Search

Introduction: Although mortality has decreased for fetuses with lower urinary tract obstruction treated with vesicoamniotic shunt (VAS) placement, survivors remain at risk for long-term renal impairment. We tested the association of fetal serum β2-microglobulin (fsβ2M) with postnatal renal function in these patients, hypothesizing that fsβ2M may predict such renal impairment., Materials and Methods: fsβ2M was obtained in patients undergoing VAS placement. The primary outcome was renal function at 3-12 months of life, as assessed by a pediatric nephrologist using medical records. Patients were divided into two groups: (1) 'stable renal function' - probable stable long-term renal function and reasonable growth - and (2) 'loss of renal function' - early loss of renal function and failure to thrive., Results: Nineteen patients with preoperative fsβ2M received a VAS. Of the 14 survivors, those with fsβ2M ≤5.6 mg/l tended to have stable renal function compared to those with fsβ2M >5.6 mg/l [5/6 (83.3%) vs. 2/8 (25.0%), OR = 15.00, 95% CI 0.70-709.89; p = 0.1026]. Eight of 9 patients followed for >12 months of age had outcomes consistent with the initial renal assessments., Discussion: Patients with initial fsβ2M >5.6 mg/l and treated with VAS tended to have poor renal outcomes., (© 2016 S. Karger AG, Basel.)

Posterior urethral valve or pleural effusion accompanied with Down syndrome have been described previously; however, there is no reported case of posterior urethral valve and pleural effusion with Down syndrome. A 45-year-old multigravida woman was transferred due to bilateral fetal hydrothorax, polyhydramnios, and threatened preterm labor at 32 weeks' gestation. Transabdominal ultrasonography revealed additional abnormality, posterior urethral valve. Amniocentesis was repeated due to intense amniotic fluid and patient's dyspnea. Ultrasound-guided thoracoamniotic shunt and vesicoamniotic shunt were done successfully. At 33 weeks' gestation, intrauterine infection was suspected by the laboratory findings of amniotic fluid. A 2.25-kg male baby with characteristic phenotypic findings of Down syndrome was delivered by Cesarean section. Cytogenetic analysis after birth revealed a karyotype of 47,XY,+21. The present case reinforces the notions that fetuses with multiple anomalies, like congenital pleural effusion and posterior urethral valve, are at risk of chromosomal abnormalities, in spite of their low possibilities of association with chromosomal abnormalities., (Copyright 2008 S. Karger AG, Basel.)

Thoracoamniotic shunting in fetal pleural effusion has poor results, with an overall survival of less than 50% when hydrops is associated. Those cases without hydrops presented the best evolution, but the indication in these cases remains controversial. We present a dichorionic diamniotic twin pregnancy at 29 weeks' with right pleural effusion and hydrops, in which one fetus presented diaphragm inversion and mediastinal shift, both considered as ultrasonographic signs of thoracic hypertension. A thoracoamniotic shunt reversed these signs immediately. Nevertheless, 1 week after, these signs and skin edema reappeared and the effusion increased, leading to a second thoracocenthesis for drainage and intrathoracic pressure measurement. Amniotic fluid and intrathoracic pressures were found at 21 and 39 mm Hg, respectively. A cesarean section was performed and the catheter was found to be obliterated by cellular detritus of leukocytes and fibrin. Our conclusion is that ultrasonographic signs of elevated thoracic pressure are a keystone for the shunt indication, and that this could be corroborated by direct measurements. This can be an important parameter for precise indication of thoracoamniotic shunting., ((c) 2007 S. Karger AG, Basel.)

Chorioamniotic membrane separation (CMS) means that the close attachment of amniotic and chorionic membranes is disrupted, usually following the traumatic entry into the amniotic cavity including fetal therapy. We report a case of twin-twin transfusion syndrome receiving fetoscopic guide laser therapy at gestational age of 19 weeks with partial CMS detected at the fifth week (gestational age of 24 weeks) after surgery and resealed 1 week later. There was no more CMS noted till delivery at gestational age of 34 weeks 6 days. CMS after fetoscope surgery may reseal spontaneously., ((c) 2007 S. Karger AG, Basel.)

