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  • Ovid Technologies (Wolters Kluwer Health)  (7)
  • 1
    Online-Ressource
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    Abstract 17047: Pathophysiologic Progression From the Second to Third Trimester in Fetuses With Ebstein's Anomaly or Tricuspid Valve Dysplasia: Are Early Echocardiographic Features Reliable Indicators of Late Gestation Status?
    Ovid Technologies (Wolters Kluwer Health) ; 2015
    In:  Circulation Vol. 132, No. suppl_3 ( 2015-11-10)
    Details
    In: Circulation, Ovid Technologies (Wolters Kluwer Health), Vol. 132, No. suppl_3 ( 2015-11-10)
    Kurzfassung: Background: Ebstein’s anomaly and tricuspid valve dysplasia (EA/TVD) are associated with high perinatal mortality. Poor hemodynamic status in both early and late gestation is associated with worse outcome. However, it is not known whether EA/TVD fetuses with more favorable physiology earlier in gestation progress to more severe disease in 3 rd trimester. The goal of this study was to evaluate whether initial echocardiographic indices in fetuses with EA/TVD presenting at gestational age (GA) 〈 24 wks are reliable indicators of physiologic status later in pregnancy. Methods: This multi-center, retrospective study included fetuses diagnosed with EA/TVD from 2005-11 at 23 centers. Patients with initial diagnosis at 〈 24 wks gestation and with ≥2 fetal echocardiograms ≥4 wks apart were included. A core laboratory analyzed the echocardiograms. Markers of poor outcome were defined as absence of antegrade flow across the pulmonary valve, pulmonary valve regurgitation (PR), cardiothoracic ratio 〉 0.48, left ventricular (LV) dysfunction or TV annulus Z score 〉 5.6. Results: The study included 51 fetuses. Median GA at diagnosis was 21 wks [18-24], and the median duration from 2 nd to 3 rd trimester echocardiograms was 12 wks [4-18]. There were 21 fetuses (41%) with 〉 2 markers of poor outcome at 〈 24 wks, which increased to 32 (63%) in later gestation (p=0.001). Nine of 27 fetuses (33%) with antegrade pulmonary blood flow on first echocardiogram developed anatomic or functional pulmonary atresia during follow-up, and 7 of 39 fetuses (18%) without PR initially developed it later. LV dysfunction was present in 2 patients at 〈 24 wks but present in 14 (37%) later (p 〈 0.01). The only variable associated with worsening physiology, from 〈 2 markers of poor outcome in 2 nd trimester to ≥2 markers in 3 rd trimester, was larger TV annulus Z-score (4.1±1.0 vs. 2.4±1.9, p=0.008). Conclusion: These results confirm a wide spectrum of disease presentation and progression in fetuses with EA/TVD. Thus, it is imperative to assess these fetuses serially. In this cohort, there were no early markers of progression other than larger TV annulus Z-score. Great care must be taken in counseling prior to 24 wks, as the absence of factors associated with poor outcome early in pregnancy may be falsely reassuring.
