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Objective: A trial comparing prenatal with postnatal open spina bifida (OSB) repair established that prenatal surgery was associated with better postnatal outcome. However, in the trial, fetal surgery was carried out through hysterotomy. Minimally invasive approaches are being developed to mitigate the risks of open maternal-fetal surgery. The objective of this study was to investigate the impact of a novel neurosurgical technique for percutaneous fetoscopic repair of fetal OSB, the skin-over-biocellulose for antenatal fetoscopic repair (SAFER) technique, on long-term postnatal outcome., Methods: This study examined descriptive data for all patients undergoing fetoscopic OSB repair who had available 12- and 30-month follow-up data for assessment of need for cerebrospinal fluid (CSF) diversion and need for bladder catheterization and ambulation, respectively, from eight centers that perform prenatal OSB repair via percutaneous fetoscopy using a biocellulose patch between the neural placode and skin/myofascial flap, without suture of the dura mater (SAFER technique). Univariate and multivariate logistic regression analyses were used to examine the effect of different factors on need for CSF diversion at 12 months and ambulation and need for bladder catheterization at 30 months. Potential cofactors included gestational age at fetal surgery and delivery, preoperative ultrasound findings of anatomical level of the lesion, cerebral lateral ventricular diameter, lesion type and presence of bilateral talipes, as well as postnatal findings of CSF leakage at birth, motor level, presence of bilateral talipes and reversal of hindbrain herniation., Results: A total of 170 consecutive patients with fetal OSB were treated prenatally using the SAFER technique. Among these, 103 babies had follow-up at 12 months of age and 59 had follow-up at 30 months of age. At 12 months of age, 53.4% (55/103) of babies did not require ventriculoperitoneal shunt or third ventriculostomy. At 30 months of age, 54.2% (32/59) of children were ambulating independently and 61.0% (36/59) did not require chronic intermittent catheterization of the bladder. Multivariate logistic regression analysis demonstrated that significant prediction of need for CSF diversion was provided by lateral ventricular size and type of lesion (myeloschisis). Significant predictors of ambulatory status were prenatal bilateral talipes and anatomical and functional motor levels of the lesion. There were no significant predictors of need for bladder catheterization., Conclusion: Children who underwent prenatal OSB repair via the percutaneous fetoscopic SAFER technique achieved long-term neurological outcomes similar to those reported in the literature after hysterotomy-assisted OSB repair. © 2021 The Authors. Ultrasound in Obstetrics & Gynecology published by John Wiley & Sons Ltd on behalf of International Society of Ultrasound in Obstetrics and Gynecology., (© 2021 The Authors. Ultrasound in Obstetrics & Gynecology published by John Wiley & Sons Ltd on behalf of International Society of Ultrasound in Obstetrics and Gynecology.)

