Thoracoscopic removal with fluoroscopic radiographic guidance of thoracoamniotic shunting catheters in newborns.

Fetal thoracoamniotic shunting (TAS), which drains pleural effusion, is a treatment for severe primary fetal pleural effusion. While TAS is an effective treatment, its complications include bleeding and the catheter becoming dislodged, and also penetrating the thoracic cavity or chest wall. Catheters dislodged into the thoracic cavity in TAS can be removed by thoracoscopy. However, if there are adhesions in the thoracic cavity, finding the TAS catheter with a thoracoscope can be difficult. We used fluoroscopic radiography in addition to a thoracoscope to remove a TAS catheter in four patients. A 5-mm trocar was inserted into the thoracic cavity, and a 2.7-mm scope and 3-mm forceps were inserted into the trocar. We searched for TAS catheters using a thoracoscope and fluoroscopic radiography. If there are adhesions in the thoracic cavity and removing the TAS catheter is difficult, the combined use of a thoracoscope and fluoroscopic radiography may prove helpful.

Long-term outcome of cases of fetal pleural effusion: A study at a single perinatal center in Japan.

PURPOSE: To analyze the long-term prognosis of primary and secondary fetal pleural effusion (FPE). METHODS: We investigated all cases of FPE in a single University hospital (2005-2020). Cases were classified as primary (cases with only pleural effusion) and secondary (cases with other abnormalities such as chromosomal abnormalities or fetal cardiac failure). We retrospectively reviewed the medical records from the time of diagnosis, to assess medical procedures performed, chromosomal test results, and clinical outcomes. RESULTS: Among 18 027 deliveries, 17 FPEs were identified (primary FPE: 8, secondary FPE: 9). Most primary FPEs were diagnosed in the second trimester of pregnancy, while all secondary FPEs were diagnosed in the third trimester. Secondary FPE was often associated with chromosomal abnormalities, including trisomy 21. The prognosis of pleural effusion caused by trisomy 21 was relatively good, except for cases with TAM. Cases of secondary FPE without trisomy 21 were of cardiac origin, and the neonatal prognosis was poor. The short-term prognosis was better in the primary FPE group, but long-term follow-up identified conditions such as acute encephalitis with refractory, repetitive partial seizures, developmental delay and attention deficit hyperactivity disorder. CONCLUSION: Fetal pleural effusion without the presence of chromosomal abnormalities or morphologies has a good short-term prognosis, but the long-term prognosis is poor. Thus, long-term follow-up is necessary for all cases of fetal pleural effusion.

Risk factors associated with fetal pleural effusion in prenatal diagnosis: a retrospective study in a single institute in Southern China.

The aim of this study was to analyse the factors associated with fetal pleural effusion over the past five years in a single institute in the South of China. Between January 2011 and May 2016, 129 foetuses with pleural effusion were referred to the Fetal Medicine Unit in Guangzhou's Women and Children's Medical Center. Seventy-nine women accepted an invasive procedure to rule out chromosomal abnormalities, fetal anaemia, intrauterine infections or some of the submicroscopic chromosomal abnormalities. Our results showed that chromosomal anomalies occurred in 15.2% (12/79) of cases including 8 Turner syndrome (45, X) (10.1%), 3 trisomy 21 (3.8%) and 1 trisomy 13 (1.3%). Pathological microdeletion or microduplication syndrome occurred in 3 out of 36 (8.3%) prenatal samples with normal karyotype and structural defects. Eight foetuses (10.1%) affected with haemoglobin Bart's disease showed pleural effusion at second or third trimester. Two cases (2.5%) were found to have an intrauterine infection. In conclusion, fetal pleural effusion has a close correlation with chromosomal abnormality. CMA may increase the detection rate of chromosomal aberrations, especially for micro-deletion or micro-duplication syndromes. In the South of China, Thalassemia must be considered when a fetal pleural effusion is detected.Impact statementWhat is already known on this subject? The aetiology of fetal pleural effusion includes a chromosomal abnormality, a congenital heart disease, congenital infections and a number of genetic syndromes.What do the results of this study add? This is the first retrospective study to analyse the aetiology of fetal pleural effusion in one institute in the South of China.What are the implications of these findings for clinical practice and/or further research? Besides the chromosomal abnormality, micro-deletion and micro-duplication syndromes were also detected in our study. We feel that thalassemia must be considered when fetal pleural effusion is detected in South China.

Clinical outcomes of thoracentesis for severe primary fetal pleural effusion. / èƒ¸è ”ç©¿åˆºæœ¯ç”¨äºŽé‡åº¦åŽŸå‘æ€§èƒ¸è ”ç§¯æ¶²èƒŽå„¿çš„ä¸´åºŠç»“å±€.

