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1.
J Pediatr Urol ; 2024 May 31.
Article in English | MEDLINE | ID: mdl-39232867

ABSTRACT

INTRODUCTION: Since the Management of Myelomeningocele Study (MOMS) publication in 2011 we have designed a prospective protocol for surveillance of neurogenic bladder according to the Leal da Cruz categorization system (J Urol, 2015) that guides us on treatment. Our institution was the precursor of in-utero myelomeningocele (MMC) in Latin America, so our database was biased mostly for patients that underwent fetal surgery for MMC closure. We have demonstrated that in-utero MMC closure does not improve bladder function in opposition to the data from the urological branch of the MOMS study (10), but our control group was based on a historical cohort, before the onset of in-utero MMC repair in our division (15) Since 2018, we have identified an increasing number of referrals of postnatal operated MMC patients to our group just for urological follow up. We decided then to start this prospective protocol for all neurogenic patients and provide a contemporary database to record differences in early bladder function, presence of hydronephrosis and vesicoureteral reflux, treatment, initial outcome and indication of surgery among three mains groups (in-utero MMC repair, post-natal repair and miscellaneous other cases of neurogenic bladder). We want to present preliminary data of this cohort in the period of time 2018 to 2023 (5 years). There was not an exclusion criteria, all patients with neuropathic bladder were included in this study. MATERIALS AND METHODS: We evaluated our database of all neurogenic bladder (NB) patients aged <1 year who started urological treatment in our institution from 2018. We evaluated diagnosis, age at first visit, clinical data, prevalence of hydronephrosis and (vesicoureteral reflux) VUR, bladder pattern according to the Leal da Cruz categorization system (1), treatment, time of follow-up, number of clinical visits and (urodynamic evaluation) UE performed, final bladder status and surgeries performed. RESULTS: We identified 43 were aged <1 year with a mean age of 4.5 months (median 3.5) at first urological appointment. Diagnosis was myelomeningocele in 33 patients and miscellaneous in 10. From the MMC group, 24 were operated in-utero and 9 post-natal. The initial bladder pattern in the whole group showed 23 (53.5%) high risk, 11 normal (25.6%), 5 underactive bladder (11.6%) and 4 incontinent (9.3%). Mean follow-up was 24 months, mean age at last UE (cases with minimum of 2): 37 months, mean UE per patient: 2. At present, 28 patients perform clean intermittent catheterization (CIC), 23 with anticholinergics and 15 are only under surveillance. VUR was seen in 11/43 cases: 25.6%. Urological surgery has been performed in 4 patients: bladder augmentation in 3 and vesicostomy in 1. Bladder categorization for high-risk and normal patterns were respectively 62.5% and 25% for in utero and 44.4% and 22.2% for postnatal repair and detailed statistical analysis did not present statistical significance of in utero and postnatal groups. CONCLUSION: Our study can conclude in a contemporary prospective study the findings of elevated incidence of high-risk patterns irrespectively of in-utero or post-natal repair (62.5% and 44.4%). We acknowledge that even though this is not new information and besides the still limited follow up, this cohort is one of the very few that follow and compare in one single institution with comparable UE for in-utero and post-natal MMC repair population, having also a miscellaneous group of NB as a pictorial report of also a similar initial distribution of bladder patterns. We plan to report in the future in a larger cohort the continuation of this study.

2.
World J Pediatr Congenit Heart Surg ; : 21501351241269881, 2024 Sep 10.
Article in English | MEDLINE | ID: mdl-39252613

ABSTRACT

Background: With significant advancements in fetal cardiac imaging, patients with complex congenital heart disease (CHD) carrying a high risk for postnatal demise are now being diagnosed earlier. We sought to assess an interdisciplinary strategy for delivering these children in an operating room (OR) adjacent to a cardiac OR for immediate surgery or stabilization. Methods: All children prenatally diagnosed with CHD at risk for immediate postnatal hemodynamic instability and cardiogenic shock who were delivered in the operating room (OR) between 2012 and 2023 in which the senior author was consulted were included. Results: Eight patients were identified. Six (75%) patients were operated on day-of-life zero, all requiring obstructed total anomalous pulmonary venous return (TAPVR) repair. Of these six patients, 2 (33%) required a simultaneous Norwood procedure, 2 (33%) required pulmonary artery unifocalization and modified Blalock-Taussig-Thomas shunt, and 2 (33%) patients had repair of obstructed mixed TAPVR. The remaining 2 patients potentially planned for immediate surgery had nonimmune hydrops fetalis and went into cardiogenic shock at 12 and 72 hours postnatally, requiring a novel Norwood procedure with left-ventricular exclusion for severe aortic/mitral valve insufficiency. The median ventilation and inpatient durations were 19 [IQR: 11-26] days and 41 [IQR: 32-128] days, respectively. Three(38%) patients required one or more in-hospital reoperations. Subsequent staged procedures included Glenn (n = 5), Fontan (n = 3), biventricular repair (n = 2), ventricular assist device placement (n = 1), and heart transplant (n = 1). Median follow-up was 5.7 [IQR:1.3-7.8] years. The five-year postoperative survival was 88% (n = 7/8). Conclusion: While children with these diagnoses have historically had poor survival, the strategy of birth in the OR adjacent to a cardiac OR where emergent surgery is planned is a potentially promising strategy with excellent clinical outcomes. However, this is a high-resource strategy whose feasibility in any program requires thoughtful assessment.

