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Obstetrics and Gynecology Aug 2023To review and perform a meta-analysis of observational studies that examined associations between prenatal cannabis exposure and major structural birth defects. (Meta-Analysis)
Meta-Analysis
OBJECTIVE
To review and perform a meta-analysis of observational studies that examined associations between prenatal cannabis exposure and major structural birth defects.
DATA SOURCES
Information sources included Google Scholar, BIOSIS, PubMed/MEDLINE, EMBASE CINAHL, and ClinicalTrials.gov.
METHODS OF STUDY SELECTION
Study titles and abstracts were reviewed with Abstrackr software. We included observational studies that examined the risk of major structural birth defects in people who used cannabis during pregnancy compared with those who had not used cannabis. We excluded case reports, ecologic studies, conference abstracts, manuscript preprints, studies designed to examine effects of cannabis used concurrently with other drugs, and studies that included synthetic cannabinoids. This process yielded 23 studies that analyzed data from birth years 1968-2021.
TABULATION, INTEGRATION, AND RESULTS
We clustered and meta-analyzed measures of association for birth defects by anatomic group. Eleven articles reported an association between cannabis use and the risk of a nonspecific outcome (eg, congenital anomaly). We estimated a pooled odds ratio of 1.33 (95% CI 1.14-1.56) and a pooled adjusted odds ratio (aOR) of 1.22 (95% CI 1.00-1.50). Anatomic groups examined were cardiac (nine studies), oral cleft (three studies), digestive (four studies), genitourinary (three studies), musculoskeletal (seven studies), and nervous system (five studies). Across most outcomes, we reported positive pooled unadjusted associations that were usually attenuated after the inclusion of only adjusted estimates. Two specific anomalies, with limited data, had pooled effect estimates that did not attenuate to the null after adjustment: Ebstein anomaly (two studies, aOR 2.19, 95% CI 1.25-3.82) and gastroschisis (five studies, aOR 2.50, 95% CI 1.09-5.740).
CONCLUSION
Studies examining associations between prenatal exposure to cannabis and major structural birth defects were heterogeneous. Most published effect estimates were unadjusted and scored low on our risk-of-bias assessment. Overall, we found inconsistent evidence to suggest that prenatal cannabis exposure is associated with birth defects. However, findings related to specific anomalies should be considered in further research.
SYSTEMATIC REVIEW REGISTRATION
PROSPERO, CRD42022319041.
Topics: Pregnancy; Female; Humans; Cannabis; Prenatal Exposure Delayed Effects; Odds Ratio; Bias
PubMed: 37473409
DOI: 10.1097/AOG.0000000000005252 -
Journal of the American Society of... Feb 2024Severe neonatal Ebstein's anomaly (EA) and tricuspid valve dysplasia (TVD) are associated with high perinatal morbidity and mortality. The authors recently demonstrated...
BACKGROUND
Severe neonatal Ebstein's anomaly (EA) and tricuspid valve dysplasia (TVD) are associated with high perinatal morbidity and mortality. The authors recently demonstrated left ventricular (LV) dysfunction and dyssynchrony to be prevalent in affected newborns and to contribute to poor outcomes. The aim of this study was to investigate the impact of patent ductus arteriosus (PDA) closure, spontaneous or surgical ligation, or right ventricular exclusion (Starnes procedure) on LV performance in neonatal EA and TVD.
METHODS
Neonates with EA or TVD encountered from 2004 to 2018 at three institutions were identified. Pre- and postoperative LV function was assessed using two-dimensional, Doppler-derived deformation (six-segment vector velocity imaging) and two measures of mechanical dyssynchrony (the SD of time to peak and global dyssynchrony index), and values were compared using paired t test analysis or the Wilcoxon rank sum test.