    Materialart: Online-Ressource
    ISSN: 0009-7322 , 1524-4539
    URL: Article
    DOI: 10.1161/circ.132.suppl_3.17047
    Sprache: Englisch
    Verlag: Ovid Technologies (Wolters Kluwer Health)
    Publikationsdatum: 2015
    ZDB Id: 1466401-X
    Standort Signatur Einschränkungen Verfügbarkeit
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  • 2
    Online-Ressource
    Online-Ressource
    Abstract 17203: Postnatal Management of Fetuses With Ebstein Anomaly or Tricuspid Valve Dysplasia in the Current Era: A Multi-center Study
    Ovid Technologies (Wolters Kluwer Health) ; 2015
    In:  Circulation Vol. 132, No. suppl_3 ( 2015-11-10)
    Details
    In: Circulation, Ovid Technologies (Wolters Kluwer Health), Vol. 132, No. suppl_3 ( 2015-11-10)
    Kurzfassung: Background: A recent multi-center study of perinatal outcome in fetuses with Ebstein anomaly or tricuspid valve dysplasia (EA/TVD) found that 1/3rd of live-born patients (pts) died prior to hospital discharge. The purpose of this study was to explore differences in postnatal management and the relationship to outcome. Methods: This 23-center, retrospective study included 243 fetuses with EA/TVD from 2005 to 2011. Neonatal procedure (NP) was defined as surgery or interventional catheterization (cath) prior to discharge. Associations between postnatal management and outcome at discharge were explored. Results: Of 176 live-born pts, 7 received comfort care only, 11 died 〈 24 hrs of life, and 4 had insufficient data. Among 154 remaining pts, 38 (25%) did not survive to discharge. Pts who required ECMO at any point (n=18) had 83% mortality. More than half of pts (54%) did not have an NP, 34% had surgery, 8% had interventional cath, and 4% had both. The median age at 1st NP was 6 days (quartiles: 1-11). Survival did not differ between pts who had an NP and those who did not (70% vs. 80%; p=0.19) or between pts who had surgery and those who did not (68% vs. 80%; p=0.09). However, mortality differed by NP performed and whether pulmonary regurgitation, an indicator of high risk, was present prenatally (Figure). No pts with a right ventricular exclusion (RVE) died. Of 49 surviving neonates with ≥1 procedure, 28 (57%) were palliated with a shunt or RVE and 21 (43%) had a biventricular circulation. Thus, in total, 86 of 154 live-born pts (56%) survived with a biventricular circulation: 65 with medical management only and 21 with ≥1 NP. Conclusion: Among live-born pts diagnosed with EA/TVD in utero, a variety of postnatal management strategies were employed with overall poor outcomes. If surgery beyond PDA ligation is necessary, then RVE or other palliative procedure may need to be considered. A prospective, multi-center study utilizing a management algorithm would help elucidate the optimal strategy.
    Materialart: Online-Ressource
    ISSN: 0009-7322 , 1524-4539
    URL: Article
    DOI: 10.1161/circ.132.suppl_3.17203
    Sprache: Englisch
    Verlag: Ovid Technologies (Wolters Kluwer Health)
    Publikationsdatum: 2015
    ZDB Id: 1466401-X
    Standort Signatur Einschränkungen Verfügbarkeit
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  • 3
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    Abstract 11382: Left and Right Ventricular Strain Mechanics in Fetal Arrhythmia
    Ovid Technologies (Wolters Kluwer Health) ; 2021
    In:  Circulation Vol. 144, No. Suppl_1 ( 2021-11-16)
    Details
    In: Circulation, Ovid Technologies (Wolters Kluwer Health), Vol. 144, No. Suppl_1 ( 2021-11-16)
    Kurzfassung: Introduction: Assessment of ventricular mechanics in fetuses with tachyarrhythmia poses significant challenges. Strain analysis measures myocardial deformation and has been shown to be a very sensitive marker of ventricular systolic dysfunction compared to traditional measurements. Global and regional myocardial deformation has not been well described in fetuses with arrhythmias. Hypothesis: We hypothesize that left and right ventricular myocardial mechanics are impaired in fetuses with tachyarrhythmias compared to normal fetuses in sinus rhythm. Methods: Retrospective review of fetuses with tachyarrhythmia (groups 1 and 2) and gestational age (GA) matched normal fetuses with normal cardiac anatomy (group 3) from 2000 to 2018 at a single institution, with IRB approval. Fetuses with isolated ectopy and congenital heart disease were excluded from all groups. Strain analysis was performed using VVI software. Arrhythmia type, medications, cardiovascular profile score and postnatal outcome were collected. Results: 18 fetuses with tachyarrhythmia with GA between 21 to 36.5 weeks were analyzed; 18 fetuses of similar GA with normal cardiac anatomy in sinus rhythm served as controls. 3 fetuses were delivered close to term and did not have strain assessment in sinus rhythm. Both LV global longitudinal strain (-4.44 ± 2.82%) and strain rate (-1.24 ± 0.85%) and RV global longitudinal strain (-6.76 ± 2.09%) and strain rate (-1.41 ± 0.55%) were reduced in tachyarrhythmia (Group 1) and recovered when in sinus rhythm (Group 2) but still reduced compared to controls (Group 3). Global longitudinal velocity was not different between patients in Group 2 and 3 (Table & Fig 1). Conclusions: Strain parameters remain abnormal in fetuses with tachyarrhythmia despite conversion to sinus rhythm when compared to GA matched controls. Serial evaluation of strain will help guide therapy and monitor ventricular function in this patient population.