3.
J Pediatr Surg ; : 161656, 2024 Jul 30.
Article in English | MEDLINE | ID: mdl-39181781

ABSTRACT

BACKGROUND: The nitrofen model of congenital diaphragmatic hernia (CDH) is widely used in translational research. However, the molecular pathways associated with pulmonary hypoplasia in this model compared to the human CDH phenotype have not been well described. The aim of this study was to investigate differentially expressed genes (DEG) and signaling pathways in early stage fetal lungs in mouse and human CDH. METHODS: CDH lung tissue was obtained from human fetuses (21-23 weeks gestation) and nitrofen mouse pups (E15.5). NovaSeq Flowcell RNA-seq was performed to evaluate differentially expressed transcriptional and molecular pathways (DEGs) in fetal mice with CDH, compared with age-matched normal mouse lungs and human CDH samples. RESULTS: There were thirteen overlapping DEGs in human and mouse CDH lung samples compared to controls. These genes were involved in extracellular matrix, myogenesis, cilia, and immune modulation pathways. Human CDH was associated with an upregulation of collagen formation and extracellular matrix reorganization whereas mouse CDH was associated with an increase in muscular contraction. The most common cell types upregulated in human and mouse CDH samples were ciliated airway cells. CONCLUSIONS: This study highlights the unique gene transcriptional patterns in early fetal mouse and human lungs with CDH. These data have implications when determining the translational potential of novel therapies in CDH using nitrofen-based animal models. LEVEL OF EVIDENCE: Level IV. STUDY TYPE: Basic science/case series.

4.
Article in English | MEDLINE | ID: mdl-39045747

ABSTRACT

This review describes an in-depth analysis of the neurotoxicity associated with the anesthetic agents used during fetal surgery, intending to highlight the importance of understanding the effects of general anesthetics on the developing brain, particularly in the context of open fetal surgery, where high doses are applied to facilitate surgical access and augment uterine relaxation. We examined evidence from preclinical studies in rodents and primates, along with studies in human subjects, with the results collectively suggesting that general anesthetics can disrupt brain development and lead to long-lasting neurological deficits. Our review underscores the clinical implications of these findings, indicating an association between extensive anesthetic exposure in early life and subsequent cognitive deficits. The current standard of anesthetic care for fetal surgical procedures was scrutinized, and recommendations have been proposed to mitigate the risk of anesthetic neurotoxicity. These recommendations emphasize the need for careful selection of anesthetic techniques to minimize fetal exposure to potentially harmful agents. In conclusion, while the benefits of fetal surgery in addressing immediate risks often outweigh the potential neurotoxic effects of anesthesia, the long-term developmental impacts nevertheless warrant consideration. Our analysis suggests that the use of general anesthetics in fetal surgery, especially at high doses, poses a significant risk of developmental neurotoxicity. As such, it is imperative to explore safer alternatives, such as employing different methods of uterine relaxation and minimizing the use of general anesthetics, to achieve the necessary surgical conditions. Further research, particularly in clinical settings, is essential to fully understand the risks and benefits of anesthetic techniques in fetal surgery.

5.
Diagnostics (Basel) ; 14(14)2024 Jul 12.
Article in English | MEDLINE | ID: mdl-39061640

ABSTRACT

Timely identification of fetal conditions enables comprehensive evaluation, counseling, postnatal planning, and prenatal treatments. This study assessed the existing evidence on how social determinants of health (SDOH) influence diagnosis timing of fetal conditions appropriate for care in fetal care centers (FCCs). Eligible studies were conducted in the U.S. and published in English after 1999. We employed the Healthy People 2020 SDOH framework to categorize and analyze data from 16 studies, where 86% focused solely on congenital heart disease (CHD). Studies primarily focused on individual-level SDOH, with only 36% addressing structural-level factors. A total of 31 distinct indicators of SDOH were identified, with 68% being unique to individual studies. Indicators often varied in definition and specificity. Three studies covered all five SDOH categories in the Healthy People 2020 Framework. Studies revealed varying and often conflicting associations with SDOH indicators, with race and ethnicity being the most explored (100%), followed by socioeconomic status (69%), maternal age (57%), residence (43%), and structural factors (29%). Our findings highlight the need for more comprehensive research, including conditions beyond CHD, and the establishment of consensus on indicators of SDOH. Such efforts are necessary to gain a deeper understanding of the underlying factors driving disparities in fetal diagnosis and treatment.