RESULTS
Before the intervention, LV function was impaired in the PDA (n = 18) and Starnes (n = 6) groups and was similar between groups. After PDA closure, LV performance did not change. After the Starnes procedure, however, LV function, including synchrony, improved significantly: fractional area change from 45 ± 5% to 58 ± 8% (P = .003), global circumferential strain from -18.2 ± 5.0% to -32.5 ± 5.5% (P = .01), cardiac index from 1.9 ± 0.3 to 3.9 ± 1.5 L/min/m (P = .05), and circumferential strain dyssynchrony (dyssynchrony index from 0.19 ± 0.09 to 0.04 ± 0.02 [P = .009] and SD of time to peak from 59.8 ± 18.5 to 29.9 ± 8.2 [P = .02]).
CONCLUSION
The Starnes procedure results in early improvements in LV dysfunction and dyssynchrony, not observed after PDA closure in neonatal severe EA and TVD, which may benefit critically unwell neonates.
Topics: Pregnancy; Female; Humans; Infant, Newborn; Ebstein Anomaly; Tricuspid Valve; Heart Defects, Congenital; Heart Valve Diseases; Ventricular Dysfunction, Left
PubMed: 38013061
DOI: 10.1016/j.echo.2023.11.019 -
The Journal of Thoracic and... Aug 2023
Commentary: Ventricular function improvement after the cone for Ebstein anomaly: It is time to incorporate magnetic resonance studies into every long-term postoperative protocol.
Topics: Humans; Ebstein Anomaly; Tricuspid Valve; Cardiac Surgical Procedures; Ventricular Function; Magnetic Resonance Spectroscopy
PubMed: 33494914
DOI: 10.1016/j.jtcvs.2020.12.106 -
Journal of Clinical Medicine Jul 2023The aim of the present review is to highlight the strengths and limitations of echocardiographic parameters and scores employed to predict favorable outcome in complex... (Review)
Review
Echocardiographic Parameters for Risk Prediction in Borderline Right Ventricle: Review with Special Emphasis on Pulmonary Atresia with Intact Ventricular Septum and Critical Pulmonary Stenosis.
The aim of the present review is to highlight the strengths and limitations of echocardiographic parameters and scores employed to predict favorable outcome in complex congenital heart diseases (CHDs) with borderline right ventricle (RV), with a focus on pulmonary atresia with intact ventricular septum and critical pulmonary stenosis (PAIVS/CPS). A systematic search in the National Library of Medicine using Medical Subject Headings and free-text terms including echocardiography, CHD, and scores, was performed. The search was refined by adding keywords "PAIVS/CPS", Ebstein's anomaly, and unbalanced atrioventricular septal defect with left dominance. A total of 22 studies were selected for final analysis; 12 of them were focused on parameters to predict biventricular repair (BVR)/pulmonary blood flow augmentation in PAIVS/CPS. All of these studies presented numerical (the limited sample size) and methodological limitations (retrospective design, poor definition of inclusion/exclusion criteria, variability in the definition of outcomes, differences in adopted surgical and interventional strategies). There was heterogeneity in the echocardiographic parameters employed and cut-off values proposed, with difficultly in establishing which one should be recommended. Easy scores such as TV/MV (tricuspid/mitral valve) and RV/LV (right/left ventricle) ratios were proven to have a good prognostic accuracy; however, the data were very limited (only two studies with <40 subjects). In larger studies, RV end-diastolic area and a higher degree of tricuspid regurgitation were also proven as accurate predictors of successful BVR. These measures, however, may be either operator and/or load/pressure dependent. TV Z-scores have been proposed by several authors, but old and heterogenous nomograms sources have been employed, thus producing discordant results. In summary, we provide a review of the currently available echocardiographic parameters for risk prediction in CHDs with a diminutive RV that may serve as a guide for use in clinical practice.
PubMed: 37510714
DOI: 10.3390/jcm12144599 -
Heart (British Cardiac Society) Nov 2023Bioprosthetic valves are often used for pulmonary valve replacement (PVR) and tricuspid valve replacement (TVR) because of concerns about mechanical valve thrombosis in...
BACKGROUND
Bioprosthetic valves are often used for pulmonary valve replacement (PVR) and tricuspid valve replacement (TVR) because of concerns about mechanical valve thrombosis in the right heart. The purpose of this study was to assess prosthetic valve function and outcomes (prosthetic valve dysfunction, reoperations and major bleeding events) after mechanical PVR and TVR and to compare these to bioprostheses implanted in the same positions.