Objective: We have described previously our percutaneous fetoscopic technique for the treatment of open spina bifida (OSB). However, approximately 20-30% of OSB defects are too large to allow primary skin closure. Here we describe a modification of our standard technique using a bilaminar skin substitute to allow closure of large spinal defects. The aim of this study was to report our clinical experience with the use of a bilaminar skin substitute and a percutaneous fetoscopic technique for the prenatal closure of large OSB defects., Methods: Surgery was performed between 24.0 and 28.9 gestational weeks with the woman under general anesthesia, using an entirely percutaneous fetoscopic approach with partial carbon dioxide insufflation of the uterine cavity, as described previously. If there was enough skin to be sutured in the midline, only a biocellulose patch was placed over the placode (single-patch group). In cases in which skin approximation was not possible, a bilaminar skin substitute (two layers: one silicone and one dermal matrix) was placed over the biocellulose patch and sutured to the skin edges (two-patch group). The surgical site was assessed at birth, and long-term follow-up was carried out., Results: Percutaneous fetoscopic OSB repair was attempted in 47 consecutive fetuses, but surgery could not be completed in two. Preterm prelabor rupture of membranes (PPROM) occurred in 36 of the 45 (80%) cases which formed the study group, and the mean gestational age at delivery was 32.8 ± 2.5 weeks. A bilaminar skin substitute was required in 13/45 (29%) cases; in the remaining 32 cases, direct skin-to-skin suture was feasible. There were 12 cases of myeloschisis, of which 10 were in the two-patch group. In all cases, the skin substitute was located at the surgical site at birth. In five of the 13 (38.5%) cases in the two-patch group, additional postnatal repair was needed. In the remaining cases, the silicone layer detached spontaneously from the dermal matrix (on average, 25 days after birth), and the lesion healed by secondary intention. The mean operating time was 193 (range, 83-450) min; it was significantly longer in cases requiring the bilaminar skin substitute (additional 42 min on average), although the two-patch group had similar PPROM rate and gestational age at delivery compared with the single-patch group. Complete reversal of hindbrain herniation occurred in 68% of the 28 single-patch cases and 33% of the 12 two-patch cases with this information available (P < 0.05). In four cases there was no reversal; half of these occurred in myeloschisis cases., Conclusions: Large OSB defects may be treated successfully in utero using a bilaminar skin substitute over a biocellulose patch through an entirely percutaneous approach. Although the operating time is longer, surgical outcome is similar to that in cases closed primarily. Cases with myeloschisis seem to have a worse prognosis than do those with myelomeningocele. PPROM and preterm birth continue to be a challenge. Further experience is needed to assess the risks and benefits of this technique for the management of large OSB defects. Copyright © 2018 ISUOG. Published by John Wiley & Sons Ltd., (Copyright © 2018 ISUOG. Published by John Wiley & Sons Ltd.)

Objective: To determine, by expert consensus, the essential substeps of fetoscopic laser surgery (FLS) for twin-twin transfusion syndrome (TTTS) that could be used to create an authority-based curriculum for training in this procedure among fetal medicine specialists., Methods: A Delphi survey was conducted among an international panel of experts (n = 98) in FLS. Experts rated the substeps of FLS on a five-point Likert-type scale to indicate whether they considered them to be essential, and were able to comment on each substep, using a dedicated online platform accessed by the invited tertiary care facilities that specialize in fetal therapy. Responses were returned to the panel until consensus was reached (Cronbach's α ≥ 0.80). All substeps that were rated  ≥ 4 by 80% of the experts were included in the evaluation instrument., Results: After the first iteration of the Delphi procedure, a response rate of 74% (73/98) was reached, and in the second and third iterations response rates of 90% (66/73) and 81% (59/73) were reached, respectively. Among a total of 81 substeps rated in the first round, 21 substeps had to be re-rated in the second round. Finally, from the initial list of substeps, 55 were agreed by experts to be essential. In the third round, the 18 categorized substeps were ranked in order of importance, with 'coagulation of all anastomoses that cross the equator' and 'determination of fetoscope insertion site' as the most important., Conclusions: A total of 55 substeps of FLS for TTTS were defined by a panel of experts to be essential in the procedure. This list is the first authority-based evidence to be used in the development of a final training model for future fetal surgeons., (Copyright © 2014 ISUOG. Published by John Wiley & Sons Ltd.)

Objective: This study aimed to analyze perinatal outcome in monoamniotic (MA) pregnancies that underwent antenatal surgical interventions for fetal complications., Methods: Review of all MA pregnancies treated with antenatal surgical interventions in three fetal treatment centers between 2000 and 2013. Indications were twin-twin transfusion syndrome, twin reversed arterial perfusion sequence, discordant anomalies, or elective reduction. We analyzed associations between indication, type of intervention, perinatal survival, and gestational age (GA) at birth and compared our results with a systematic review of the literature., Results: Fifty-eight MA pregnancies were included. Median GA at treatment was 18.0 weeks (range: 13.1-33.0). Procedures included cord coagulation plus transection (n = 42), cord coagulation without transection (n = 7), laser coagulation of placental anastomoses (n = 7), and one case each with interstitial laser and radiofrequency ablation. Median GA at birth was 34 weeks (range 16.0-41.0), and 75% (53/71) of fetuses intended to survive indeed survived. Literature review included 20 articles, reporting on a total of 45 cases of surgically treated MA pregnancies, showing similar outcome results., Conclusion: We present the largest series concerning surgical interventions in complicated MA pregnancies. Despite being rare in experienced hands, a 75% survival is achieved. Collaboration between centers, data sharing, and benchmarking may further improve outcome., (© 2014 John Wiley & Sons, Ltd.)