6.
Fetal Diagn Ther ; : 1-9, 2024 Jul 30.
Article in English | MEDLINE | ID: mdl-39079511

ABSTRACT

INTRODUCTION: Limited data exist regarding outcomes when pre- and periviable PPROM (PPROM ≤26 weeks of gestation) occurs as a complication of fetoscopic laser surgery (FLS) for twin-twin transfusion syndrome (TTTS). METHODS: This is a retrospective cohort study of FLS cases performed at a single institution between January 2015 and May 2021. Study inclusion was limited to patients with monochorionic-diamniotic twin pregnancies complicated by TTTS who underwent FLS. Patients were grouped by pPPROM status, and further stratified to those continuing with expectant management, and outcomes were compared between groups. The primary outcome was survival to live birth of at least one twin. RESULTS: During the study period, 171 patients underwent FLS and a total of 96 (56.1%) subjects satisfied inclusion criteria. Among included subjects, 18 (18.8%) experienced pPPROM after FLS and 78 (81.2%) did not. Baseline characteristics were similar between groups. Among patients with pPPROM, 11 (61.1%) pursued expectant management and 7 (38.9%) opted for pregnancy termination. Among expectantly managed subjects, median pPPROM-to-delivery interval was 47.0 days (6.0-66.0 IQR) with a median gestational age at delivery of 29+1 weeks (24 + 4-33 + 6 IQR). Rates of survival to live birth of at least one twin (90.9% vs. 96.2% p = 0.42) were similar between those with pPPROM undergoing expectant management and those without pPPROM. Dual survivorship (45.5% vs. 78.2%, p = 0.03), perinatal survival to live birth (68.2% vs. 87.2%, p = 0.05), and perinatal survival to newborn hospital discharge (59.1% vs. 85.9%, p = <0.01) were all significantly lower among those with pPPROM. Gestational age at delivery was lower among those continuing with pregnancies complicated by pPPROM (29 + 1 vs. 32+5 weeks, p = <0.01). CONCLUSION: Survival of at least one twin to live birth remained high among those pursing expectant management after experiencing post-FLS pPPROM, suggesting that the outlook after this complication is not necessarily poor. However, this complication was associated with lower chances of dual survival and greater prematurity.

7.
Fetal Diagn Ther ; : 1-9, 2024 Jul 27.
Article in English | MEDLINE | ID: mdl-39068910

ABSTRACT

INTRODUCTION: Congenital hydrocephalus often results in irreversible and severe damage to the brain despite postnatal interventions. The potential for prenatal intervention to mitigate these deleterious effects underscores the importance of a suitable animal model. We aimed assess the results of an ultrasound-guided transuterine approach to replicate the BioGlue injection fetal hydrocephalus model. METHODS: Pregnant ewes were anesthetized at 95 days of gestation and BioGlue was injected into the fetal cisterna magna under ultrasound guidance through the uterus. Ventriculomegaly was assessed by MRI and histology. RESULTS: Nine pregnant ewes were included in the study, and their fetuses were divided into the BioGlue intervention group (n = 9 fetuses) or the control group (n = 7 fetuses) who were not injected. Although hydrocephalus was noted in 5 of 9 fetuses in the intervention group, the ability to induce hydrocephalus went from 0% to 100% in the last 3 fetuses following technical modifications. None of the controls developed hydrocephalus. Fetal brains with hydrocephalus demonstrated increased IBA1+ compared to control animals. CONCLUSIONS: While technical challenges were noted, the ultrasound-guided transuterine approach to replicate the BioGlue fetal hydrocephalus model in sheep showed consistent and reproducible results. This model offers the advantage of directly visualizing the location of the needle tip and injection of the BioGlue. This technique offers an alternative for testing novel approaches for prenatal congenital hydrocephalus treatment.

8.
Fetal Diagn Ther ; : 1-11, 2024 Jul 26.
Article in English | MEDLINE | ID: mdl-39068923

ABSTRACT

INTRODUCTION: Chorioamniotic membrane separation (CMS) is a known complication after fetal spina bifida (fSB) repair. This study's goal was to analyze women's outcomes with open fSB repair and CMS (group A) compared to the ones without (group B) and to assess the influence of CMS size and patient management. METHODS: A total of 194 women with open fSB repair at our center were included in this retrospective study. Outcomes of group A were compared to the ones of group B. Regression analysis was performed to assess risk factors for CMS. Two subgroup analyses assessed the impact of CMS size (small [A-small] vs. large [A-large]) as well as patient management (A1 = hospitalization vs. A2 = no hospitalization) on pregnancy outcomes. RESULTS: Of 194 women, 23 (11.9%) were in group A and 171 (88.1%) in group B. Preterm premature rupture of membranes (PPROMs) (69.6% vs. 24.1%, p = <0.001), amniotic infection syndrome (AIS) (22.7% vs. 7.1%, p = 0.03), histologically confirmed chorioamnionitis (hCA) (40.0% vs. 14.7%, p = 0.03), length of hospital stay (LOS) after fSB repair (35 [19-65] vs. 17 [14-27] days), and overall LOS (43 [33-71] vs. 35 [27-46] days, p = 0.004) were significantly more often/longer in group A. Gestational age (GA) at delivery was significantly lower in group A compared to group B (35.3 [32.3-36.3] vs. 36.7 [34.9-37.0] weeks, p = 0.006). Regression analysis did not identify risk factors for CMS. Subgroup analysis comparing CMS sized in group A-small versus A-large showed higher AIS rate (42% vs. 0%, p = 0.04), lower LOS (22.0 [15.5-42.5] vs. 59.6 ± 24.1, p = 0.003). Comparison of group A1 versus A2 showed longer LOS (49.3 ± 22.8 vs. 15 [15-17.5] days, p < 0.001), lower planned readmission rate (5.6% vs. 80%, p = 0.003). CONCLUSION: CMS significantly increased the risk of PPROM, AIS, hCA, caused longer LOS, and caused lower GA at delivery. Women with small CMS had higher AIS rates but shorter LOS compared to women with large CMS, while apart from LOS pregnancy outcomes did not differ regarding patient management (hospitalization after CMS yes vs. no).

