline characteristics, sonographic findings, procedure details, and neonatal outcomes were compared between groups. Results The cohort included 278 women with 31 in the study group and 247 in the control group. Sonographic parameters, including fetal biometry and TTTS stage, were comparable between study groups. Gestational age at delivery did not differ between the groups (29.22 ± 4.55 weeks in the study group vs. 30.62 ± 4.3 weeks in the control group, p = 0.09). There were no differences in neonatal survival rates at birth and at 30 days between both groups. A subanalysis comparing the laparoscopy-assisted group to only those patients with anterior placenta in the control group, showed a lower rate of incomplete Solomonization in the laparoscopy-assisted study group (3.4% vs. 33%, p = 0.01). Conclusion Laparoscopy-assisted FLPC is a reasonable and safe option that may be offered in cases of FLPC where an anterior placenta restricts adequate surgical access.

Objective Selective fetal growth restriction (sFGR) complicating twin to twin transfusion syndrome (TTTS) is associated with 3 to 6 fold increased risk of fetal demise after fetoscopic laser surgery (FLS). Identifying these patients is challenging due to varying definitions used in the literature. Our objective was to determine the association of three currently used definitions for sFGR with demise of the smaller twin, typically the donor, following FLS METHODS: Retrospective cohort of monochorionic diamniotic pregnancies undergoing FLS for TTTS between January 2015 to December 2018. The entire cohort was classified as sFGR and non sFGR using 3 different criteria: 1- International Society of Ultrasound in Obstetrics and Gynecology (ISUOG) as estimated fetal weight (EFW) of either twin 25% (Group A); 2- Delphi methods, EFW of either twin 95th centile (Group C). Pearson chi square, univariate and multivariate logistic regression analyses were performed. Results One hundred and twenty four patients underwent FLS between January 2015 and December 2018 for TTTS. 46/124 (37.1%) cases were identified as sFGR by ISUOG criteria (Group A), 57/124 (46%) cases were identified based on EFW Conclusions Fetal demise following FLS is increased in the presence of sFGR. Improving diagnosis of sFGR, will improve counseling and may affect management. Delphi method of defining sFGR based on presence of 2 of the 4 criteria had the highest predictive value of donor demise following FLS. This article is protected by copyright. All rights reserved.

To assess outcomes in TTTS according to MCA-PI prior to FLPC surgery.A retrospective cohort analysis of monochorionic-twin pregnancies complicated by TTTS who underwent FLPC at two fetal centers (2012-2021). The cohort was stratified according to abnormal MCA-PI of the donor twin, defined as below 5Abnormal MCA-PI of the donor twin was detected in 46(17.7%) cases compared to 213(83.3%) controls with no such abnormality. The abnormal PI group presented with higher rates of sFGR(56.5% vs.36.8% in controls, p=0.014) and lower donor survival rate within 48 hours after FLPC(73.9 vs.86.8%, p=0.029). Donor twin survival rates at time of delivery and 30 days after birth were lower in the abnormal MCA-PI. Multivariate logistic regression analysis controlling for sFGR and MCA-PI5Donor survival at time of delivery and 30 days after birth were lower in TTTS cases with MCA-PI5

OBJECTIVE To compare the perinatal outcomes between pregnancies with and those without iatrogenic chorioamniotic separation (iCAS) following fetoscopic intervention. METHODS We performed a search in PubMed, EMBASE, Scopus, Web of Science and Google Scholar from inception up to December 2020 for studies comparing perinatal outcomes between pregnancies that developed and those that did not develop iCAS after fetoscopic intervention for twin-to-twin transfusion syndrome (TTTS), open neural tube defect (ONTD) or congenital diaphragmatic hernia. A random-effects model was used to pool the mean differences (MD) or odds ratios (OR) and the corresponding 95% CI. The primary outcome was neonatal survival. Secondary outcomes included gestational age (GA) at intervention and at delivery, interval from intervention to delivery and incidence of preterm prelabor rupture of membranes (PPROM) and preterm delivery. The methodological quality of the included studies was evaluated using the Newcastle-Ottawa scale. RESULTS The search identified 348 records, of which seven studies (six on fetoscopic laser photocoagulation (FLP) for TTTS and one on fetoscopic repair for ONTD) assessed the perinatal outcomes of pregnancies that developed iCAS after fetoscopic intervention. Given that only one study reported on fetoscopic ONTD repair, the meta-analysis was limited to TTTS pregnancies and included six studies (total of 1881 pregnancies). Pregnancies that developed iCAS after FLP for TTTS, compared with those that did not, had significantly lower GA at the time of intervention (weeks) (MD, -1.07 (95% CI, -1.89 to -0.24); P = 0.01) and at delivery (weeks) (MD, -1.74 (95% CI, -3.13 to -0.34); P = 0.01) and significantly lower neonatal survival (OR, 0.41 (95% CI, 0.24-0.70); P = 0.001). In addition, development of iCAS after FLP for TTTS increased significantly the risk for PPROM