Operative fetoscopy is a surgical approach to the in utero treatment of fetal or placental abnormalities. The approach is on the basis of combined use of ultrasound and endoscopy and minimally invasive access into the womb. Operative fetoscopy is associated with significantly less risk of preterm birth, neonatal morbidity, and maternal morbidity as compared with open fetal surgery. A wide variety of fetal conditions have been surgically managed via operative fetoscopy, particularly complicated monochorionic twin gestations. The aim of this chapter is to review the current status and future direction of operative fetoscopy for the treatment of complicated monochorionic twin pregnancies.

A case of prenatal diagnosis of bilateral congenital cystic adenomatoid malformation of the lung (CCAM) is presented. The differential diagnosis among CCAM, pulmonary sequestration, bronchial obstruction, and laryngeal atresia is discussed. The role of diagnostic fetoscopy and fetal lung biopsy as an adjunct to ultrasound is addressed.

Until recently, fetoscopic laser surgery to seal the placental anastomoses that cause severe twin-to-twin transfusion syndrome has been available in only a few centres worldwide. The technique typically takes a long time to learn. We have used a dedicated Internet Protocol (IP) connection for tele-education to assist the introduction of fetoscopic laser surgery to Australia. During the implementation of the international telemedicine link, there were multiple clinical and technical problems, which were eventually overcome. The quality of images and of video-sequences was comparable to that supported by an ISDN connection. Pictures of live surgery performed by an expert in Florida, USA, were transmitted and viewed by a novice team in Brisbane, Australia. The Australian team has performed 19 fetoscopic laser operations to date. Preliminary results are comparable to those from centres that have performed over 100 procedures.

We describe a case in which telesurgical consultation from Tampa, Florida, USA was used to accomplish operative fetoscopy in Santiago, Chile for the treatment of a twin pregnancy involving an acardiac twin. The procedure was successful and a healthy infant was delivered at 37.5 weeks. Operative fetoscopy, a surgical approach to correct birth defects in utero via combined ultrasound and endoscopy, is only available in a handful of centers worldwide. Telesurgery makes use of telecommunication to allow a surgeon at a primary operating site to consult with another experienced surgeon for complex surgical cases. This case illustrates the potential for ultrasound and telesurgery to expand the horizons of operative fetoscopy.

Objective: The purpose of this report is to document endoscopically the occurrence of unintentional piercing of the dividing membrane in a monochorionic twin pregnancy with twin-twin transfusion syndrome (TTTS)., Materials and Methods: The dividing membrane was visualized endoscopically during laser therapy in a patient with TTTS who had previously undergone therapeutic amniocentesis. The donor twin had no visible bladder, but the amniotic fluid volume had normalized after amniocentesis., Results: The suspected membrane perforation was identified endoscopically during laser surgery. Five areas of vascular communications were identified and photocoagulated., Conclusions: Sonographic evidence of normalization of the amniotic fluid volumes after therapeutic amniocentesis with persistent nonvisualization of the bladder of the donor twin should not be interpreted as a sign of improvement of TTTS, but rather as indirect evidence of unintentional perforation of the dividing membrane. This complication can be confirmed endoscopically.

