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Introduction: We aimed to identify maternal and fetal complications and investigate postnatal and long-term outcomes of fetal hydrothorax (FHT) treated with pleuro-amniotic shunting (shunt)., Methods: Single-center retrospective observational cohort of shunt cases performed from 2000 to 2021. Risk factors for maternal complications, fetal demise, neonatal death (NND), and postnatal outcomes were identified., Results: Out of 88 cases, 70 (79.5%) were complicated by hydrops, with an average gestational age (GA) at diagnosis of 27 weeks (range 16-34). In 16 cases, definitive etiology of FHT was identified; five cases of Noonan syndrome and three cases of monogenic disorders diagnosed by whole-exome sequencing (EPHB4, VEGFR3, RASA1). Shunt was performed at an average GA of 28 weeks (20-34), with a dislodgement in 10 cases (11.4%). Maternal: Complications occurred in three cases; survival rate was 76.1% (67/88). Follow-up data were available for 57/67 (85.1%) children. Incidence of severe neurodevelopmental impairment and pneumopathy (broncho dysplasia, persistent pulmonary hypertension of newborn, and asthma) was 5.3% and 8.8%, respectively. Post-treatment persistence of hydrops, FHT associated with genetic syndromes, and GA at birth were risk factors for fetal demise, NND, and postnatal complications., Conclusion: In truly isolated FHT, whenever indicated, pleuro-amniotic shunting is a safe procedure associated with good survival rate and long-term outcome., (© 2023 S. Karger AG, Basel.)

Objective: The objective of this study was to assess the predictive performance of preoperative cervical length (CL) for delivery within 1 week after pleuroamniotic shunting (PAS) in fetuses with severe hydrothorax., Methods: A prospective cohort of fetuses with severe hydrothorax referred to our fetal surgery center in Querétaro, Mexico from January 2012 to July 2020. Severe fetal hydrothorax was diagnosed as an accumulation of fluid within the fetal pleural space accompanied with severe bilateral lung compression, mediastinal shift, polyhydramnios, and/or hydrops. Transvaginal CL was measured immediately before PAS, and a short cervix was defined as that <25 mm. The interval from fetal intervention to delivery, prevalence of preterm prelabor rupture of membranes (PPROMs), and associations with delivery within the first week after PAS according to a short or a normal CL, were evaluated., Results: Thirty-five pregnancies with severe fetal hydrothorax treated with PAS were evaluated. Median gestational age at PAS was (weeks + days) 31+2 (range, 26+0-36+1). Two (5.7%) and 7 (20.0%) cases delivered within the first 24 h and 1 week after PAS, respectively. Ten (28.6%) women had a short cervix before PAS, while 25 (71.4%) had normal preoperative CL. Women with a short cervix showed lower mean interval between fetal intervention and delivery (2.4 vs. 5.5 weeks, p = 0.01), and higher prevalence of PPROM (50 vs. 12%, p = 0.01), as compared to women with a nonshort cervix. Preoperative short cervix was associated with significantly higher risk of delivery within the first 24 h (20.0 vs. 0%, respectively, p < 0.05) and 1 week after PAS (50.0 vs. 8.0%, respectively, p < 0.01) compared with pregnancies with normal preoperative CL., Conclusion: In pregnancies with severe fetal hydrothorax candidates for pleuroamniotic shunt, identification of a short cervix before fetal intervention can predict delivery within 1 week after the surgical procedure., (© 2021 S. Karger AG, Basel.)

Objective: The objective of this study was to describe the feasibility of single percutaneous uterine access for bilateral pleuroamniotic shunting (PAS) in fetuses with severe hydrothorax by using an internal rotational maneuver and to compare perinatal outcomes between successful and failed procedures., Methods: A prospective cohort of 25 fetuses with isolated bilateral hydrothorax and hydrops were referred to our fetal surgery center in Queretaro, Mexico during an 8-year period. Bilateral PAS was first attempted through a percutaneous single uterine access by internal rotation of the fetus, which was achieved by using the blunt tip of the same cannula, and in case of a failed procedure, a second uterine port was used to place the second shunt. The perinatal outcomes between successful (single uterine port) and failed (2 uterine ports) fetal procedures were compared., Results: Placing of bilateral shunts through a percutaneous single uterine access was feasible in 15/25 (60%) cases. Overall, median GA at delivery was 35.2 weeks with a survival rate of 64.0% (16/25). Three cases were excluded due to shunt dislodgement, leaving a final population of 22 fetuses; 13/22 (59.1%) and 9/22 (40.9%) managed using 1 and 2 uterine ports, respectively. The group with bilateral PAS placement through a successful single uterine port showed a significantly higher GA at birth (36.5 vs. 32.8 weeks, p = 0.001), lower surgical time (11.0 vs. 19.0 min, p = 0.01), longer interval between fetal intervention and delivery (5.7 vs. 2.7 weeks, p = 0.01), lower risk of preterm delivery (46.2 vs. 100%, p < 0.01), and lower rate of perinatal death (15.4 vs. 55.6%, p < 0.05) than the failed procedures requiring 2 uterine ports., Conclusion: In fetuses with severe bilateral hydrothorax and hydrops, bilateral pleuroamniotic shunting through a successful single percutaneous uterine access is feasible in up to 60% of cases and is associated with better perinatal outcomes., (© 2021 S. Karger AG, Basel.)