To evaluate the association of laser photocoagulation of placental anastomoses (LPA) prior to 18 weeks' gestation (early LPA) with very preterm delivery and neonatal survival in pregnancies with twin-twin transfusion syndrome (TTTS).This was a retrospective cohort study of monochorionic diamniotic twin pregnancies with TTTS undergoing LPA between 2002 and 2018 at two institutions. The rates of delivery 28, 30 and 32 weeks' gestation, preterm prelabor rupture of membranes (PPROM) and 30-day survival of one or both infants were compared between pregnancies undergoing early LPA and those undergoing LPA ≥ 18 weeks' gestation. Regression analysis was performed to determine the association of early LPA with very preterm delivery and 30-day survival, adjusted for Quintero stage, study phase, selective fetal growth restriction, gestational age at delivery, maternal age ≥ 35 years, body mass index 35 kg/mA total of 414 TTTS pregnancies were included in the study, of which 68 (16.4%) underwent early LPA. In the total cohort, the incidence of delivery at 28, 30 and 32 weeks' gestation was 22.7%, 39.6% and 53.4%, respectively. Survival of both twins and survival of at least one twin at 30 days were 67.5% and 90.8%, respectively. No significant difference was noted between pregnancies that underwent early LPA and those that had LPA ≥ 18 weeks in the rate of delivery 28 weeks (19.1% vs 23.4%; P = 0.4), 30 weeks (38.2% vs 39.9%; P = 0.8) and 32 weeks (44.1% vs 55.2%; P = 0.1) and PPROM (29.0% vs 24.1%; P = 0.4), or in the incidence of double-twin survival (63.9% vs 68.1%; P = 0.5) and survival of at least one infant (91.8% vs 90.6%; P = 0.7) at 30 days. Early LPA was not associated with very preterm delivery or neonatal survival in the regression analyses. Early LPA was associated with a longer LPA-to-delivery interval compared with LPA performed ≥ 18 weeks (median, 106.9 days (range, 2-164 days) vs median, 69.3 days (range, 0-146 days); P 0.001) when adjusted for confounding variables (hazard ratio, 2.56 (95% CI, 1.76-3.73); P 0.001).Laser surgery before 18 weeks is not associated with an increased rate of very preterm delivery and PPROM or with reduced neonatal survival when compared with LPA after 18 weeks. © 2020 International Society of Ultrasound in Obstetrics and Gynecology.

OBJECTIVE To determine the impact of the lesion type (cystic [myelomeningocele] or flat [myeloschisis]) on the fetal motor function (MF) in cases candidates for prenatal open neural tube defect (ONTD) repair. METHODS Retrospective cohort study of patients with ONTD who underwent prenatal repair at a single institution between 2011 and 2019. The lesion type and the measurements of the length and width of the lesions to calculate the surface of the ellipsoid lesion were performed using MR scans. Prenatal MF of the lower extremities was evaluated by ultrasound following a metameric distribution at the time of referral. Intact MF was defined as the observation of plantar flexion of the ankle. Logistic regression was performed to determine the predictive value of the type of lesion for having an intact MF at the time of referral. RESULTS 103 patients were included at 22.9 (19-25.4) weeks; 65% had cystic and 35% had flat lesions. At the time of referral, there was a higher proportion of cases with an intact MF in the presence of flat lesions (34/36; 94.4%) as compared to cystic lesion (48/67; 71.6%, p

In utero repair of open neural tube defect (ONTD) using an open hysterotomy approach (hereafter referred to as "Open") has been shown to reduce the need for ventriculo-peritoneal shunting and to improve motor outcomes for affected infants. Laparotomy-assisted fetoscopic repair (hereafter referred to as "Hybrid") is an alternate approach that may confer similar neurological benefits, while reducing the incidence of hysterotomy related complications. In this study we aimed to analyze procedure related maternal and fetal complications of in-utero repair using the Clavien-Dindo classification, and to compare the outcomes of the Hybrid and Open approaches.This was a retrospective cohort study conducted in a single center between September 2011 and July 2021. All patients who met the MOMS criteria and who underwent either Hybrid or Open fetal surgery were included, Maternal complications were classified using a unique adapatation of the Clavien-Dindo scoring system allowing the development of a comprehensive complication index score specific to fetal surgery. Primary fetal outcome was defined as gestational age at delivery and summarized according to the World Health Organization definitions of preterm delivery.There were 146 fetuses with ONTD who were eligible for, and underwent, in utero repair during the study period. Of these, 102 underwent Hybrid fetoscopic repair and 44 underwent Open hysterotomy repair. Gestational age at the time of surgery was higher in the Hybrid group when compared with the Open group (25.1 vs. 24.8 weeks, p=0.004). Maternal body mass index was lower in the Hybrid compared to the Open group (25.4 vs 27.1 kg\mUtilization of our adaptation of the standardized Clavien-Dindo classification to assess the maternal complications associated with in utero ONTD repair provides a new method to objectively assess different fetal surgical approaches. It also provides a much-needed standardized tool to allow objective comparisons between methods, which can be used when counseling patients. The Hybrid ONTD repair was associated with a lower rate of maternal adverse events than the Open approach, and was associated with later gestational age at delivery and less preterm delivery.

In fetuses with severe congenital diaphragmatic hernia, fetal endoluminal tracheal occlusion (FETO) with balloon increases survival and reduces morbidity. Balloon removal is often scheduled electively. In urgent cases, in-utero removal is impossible and removal immediately after delivery has to occur, posing risk of death from airway obstruction. Medical staff need training in urgent removal. Ideal training method is unclear; thus, we compared the performance of two groups trained by different methods.24 medical students were randomly assigned to two different learning methods for removal: Group 1 (in-person lecture) and Group 2 (online video). Both methods presented the same information: endoscopic instrument set-up, anatomical landmarks for intubation, and balloon removal. All participants were evaluated using the same instruments and high-fidelity simulator, comparing time for instrument set-up and simulate balloon removal (including removal attempts).Group 1 took significantly less time for instrument set-up compared to Group 2 [62 (30-92) secs vs 81 (57-108) secs; p 0.01)]; no difference in time to intubate and locate the balloon [75 (50-173) secs vs 92 (32-232) secs; p 0.42], or number of attempts.There was no difference between video training and in-person training with regards to the time taken to locate the FETO balloon in the trachea and to simulate its removal.