Objective: To describe the prenatal diagnosis of trichothiodystrophy (TTD) through endoscopically-guided fetal eyebrow biopsy., Materials and Methods: A 32-year-old patient, gravida 4, para 3, with a history of 2 previous infants affected with TTD was referred at 17(5)/(7) weeks for fetal hair biopsy. DNA repair studies had been normal in the previous children. Four 1-mm biopsies were obtained from the external aspect of the fetal eyebrows under direct endoscopic guidance. Fetal hair samples were assessed with polarized microscopy, electron microscopy, hematoxylin and eosin staining, and were also sent for analysis of sulfur content (cystine levels)., Results: The fetal eyebrows were the only adequate source of hair in the early second trimester. The biopsy samples yielded adequate material for all tests. Polarized microscopy showed characteristic banding patterns, but trichoschisis was not apparent. Cystine levels (19 micromol/l) in the biopsy sample were significantly lower than an age-matched (fresh spontaneous abortion) control (368 micromol/l)., Conclusion: Prenatal diagnosis of TTD is possible in the second trimester through endoscopically-guided eyebrow biopsy. An adequate amount of hair is present in the eyebrows by then, and the disease is already manifest. Analysis of sulfur content of the hair samples is preferred over polarized or electron microscopy, as many classic microscopic findings of TTD may not be present in the early second trimester., (Copyright 2000 S. Karger AG, Basel.)

Selective laser photocoagulation of communicating vessels (SLPCV) is an application of minimally invasive endoscopic fetal surgery (i.e., surgical fetoscopy) performed for severe cases of twin-to-twin transfusion syndrome (TTTS), a complication of monochorionic twin pregnancies. Advances in ultrasound and endoscopy have aided the identification and treatment of this potentially lethal or fatal condition. Surgical fetoscopy, an innovation in the field of surgery and maternal-fetal medicine, benefits patients by reducing the morbidity and mortality associated with TTTS, with minimal risks to the mother.

Malpositioning of the fingers is a characteristic anomaly in fetuses with trisomy 18. The defect results in part from muscle variations along the radial margin of forearm and hand, absence of the thenar muscles, anomalous tendons and attachments among the forearm groups, and fusions among the arm flexor group. These variations result in radial or ulnar displacement of the tendons of extensor digitorum and digiti minimi, with overlapping of the fourth and fifth fingers radially and second finger in an ulnar direction. The ontogeny of these changes is unknown. We performed serial transabdominal thin-gauge fetoscopy in a patient with increased nuchal thickening at 12 weeks of pregnancy at the time of genetic testing and again at 14 weeks at the time of termination of pregnancy. Changes in the positioning of the fingers were not apparent at 12 weeks, but were evident at 14 weeks. The findings were beyond the resolution of ultrasound. We conclude that malpositioning of the fingers in trisomy 18 occurs some time between 12 and 14 weeks of gestation. Noninvasive confirmation of these findings may be possible with new and improved ultrasound imaging capabilities or perhaps with three-dimensional ultrasound.

Objective: Our purpose was to describe the endoscopic characteristics of the site of rupture in vivo in patients with spontaneous premature rupture of membranes., Study Design: Patients with preterm premature rupture of membranes between 16 and 26 weeks of gestation, without evidence of intra-amniotic infection, and with a normal karyotype underwent transabdominal endoscopic examination of the amniotic cavity. Subsequently, an amniopatch of a combination of platelets and cryoprecipitate to seal the membrane defect was administered. The study was approved by the Institutional Review Board of St. Joseph's Hospital in Tampa, Florida, and all patients gave written informed consent., Results: Four patients underwent endoscopic examination and amniopatch administration; three had spontaneous preterm premature rupture of membranes, and in the other the membranes ruptured after an early amniocentesis. The location of the site of rupture was over the internal os in the 3 cases with spontaneous preterm premature rupture of membranes. This area was normal in the patient with iatrogenic preterm premature rupture of membranes. The longer the time between preterm premature rupture of membranes and fetoscopy, the larger and less defined was the site of rupture. The amniopatch restored amniotic integrity for a maximum of 72 hours., Conclusions: This is the first in vivo endoscopic visualization of the site of spontaneous rupture of membranes from within the uterine cavity. The defect is located over the internal cervical os in patients with spontaneous preterm premature rupture of membranes. There appear to be time-related changes in the morphologic characteristics of the site of rupture. Endoscopic visualization of the site of rupture has the potential for improving our understanding of spontaneous preterm premature rupture of membranes and in the development of possible therapeutic alternatives.