Introduction: The management of primary fetal pleural effusion remains a challenge for clinicians given the paucity of clinical information to guide practice., Materials and Methods: A retrospective descriptive study of cases referred for management to our fetal therapy center over a 10-year period. Survival to hospital discharge was evaluated against case characteristics and prenatal intervention. For this study, we categorized the severity of the pleural effusion at diagnosis as mild, moderate or severe, and the clinical course as regression, stable or progression., Results: Forty-five of the 103 pregnancies complicated by fetal pleural effusions during the study period were managed for primary effusions. Termination of pregnancy was requested in 6 cases. Thirty-nine pregnancies continued management, with 14 undergoing prenatal intervention. The overall survival rate to hospital discharge was 51%, including 7 survivors after prenatal intervention. The rate of survival was low if the effusion was categorized as severe at diagnosis or if there was progression of the clinical course., Conclusions: Case characteristics at the time of diagnosis and clinical course can be used to guide patient counseling and decision-making regarding fetal therapy. Prenatal intervention may improve the chance of survival in cases with characteristics associated with a poor prognosis., (Copyright © 2013 S. Karger AG, Basel.)

Background: Primary fetal pleural effusion can be associated with high perinatal morbidity and mortality, especially when it is associated with the presence of fetal hydrops. Pleuroamniotic shunting results in effective drainage and lung expansion which prolong the pregnancy and improve neonatal survival. Intrathoracic displacement of the shunt is a rare but a known complication of shunt insertion and can cause some infant morbidity. We present a case of successful antenatal treatment of primary fetal pleural effusion complicated with intrathoracic displacement of the shunt without any fetal and long-term infant morbidity., Case Report: Our case presented with severe bilateral fetal pleural effusion, causing fetal hydrops treated successfully by pleuroamniotic shunting at 25 weeks of gestation which was complicated by dislodgement of the shunt into the fetal chest documented by antenatal ultrasound at 30 weeks. There was no recurrence of fetal pleural effusion or other antenatal complication. After delivery there were no postnatal complications, so removal of the shunt was not considered, and clinical follow-up of the infant to the age of 12 months was uneventful., Conclusion: The complication of intrathoracic displacement of a pleuroamniotic shunt can be recognized antenatally by ultrasound. Since there are no postnatal pulmonary complications related to the presence of the shunt inside the infant chest, conservative management in these cases appears to be a safe approach., ((c) 2009 S. Karger AG, Basel.)

Internalization of a pleuroamniotic shunt previously has not been associated with significant fetal or neonatal problems. We report a case in whom the shunt internalized and compressed the lung hilum. A review of the complications of antenatal pleuroamniotic shunting is also presented.

The management of fetal hydrops in the second trimester is changing as the underlying etiologies are better understood. We report a case which was diagnosed at 18 weeks gestation. There was no underlying anatomical abnormality. Fetal blood sampling confirmed a normal karyotype and there was no evidence of fetal infection. Bilateral thoracocentesis and paracentesis caused temporary improvement of the fetal condition but subsequent fluid reaccumulation was noted within the left pleural cavity. A pleuroamniotic shunt inserted at 22 weeks caused permanent resolution of the hydrops. The infant was normal at 1-year follow-up.

A male newborn underwent a myelomeningocele repair, with subsequent placement of a ventriculoperitoneal shunt for treatment of hydrocephalus. Five days after shunt surgery, the infant acutely developed a deeply sunken fontanel, pallor, tachypnea, bradycardia, and irritability. Chest radiographs revealed intrathoracic migration of the distal shunt tubing and a tension hydrothorax. Treatment consisted of tube thoracostomy and temporary externalization of the distal shunt tubing. The patient fully recovered. The acute onset of shock in association with a sunken fontanel in a neonate with a shunt should raise the suspicion of tension hydrothorax. For critically ill infants immediate needle aspiration or thoracostomy is suggested. In less severely ill children, exposure of the shunt tubing in the neck and withdrawal of the pleural effusion by the distal shunt tubing may be performed as an emergency measure. The early recognition and urgent management of this problem are emphasized.