Objectives To evaluate the association of intertwin differences in umbilical artery pulsatility index (DUAPI) and infant survival in twin-to-twin transfusion syndrome (TTTS). Methods Absolute DUAPI was calculated prior to laser surgery. Receiver-operating characteristics (ROC) curve analysis provided an intertwin DUAPI cutoff of 0.4 for the prediction of double twin survival to 30 days of life. Infant survival was compared between women with an intertwin DUAPI Results In total, 349 TTTS cases were included. Double twin survival to 30 days was observed in 67% (234/349) of cases. Significant differences in double twin survival was seen between intertwin DUAPI groups in the whole cohort (76.8 vs. 52.2%; p Conclusions Small intertwin DUAPI is associated with increased double infant survival in early and advanced TTTS stages.

To investigate the association of the myelomeningocele (MMC) volume with prenatal and postnatal motor function (MF) in cases who underwent a prenatal repair. Retrospective cohort study (11/2011 to 03/2019) of 63 patients who underwent a prenatal MMC repair (37 fetoscopic, 26 open-hysterotomy). At referral, measurements of the volume of MMC was performed based on ultrasound scans. A large MMC was defined as greater than the optimal volume threshold (ROC analysis) for the prediction of intact MF at referral (2.7 cc). Prenatal or postnatal intact motor function (S1) was defined as the observation of plantar flexion of the ankle based on ultrasound scan or postnatal examination. 23/63 participants presented a large MMC. Large MMC lesions was associated with an increased risk of having clubfeet by 9.5 times (CI%95[2.1–41.8], p Trial registration: Clinicaltrials.gov NCT02230072 and NCT03794011 registered on September 3rd, 2014 and January 4th, 2019.

AIM To evaluate perinatal outcomes of incidental septostomy (IS) after laser photocoagulation for twin-to-twin transfusion syndrome (TTTS), and to compare the outcomes with those who had intact intertwin membrane. METHODS Databases such as PubMed, Web of Science, Scopus, and Embase were systematically searched from inception up to August 2020. The random-effects model was used to pool the mean difference (MD) or odds-ratio (OR) with the corresponding 95% confidence intervals (CIs). Primary outcome was incidence of preterm delivery, and preterm premature rupture of membranes (PPROM), while secondary outcomes included gestational age (GA) at intervention, GA at delivery, neonatal survival, and incidence of pseudoamniotic band syndrome (PABS). RESULTS Four studies (1442 patients) met our inclusion criteria. Postoperative identification of IS was associated with earlier GA at delivery (MD = -2.52; 95% CI: -3.22, -1.82; p

To estimate the significance of the association between mid-trimester maternal serum alpha-fetoprotein (MSAFP) level and fetal neuroanatomic findings in cases of open neural tube defect (ONTD).Retrospective study of patients referred for prenatal ONTD repair between 2012 and 2018. Cases were classified into three groups based on their MSAFP level: 1)High MSAFP (3.8MoM - n = 22), 2)Moderately high MSAFP (≤3.8 and ≥2.5MoM - n = 28), 3)Normal MSAFP (2.5MoM - n = 18). MRI scans at the time of referral were used to assess the relationship between MSAFP and: A)Type of ONTD; B) Ventriculomegaly; C) Size of the myeloschisis lesion; D) Volume of myelomeningocele; E) Anatomical level of the lesion (LL).Having a high MSAFP level was more likely to be associated ventriculomegaly at mid-gestation than a moderately high or normal MSAFP level (OR = 8.4;CI95[0.9-73.4];p = 0.05 and OR = 2.8;CI95[0.9-8.8];p = 0.07). There were no differences between the three groups regarding type of lesion, size of the myeloschisis lesion, anatomic LL, or volume of the myelomeningocele sac. Myeloschisis cases with normal MSAFP had a larger surface area when compared to myeloschisis cases with moderately high MSAFP (219.8[104.4-551] vs 155.4[38.5-502.4] mm², p = 0.04).A 2nd trimester MSAFP level3.8MoM in a fetus with ONTD is associated with mid-gestation ventriculomegaly.

Background Ex Utero Intrapartum Treatment (EXIT) is utilized for safe delivery when a baby has a compromised airway. The purpose of this retrospective study was to examine the indications and outcomes of 11 children presenting with airway occluding oropharyngeal and cervical teratomas. Methods Study of all children with an airway occluding teratoma delivered via EXIT (2001-2018) in our unit. Primary outcomes included survival and tracheostomy at discharge. Data are reported using descriptive statistics as median [range] and rate (%). Results We performed 45 EXIT procedure performed between January 2001 and April 2018. Of these, eleven were for cervical and/or upper airway teratoma. Ten (91%) cases had associated polyhydramnios, two (18%) developed nonimmune hydrops, and eight (72%) delivered preterm. Six (45.5%) were performed as an emergency. Estimated blood loss was 1000 mL (500, 1000). The neonatal mortality rate was 18% (2/11) and 33% (3/9) of the survivors were discharged with a tracheostomy. Conclusion EXIT is a reasonable option for delivery of babies with an occlusive upper airway mass. Neonatal survival depends on individualized factors but may be as high as 82% in those with teratoma. This article is protected by copyright. All rights reserved.