METHOD
Case-control study of adults with congenital heart disease that underwent mechanical TVR or PVR (2003-2021) at Mayo Clinic Rochester, Minnesota. For each mechanical prosthesis, we identified two patients that received bioprosthesis in the same position (1:2 matching).
RESULTS
We identified 48 consecutive patients that underwent mechanical PVR (n=39, age 32 (26-38) years, men 22 (56%)) and/or mechanical TVR (n=17, age 36 (31-42) years, men 9 (53%)), as control group of 78 patients (age 30 (24-36) years, men 44 (56%)) and 34 patients (age 34 (29-39) years, men 18 (53%)) that underwent bioprosthetic PVR and TVR, respectively. The most common diagnoses in patients that received mechanical prosthesis were: tetralogy of Fallot (n=14, 19%), aortic stenosis status post Ross operation (n=11, 23%), truncus arteriosus (n=5, 11%), atrioventricular canal defect (n=4, 8%), Ebstein anomaly (n=3, 6%), double outlet right ventricle (n=2, 4%), valvular pulmonic stenosis (n=2, 4%). Compared with the bioprosthesis group, the mechanical prosthesis group had lower temporal increase in Doppler systolic mean gradient after PVR (∆ -1±2 vs 3±2 mm Hg, p<0.001) and Doppler diastolic mean gradient after TVR (∆ 0±1 vs 2±1 mm Hg, p=0.005). The mechanical prosthesis group also had lower risk of prosthetic valve dysfunction after PVR (1.0% vs 2.8% /year, p=0.02) and after TVR (2.6% vs 4.3% /year, p=0.008), but higher risk of major bleeding events (2.2% vs 0.1% /year, p<0.001).
CONCLUSIONS
Patients that received right-sided mechanical valve prostheses had lower temporal increase in valve gradient, lower risk of prosthetic valve dysfunction, but higher risk of bleeding complications compared with those that underwent right-sided bioprosthetic valve implantation.
Topics: Adult; Male; Humans; Heart Valve Prosthesis Implantation; Case-Control Studies; Heart Defects, Congenital; Heart Valve Prosthesis; Bioprosthesis; Retrospective Studies; Hemorrhage; Treatment Outcome
PubMed: 37407219
DOI: 10.1136/heartjnl-2023-322666 -
Journal of Clinical Medicine Apr 2024: Ebstein anomaly (EA) is a rare congenital heart disease characterized by the apical displacement of the tricuspid leaflets, creating an enlarged functional right...
A Twenty-Year Follow-Up of Adults with Ebstein Anomaly with Special Focus on Supraventricular Arrhythmias, Supraventricular Arrhythmias and Effectiveness of Catheter Ablation in 20-Year Follow-Up of Adults with Ebstein Anomaly.
: Ebstein anomaly (EA) is a rare congenital heart disease characterized by the apical displacement of the tricuspid leaflets, creating an enlarged functional right atrium. Supraventricular arrhythmias (SVA) are common, and catheter ablation remains challenging. SVA is considered a risk factor for sudden cardiac death in this population. Still, there are very few real-life data on the impact of SVA treated invasively or conservatively on a patient's prognosis. We aimed to analyze the incidence of SVA in adults with EA, evaluate the effectiveness of catheter ablation, and analyze the impact of SVA and catheter ablation on survival in this population. : 71 pts (median age 53 years; range 24-84 years) with EA were evaluated retrospectively from 1988 to 2020. Forty patients (56.3%) had SVA, and eighteen of them (45.0%) required at least one catheter ablation (35 procedures in total). Indications for ablation were mostly intra-atrial reentrant tachycardia (IART) and atrioventricular reentrant tachycardia (AVRT) (14 pts [77.8% and 9 pts [50.0%], respectively. IART and AVRT coexisted in nine pts. One patient suffered from persistent atrial fibrillation. Procedural effectiveness was reported in 28 (80%) cases; over a longer follow-up (mean 12.6 ± 5.4 years), only eight (44.4%) patients were completely free from SVA after the first ablation. In total, 10 patients (14%) died due to cardiovascular events. There was no difference in survival between patients with or without SVA ( = 0.9) and between ablated and non-ablated EA individuals ( = 0.89). : Supraventricular arrhythmia is frequent in adults with Ebstein anomaly. Patients often require more than one catheter ablation but eventually become free from arrhythmias. The imaging parameters assessed by echocardiography or cardiac magnetic resonance do not seem to be associated with ablation outcomes. The impact of supraventricular arrhythmia itself or treatment with radiofrequency ablation is questionable and should be thoroughly investigated in this population.