In this study we aimed to set up an in vitro culture of the rabbit amnion in order to support in vivo fetal membrane healing capacity following fetoscopy. Fetal membranes were collected from a mid-gestational rabbit, and cultured on collagen support material for 14 days. 34 rabbits at 22-23 days gestational age (GA) underwent fetoscopy. The entry site was randomly allocated to 4 closure technique study groups: group I, human amnion membrane (n = 23); group II, collagen foil (n = 16); group III, collagen plug (n = 19), and group IV, collagen plug with cultured amnion cells (n = 19). In all groups membrane access sites were additionally sealed with fibrin sealant, and the myometrium was closed with sutures. Fetal survival, amnion membrane integrity, and the presence of amniotic fluid were evaluated at 30 days GA. Cultures showed good survival in the collagen support material. Increased cellularity, survival and proliferations were observed. The amnion at the access site resealed in 58-64% of cases in groups II-IV, but none of the tested techniques was significantly better than the other. Histological examination indirectly revealed the anatomic repair of the membranes, since no entrapment of the membranes could be demonstrated in the myometrial wound.

Background: Intrathoracic shunt dislodgement is a rare, although significant, complication of pleuro-amniotic shunting. In a previously reported case, the abnormally located shunt caused constriction of the pulmonary hilum resulting in neonatal death., Cases: Three cases of severe fetal pleural effusion treated by pleuro-amniotic shunting at 33, 26 and 30 weeks of gestation and complicated by dislodgement of the shunt into the fetal chest are presented. These cases were collected from three fetal medicine referral centers, where 15 pleuro-amniotic shunts have been inserted in a total of 13 fetuses (11 unilateral and 2 bilateral procedures), giving prevalence for this complication of 20%. The indication for shunting was severe bilateral pleural effusions and hydrops in one case, and unilateral pleural effusion with marked mediastinal shift in 2 others. Intrathoracic dislodgement was diagnosed by ultrasound at 33, 29 and 39 weeks, and the infants were delivered at 35, 34 and 39 weeks of gestation, respectively. There were no complications associated with the malposition of the shunt within the fetal chest and clinical follow-up from 10 months to 2 years of age has demonstrated asymptomatic infants., Conclusion: The complication of intrathoracic dislodgement of a pleuro-amniotic shunt can be recognized prenatally by ultrasound. Prevention of this complication seems difficult, but if recognized, a conservative approach appears to be a sensible management option. Since the material of the shunt is inert, its presence within the pleural cavity is unlikely to be associated with an inflammatory foreign body response and, therefore, it should not cause long-term pulmonary complications., (Copyright (c) 2005 S. Karger AG, Basel.)

Objective: To determine whether fetuses that underwent thoracoamniotic shunt placement for treatment of pleural effusion (PE) or macrocystic congenital cystic adenomatoid malformation (CCAM) have an improved outcome as compared with an untreated population., Methods: A retrospective review from a single tertiary center was performed using thoracoamniotic shunt placement to treat PE or macrocystic CCAM between 1998 and 2001. Thoracoamniotic shunts were used on 26 occasions in 19 pregnancies., Results: The average gestational age at the diagnosis of PE and CCAM was 22 + 4 and 20 + 0 weeks, respectively. Shunts were offered in pregnancies complicated by hydrops or at significant risk for pulmonary hypoplasia. Shunts were placed at 26 + 2 weeks (average) and 23 + 1 weeks (average) in the PE and CCAM groups, respectively. In CCAM patients, the mean pre- and postshunting mass volumes were 50.5 and 25.7 cm(3), representing a 51% reduction in mass volume following shunt placement. In the PE group, the average delivery age was 33 + 5 weeks, with an average shunt placement to delivery time of 7 + 3 weeks. In the CCAM group, the average delivery was 33 + 3 weeks, with an average shunt placement to delivery time of 10 + 2 weeks. The postnatal survival rates were 67% (6/9) and 70% (7/10) in the PE and CCAM groups, respectively., Conclusions: (1) Thoracoamniotic shunts should be considered as a treatment option for selected PE or macrocystic CCAM fetuses with hydrops or a significant risk for pulmonary hypoplasia; (2) the neonatal survival with shunting was similar for PE and CCAM groups and was improved as compared with literature reports, and (3) fetuses with CCAM presented earlier with hydrops than those with PE. Successful shunting resulted in a prolongation of pregnancy into the 3rd trimester in both groups., (Copyright 2004 S. Karger AG, Basel)

Thoracoamniotic shunting has been described as having a beneficial role in the antenatal management of primary pleural effusions in singleton pregnancies. We report a case of a twin pregnancy in which progressive pleural effusions and hydrops were diagnosed in one of the fetuses at 16 weeks of gestation. An initial evaluation ruled out underlying genetic and anatomic abnormalities in both twins. At 19 weeks gestation, the first procedure of bilateral thoracoamniotic shunting was performed in the affected fetus, subsequent to which the lungs re-expanded and the hydrops resolved. Three additional shunt replacements and one therapeutic amniocentesis were required on follow-up. At 35 weeks, labor was induced. The first fetus (healthy) was delivered vaginally and the second fetus (affected) was delivered by cesarean section. Both neonates are healthy at one year follow-up., (Copyright 2000 S. Karger AG, Basel.)