Objectives Our primary aim was to confirm whether intertwin discordance in ventricular strain and ductus venosus (DV) time intervals predicts twin–twin transfusion syndrome (TTTS). Secondary aims were to create gestational‐age ranges for ventricular strain in uncomplicated monochorionic diamniotic (MCDA) twin pregnancies without selective intrauterine growth restriction (sIUGR) and to characterize the relationship of ventricular strain with gestational age in MCDA twin pregnancies with sIUGR that did not develop TTTS. Methods In the period 2015–2018, we enrolled 150 MCDA twin pregnancies consecutively into this prospective, blinded study of global longitudinal left and right ventricular strain. With the observer blinded to twin pairing and pregnancy outcome, videoclips of the four‐chamber view, which had been recorded during ultrasound surveillance in the usual window for development of TTTS (16–26 completed gestational weeks), underwent offline measurement of strain. Uncomplicated MCDA twin pregnancies, without sIUGR, were used to test the association between strain, gestational age and estimated fetal weight using mixed‐effects multilevel regression. Inter‐rater reliability was tested in 208 strain measurements in 31 fetuses from pregnancies which did not develop TTTS and within‐fetus variation was assessed in 16 such fetuses, in which multiple four‐chamber views were taken on the same day. The effect of sIUGR on strain in otherwise uncomplicated MCDA twin pregnancy was analyzed. MCDA twin pregnancies were defined as ‘pre‐TTTS’ when, having been referred for TTTS evaluation, they did not satisfy Quintero staging criteria, but subsequently developed TTTS requiring laser treatment. MCDA pregnancies which did not develop TTTS comprised the ‘non‐TTTS’ group. Cardiovascular parameters measured in these cases included tissue Doppler parameters and DV early filling time as a percentage of the cardiac cycle (DVeT%). Intertwin strain and DVeT% discordance was compared between non‐TTTS and pre‐TTTS cases, matched for gestational age. Results Paired strain data were available for intertwin comparison in 127/150 MCDA twin pregnancies, comprising 14 pre‐TTTS and 113 non‐TTTS pregnancies, after exclusions. Scans were collected at a median frame rate of 97 (range, 28–220) Hz. Laser therapy was performed at a median gestational age of 20.6 (range, 17.2–26.6) weeks. There were no group differences in right (RV) or left (LV) ventricular strain discordance between 68/113 non‐TTTS and 13/14 pre‐TTTS MCDA twin pregnancies 3.6% was found in eight of 13 pre‐TTTS pregnancies. In non‐TTTS pregnancies, the estimated variability in ventricular strain within each twin during the day was high (RV, 19.7; LV, 12.9). However, within each pair (intertwin variation), variability was low (RV, 5.5; LV, 2.9). Interclass correlation reflecting the proportion of total variability represented by the variability between twin pairs was low (RV, 0.22; LV, 0.18). Both RV (P

OBJECTIVES Fetal surgery for repair of open neural tube defect (ONTD) typically results in decreased need for a ventriculoperitoneal shunt (VPS). Our objectives were to determine the trend in ventricle size (VS) during pregnancy and whether VS and change in VS, as assessed by ultrasound, were predictive of the need for VPS in pregnancy with ONTD. METHODS This was a retrospective analysis of prospectively collected data of consecutive pregnancies with ONTD, evaluated in a single center from January 2012 to May 2018. Two groups were identified: the first consisted of pregnancies that underwent in-utero repair (IUR) and the second those that had postnatal repair (PNR). Penalized B splines were used to determine the trend in VS, across 2-week gestational-age (GA) epochs, between 24 and 36 weeks of gestation. VS at each GA epoch and the change in VS between each GA epoch were compared between the IUR and PNR groups. To determine whether VS at any GA was predictive of VPS, receiver-operating-characteristics (ROC) curves were used and the optimal cut-off at each GA epoch was identified. Univariate analysis and multiple logistic regression were used for further analysis. RESULTS ONTD was diagnosed in 110 fetuses, of whom 69 underwent IUR and 41 had PNR. Fetuses in the IUR group were more likely to have Chiari II malformation (100.0% vs 82.9%; P

Objectives To determine the accuracy of ultrasound estimation of fetal weight among fetuses with gastroschisis and how the diagnosis of fetal growth restriction (FGR) affects the timing of delivery. Methods This was a retrospective cohort study including all fetuses with a diagnosis of gastroschisis at our institution from November 2012 through October 2017. We excluded multiple gestations, pregnancies with major structural or chromosomal abnormalities, and those for which prenatal and postnatal follow-up were unavailable. Performance characteristics of ultrasound to predict being small for gestational age (SGA) were calculated for the first and last ultrasound estimations of fetal weight. Results Our cohort included 75 cases of gastroschisis. At the initial ultrasound estimation, 15 of 58 (25.9%) fetuses met criteria for FGR; 48 of 70 (68.6%) met criteria at the time of the last ultrasound estimation (median, 34.7 weeks). Cesarean delivery was performed for 37 of 75 (49.3%), with FGR and concern for fetal distress as the indication for delivery in 17 of 37 (45.9%). Only 6 of 17 (35.3%) of the neonates born by cesarean delivery for an indication of FGR and fetal distress were SGA. The initial ultrasound designation of FGR corresponded to SGA at birth in 8 of 15 (53.3%), whereas the last ultrasound estimation corresponded to SGA in 17 of 48 (35.4%). The initial ultrasound estimation agreed with the last ultrasound estimation before delivery with the diagnosis of FGR in 13 of 15 (86.7%). Conclusions Ultrasound in the third trimester was sensitive but had a low positive predictive value and low accuracy for the diagnosis of SGA at birth for fetuses with gastroschisis. A large proportion of fetuses were born by cesarean delivery with indications related to FGR or fetal concerns.