OBJECTIVES: To investigate the prenatal diagnosis of severe primary fetal hydrothorax and the clinical outcome after intrauterine thoracentesis. METHODS: A total of 12 patients with severe PFHT and thoracentesis, who were admitted to Xiangya Hospital, Central South University from January 2016 to December 2018, were enrolled. The clinical data were retrospectively analyzed. RESULTS: Five cases were bilateral pleural effusion, 6 on the right side, and 1 on the left side. All cases were accompanied with polyhydramnios, 4 cases with ascites, and 3 cases with skin edema. Ten patients underwent 1 thoracentesis puncture, 1 case for twice, and 1 case for 5 times. Hydrothorax test results of all cases were consistent with primary pleural effusion. Three patients underwent labor induction, 4 of 9 live births had mild asphyxia, 8 required respiratory support, and 7 needed the closed thoracic drainage. All children were fed with medium-chain fatty acid milk powder. CONCLUSIONS: Thoracentesis is one of the measures for intrauterine intervention in severe PFHT, which can improve the prognosis of children. Respiratory support, closed thoracic drainage, medium-chain fatty acid feeding are given to newborn. After these treatments, the survival rate is high and the prognosis is good.

New terminology for adverse events of fetal therapy: Re-evaluation of the thoraco-amniotic shunting in a Japanese study.

AIM: Since fetal therapy has been newly developed, objective evaluation of adverse events (AE), all harmful events that are not always related to the procedures, has not yet been adequately reported. We established new terminology and tried to re-evaluate it based on the Japanese prospective thoraco-amniotic shunting (TAS) study. METHODS: From the literature, all complications that occurred with all fetal therapies were identified as a basis for developing the terminology. Grading was set from 0 to 5. Grade 3 was defined as the need for invasive treatment, such as surgery. Grade 4 was defined as life-threatening, and Grade 5 was defined as death of the mother or fetus. Then, one series of TAS that we had already reported was re-evaluated, including 24 cases with 37 procedures and 200 opportunities those could be evaluated AE after procedures. RESULTS: Grade 4 preterm rupture of the membranes was reported in only 1 of 24 cases. Catheter displacement was reported in 7, 2 and 11 cases, of Grades 1, 2 and 3, respectively. It was found that the double-basket catheter had some association with catheter displacement, but it was clear that there were no life-threatening AE. CONCLUSION: Newly developed AE criteria are now available for fetal therapy. They will contribute to the objective evaluation of AE of new fetal therapies in the future.

Spontaneous resolution of prenatally diagnosed isolated pleural effusion: An unusual early sign of a newborn disease.

We present a case report of a fetus with a diagnosed pleural effusion in the first trimester on nuchal translucency scan. The effusion resolved spontaneously by 17 weeks of pregnancy. Toxoplasmosis, rubella, cytomegalovirus, herpes simplex (TORCH) - negative. Array comparative genomic hybridization (aCGH) - normal. Serial Doppler scans normal - no prenatal signs of anemia. Maternal antibodies against red cell antigens - negative. Delivery at term by cesarean section because of macrosomia. Neonate suffered from prolonged jaundice. At 3 weeks of life diagnosed with hereditary spherocytosis. Literature review shows that this may be the first connection between this disease and prenatal life.

Long-Term Neurodevelopmental and Respiratory Outcome after Intrauterine Therapy for Fetal Thoracic Abnormalities.

INTRODUCTION: The aim of this study is to evaluate long-term neurodevelopmental and respiratory outcome after fetal therapy for fetal pleural effusion, congenital cystic adenomatoid malformation, and bronchopulmonary sequestration. METHODS: Children ≥18 months of age underwent an assessment of neurologic, motor, and cognitive development. Medical records were reviewed to determine respiratory outcome. Behavioral outcome was assessed using the Child Behavioral Checklist. RESULTS: Between 2001 and 2016, 63 fetuses with fetal hydrops secondary to thoracic abnormalities were treated at our center. Overall perinatal survival was 64% (40/63). Twenty-six children were included for follow-up (median age 55 months). Severe neurodevelopmental impairment (NDI) was detected in 15% (4/26). Three out of 4 children with severe NDI had associated causes contributing to the impairment. Overall adverse outcome, including perinatal mortality or NDI, was 55% (27/49). Fifteen percent (4/26) had severe respiratory sequelae. Parents did not report more behavioral problems than Dutch norms. DISCUSSION: Our results suggest that severe NDI in this specific high-risk cohort occurs in 15%, which is above the range of the incidence of NDI reported in case series treated with other fetal therapies (5-10%). Large multicenter studies and an international web-based registry are warranted to prospectively gather outcome data at fixed time points.