    Materialart: Online-Ressource
    ISSN: 0009-7322 , 1524-4539
    URL: Article
    DOI: 10.1161/circ.144.suppl_1.11382
    Sprache: Englisch
    Verlag: Ovid Technologies (Wolters Kluwer Health)
    Publikationsdatum: 2021
    ZDB Id: 1466401-X
    Standort Signatur Einschränkungen Verfügbarkeit
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  • 4
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    Abstract 17117: Prevalence of Systemic AV Valve Regurgitation in Patients With Congenitally Corrected Transposition of the Great Arteries: Anatomic versus Physiologic Repair
    Ovid Technologies (Wolters Kluwer Health) ; 2020
    In:  Circulation Vol. 142, No. Suppl_3 ( 2020-11-17)
    Details
    In: Circulation, Ovid Technologies (Wolters Kluwer Health), Vol. 142, No. Suppl_3 ( 2020-11-17)
    Kurzfassung: Background: Congenitally corrected transposition of the great arteries (ccTGA) is a rare anomaly with highly variable time of diagnosis. Depending on time of diagnosis and associated lesions, multiple therapies exist, directed towards surgical correction. Anatomic repair (AR) involving double switch or atrial switch/Rastelli and physiologic repair (PR) leaving the right ventricle as the systemic ventricle are the two main surgical options. A subset of patients without associated lesions exist who can be followed expectantly over time. Systemic atrioventricular valve (SAVV) function is a major anatomic variable, determining eligibility for double switch and durability of physiologic repair or expectant management (medical group). We therefore sought to assess the prevalence of AV valve regurgitation in the different subgroups and its determinants, hypothesizing that SAVV regurgitation (SAVVR) was reduced in the AR group. Methods: Retrospective record review coupled with prospective cross-sectional follow-up of 240 patients with ccTGA treated at our center was performed. Echocardiographic imaging review was conducted to assess SAVV function and systemic ventricular function among the 3 subgroups. Results: Of 240 patients with ccTGA, 162 met inclusion. Median follow up was 10 years (mean +/- SD 11+/- 9 years). In the AR group majority (85%) had none/mild SAVVR. Prior PA band did not influence the severity of SAVVR following double switch. In the PR group, 22% underwent SAVV repair/replacement, and 75% had none/mild SAVVR; as opposed to only 65% with none/mild SAVVR in the medical group. Only 14% of patients were free of systemic ventricle dysfunction in PR group as opposed to 72% in the AR group. Conclusions: SAVV function is preserved in the AR group. In contrast patients in the PR group had higher prevalence of more than mild SAVVR followed by patients who were managed expectantly.
    Materialart: Online-Ressource
    ISSN: 0009-7322 , 1524-4539
    URL: Article
    DOI: 10.1161/circ.142.suppl_3.17117
    Sprache: Englisch
    Verlag: Ovid Technologies (Wolters Kluwer Health)
    Publikationsdatum: 2020
    ZDB Id: 1466401-X
    Standort Signatur Einschränkungen Verfügbarkeit
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  • 5
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    Risk Factors for Mortality and Circulatory Outcome Among Neonates Prenatally Diagnosed With Ebstein Anomaly or Tricuspid Valve Dysplasia: A Multicenter Study
    Ovid Technologies (Wolters Kluwer Health) ; 2020
    In:  Journal of the American Heart Association Vol. 9, No. 21 ( 2020-11-03)
    Details
    In: Journal of the American Heart Association, Ovid Technologies (Wolters Kluwer Health), Vol. 9, No. 21 ( 2020-11-03)
    Kurzfassung: In a recent multicenter study of perinatal outcome in fetuses with Ebstein anomaly or tricuspid valve dysplasia, we found that one third of live‐born patients died before hospital discharge. We sought to further describe postnatal management strategies and to define risk factors for neonatal mortality and circulatory outcome at discharge. Methods and Results This 23‐center, retrospective study from 2005 to 2011 included 243 fetuses with Ebstein anomaly or tricuspid valve dysplasia. Among live‐born patients, clinical and echocardiographic factors were evaluated for association with neonatal mortality and palliated versus biventricular circulation at discharge. Of 176 live‐born patients, 7 received comfort care, 11 died 〈 24 hours after birth, and 4 had insufficient data. Among 154 remaining patients, 38 (25%) did not survive to discharge. Nearly half (46%) underwent intervention. Mortality differed by procedure; no deaths occurred in patients who underwent right ventricular exclusion. At discharge, 56% of the cohort had a biventricular circulation (13% following intervention) and 19% were palliated. Lower tricuspid regurgitation jet velocity (odds ratio [OR] , 2.3 [1.1–5.0], 95% CI, per m/s; P =0.025) and lack of antegrade flow across the pulmonary valve (OR, 4.5 [1.3–14.2]; P =0.015) were associated with neonatal mortality by multivariable logistic regression. These variables, along with smaller pulmonary valve dimension, were also associated with a palliated outcome. Conclusions Among neonates with Ebstein anomaly or tricuspid valve dysplasia diagnosed in utero, a variety of management strategies were used across centers, with poor outcomes overall. High‐risk patients with low tricuspid regurgitation jet velocity and no antegrade pulmonary blood flow should be considered for right ventricular exclusion to optimize their chance of survival.