Objectives To provide an overview of perinatal outcomes in prenatally diagnosed spontaneous chorioamniotic separation (sCAS). Methods A systematic search of the literature was performed from inception to July 2019, including PubMed, Ovid MEDLINE, and Ovid EMBASE. All studies reporting prenatally diagnosed sCAS after 16 weeks' gestation in singleton pregnancies were eligible. Two independent reviewers used standardized forms for data abstraction. Results Of 408 screened abstracts, 17 studies reporting 118 cases of sCAS were included. Among 113 cases with delivery outcomes, preterm birth (PTB) occurred in 60 (53.1%, 95% confidence interval [CI] 43.9-62.3%). Intrauterine fetal demise (IUFD) occurred in seven (6.2%, 95% CI 1.8-10.6%) cases, with four due to cord strangulation. Spontaneous abortion occurred in one (0.88%, 95% CI -0.84-2.6%) case. Among 104 cases with postnatal follow-up, there were six (5.8%, 95% CI 1.3-10.3%) neonatal deaths and one (0.96%, 95% CI -0.91-2.8%) infant death. Perinatal mortality (IUFD and neonatal deaths) was 11.0% (95% CI 5.4-16.7%). Conclusions sCAS may be associated with increased risk of PTB, however, the available data are largely case reports and series. Antepartum surveillance after viability can be considered due to risk of cord accidents. Prospective study is necessary to understand the clinical implications of sCAS.

Objective Preterm delivery following fetoscopic laser surgery (FLS) of twin–twin transfusion syndrome (TTTS) is associated with severe perinatal morbidity and mortality. The role of steroid hormones in amniotic fluid (AF) after FLS remains unknown. Study Design A prospective cohort study of consecutive case series of FLS for TTTS was performed from April 2012 to February 2017. Cases were divided into early (≤27 weeks) spontaneous preterm delivery (ED) and late delivery (LD; ≥34 weeks) following FLS and compared. AF supernatants were assessed for protein, estradiol, progesterone and cortisol levels (using the ELISA kit), and normalized to total protein levels to adjust for dilution. Results A total of 294 consecutive cases of FLS for TTTS in monochorionic–diamniotic twins were performed during the study period. AF was available in 44 ED patients and 50 LD patients. On logistic regression, ED was associated with higher normalized progesterone levels (odds ratio [OR]: 1.25; 95% confidence interval [CI]: 1.12–1.41), lower normalized cortisol (OR: 0.78; 95% CI: 0.64–0.96), and higher estradiol levels (OR: 1.3; 95% CI: 1.03–1.63). Conclusion Elevated AF normalized progesterone and estradiol, and lower normalized cortisol levels were associated with ED. This novel finding requires further exploration to establish the molecular mechanism operational in pregnancies complicated by TTTS to potentially prevent early preterm birth after fetal surgery.

Congenital lung malformation (CLM) volume ratio (CVR) of ≥1.1 has been shown to be highly predictive of the need for urgent, perinatal surgical intervention. The purpose of this study was to utilize this information to propose a delivery planning and clinical management algorithm based on this threshold.A retrospective cohort study was performed for all fetuses evaluated at our fetal center between 5/2015 and 11/2020. Demographics, ultrasound findings, late gestation CVR (≥27 weeks gestational age), prenatal and postnatal treatment, and outcomes were analyzed with nonparametric univariate analysis based on late gestation CVR of 1.1. Receiver operating characteristic curve analysis was performed to evaluate association between late gestation CVR, hydrops, need for fetal intervention, and need for urgent perinatal surgery.Of the 90 CLMs referred to our fetal center, 65 had late gestation CVR with a majority1.1 (47/65, 72%). All patients with late gestation CVR ≥ 1.1 were managed with resection (18/18) with most resections requiring fetal intervention or urgent neonatal resection (13/18). Late gestation CVR 1.1 were managed with elective resection (36/47, 77%) or non-operative observation (11/47, 23%). Late gestation CVR ≥ 1.1 had 100% sensitivity and NPV for hydrops, need for fetal intervention, and need for urgent perinatal surgery.CLM with CVR ≥ 1.1 were associated with urgent perinatal surgical intervention and expectant mothers should plan for delivery at centers equipped to manage neonatal resuscitation and potential urgent neonatal resection. Conversely, CLM with CVR 1.1 may be safe to deliver at patient hospital of choice.