9.
Rev. obstet. ginecol. Venezuela ; 84(2): 204-211, jun. 2024. ilus, tab
Article in Spanish | LILACS, LIVECS | ID: biblio-1568547

ABSTRACT

El secuestro broncopulmonar es una malformación del aparato respiratorio que consiste en tejido bronquial y pulmonar no funcionante, separado del árbol tráqueo-bronquial y alimentado por un vaso sanguíneo proveniente de la circulación sistémica. La incidencia es de 1 por cada 5000 nacimientos, comprende entre 0,15 % y 6,45 % de las patologías pulmonares. El pronóstico es, generalmente, favorable, reportándose regresión espontánea de la lesión en 50 % a 75 % de los pacientes. Puede ocasionar efecto de masa, comprimiendo el corazón y el pulmón hasta generar cambios hemodinámicos y falla cardíaca. Hay múltiples procedimientos para el tratamiento y manejo, principalmente en casos de gran tamaño y fetos hidrópicos, para mejorar el pronóstico perinatal. Se presentan los dos primeros casos de secuestro broncopulmonar tratados en Venezuela mediante fotocoagulación láser del vaso nutricio y su evolución perinatal, con sobrevida del 100 % y sin ninguna complicación registrada en el periodo perinatal(AU)


Bronchopulmonary sequestration is a malformation of the respiratory system consisting of non-functioning bronchial and pulmonary tissue, separated from the tracheo-bronchial tree and fed by a blood vessel from the systemic circulation. The incidence is 1 in 5000 births, ranging from 0.15% to 6.45% of pulmonary pathologies. The prognosis is generally favorable, with spontaneous regression of the lesion reported in 50% to 75% of patients. It can cause mass effect, compressing the heart and lung to the point of generating hemodynamic changes and heart failure. There are multiple procedures for treatment and management, mainly in large cases and hydropic fetuses, to improve perinatal prognosis. We present the first two cases of bronchopulmonary sequestration treated in Venezuela by laser photocoagulation of the nutrient vessel and their perinatal evolution, with 100% survival and without any complications recorded in the perinatal period(AU)


Subject(s)
Humans , Female , Pregnancy , Adult , Ultrasonics , Bronchopulmonary Sequestration , Laser Coagulation , Perinatology , Respiratory System
10.
J Clin Med ; 13(12)2024 Jun 18.
Article in English | MEDLINE | ID: mdl-38930102

ABSTRACT

Background: We aimed to conduct a systematic review and meta-analysis to evaluate the fetoscopic tracheal occlusion in patients with isolated severe and left-sided diaphragmatic hernia. Methods: Cochrane Library, Embase, and PubMed (Medline) databases were searched from inception to February 2024 with no filters or language restrictions. We included studies evaluating the outcomes of fetoscopic intervention compared to expectant management among patients with severe congenital diaphragmatic hernia exclusively on the left side. A random-effects pairwise meta-analysis was performed using RStudio version 4.3.1. Results: In this study, we included 540 patients from three randomized trials and five cohorts. We found an increased likelihood of neonatal survival associated with fetoscopic tracheal occlusion (Odds Ratio, 5.07; 95% Confidence Intervals, 1.91 to 13.44; p < 0.01) across general and subgroup analyses. Nevertheless, there were higher rates of preterm birth (OR, 5.62; 95% CI, 3.47-9.11; p < 0.01) and preterm premature rupture of membranes (OR, 7.13; 95% CI, 3.76-13.54; p < 0.01) in fetal endoscopic tracheal occlusion group compared to the expectant management. Conclusions: Our systematic review and meta-analysis demonstrated the benefit of fetoscopic tracheal occlusion in improving neonatal and six-month postnatal survival in fetuses with severe left-sided CDH. Further studies are still necessary to evaluate the efficacy of tracheal occlusion for isolated right-sided CDH, as well as the optimal timing to perform the intervention.