Objective: To report our results of the measurement of the amniotic fluid pressure in twin-to-twin transfusion syndrome before and after amniodrainage and the separate measurement in the cavity of the stuck twin and the recipient, respectively, in 2 cases., Method: Six patients with severe TTS underwent therapeutic amniodrainage. Amniotic fluid pressure was obtained before and after reduction of the fluid volume. In 2 of these cases we were able to calculate the pressure separately in both amniotic cavities, the donors (stuck twin) and the recipients., Results: In all cases of TTS, amniotic pressure was high before and reduced after amniodrainage. The pressure was equal in the donors and in the recipients cavity., Conclusion: Amniotic pressure seems to be high in TTS, but equal in both cavities. Therefore, amniotic septostomy is probably of low value, especially if a high-risk monoamniotic situation is artificially created., (Copyright 2000 S. Karger AG, Basel.)

We report our experience in a woman with a twin pregnancy. The patient suffered severe Escherichia coli chorioamnionitis and the outcomes were different between the two babies after birth. The first baby had only a mild infection, but the second suffered sepsis and subsequent perinatal death. These differences in outcome appeared to be due to amniotomy performed for the first baby after late labor stage I to augment uterus contractions. Removal of infectious amniotic fluid from the amniotic cavity might thus have prevented the spread of the chorioamnionitis. E. coli sometimes causes severe infection during pregnancy and the perinatal period. In this case, a large number of enteropathogenic E. coli (serotype O-6) was cultured from blood, stool, pharyngeal swab, gastric juice and puncture fluid from the thoracic cavity of the second baby. O-6 is classified an enterotoxigenic strain mainly causing diarrhea because of endotoxin released from bacteria. O-6 has not hitherto been reported as a cause of severe infection in chorioamnionitis and perinatal sepsis., (Copyright 2000 S. Karger AG, Basel.)

Introduction: Amniotic septostomy has been described as a method to treat twin-twin transfusion syndrome. A case report of 3 patients treated in this way is described., Case Report: Three subsequent patients, who presented with twin-twin transfusion syndrome, were treated by amniotic septostomy. All 3 showed initial improvement in the amniotic fluid volume and mobility of the donor fetus. However, all three pregnancies were lost within 5 days of the amniotic septostomy due to ruptured membranes and premature labour., Conclusion: In our experience, amniotic septostomy did not improve the pregnancy outcome in twin-twin transfusion syndrome. Possible reasons for this are discussed., (Copyright 1999 S. Karger AG, Basel)

Alimentary tract duplication cysts are rarely diagnosed in utero. We report two fetal patients that presented with intrathoracic alimentary tract duplication cysts, mediastinal shift and hydrops. In one fetus, the cyst continued into the fetal abdomen and pelvis through a left diaphragmatic hernia. Despite successful ultrasound-guided needle aspirations in both fetuses, there was rapid reaccumulation of the fluid and recurrence of the mediastinal shift, prompting the placement of a thoracoamniotic shunt. In one fetus, there was rapid resolution of the mediastinal shift with the disappearance of the hydrops within 2 weeks. The second fetus suffered an intrauterine demise 2 days after the shunt placement. Postnatal resection in the surviving infant revealed a large cyst consistent with an intrathoracic duplication of the stomach. The autopsy of the second fetus revealed an intrathoracic duplication cyst of the stomach and proximal small intestine.

