Short- and long-term outcome following thoracoamniotic shunting for fetal hydrothorax.

OBJECTIVES: To assess short- and long-term outcome in a cohort of fetuses diagnosed with hydrothorax (FHT) which underwent thoracoamniotic shunting in utero, and to examine the antenatal predictors of survival and of survival with normal neurodevelopmental outcome. METHODS: This was a retrospective analysis of 132 fetuses that underwent thoracoamniotic shunting at our center between 1991 and 2014. Data were extracted from hospital obstetric and relevant neonatal intensive care and neonatal developmental follow-up databases. Outcomes included survival to discharge and survival with normal neurodevelopmental outcome beyond 18 months. Information on malformations, syndromes and genetic abnormalities were obtained from antenatal, postnatal and pediatric hospital records or by parent report. We compared pregnancy characteristics among those who survived vs non-survivors and among those with normal neurodevelopmental outcome vs those who were abnormal or died. We explored whether there was a trend in survival over the study period. RESULTS: The mean gestational age at diagnosis of FHT was 25.6 weeks. The fetus was hydropic at diagnosis in 61% of cases, 69% had bilateral effusions and 55% had bilateral shunts inserted. Other diagnoses were present in 24% of cases, two-thirds of which were discovered only postnatally. There were 16 intrauterine and 30 neonatal deaths, with a 65% survival rate overall. The mean gestational age at delivery of liveborns was 35.4 (range, 26.9-41.6) weeks, and 88/116 (76%) were preterm (< 37 weeks). Of 87 liveborn at the treatment center, 75% experienced some respiratory and/or cardiovascular morbidity after birth, many with a lengthy hospital stay (mean, 36 (range, 1-249) days). Overall, 84% of survivors were developmentally normal beyond 18 months and outcomes were better when pleural effusions were isolated, 92% of these cases being neurodevelopmentally normal. There was no trend in survival or neurodevelopmental outcome over time. Despite the presence of FHT and neonatal respiratory issues, most (89%) of the 55 survivors with relevant follow-up had no long-term pulmonary complications. Gestational age at delivery was the only factor independently predictive of both survival and survival with normal neurodevelopmental outcome. CONCLUSIONS: FHT is associated with other pathologies in a quarter of cases and carries a significant risk of prematurity, mortality and neonatal morbidity. The outcome is good in survivors but is best in isolated cases. Predictors of outcome at diagnosis are poor. Future improvement in diagnostics at time of identification of FHT may help to identify those that would benefit most from thoracoamniotic shunting. © 2020 International Society of Ultrasound in Obstetrics and Gynecology.

[Prenatal diagnosis and intervention to fetal hydrothorax: five cases analysis].

OBJECTIVE: To research the important of prenatal diagnosis and effect of intervention to fetal hydrothorax. METHODS: The cases of fetal hydrothorax (n=5) were obtained from the Shengjing Hospital, China Medical University between December 2014 and May 2015. All pregnancies were uncomplicated, excluded congenital organic and chromosomal abnormalities during prenatal diagnosis and with a 37 average gestational weeks. The case 1, 2, 4 were unilateral hydrothorax and the case 3, 5 were bilateral. We performed an antenatal thracocentesis to case 1, 2, 3. In case1, the hydrothorax increased rapidly after 5 days, and the patient underwent a cesarean section and ex utero intrapartum treatment (EXIT); in case 2, the fetal heart rate was decreased to 40-50 bpm suddenly during thracocentesis, and we performed an emergent cesarean section and EXIT for the patient; in case 3, the patient underwent thracocentesis and a meanwhile cesarean section and EXIT procedure. We performed a conservative management to case 4, 5, the hydrothorax resolved spontaneously during the pregnancy and after birth, both patients underwent cesarean section. RESULTS: All fetuses were survived, the neonates of case 1, 2 and 3 underwent assited mechanical ventilation, thoracic close drainage, then discharged after hydrothorax resolved and feeding tolerance; in case 4, there was no respiratory distress and hospital treatment; in case 5, the neonate underwent assited mechanical ventilation and conservative management, the hydrothorax has resolved gradually. CONCLUSIONS: The prenatal diagnosis and antenatal intervention (thracocentesis) may play an important role in fetal hydrothorax treatment. In clinical, we should choose different plan according to the gestation weeks and classification of hydrothorax of the patient.