11.
Fetal Diagn Ther ; : 1-6, 2024 Jun 18.
Article in English | MEDLINE | ID: mdl-38889699

ABSTRACT

INTRODUCTION: Maternal laparotomy-assisted fetoscopic surgery for in-utero myelomeningocele repair has shown that a trans-amniotic membrane suture during fetoscopic port placement can reduce postsurgical complications. Fetoscopic laser photocoagulation (FLP) for complex twins is typically performed percutaneously without a transmembrane stitch. However, in scenarios without a placental-free window, maternal laparotomy may be used for recipient sac access. Here, we present the outcomes of our series of laparotomy-assisted FLP cases, including a trans-amniotic membrane suturing of the fetoscopic port. METHODS: Retrospective series of twin-twin transfusion syndrome or twin anemia-polycythemia sequence (TAPS) cases treated at 2 fetal centers that underwent maternal laparotomy to FLP from September 2017 to January 2023. We recorded preoperative and operative characteristics, as well as pregnancy and neonatal outcomes. RESULTS: During the study period, 9 maternal laparotomy to FLP cases were performed. Two were excluded for prior percutaneous FLP in the pregnancy. The remaining seven utilized a maternal laparotomy to trans-amniotic membrane stitch with confirmation of proper suture placement under ultrasound guidance, and all surgeries were performed with a single 10 F Check-Flo® cannula. Mean gestational age (GA) at surgery was 19.1 weeks (range 16 weeks 4 days-23 weeks 3 days), with delivery occurring at a mean GA of 35.0 weeks (range 32 weeks 0 days-37 weeks 1 day), resulting in a mean latency of 15.8 weeks, significantly longer than what is reported in the literature and our own data (mean latency for percutaneous FLP 10.2, 95% CI 9.9-10.5). Furthermore, all cases underwent iatrogenic delivery before labor onset, with the lone delivery prior to 34 weeks due to concern for post-laser TAPS. CONCLUSION: This case series of laparotomy to FLP with trans-amniotic stitch, demonstrated no cases of spontaneous preterm birth and a longer-than-expected latency from surgery to delivery. Larger studies are warranted to investigate this approach.

12.
Magn Reson Imaging Clin N Am ; 32(3): 431-442, 2024 Aug.
Article in English | MEDLINE | ID: mdl-38944432

ABSTRACT

This review covers the embryology, definition, and diagnosis of open spinal dysraphism with a focus on fetal ultrasound and MR imaging findings. Differentiating open versus closed spinal dysraphic defects on fetal imaging will also be discussed. Current fetal surgery practices and imaging findings in the context of fetal surgery are also reviewed.


Subject(s)
Magnetic Resonance Imaging , Prenatal Diagnosis , Spine , Humans , Magnetic Resonance Imaging/methods , Female , Spine/diagnostic imaging , Spine/abnormalities , Spine/surgery , Pregnancy , Prenatal Diagnosis/methods , Spinal Dysraphism/diagnostic imaging , Spinal Dysraphism/surgery , Ultrasonography, Prenatal/methods , Fetal Diseases/diagnostic imaging , Fetal Diseases/surgery
13.
Am J Obstet Gynecol ; 231(3): 365.e1-365.e15, 2024 Sep.
Article in English | MEDLINE | ID: mdl-38897340

ABSTRACT

BACKGROUND: Intraoperative blood transfer between twins during laser surgery for twin-twin transfusion syndrome can vary by surgical technique and has been proposed to explain differences in donor twin survival. OBJECTIVE: This trial compared donor twin survival with 2 laser techniques: the sequential technique, in which the arteriovenous communications from the volume-depleted donor to the volume-overloaded recipient are laser-occluded before those from recipient to donor, and the selective technique, in which the occlusion of the vascular communications is performed in no particular order. STUDY DESIGN: A single-center, open-label, randomized controlled trial was conducted in which twin-twin transfusion syndrome patients were randomized to sequential vs selective laser surgery. Nested within the trial, a second trial randomized patients with superficial anastomoses (arterioarterial and venovenous) to ablation of these connections first (before ablating the arteriovenous anastomoses) vs last. The primary outcome measure was donor twin survival at birth. RESULTS: A total of 642 patients were randomized. Overall donor twin survival was similar between the 2 groups (274 of 320 [85.6%] vs 271 of 322 [84.2%]; odds ratio, 1.12 [95% confidence interval, 0.73-1.73]; P=.605). Superficial anastomoses occurred in 177 of 642 cases (27.6%). Donor survival was lower in the superficial anastomosis group vs those with only arteriovenous communications (125 of 177 [70.6%] vs 420 of 465 [90.3%]; adjusted odds ratio, 0.33 [95% confidence interval, 0.20-0.54]; P<.001). In cases with superficial anastomoses, donor survival was independent of the timing of ablation or surgical technique. The postoperative mean middle cerebral artery peak systolic velocity was lower in the sequential vs selective group (1.00±0.30 vs 1.06±0.30 multiples of the median; P=.003). Post hoc analyses showed 2 factors that were associated with poor overall donor twin survival: the presence or absence of donor twin preoperative critical abnormal Doppler parameters and the presence or absence of arterioarterial anastomoses. Depending on these factors, 4 categories of patients resulted: (1) Category 1 (347 of 642 [54%]), no donor twin critical abnormal Doppler + no arterioarterial anastomoses: donor twin survival was 91.2% in the sequential and 93.8% in the selective groups; (2) Category 2 (143 of 642 [22%]), critical abnormal Doppler present + no arterioarterial anastomoses: donor survival was 89.9% vs 75.7%; (3) Category 3 (73 of 642 [11%]), no critical abnormal Doppler + arterioarterial anastomoses present: donor survival was 94.7% vs 74.3%; and (4) Category 4 (79 of 642 [12%]), critical abnormal Doppler present + arterioarterial anastomoses present: donor survival was 47.6% vs 64.9%. CONCLUSION: Donor twin survival did not differ between the sequential vs selective laser techniques and did not differ if superficial anastomoses were ablated first vs last. The donor twin's postoperative middle cerebral artery peak systolic velocity was improved with the sequential vs the selective approach. Post hoc analyses suggest that donor twin survival may be associated with the choice of laser technique according to high-risk factors. Further study is needed to determine whether using these categories to guide the choice of surgical technique will improve outcomes.