Outcome and Treatment of Antenatally Diagnosed Nonimmune Hydrops Fetalis.

INTRODUCTION: The objectives of this study were to evaluate the outcome of nonimmune hydrops fetalis in an attempt to identify independent predictors of perinatal mortality. MATERIAL AND METHODS: A retrospective cohort study was conducted including all cases of nonimmune hydrops from two tertiary care centers. Perinatal outcome was evaluated after classifying nonimmune hydrops into ten etiological groups. We examined the effect of etiology, site of fluid accumulation, and gestational age at delivery on postnatal survival. Neonatal mortality and hospital discharge survival were compared between the expectant management and fetal intervention groups among those with idiopathic etiology. RESULTS: A total of 142 subjects were available for analysis. Generally, nonimmune hydrops carried 37% risk of neonatal mortality and 50% chance of survival to discharge, which varies markedly based on the underlying etiology. Ascites was an independent predictor of perinatal mortality (p value = 0.003). There was nonsignificant difference in neonatal mortality and hospital discharge survival among idiopathic cases that were managed expectantly versus those in whom fetal intervention was carried out. DISCUSSION: The outcome of nonimmune hydrops varies largely according to the underlying etiology and the presence of ascites is an independent risk factor for perinatal mortality. In our series, fetal intervention did not offer survival advantage among fetuses with idiopathic nonimmune hydrops.

Chromosomal abnormalities associated with fetal pleural effusion (II): Specific and non-specific chromosome aberrations.

Fetal pleural effusion has been reported to be associated with chromosomal abnormalities, genetic syndromes, obstructive uropathy, lymphatic vessel abnormalities such as Noonan syndrome, RASopathy and congenital lymphatic anomalies, thoracic cavity defects, Rh or ABO incompatibility, non-immune hydrops fetalis, infections, congenital cardiac anomalies, metabolic diseases and hematologic diseases such as α-thalassemia. This review provides a comprehensive view of specific and non-specific chromosome aberrations associated with fetal pleural effusion which is useful for genetic counseling and fetal therapy at prenatal diagnosis of fetal pleural effusion.

Spontaneous regression of fetal pleural effusion in pregnancy complicated with Herpes simplex infection: Clinical presentation and literature review.

Fetal primary hydrothorax is a rare congenital anomaly with an estimated incidence of 1:10,000-15,000 pregnancies, with an unpredictable clinical course, ranging from spontaneous resolution to fetal death. A case of unilateral fetal pleural effusion was diagnosed at 35th week of gestation during a routine ultrasonographic fetal assessment in an uncomplicated pregnancy. A large echogenic collection of fluid was revealed in the right pleural cavity, together with atelectasis of the right lung, as well as displacement of heart and mediastinal structures to the left side of thorax. The patient was also diagnosed with polyhydramnios and there was a disproportion of heart ventricles volume. No other fetal structural abnormalities were detected and there were no symptoms of edema. Fetal biometrics was consistent with the gestational age. In echocardiography, fetal heart was structurally and functionally normal. Screening tests for congenital infections of the fetus were negative. Autoimmune fetal hydrops was excluded after laboratory tests. There was no parents' consent for the analysis of the karyotype. The patient presented clinical symptoms and was diagnosed with Herpes simplex virus infection and was treated with oral acyclovir. Serial fetal ultrasound exams showed gradual decrease in pleural fluid volume up to complete resolution in 38th week of pregnancy. Pregnancy was ended in the 38th week of gestation with a cesarean delivery of a healthy neonate. It is yet to be determined if there is a direct association between Herpes simplex virus infection in pregnancy and the risk of fetal pleural effusion. The incidence of fetal pleural effusion is low and the neonatal outcome difficult to be predicted. The optimum management of fetal pleural effusion should be subject to further studies to determine the best clinical practice.

Case report: Minimally invasive removal of a dislodged thoracoamniotic shunt with an integral cystoscope in a preterm infant.

Introduction: Fetal pleural effusion is a rare condition that is associated with significant mortality. Although the insertion of fetal thoracoamniotic shunts can improve perinatal outcomes, there are several associated complications, such as intrathoracic dislodgement of the shunts. The optimal neonatal treatment for retained shunts remains uncertain. Case Description: A male infant was born at 32 weeks of gestation. He had antenatal hydrothorax that was detected at 27 weeks of gestation and was managed by intrauterine thoracoamniotic shunting. However, the shunt catheter dislodged into the fetal chest, which caused reaccumulated pleural effusion and respiratory distress requiring ventilatory support after birth. After the patient's condition stabilized, minimally invasive removal of the retained catheter was performed on day 17 of life using an integral pediatric cystoscope via a 3-mm thoracic incision. The procedure took approximately 5 min. The postoperative course was uneventful, and the patient, who was discharged 39 days postnatally, is thriving at the 6-month follow-up. Conclusions: We present a novel and effective approach to the management of an intrathoracic shunt using an integral cystoscope. This approach may offer a valuable alternative to traditional thoracoscopy in the neonatal period.