Outcomes of fetal intervention for primary hydrothorax.

OBJECTIVE: Primary hydrothorax is a rare congenital anomaly with outcomes ranging from spontaneous resolution to fetal demise. We reviewed our experience with fetuses diagnosed with primary hydrothorax to evaluate prenatal management strategies. METHODS: We reviewed the records of patients evaluated for fetal pleural effusions at our Fetal Treatment Center between 1996 and 2013. To define fetuses with primary hydrothorax, we excluded those with structural or genetic anomalies, diffuse lymphangiectasia, immune hydrops, and monochorionic diamniotic twin gestations. RESULTS: We identified 31 fetuses with primary hydrothorax, of whom 24 had hydrops. Hydropic fetuses were more likely to present with bilateral effusions. Of all fetuses with primary hydrothorax, 21 had fetal interventions. Survival without hydrops was 7/7 (100%), whereas survival with hydrops depended on whether or not the patient had fetal intervention: 12/19 (63%) with intervention and 1/5 (20%) without intervention. Premature delivery was common (44%) among those who had fetal intervention. CONCLUSIONS: Fetal intervention for primary hydrothorax may lead to resolution of hydrops, but preterm birth and neonatal demise still occur. Understanding the pathophysiology of hydrops may provide insights into further prenatal management strategies, including targeted therapies to prevent preterm labor.

Fetal lung volume measurements using 3D ultrasonography.

PURPOSE: The aim of the study was to establish fetal lung, thoracic and heart volume nomograms using three-dimensional (3D) ultrasonographic measurements. MATERIALS AND METHODS: For this purpose a total of 300 fetuses were examined between 18 and 34 weeks of gestation using Voluson 530 ME and Voluson 730 PRO (General Electic, USA) ultrasound devices with 5 MHz three-dimensional annular volume transducers. To determine fetal lung volume, each lung was measured separately using a transversal sectional plane in the multiplanar mode. Measurements were performed by area tracing around the fetal lung in cross-sectional planes in different slices. The distance between two represented slices was calculated by computer. Calculated volume data was plotted against gestational age in order to obtain nomograms of fetal lung, heart and thorax volumes. RESULTS: Our nomograms revealed increasing lung, heart and thoracic volume growth between 22 and 34 weeks of gestation. Furthermore, we could demonstrate that there is a statistically significant difference between the growth of the right and left lung volume (right > left). In a group of 12 fetuses with skeletal dysplasias or hydrothorax pulmonary hypoplasia was suspected by ultrasound. Comparing two-dimensional (2D) sonographic measurements of oblique lung diameter and 3D lung volumetry, it was found that these two methods were complementary for the recognition of pulmonary hypoplasia before the 24 weeks of gestation. CONCLUSION: Using 3D ultrasound it is easy to perform fetal lung volumetry especially before 30 weeks of gestation. The encouraging results suggest that this method could be useful for the early detection of pulmonary hypoplasia even before 24 weeks of gestation.

A rare case of congenital pulmonary lymphangiectasia, hydrothorax and ascites in a male embryo aborted at 20 weeks of gestation.

A case of a male embryo aborted at the 20th week of gestation with extensive ascites, hydrothorax, pulmonary lymphangiectasia and pulmonary hypoplasia is presented together with the pathological findings, the etiology, differential diagnosis, course and therapy of this pathologic entity. Also a short review of the literature is discussed.

In-utero stenting: development of a low-cost high-fidelity task trainer.

OBJECTIVE: To develop an in-utero stent placement training model. METHODS: The in-utero stent task trainer was constructed using a formalin-preserved gravid pig uterus. Altering the size of the uterine segment, changing the fluid level in the uterus and addition of a large Ziploc freezer bag variably filled with differing amounts of ultrasound gel can vary the procedural skill required. RESULTS: Thoracoamniotic and vesicoamniotic shunts can be simulated using this life-like model. The cost of eight to 10 learning stations is approximately US $ 60. Fetal position, maternal size and amniotic fluid status can be altered rapidly to increase the complexity of the procedure. CONCLUSIONS: This low-cost and realistic task trainer can provide the opportunity to practice in-utero shunt procedures in a non-clinical environment. This model should enhance learning and reinforce acquired skills.