Subject(s)
Fetofetal Transfusion , Laser Therapy , Humans , Fetofetal Transfusion/surgery , Female , Pregnancy , Laser Therapy/methods , Adult , Arteriovenous Anastomosis
14.
J Clin Med ; 13(9)2024 Apr 30.
Article in English | MEDLINE | ID: mdl-38731178

ABSTRACT

Background: This study aims to evaluate the outcomes of fetal sacrococcygeal teratoma (SCT) submitted to prenatal interventions. Methods: We performed a systematic literature review of fetal SCT patients and compared the outcomes between open fetal surgery and percutaneous intervention. In addition, we also compared the results of SCT fetuses who did not undergo any surgical intervention (NI). Results: We identified 16 cases of open fetal surgery (OS), 48 cases of percutaneous fetal intervention (PI), and 93 NI patients. The survival rate was 56.2% in OS, 45.8% in PI (p = 0.568), and 71.0% in NI patients. The gestational age at delivery was earlier in cases where there was no survival compared to cases where the fetuses did survive across all evaluated cohorts (OS: p = 0.033, PI: p < 0.001, NI: p < 0.001). The gestational weeks at delivery in OS and PI fetuses were more similar; however, OS tended to be performed later on in pregnancy, and the affected fetuses had more severe presented findings. In our evaluation, we determined that the presence of fetal hydrops and cardiac failure had no significant impact on survival in SCT cases. In NI patients, polyhydramnios was much higher in fetuses who did not survive compared to their surviving cohorts (p < 0.001). Conclusions: In conclusion, gestational age at delivery can affect the short-term prognosis of fetuses affected with sacrococcygeal teratomas. Regardless of the mode of delivery or the necessity for intervention during the fetal period, monitoring for complications, including polyhydramnios, can prevent premature delivery.

15.
Article in English | MEDLINE | ID: mdl-38748926

ABSTRACT

OBJECTIVE: There are conflicting data on whether fetoscopic laser photocoagulation of placental anastomoses (FLP) for treating twin-to-twin transfusion syndrome (TTTS) is associated with lower rates of overall survival. The objective of this study is to characterize survival and other associated morbidity after FLP across gestational ages of FLP. METHODS: This is a secondary analysis of prospectively collected data on patients with monochorionic-diamniotic twins that had FLP for TTTS at two centers between 2011 and 2022. Patients were divided into gestational age epochs for FLP before 18 wks, 18 0/7 - 19 6/7 wks, 20 0/7 - 21 6/7 wks, 22 0/7 - 23 6/7 wks, 24 0/7 - 25 6/7 wks and after 26 wks. Demographic characteristics, sonographic characteristics of TTTS and operative characteristics were compared across the gestational age epochs. Outcomes including overall survival, preterm delivery, preterm prelabor rupture of membranes (PPROM), intrauterine fetal demise (IUFD) and neonatal demise (NND) were also compared across gestational age epochs. Multivariate analysis was performed by fitting logistic regression models for these outcomes. Kaplan-Mejer curves were constructed to compare the interval from PPROM to delivery for each gestational age epoch. RESULTS: There were 768 patients that met inclusion criteria. The dual survival rate was 61.3% for FLP performed prior to 18 weeks compared to 78.0% - 86.7% across later gestational age epochs. This appears to be related to increased rates of donor IUFD following FLP performed before, versus after 18 weeks (28.0% vs. 9.3% - 14.1%). Rates of recipient IUFD/NND and donor NND were similar regardless of gestational age of FLP. Rates of PPROM were higher for earlier FLP, ranging from 45.6% for FLP before 18 weeks to 11.9% for FLP at 24 - 26 weeks gestational age. However, the gestational age of delivery was similar across gestational age epochs with a median of 31.7 weeks. In multivariate analysis, donor loss was independently associated with FLP before 18 weeks after adjusting for selective fetal growth restriction, Quintero stage and other covariates. PPROM and PTD were also associated with FLP before 18 weeks after adjusting for cervical length, placental location, trocar size, laser energy and amnioinfusion. CONCLUSION: FLP performed at earlier gestational ages is associated with lower overall survival, which is driven by higher risk of donor IUFD, as opposed to differences in PPROM or PTD. Counseling regarding survival should account for gestational age of presentation. This article is protected by copyright. All rights reserved.