PubMed: 38610805
DOI: 10.3390/jcm13072039 -
The Journal of Maternal-fetal &... Dec 2023Ebstein anomaly (EA) is a cardiac malformation with highly variable presentation and severity with limited perinatal management options. We present incorporation of...
OBJECTIVE
Ebstein anomaly (EA) is a cardiac malformation with highly variable presentation and severity with limited perinatal management options. We present incorporation of fetal lung measurements into a multidisciplinary evaluation for counseling and predicting postnatal outcomes in patients with severe EA.
METHODS
Five fetuses with severe fetal EA were reviewed. Third trimester sonographic observed/expected total lung area (O/E TLA) and lung to head ratio (O/E LHR), fetal MRI total fetal lung volume ratio (O/E-TFLV), echocardiographic cardio-thoracic ratio (CT ratio), sonographic estimated fetal weight (EFW) by Hadlock formula and presence of hydrops, were used to guide perinatal management.
RESULTS
Three of five had appropriate fetal growth, were delivered at term in a cardiac operative suite, and underwent immediate intervention with good neonatal outcomes. Two had severe fetal growth restriction (FGR), CT ratios > 0.8 and O/E LHR and TLA < 25%. One of which delivered prematurely with neonatal demise and one suffered in utero demise at 34 weeks.
CONCLUSIONS
FGR, hydrops, increased CT ratio and reduced O/E LHR and TFLV are potential prognosticators of poor outcomes in severe EA, and should be validated in larger cohorts that would allow for a statistical analysis of the predictive utility of these measurements.
Topics: Pregnancy; Infant, Newborn; Female; Humans; Hernias, Diaphragmatic, Congenital; Ebstein Anomaly; Lung; Fetus; Edema; Ultrasonography, Prenatal; Retrospective Studies; Gestational Age
PubMed: 37904503
DOI: 10.1080/14767058.2023.2271626 -
Archives of Gynecology and Obstetrics Apr 2024Ebstein anomaly (EA) and tricuspid valve dysplasia (TVD) represent uncommon congenital malformations of the tricuspid valve. The purpose of this study is to report on...
PURPOSE
Ebstein anomaly (EA) and tricuspid valve dysplasia (TVD) represent uncommon congenital malformations of the tricuspid valve. The purpose of this study is to report on current perinatal outcomes of EA/TVD in our center and to investigate clinical and fetal echocardiographic predictors of perinatal mortality.
METHODS AND RESULTS
We performed a retrospective study among fetuses diagnosed from January 2014 to December 2023. Clinical and echocardiographic data were obtained from hospital records of Research and Education Hospital. The primary outcome was perinatal mortality. Of 21 fetuses diagnosed, there were 1 lost to follow-up, 1 termination, and 7 demises. In the live-born cohort of 12 live-born patients, 2 died before discharge, yielding an overall perinatal mortality of 50%. The median gestational age at diagnosis was 23 for non-survivors and 24 weeks for survivors. Birth weight was lower in non-survivors (2430 g vs 2990 g). Tricuspid insufficiency severity varied insignificantly. Non-survivors exhibited higher rates of hydrops, functional atresia, and absent antegrade flow (p < 0.05). Two infants with severe tricuspid insufficiency and congenital abnormalities died postnatally. The limited dataset enables further analysis for a predictive model. Notably, all non-survivors displayed hydrops, functional atresia, and absent antegrade flow, hindering definitive determination of the most impactful parameter on survival estimation.