Percutaneous fetal cystoscopy was performed in a male fetus with ultrasonographic evidence of lower urinary tract obstruction at 19 weeks of gestation. The diagnosis of posterior urethral valves was confirmed. Percutaneous endoscopic fulguration of the valves was successfully performed at 22 weeks of gestation, and urethral patency was established. This case illustrates the feasibility of performing diagnostic and therapeutic endoscopic procedures within the human fetus for the management of a congenital anomaly. While we believe that fetal cystoscopy may improve our diagnostic, prognostic, and therapeutic capabilities in the management of fetuses with lower obstructive uropathy, studies are needed to establish the actual value, risks, and limitations of this new approach in fetal medicine.

Objective: Our goal was to become adept at performing laparoscopic procedures within a fluid medium and at using miniature instruments in a small animal model., Study Design: Adult female New Zealand rabbits underwent carbon dioxide laparoscopy while they were under general anesthesia with the use of 2 mm instruments. The abdominal cavity was filled with lactated Ringer's solution. Visualization of the intraabdominal organs and surgical procedures were performed below the fluid level., Results: Excellent visualization of the abdominal organs below the fluid level was obtained. Several surgical tasks were accomplished, including cutting and coagulation of the uterine horns with monopolar electrocautery, creation of a defect in the mesovarium and mesometrium, extracorporeal knot tying, and intraabdominal cutting of suture material. Relatively high amounts of energy were needed during electrocautery within the liquid medium (> 25 W). Occasional fluid and gas leakage through the skin punctures was prevented with accessory clamps., Conclusion: Hydrolaparoscopy can be performed in the rabbit with miniature instruments. As it simulates the human intraamniotic environment, it is a useful model for the development of operative fetoscopy.

The introduction of transabdominal fiberoptic thin-gauge visualization of the embryo or fetus in the first trimester allows an earlier diagnosis of congenital anomalies currently beyond the resolution of ultrasound. In addition, it has the true potential of providing access to the fetal circulation at an early age, an accomplishment that would have enormous diagnostic and therapeutic implications. In the second and third trimester, operative fetoscopic techniques promise to open a new frontier in the diagnosis and management of fetal surgical and medical conditions.

Objectives To compare the technical characteristics and perinatal outcomes of selected twin-twin transfusion syndrome (TTTS) cases treated with primary umbilical cord occlusion (UCO) of the donor vs. recipient twin. Methods TTTS patients who underwent primary UCO via ultrasound endoscopic-guided umbilical cord ligation (UCL) or endoscopic umbilical cord photocoagulation (UCP) were eligible for the study. Primary UCO was offered only if a discordant lethal anomaly was present or by patient choice in TTTS Stages III/IV on the cord of the fetus with abnormal Doppler studies or hydrops. Results Primary UCO was performed in 25 TTTS cases between July 1997 and June 2002. UCO was performed in six (24%) donor twins and 19 (76%) recipient twins. UCL was performed in 23 cases and UCP in two cases. Overall, 22 (88%) patients had liveborn infants. There were no differences in cotwin survival (100% vs. 84.2%), median gestational age at delivery (34.8 vs. 33.8 weeks) and preterm premature rupture of membranes rate (16.6% vs. 15.7%) whether the donor or recipient twin, respectively, was the subject of UCO. However, two-trocar access (50% vs. 5.3%; P = 0.03) and amnioinfusion (83.3% vs. 0%; P <0.001) were required more often in the UCO of the donor than of the recipient twin, respectively. Operating time (75 vs. 40 mm) was significantly longer in UCO of the donor twin (P = 0.04). UCP was more likely to be used in occlusion of the donor's cord (33%) than of the recipient's cord (0%; P = 0.05). Conclusion Primary UCO of the donor twin is technically more demanding than that of the recipient twin, but perinatal outcomes of the cotwins are comparable. [ABSTRACT FROM AUTHOR]