16.
Acta Obstet Gynecol Scand ; 103(8): 1550-1557, 2024 Aug.
Article in English | MEDLINE | ID: mdl-38780288

ABSTRACT

INTRODUCTION: Prenatal lower urinary tract obstruction (LUTO) is a rare and challenging condition with potential severe morbidity and mortality. Prenatal shunting methods, specifically vesicoamniotic shunting (VAS) and fetal cystoscopy, aim to manage this condition. However, comprehensive education and training are hindered by the rarity of LUTO. To address this gap, we present a low-cost 3D-printed ultrasound training model for VAS in LUTO fetuses. The aim of the study was to evaluate ultrasound and haptic fidelity of the model. MATERIAL AND METHODS: Ultrasound images of three LUTO fetuses at 12-14 weeks were utilized to create detailed 3D-printed models. Fusion360TM software generated stereo-lithography files, and the Formlabs Form3® printer, using Flexible 80A resin, produced the models. A simulation box mimicking uterine conditions and fetal anatomy was developed for testing. Ultrasound assessments determined model accuracy, and expert evaluations gauged fidelity for VAS placement. RESULTS: The 3D-printed model accurately replicated LUTO fetal anatomy, demonstrating structural integrity and realistic sonographic and haptic feedback during 20 punctures. Macroscopic visualization confirmed the model's durability and authenticity. DISCUSSION: This innovative 3D-printed model addresses the scarcity of LUTO cases and the lack of realistic training tools. Simulation models enhance skills, providing a controlled learning environment that bridges theoretical knowledge and clinical application, potentially improving patient outcomes. CONCLUSIONS: The 3D-printed training model for VAS in LUTO represents a significant advancement in surgical education, offering realistic anatomical simulation and tactile feedback. Future studies should assess its effectiveness in enhancing surgical skills and impacting patient outcomes in clinical practice.


Subject(s)
Models, Anatomic , Printing, Three-Dimensional , Ultrasonography, Prenatal , Humans , Female , Pregnancy , Urethral Obstruction/surgery , Urethral Obstruction/diagnostic imaging , Ultrasonography, Interventional/methods , Simulation Training/methods
17.
Neonatology ; : 1-7, 2024 May 23.
Article in English | MEDLINE | ID: mdl-38781940

ABSTRACT

BACKGROUND: The concept of fetal pain results from procedures conducted without anesthesia in preterm newborns and fetuses, which indicate that it is possible to examine fetal pain based on stress hormone, metabolic, and behavioral changes. Anatomical and physiological data suggest that fetuses become capable of processing nociceptive stimuli around midgestation, although the associated changes in fetal brain development remain unclear. What constitutes fetal pain remains controversial in the light of the definition of pain adopted by the International Association for the Study of Pain (IASP), which posits pain as an "unpleasant sensory and emotional experience." SUMMARY: Here, we examine the notion that human fetuses cannot "experience" pain and potential implications of this claim. We highlight the key scientific evidence related to fetal pain, including clinical studies on pain in fetuses and preterm newborns. We argue that consistent patterns of stress hormones, metabolic changes, body movements, hemodynamic changes, and pain-related facial expressions in fetuses exposed to invasive procedures overcome the need for subjective proof of pain as articulated in the IASP definition. No study to date has conclusively proven the absence of fetal pain beyond the age of viability. KEY MESSAGES: Based on the current evidence, we propose that all fetuses receive anesthesia regardless of the invasive procedures being performed to guarantee the least possible pain and physiological, behavioral, or hormonal responses without exposing the mother or her baby to unnecessary complications.