Objective: To report our experience with intentional puncture of the intervening membrane ('septostomy') for the treatment of the twin oligohydramnios-polyhydramnios sequence (TOPS)., Methods: 12 patients were diagnosed with TOPS based on ultrasonographic findings. A 20- to 22-gauge spinal needle was used to puncture the membrane between the twins without any attempt at amnioreduction in 9 patients, while the procedure was combined with amnioreductions in 3 patients., Results: Gestational age was 23.1 +/- 3.3 weeks at the time of septostomy and 31.1 +/- 4.4 weeks at delivery. Rapid accumulation of fluid around the 'stuck' fetus occurred in all cases following a single procedure. Three of the 24 fetuses died in utero and 1 died on the fifth day of life, for a combined survival of 83.3%. In the survivors, the septostomy to delivery interval ranged between 0.6 and 13 weeks (mean +/- SD 8.3 +/- 4.8)., Conclusion: Amniotic septostomy is a promising new method for the management of TOPS and is associated with survival rates that are better than, or comparable to, more invasive modalities. A multicenter trial comparing septostomy to other modalities is warranted.

Objective: To discuss the necessity of close sonographic surveillance of small, isolated fetal pleural effusions as well as the selection criteria and benefits of in utero therapy utilizing thoracoamniotic shunts., Methods: High-resolution ultrasound, fetal echocardiography, amniocentesis for viral cultures, cordocentesis, and thoracocentesis were performed to evaluate the underlying etiology of a unilateral pleural effusion. A Rodeck Rocket shunt was placed following sudden, rapid progression of the effusion and onset of severe fetal hydrops., Results: Placement of the thoracoamniotic shunt resulted in near-complete drainage of the pleural effusion with normalization of intrathoracic anatomic relationships, subsequent complete resolution of fetal hydrops, and excellent postnatal outcome., Conclusions: Apparently stable, small pleural effusions can progress rapidly to severe hydrops and need to be followed closely. In appropriately evaluated and selected cases, thoracoamniotic shunt placement may result in resolution of fetal hydrops and prevent intrauterine fetal death.

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.

Vesicoamniotic shunting is typically reserved for treatment of fetal obstructive uropathy. We report a case of congenital megacystis without anatomic urinary obstruction in whom a vesicoamniotic shunt was used to improve fetal hemodynamics as measured by Doppler velocimetry.

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 large contained urinoma measuring 12 x 8 x 8 cm3 was diagnosed in the left kidney of a female fetus at 28 weeks gestation. Analysis of fluid from serial punctures of the cyst indicated that renal function on that side was poor, but because ultrasound findings of the thorax suggested lung growth impairment, a shunt was inserted. After 3 weeks the shunt dislocated into the fetal abdominal cavity and urinous ascites developed. The ascites was found to have resolved spontaneously on ultrasonography 3 weeks later. The infant was delivered at 37 weeks gestation and appeared normal except for bilateral pelvi-ureteric obstruction. This was much worse in the left kidney, the renal function of which was only 10% of normal. A left nephrectomy was carried out when the infant was 6 months old, when the inner part of the shunt was discovered in perinephric fibrous tissue. The renal pelvis was greatly enlarged and fibrotic; no site of rupture could be identified. The infant's respiratory function was normal. This case suggests the use of intrauterine shunting to decompress a large unilateral urinoma resulting in marked elevation of the fetal diaphragm to prevent impairment of fetal lung growth.

Respiratory status was assessed at a median of 12 (range 3-60) months in 17 infants who had undergone pleuro-amniotic shunting to drain fetal pleural effusions. The shunts had been inserted at a median of 29 weeks (range 21-35) gestation; 12 fetuses were hydropic at the time of shunting. In all 17 cases effective chronic drainage of the pleural effusions was achieved. Respiratory status at follow-up was assessed by documentation of respiratory symptoms and measurement of functional residual capacity (FRC) by a helium gas dilution technique. Six infants suffered from recurrent respiratory symptoms, this incidence (37%) did not differ significantly from that found in a control group. The mean FRC of the study population was 28 ml/kg (range 19-34 ml/kg), only 2 infants' FRCs were below the reference range. These results suggest that pleuro-amniotic shunting may, be effective drainage of pleural effusion and hence prevention of chronic antenatal intrathoracic compression, avoid impairment of antenatal lung growth.

Thoraco-amniotic shunting was performed in 51 singleton pregnancies for decompression and chronic drainage of fetal pleural effusions (n = 47), pericardial effusion (n = 1), or pulmonary cysts (n = 3). Five fetuses had chromosomal defects and in 4 the parents elected termination of pregnancy. All 18 non-hydropic fetuses and 14 of the 28 with hydrops survived. Thoraco-amniotic shunting is useful for diagnostic evaluation and treatment of fetuses with pathologic collection of intrathoracic fluid.