Intrauterine Thoracoamniotic Shunting of Fetal Hydrothorax with the Somatex Intrauterine Shunt: Intrauterine Course and Postnatal Outcome.

(1) Background: Severe fetal hydrothorax can be treated by intrauterine thoracoamniotic shunting (TAS). The aim of this study was to assess perinatal outcome and complication rates of TAS with a novel Somatex intrauterine shunt. (2) Methods: This is a single-center retrospective study of all fetuses with hydrothorax treated with TAS using a Somatex shunt between 2014 and 2020. (3) Results: A total of 39 fetuses were included in the study. Mean gestational age at first intervention was 27.4 weeks (range 19-33). Of these, 51% (n = 20) of fetuses had fetal hydrops, which resolved in 65% (13/20) before delivery. The live birth rate was 97% (n = 38), and 74% (n = 29) survived the neonatal period. The rate of postnatal pulmonary complications was high, with 88% of neonates requiring any kind of ventilatory support. There were 23% (n = 9) genetic abnormalities (trisomy 21 and Noonan syndrome). (4) Conclusions: TAS with a Somatex shunt has a high technical success rate, leading to high neonatal survival rates. Pregnancy and neonatal outcome is comparable to TAS for fetal hydrothorax using different shunt types.

In utero congenital chylothorax treatment with fetal thoracoamniotic shunt: Case report.

BACKGROUND: Fetal pleural effusions are a rare fetal anomaly that may result from congenital chylothorax. Severe cases lead to chest compression with resulting pulmonary hypoplasia and possible neonatal demise. Fetal thoracoamiontic shunt (TAS) placement may decrease the amount of pleural effusion and improve lung expansion. CASE: A 30-year-old primigravida at 29 2/7 weeks' gestation presented with fetal bilateral pleural effusions with no identifiable genetic or structural abnormalities. TAS placement accomplished decompression of the left fetal chest. The neonate was delivered at 33 3/7 weeks and required minimal respiratory support with no apparent long term complications at discharge. CONCLUSION: This case demonstrated that fetal intervention with TAS placement can improve neonatal outcomes. Referral to an MFM specialist capable of TAS should be considered for isolated fetal bilateral pleural effusion.

Thoracoamniotic shunting for fetal pleural effusion with hydropic change using a double-basket catheter: An insight into the preoperative determinants of shunting efficacy.

OBJECTIVES: Although the efficacy of thoracoamniotic shunting (TAS) for fetal hydrothorax is well-recognized, the coexistence of hydrops fetalis is still a clinical challenge. The preoperative determinants of shunting efficacy are not fully understood. In this study, we aimed to investigate the perinatal and postnatal outcomes of hydrops fetalis with pleural effusion treated by TAS using a double-basket catheter, and to discuss the preoperative factors predictive of patients who will benefit from TAS. STUDY DESIGN: We conducted a retrospective study in hydropic fetuses with pleural effusion treated by TAS between 2007 and 2015. We extracted information regarding postnatal survival and pretherapeutic sonographic findings, including skin-edema thickness, pleural-effusion pocket size, and Doppler readings. RESULTS: Twelve subjects underwent TAS at a median gestational age of 29+5 weeks (range, 25+5-33+2 weeks). Skin edema disappeared or regressed in 7. Three experienced early neonatal death and the other 9 ultimately survived after a live birth at a median gestational age of 33+4 weeks (range, 29+1-38+2 weeks). All surviving children, except for 1, had a pretherapeutic pleural-effusion pocket greater than the precordial-edema thickness. All 3 children that died had precordial-edema thickness equal to or greater than the size of the pleural-effusion pocket. CONCLUSIONS: We achieved a high survival rate (75%) using the double-basket technique. A greater pretherapeutic width of skin edema compared with the pleural-effusion pocket is possibly suggestive of a treatment-resistant condition and subsequent poor postnatal outcome.

Extra-lobar Pulmonary Sequestration Requiring Intrauterine Thoracentesis.

Congenital lung malformations can result in significant morbidity and mortality in children. Pulmonary sequestration is an uncommon congenital malformation of the lung that can cause complications even in fetal life. We herein present a newborn with extra-lobar sequestration (ELS) that lead to hydrops fetalis necessitating fetal intervention.