OBJECTIVE To evaluate the efficacy of long-term indomethacin therapy (LIT) in prolonging pregnancy and reducing spontaneous preterm birth (PTB) in patients undergoing fetoscopic laser surgery (FLS) for the management of twin-to-twin transfusion syndrome (TTTS). DESIGN Retrospective cohort study of prospectively collected data. SETTING Collaborative multicentre study. POPULATION Five hundred and fifty-seven consecutive TTTS cases that underwent FLS. METHODS Long-term indomethacin therapy was defined as indomethacin use for at least 48 hours. Log-binomial regression was used to estimate the relative risk of PTB in the LIT group compared with a non-LIT group. Cox regression was used to evaluate the association between LIT use and FLS-to-delivery survival. MAIN OUTCOME MEASURES Gestational age (GA) at delivery. RESULTS Among the 411 pregnancies included, a total of 180 patients (43.8%) received LIT after FLS and 231 patients (56.2%) did not. Median GA at fetal intervention did not differ between groups (20.4 weeks). Median GA at delivery was significantly higher in the LIT group (33.6 weeks) compared with the non-LIT group (31.1 weeks; P

Objective This systematic review and meta-analysis aimed to compare the perinatal outcomes of complicated monochorionic pregnancies after selective reduction by radiofrequency ablation, bipolar cord coagulation, and interstitial laser. Data Sources We searched PubMed, Scopus, and Web of Science, from the inception of the database up to April 26, 2021. Study Eligibility Criteria Studies comparing at least 2 selective reduction techniques among complicated monochorionic pregnancies and presenting data on perinatal outcomes, including gestational age at procedure, gestational age at delivery, procedure to delivery interval, preterm premature rupture of membranes, preterm birth, survival rate, and birthweight, were eligible. Methods The random-effects model was used to pool the mean differences or odds ratios and corresponding 95% confidence intervals. Heterogeneity was assessed using the I2 value. Results A total of 10 studies with 734 cases of fetal reduction met the inclusion criteria, of which 9 studies with 674 fetuses were eligible for quantitative synthesis. In 8 studies that compared radiofrequency ablation with bipolar cord coagulation, radiofrequency ablation was associated with increased procedure to delivery interval (days) (mean difference, 13.42; 95% confidence interval, 1.90–24.94; P=.02; I2=0.0%), decreased preterm birth (odds ratio, 0.50; 95% confidence interval, 0.29–0.85; P=.01; I2=3.0%), and decreased preterm premature rupture of membranes (odds ratio, 0.45; 95% confidence interval, 0.27–0.73; P=.001; I2=0.0%). Radiofrequency ablation and bipolar cord coagulation had comparable survival rates (odds ratio, 0.85; 95% confidence interval, 0.54–1.35; P=.49; I2=0.0%). In 3 studies that compared radiofrequency ablation with interstitial laser, there was no significant difference in gestational age at delivery (P=.07) or survival (P=.15). In 3 studies that compared bipolar cord coagulation with interstitial laser, bipolar cord coagulation was associated with a higher survival rate (odds ratio, 3.21; 95% confidence interval, 1.13–9.10; P=.03; I2=0.0%), but the gestational age at delivery was comparable between groups (P=.16). Conclusion This study demonstrated that radiofrequency ablation has a greater procedure to delivery interval and decreased preterm premature rupture of membranes and preterm birth than bipolar cord coagulation. Although there was no difference in gestational age at delivery for either bipolar cord coagulation, radiofrequency ablation, or interstitial laser, survival was higher with bipolar cord coagulation than with interstitial laser.

Objective: To evaluate the efficacy of long-term indomethacin therapy (LIT) in prolonging pregnancy and reducing spontaneous preterm birth (PTB) in patients undergoing fetoscopic laser surgery (FLS) for the management of twin-to-twin transfusion syndrome (TTTS). Design: Retrospective cohort study of prospectively collected data. Setting: Collaborative multicenter study Population: 557 consecutive TTTS cases that underwent FLS Methods: LIT was defined as indomethacin use for at least 48 hours. Log-binomial regression was used to estimate the relative risk (RR) of PTB in LIT compared to non-LIT group. Cox regression was used to evaluate the association between LIT use and FLS-to-delivery survival. Main outcome measures: gestational age (GA) at delivery Results: Among the 411 pregnancies included, a total of 180 patients (43.8%) received LIT after FLS and 231 patients (56.2%) did not. Median GA at fetal intervention did not differ between groups (20.4 weeks). Median GA at delivery was significantly higher in the LIT group (33.6 weeks) compared to the non-LIT group (31.1weeks), p

To study the role of fetal endoscopic tracheal occlusion (FETO) on resolution of pulmonary hypertension (PH) in fetuses with isolated moderate left-sided diaphragmatic hernia (CDH).This retrospective study included fetuses with CDH evaluated between February 2004 and July 2017. Using the tracheal occlusion to accelerate lung growth (TOTAL) trial definition, we classified fetuses into moderate left CDH if O/E-LHR (observed/expected-lung head ratio) was 25-34.9% regardless of liver position or O/E-LHR of 35-44.9% if liver was in the chest. Postnatal echocardiograms were used to diagnose PH. Logistic regression analyses were performed to determine the relationship of FETO with study outcomes.Of 184 cases with no other major anomalies, 30 (16%) met criteria. There were nine FETO and 21 non-FETO cases. By hospital discharge, a higher proportion of infants in the FETO group had resolution of PH (87.5 (7/8) vs. 40% (8/20);Infants with moderate left-sided CDH according to O/E-LHR, FETO is associated with resolution of PH by the time of hospital discharge.