18.
Fetal Diagn Ther ; 51(4): 365-376, 2024.
Article in English | MEDLINE | ID: mdl-38569486

ABSTRACT

INTRODUCTION: In fetal surgery, successful pain management is crucial for postoperative mobilization, prophylaxis of contractions, and fast recovery. This study analyzed patient's pain experience after open fetal spina bifida (fSB) repair in comparison to pain scores after the subsequent Caesarean section (C-section). MATERIALS AND METHODS: Data were collected with a questionnaire given to 91 women, who had fSB repair and then C-section at our center between 2019 and 2022. It comprised 12 questions covering different aspects of pain experience and satisfaction with pain therapy and was answered by 67 women after fSB repair and 53 after C-section. Postoperative pain was rated on a Likert scale from 0 (slight/rarely) to 100 (strongest/always). Outcomes after fSB repair were compared to those after C-section. Additionally, subgroup analysis compared outcomes of women with different pain levels (group 1-5) after fSB repair. RESULTS: Compared to women after C-section women after fSB repair reported significantly higher maximum pain scores (MPS) (p = 0.03), higher sleep disturbance due to pain (p = 0.03), and sedation rates (p = 0.001) as side effect from pain therapy. No differences were found regarding feelings of insecurity (p = 0.20) or helplessness (p = 0.40), as well as involvement in (p = 0.3) and satisfaction with pain therapy (p = 0.5). Subgroup analysis revealed that women with higher MPS after fSB repair were significantly more often non-Caucasians (p = 0.003) and more often affected by pain while lying in bed (p = 0.007) and during mobilization (p = 0.005). Additionally, they reported higher rates of dizziness (p = 0.02) and lower satisfaction rates with pain therapy (p = 0.03). Postoperative complication rate did not differ among groups. CONCLUSION: Although women after fSB repair reported higher MPS compared to after C-section, the current pain management was generally perceived as satisfactory.


Subject(s)
Cesarean Section , Pain Measurement , Pain, Postoperative , Humans , Female , Cesarean Section/adverse effects , Adult , Pregnancy , Pain, Postoperative/etiology , Pain, Postoperative/psychology , Longitudinal Studies , Spinal Dysraphism/surgery , Spinal Dysraphism/complications , Spinal Dysraphism/psychology , Surveys and Questionnaires , Patient Satisfaction , Pain Management/methods , Cohort Studies
19.
Fetal Diagn Ther ; 51(4): 357-364, 2024.
Article in English | MEDLINE | ID: mdl-38643759

ABSTRACT

INTRODUCTION: No evidence-based protocols exist for fetal cardiac monitoring during fetoscopic myelomeningocele (fMMC) repair and intraprocedural spectral Doppler data are limited. We determined the feasibility of continuous fetal echocardiography during fMMC repair and correlated Doppler changes with qualitative fetal cardiac function during each phase of fMMC repair. METHODS: Patients undergoing fMMC repair had continuous fetal echocardiography interpreted in real-time by pediatric cardiology. Fetal data included fetal heart rate (FHR), qualitative cardiac function, mitral and tricuspid valve inflow waveforms, and umbilical artery (UA), umbilical vein (UV), ductus arteriosus (DA), and ductus venosus (DV) Dopplers. RESULTS: UA abnormalities were noted in 14/25 patients, UV abnormalities were observed in 2 patients, and DV and DA abnormalities were each noted in 4 patients. Qualitative cardiac function was normal for all patients with the exception of one with isolated left ventricular dysfunction during myofascial flap creation, concurrent with an abnormal UA flow pattern. All abnormalities resolved by the first postoperative day. CONCLUSIONS: Continuous fetal echocardiography was feasible during all fMMC repairs. Spectral Doppler changes in the UA were common during fMMC procedures but qualitative cardiac dysfunction was rare. Abnormalities in the UV, DV, and DA Dopplers, FHR, and cardiac function were less common findings.


Subject(s)
Fetoscopy , Meningomyelocele , Humans , Meningomyelocele/surgery , Meningomyelocele/diagnostic imaging , Fetoscopy/methods , Female , Pregnancy , Ultrasonography, Prenatal/methods , Fetal Monitoring/methods , Fetal Heart/diagnostic imaging , Fetal Heart/surgery , Fetal Heart/physiopathology , Echocardiography, Doppler/methods , Adult , Feasibility Studies , Umbilical Arteries/diagnostic imaging
20.
J Pediatr Surg ; 59(7): 1374-1377, 2024 Jul.
Article in English | MEDLINE | ID: mdl-38589273

ABSTRACT

BACKGROUND: The ripple effect of the Supreme Court ruling in Dobbs v. Jackson Women's Health Organization has impacted physicians and patients across numerous medical specialties. In pediatric surgery, the patient population ranges from fetus to the pregnant patient. There is a gap in the knowledge of pediatric surgeons regarding abortion laws and access. This project aims to bridge the gap by creating access to reliable resources which may be used to optimize patient care and support physicians. METHODS: We collaborated with the Reproductive Health Coalition, co-founded by the American Medical Women's Association and Doctors for America, to curate a list of resources beneficial to pediatric surgeons. RESULTS: We created a web-based toolkit with the purpose of providing easily accessible and reliable information on reproductive rights in the United States. We identified up-to-date resources on state-by-state abortion laws, legal resources, patient-centered information on obtaining abortion care, and resources for physicians interested in getting involved in advocacy. CONCLUSION: Pediatric surgery rests at a critical juncture with respect to reproductive rights in the United States. Our toolkit enables users to understand the current climate and identify next steps to advocate for patients and physicians amidst a formidable legal environment. LEVEL OF EVIDENCE: Level V.


Subject(s)
Reproductive Rights , Humans , United States , Reproductive Rights/legislation & jurisprudence , Female , Pregnancy , Pediatrics/legislation & jurisprudence , Abortion, Legal/legislation & jurisprudence , Abortion, Induced/legislation & jurisprudence
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