Objective To investigate the role of Doppler studies in predicting individual fetal demise in patients scheduled for selective laser photocoagulation of communicating vessels (SLPCV) for twin-twin transfusion syndrome (TTTS). Methods Doppler studies of the umbilical artery, umbilical vein, ductus venosus, tricuspid valve regurgitation and middle cerebral artery were performed in the donor and recipient twins before and 24 hours after SLPCV. Results were analyzed cross-sectionally and longitudinally. As multiple comparisons were made, an a priori alpha rejection was set at P < 0.001. Results One hundred and ten consecutive patients were available for analysis. Overall fetal survival was 68.6% (151/220) with at least one survivor in 88.2% (97/110) of cases. Absent or reversed end-diastolic velocity in the umbilical artery of the donor twin was the only preoperative Doppler result predictive of intrauterine fetal demise (IUFD) (10/15, 66.7%, P < 0.001). Postoperatively, reversed flow during atrial contraction in the ductus venosus of the donor twin showed a trend towards prediction of IUFD of this fetus (4/5, 80%, P = 0.007). No other Doppler studies, including the longitudinal analyses, were predictive of IUFD. Conclusions Our data suggest that preoperative absent or reversed end-diastolic velocity in the umbilical artery may be useful in predicting individual fetal demise of the donor twin in TTTS patients scheduled for SLPCV. This may reflect the role of decreased individual placental mass that may be associated with some donor twins. The inability of other Doppler studies to predict individual IUFD may be explained preoperatively by the effect of the interfetal vascular connections on the individual Doppler signals and postoperatively by the effect of surgery or the timing of the assessment. Our findings may be important in patient counseling, in furthering understanding of the disease, and perhaps in improving surgical technique. [ABSTRACT FROM AUTHOR]

Presents information on a study which compared selective laser photocoagulation of communicating vessels (S-LPCV) with non-S-LPCV of placental vessels in twin-to-twin transfusion syndrome (TTTS). Basis of LPCV in TTTS. Characteristics of the pregnancy of the patients; Outcome of the pregnancy of the patients.

Objective: Our aim was to describe the treatment of iatrogenic previable premature rupture of membranes with the intra-amniotic injection of platelets and cryoprecipitate (amniopatch).Study Design: Patients with iatrogenic previable premature rupture of membranes and without evidence of intra-amniotic infection underwent transabdominal intra-amniotic injection of platelets and cryoprecipitate through a 22-gauge needle. The study was approved by the Institutional Review Board of St Joseph's Hospital in Tampa, Florida, and all patients gave written informed consent.Results: Seven patients with iatrogenic preterm premature rupture of membranes underwent placement of an amniopatch. Membrane sealing was verifiable in 6 of 7 patients. Three patients had iatrogenic preterm premature rupture of membranes after operative fetoscopy, 3 cases were after genetic amniocentesis, and 1 was after diagnostic fetoscopy. Three pregnancies progressed well, with restoration of the amniotic fluid volume and no further leakage. Two patients had unexplained fetal death despite successful sealing. One case of bladder outlet obstruction had no further leakage, but oligohydramnios persisted and did not allow unequivocal documentation of sealing. One patient miscarried from twin-twin transfusion, but the amniotic cavity was sealed.Conclusions: Iatrogenic preterm premature rupture of membranes can be treated effectively with an amniopatch. The technique is simple and does not require knowledge of the exact location of the defect. Unexpected fetal death from the procedure may be attributable to vasoactive effects of platelets or indigo carmine. Although the appropriate dose of platelets and cryoprecipitate needs to be established, the amniopatch may mean that iatrogenic preterm premature rupture of membranes no longer needs to be considered a devastating complication of pregnancy. [ABSTRACT FROM AUTHOR]