OBJECTIVE Monochorionic twins with twin-twin transfusion syndrome (TTTS) treated with fetoscopic laser photocoagulation (FLP) are at increased risk of neurodevelopmental impairment (NDI). This meta-analysis aimed to identify the prevalence of and perinatal risk factors for NDI in TTTS survivors treated with FLP. METHODS We performed a search in PubMed, EMBASE, Scopus and Web of Science, from inception to 13 February 2021, for studies evaluating perinatal risk factors for NDI in children diagnosed prenatally with TTTS managed by FLP. Data on severity of TTTS at the time of diagnosis, defined according to the Quintero staging system, FLP-related complications and perinatal outcomes were compared between children with a history of TTTS treated with FLP with and those without NDI, which was defined as performance on a cognitive or developmental assessment tool ≥ 2 SD below the mean or a defined motor or sensory disability. A random-effects model was used to pool the mean differences or odds ratios (OR) with the corresponding 95% CIs. Heterogeneity was assessed using the I2 statistic. RESULTS Nine studies with a total of 1499 TTTS survivors were included. The overall incidence of NDI was 14.0% (95% CI, 9.0-18.0%). The occurrence of NDI in TTTS survivors was associated with later gestational age (GA) at FLP (mean difference, 0.94 weeks (95% CI, 0.50-1.38 weeks); P

Background. Fetal anemia results from several conditions; however intrauterine transfusion (IUT) remains the treatment for severe cases. The complications of this procedure are rare and yet can result in preterm delivery or fetal death. Case. 31 y/o G3P2002 with Rh alloimmunization underwent IUT from 19 to 35 weeks. Umbilical artery thrombosis was noted after her 5th IUT. Further transfusions were performed without any complications and she delivered a full term male infant with APGARS of 8 and 9 at 1 and 5 minutes, respectively. Conclusion. The complication of umbilical artery thrombosis is unusual and the optimal management is unclear. We report such a case and propose that the presence of Hyrtl’s anastomosis near the placental cord insertion may explain the reassuring fetal status throughout the pregnancy.

OBJECTIVE The optimal outcome after fetoscopic laser surgery (FLS) for twin-twin transfusion syndrome (TTTS) depends on the successful ablation of all placental anastomoses. The objective of this study was to determine the incidence of and risk factors for recurrent TTTS (rTTTS) or twin anemia-polycythemia sequence (TAPS) after FLS, focusing on the impact of cannula diameter. METHODS This was a secondary analysis of data collected prospectively at two centers from 666 consecutive patients undergoing FLS for TTTS. The main outcomes were rTTTS and TAPS following FLS. Variables assessed included gestational age at intervention, stage of disease, recipient maximum vertical pocket, anterior placenta, number of anastomoses ablated, cannula diameter/operative scopes and use of the Solomon technique. Cannula diameter and corresponding scopes used were as follows: 8 Fr and 1.3 mm/0°; 9 Fr and 2.7 mm/0°; 10 Fr and 3 mm/0°; or 12 Fr and 3.3-3.7 mm/30-70°. Cannula diameter was used as a surrogate for scopes during analysis. Multivariate logistic regression analysis was performed to identify risk factors associated with rTTTS or TAPS after FLS; 'center' was considered an independent variable to account for variations in practice. In a nested cohort of pregnancies in which both fetuses survived, placental dye injection was performed in 315 placentae. Multivariate logistic regression analysis was performed to evaluate variables associated with the presence of residual anastomoses. RESULTS rTTTS or TAPS occurred in 61 (9%) cases following FLS (rTTTS in eight (1%) and TAPS in 53 (8%)). Factors associated significantly with the risk of rTTTS/TAPS on multivariate analysis were cannula diameter (when an 8-Fr, 9-Fr, 10-Fr or 12-Fr cannula was used, there was rTTTS/TAPS in 24%, 13%, 2% or 0.8% of cases, respectively (P

Background In utero closure of meningomyelocele using an open hysterotomy approach is associated with preterm delivery and adverse neonatal outcomes. Objective This study compared the neonatal outcomes in in utero meningomyelocele closure using a 2-port, exteriorized uterus, fetoscopic approach vs the conventional open hysterotomy approach. Study Design This retrospective cohort study included all consecutive patients who underwent in utero meningomyelocele closure using open hysterotomy (n=44) or a 2-port, exteriorized uterus, fetoscopic approach (n=46) at a single institution between 2012 and 2020. The 2-port, exteriorized uterus, fetoscopic closure was composed of the following 3 layers: a bovine collagen patch, a myofascial layer, and a skin. The frequency of respiratory distress syndrome and a composite of other adverse neonatal outcomes, including retinopathy of prematurity, periventricular leukomalacia, and perinatal death, were compared between the study groups. Regression analyses were performed to determine any association between the fetoscopic closure and adverse neonatal outcomes, adjusted for several confounders, including gestational age of Results The fetoscopic closure was associated with a lower rate of respiratory distress syndrome than the open hysterotomy closure (11.5% [5 of 45] vs 29.5% [13 of 44]; P=.037). The proportion of neonates with a composite of other adverse neonatal outcomes in the fetoscopic group was half of that observed patients in the open hysterotomy group; however, this difference did not reach statistical significance (4.3% [2 of 46] vs 9.1% [4 of 44]; P=.429). Here, regression analysis has demonstrated that fetoscopic meningomyelocele closure was associated with a lower risk of respiratory distress syndrome (adjusted odds ratio, 0.23; 95% confidence interval, 0.06–0.84; P=.026) than open hysterotomy closure. Conclusion In utero meningomyelocele closure using a 2-port, exteriorized uterus, fetoscopic approach was associated with a lower risk of respiratory distress syndrome than the conventional open hysterotomy meningomyelocele closure.