CONCLUSION
Perinatal mortality remains notably elevated in fetuses with EA/TVD. The individuals at the highest risk are those with antegrade flow loss and functional atresia of pulmonary valve, this high-risk subgroup could benefit from targeted interventions, such as novel prenatal therapies or a more comprehensive perinatal approach involving optimized timing of delivery and postnatal interventional strategies.
PubMed: 38683395
DOI: 10.1007/s00404-024-07509-y -
Current Problems in Cardiology Jan 2024Complex congenital heart disease (CCHD) is a group of heart defects present at birth. Some imaging methods can support the diagnosis of these pathologies, such as... (Review)
Review
Complex congenital heart disease (CCHD) is a group of heart defects present at birth. Some imaging methods can support the diagnosis of these pathologies, such as echocardiography, computed tomography, and magnetic resonance imaging. This study aimed to perform a bibliometric analysis of the top 100 articles cited on CCHD. Articles from 2013 to 2023 found in Scopus were scanned using 15 CCHD topics titles crossed with echocardiography, computed tomography, and magnetic resonance imaging. Duplicated manuscripts were eliminated using Microsoft Excel software, and the publications were ranked according to their citation count. This study selected and analyzed the top 100 most cited English-language papers. The top 100 most cited publications accumulated 16,563 citations. The manuscript with the most citations obtained 1086, representing 6.55% of the total citations. The year 2014 had the highest number of publications, with 27 papers. The first authors had affiliations from 44 countries; the United States was the country that contributed the most, with 54 manuscripts. Boston Children's Hospital was the institution that provided more articles to the top 100. Finally, the Ebstein anomaly was the topic with the highest number of citations. This study identified the 100 most cited on CCHD, and the results obtained can provide practical guidance to clinicians and researchers to familiarize themselves with the most influential publications in this field.
Topics: Infant, Newborn; Child; Humans; United States; Bibliometrics; Tomography, X-Ray Computed; Magnetic Resonance Imaging
PubMed: 37553062
DOI: 10.1016/j.cpcardiol.2023.102028 -
Prenatal Diagnosis Jun 2024Ebstein anomaly (EA) is a rare congenital cardiac malformation associated with high perinatal mortality. In this systematic review and meta-analysis, we aimed to... (Meta-Analysis)
Meta-Analysis Review
Ebstein anomaly (EA) is a rare congenital cardiac malformation associated with high perinatal mortality. In this systematic review and meta-analysis, we aimed to investigate the outcomes of pregnancies affected by EA or tricuspid valve dysplasia (TVD) with circular shunt, focusing on two prenatal management approaches: (1) expectant management (EM) and (2) transplacental non-steroidal anti-inflammatory drugs (NSAID) therapy. We searched PubMed, Scopus, and Web of Science systematically from its inception until June 2023. The random-effect model was used to pool the data. Heterogeneity was assessed using the I value. Twenty-one studies with a total of 610 fetuses with EA/TVD with circular shunt were included in the synthesis, of which 17 studies (583 fetuses) were on EM and 4 studies (27 fetuses) used transplacental NSAID therapy. The NSAID group had higher rates of moderate to severe tricuspid regurgitation, hydrops, and pericardial effusion on prenatal ultrasound compared with the EM group. However, ductal constriction was achieved in 81% of NSAID cases, mitigating the disease pathophysiology, although 65% of them experienced oligohydramnios. Notably, the NSAID group showed significantly higher rates of live birth (86%) and survival to hospital discharge (89%) compared with the EM group (67% and 43%, respectively). Despite these promising results, it's important to acknowledge that the number of cases treated with NSAIDs was small, with limited safety data. Therefore, caution is advised in interpreting these findings, and patients considering NSAID therapy should be informed about these limitations. Future multicenter studies are necessary to further explore the safety and effectiveness of NSAID therapy in this particular population.
Topics: Humans; Ebstein Anomaly; Pregnancy; Female; Anti-Inflammatory Agents, Non-Steroidal; Watchful Waiting; Ultrasonography, Prenatal
PubMed: 37902170
DOI: 10.1002/pd.6446