Single-needle laser treatment with drainage of hydrothorax in fetal bronchopulmonary sequestration with hydrops.

Bronchopulmonary sequestration (BPS) is sometimes associated with hydrothorax and hydrops in utero. In the absence of fetal hydrops, perinatal outcome is favorable and justifies expectant management. In the presence of fetal hydrops, perinatal outcome is reported to be extremely poor and intervention should be considered. Therapeutic options include open fetal surgery, minimally invasive coagulation of the blood supply and thoracoamniotic shunting. We present the first case of fetal hydrops and a large hydrothorax due to BPS treated successfully with one ultrasound-guided thin needle insertion, through which both laser coagulation of the feeding artery and drainage of the hydrothorax were performed. Following the procedure the hydrothorax and hydrops gradually disappeared and the BPS diminished in size. A healthy neonate was delivered uneventfully at term. We describe the case and discuss the different therapeutic options.

Extrauterine Intrapartum Treatment (EXIT) in bilateral primary fetal hydrothorax.

Primary Foetal Hydrothorax (PFHT), is an intrathoracic collection of fluid in the fetus, which may be present on either side or even bilaterally. Advances in foetal diagnostics now allow consideration of the Ex-utero Intrapartum Treatment (EXIT) procedure for PFHT. Ex-utero Intrapartum Treatment (EXIT) allows therapeutic interventions on the neonate while maintaining fetoplacental circulation and thereby maintaining oxygenation. We report two cases of bilateral PFHT managed successfully with EXIT procedure.

Hidrotórax fetal: evaluación y manejo prenatal / Fetal hydrothorax: evaluation and prenatal management

El hidrotórax fetal es la acumulación de liquido libre en la cavidad pleural fetal. Aunque esta condición frecuentemente en el contexto de hidrops fetalis, el hidrotórax fetal primario es una patología rara, estimándose su incidencia en 1 de cada 15.000 embarazos. El objetivo de este trabajo es presentar la experiencia de nuestro grupo en relación a terapia fetal invasiva en el manejo del hidrotórax fetal. Se identificaron los casos de hidrotórax fetal diagnosticados prenatalmente por los autores durante periodo comprendido entre Enero 1997 y Julio del 2001. Estos casos fueron diagnosticados como parte de un estudio multicéntrico destinado a la evaluación prenatal de anomalías estructurales fetales. Durante el periodo estudiado se manejaron nueve casos de hidrotórax fetal que cumplieron con los requisitos de inclusión al estudio. De éstos, cinco se asociaban a hidrops al momento de la evaluación inicial y ocho casos eran bilateral. De los cuatro casos de hidrotórax fetal aislado, uno fue unilateral y tres bilaterales. Solo en dos casos no se realizó terapia prenatal invasiva, uno de ellos por presentar una cardiopatía congénita y el otro por encontrarse una aneuploidia. En los seis restantes se realizó terapia prenatal, toracocentesis y/o shunt pleuroamniótico. En nuestra serie hubo seis muertes perinatales, cinco de los cuales presentaban hidrops al momento de la intervención y uno tenía una cardiopatía severa que contraindicaba terapia fetal. Se analizan los casos y se presenta una sugerencia de manejo hidrotórax fetal a partir de la experiencia presentada y de publicaciones extranjeras

Feto-amniotic shunting--report of the experience of four European centres.

Few reports concerning intrauterine shunting are available. We investigated the impact of this method. In order to evaluate intrauterine shunting and the complication rate for different indications, we sent a questionnaire to all German-speaking level 3 centres. In four level 3 centres, 52 intrauterine catheters were inserted in 34 fetuses. The indications were uni- or bilateral hydrothorax in nine cases, cystic adenomatoid malformation of the lung in four cases, infravesical stenosis in 13 cases, and fetal ascites in eight cases. In three cases (6 per cent), difficulties occurred when the drain was inserted. In 15 cases (29 per cent), the function of the drain was reduced by dislocation or occlusion. The mortality rate caused by shunting was as high as eight per cent (four cases). The application of an intrauterine shunt currently represents a rarely performed ultrasound-guided therapeutic intervention in the fetus. In all cases, the indication for shunting is to avoid compression of normal tissue by cystic structures. A high complication rate restricts the application of drainage to selected cases.