Objectives To evaluate a novel scoring system that combines several prenatal parameters for selecting ideal candidates for fetal intervention, and for predicting postnatal survival in patients with severe fetal lower urinary tract obstruction (LUTO). Methods We retrospectively reviewed all cases of severe LUTO evaluated for fetal intervention in a single large fetal center between January 2013 and December 2017. A scoring system for determining fetal candidacy for intervention was retrospectively developed based on postnatal outcomes. The proposed scoring system included fetal urinary biochemistry, renal ultrasound parameters, initial bladder volume, and degree of bladder refill. Relevant demographic characteristics, ultrasound reports and laboratory results were reviewed. Receiver operating characteristic (ROC) curves were used to select the cut-off values for initial bladder volume and degree of bladder refill and to evaluate the performance of the scoring system in predicting postnatal death. Results Of the 79 LUTO patients evaluated, 31 were eligible for the study. The overall 6-month postnatal survival was 64.5 % (20/31). A scoring system (0−8) was suggested with 2 points for unfavorable biochemistry, 4 points for ultrasound evidence of dysplastic kidneys, 1 point for inadequate initial bladder volume and 1 point for inadequate bladder refill. Scores>3 (N = 7) were associated with 0 % 6-month survival. The ROC curve for predicting postnatal mortality showed area under curve (AUC) of 0.82 (95 % CI 0.65−0.99). Subgroup analysis within subjects who underwent fetal intervention (N = 22) also confirmed the significance of the distribution of the scoring system between groups who survived and those who did not after adjustment for GA at delivery (p = 0.01). Conclusion We propose a novel scoring system for antenatal evaluation of patients with severe LUTO which may be useful in selecting those candidates most appropriate for intervention and in counseling parents about predicted postnatal outcome.

Objective To evaluate if magnesium sulfate (MgSO4 ) titration following fetoscopic spina bifida closure is associated with fewer maternal complications than the Management of Myelomeningocele Study (MOMS) tocolytic regimen. Methods This prospective cohort study included 73 consecutive patients undergoing fetoscopic closure of spina bifida between 2015 and 2020. A policy of using the MgSO4 regimen per the MOMS trial was changed to a flexible one in which MgSO4 was titrated according to the frequency of the uterine contractions following surgery. The frequency of maternal pulmonary edema, low maternal oxygen saturation requiring oxygen supplementation, atelectasis, hypocalcemia, and preterm delivery was compared before and after the policy was changed. Results A higher proportion of women in the group that used the MOMS MgSO4 regimen had pulmonary edema compared to those in the flexible one (26.1% [6/23] vs. 6% [3/50]; p = 0.024). Multivariate analysis showed that the MOMS tocolytic regimen was independently associated with a higher risk of pulmonary edema (adjusted odds ratio: 8.57; 95% confidence interval: 1.54-47.7; p = 0.014) than a flexible one. There was no difference in the rate of preterm delivery. Conclusion Following fetoscopic closure of spina bifida, the MOMS MgSO4 regimen is associated with an increased risk of pulmonary edema than a more flexible regimen.

Objective: To evaluate the role of intertwin differences in umbilical artery (UA) pulsatility index (DUAPI) in predicting infant survival in twin-to-twin transfusion syndrome (TTTS) Design: Retrospective cohort study Setting: Baylor College of Medicine, Texas Children’s Hospital and University of Maryland School of Medicine Population: Women who underwent laser surgery due to TTTS between 2002 and 2018. Methods: Absolute DUAPI were calculated prior to laser surgery. ROC analysis was done to derive a DUAPI cutoff value of 0.4 for the prediction of double infant survival. Regression analyses were performed to evaluate the association of DUAPI

OBJECTIVE To compare perinatal outcomes between acute single fetal demise following fetoscopic laser photocoagulation to planned selective reduction (SR) in complicated monochorionic twin pregnancies. METHODS This was a secondary analysis of prospectively collected data in complicated monochorionic twin pregnancies from 2 fetal centers from 2011 to 2016. Group 1 included women undergoing fetoscopic laser photocoagulation for twin-twin transfusion syndrome (TTTS) who experienced a single fetal demise within 24 hours of the procedure. Group 2 consisted of women undergoing planned selective reduction (SR) with TTTS, and Group 3 SR for indications other than TTTS. RESULTS Survival of the remaining co-twin at birth was highest in Group 1 (n = 77 patients; 95%) compared with that in Group 2 (n = 15; 80%) and Group 3 (n = 32; 78%; P = .047). The preterm premature rupture of membrane rate was higher in Group 1 (47%) compared with that in Group 2 (33%) and Group 3 (7%; P

Acute papillary muscle rupture during pregnancy is a rare cardiac condition with potential for 200% mortality. We describe a 28-year-old morbidly obese woman at 27 weeks gestation who presented with acute decompensated mitral regurgitation secondary to spontaneous papillary muscle rupture. After hemodynamic stabilization and caesarean delivery, we performed an emergent mitral valve repair through a minimally invasive right thoracotomy.