    Materialart: Online-Ressource
    ISSN: 2047-9980
    URL: Article
    DOI: 10.1161/JAHA.120.016684
    Sprache: Englisch
    Verlag: Ovid Technologies (Wolters Kluwer Health)
    Publikationsdatum: 2020
    ZDB Id: 2653953-6
    Standort Signatur Einschränkungen Verfügbarkeit
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  • 6
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    Outcomes and Predictors of Perinatal Mortality in Fetuses With Ebstein Anomaly or Tricuspid Valve Dysplasia in the Current Era : A Multicenter Study
    Ovid Technologies (Wolters Kluwer Health) ; 2015
    In:  Circulation Vol. 132, No. 6 ( 2015-08-11), p. 481-489
    Details
    In: Circulation, Ovid Technologies (Wolters Kluwer Health), Vol. 132, No. 6 ( 2015-08-11), p. 481-489
    Kurzfassung: Ebstein anomaly and tricuspid valve dysplasia are rare congenital tricuspid valve malformations associated with high perinatal mortality. The literature consists of small, single-center case series spanning several decades. We performed a multicenter study to assess the outcomes and factors associated with mortality after fetal diagnosis in the current era. Methods and Results— Fetuses diagnosed with Ebstein anomaly and tricuspid valve dysplasia from 2005 to 2011 were included from 23 centers. The primary outcome was perinatal mortality, defined as fetal demise or death before neonatal discharge. Of 243 fetuses diagnosed at a mean gestational age of 27±6 weeks, there were 11 lost to follow-up (5%), 15 terminations (6%), and 41 demises (17%). In the live-born cohort of 176 live-born patients, 56 (32%) died before discharge, yielding an overall perinatal mortality of 45%. Independent predictors of mortality at the time of diagnosis were gestational age 〈 32 weeks (odds ratio, 8.6; 95% confidence interval, 3.5–21.0; P 〈 0.001), tricuspid valve annulus diameter z-score (odds ratio, 1.3; 95% confidence interval, 1.1–1.5; P 〈 0.001), pulmonary regurgitation (odds ratio, 2.9; 95% confidence interval, 1.4–6.2; P 〈 0.001), and a pericardial effusion (odds ratio, 2.5; 95% confidence interval, 1.1–6.0; P =0.04). Nonsurvivors were more likely to have pulmonary regurgitation at any gestational age (61% versus 34%; P 〈 0.001), and lower gestational age and weight at birth (35 versus 37 weeks; 2.5 versus 3.0 kg; both P 〈 0.001). Conclusion— In this large, contemporary series of fetuses with Ebstein anomaly and tricuspid valve dysplasia, perinatal mortality remained high. Fetuses with pulmonary regurgitation, indicating circular shunt physiology, are a high-risk cohort and may benefit from more innovative therapeutic approaches to improve survival.
    Materialart: Online-Ressource
    ISSN: 0009-7322 , 1524-4539
    URL: Article
    DOI: 10.1161/CIRCULATIONAHA.115.015839
    Sprache: Englisch
    Verlag: Ovid Technologies (Wolters Kluwer Health)
    Publikationsdatum: 2015
    ZDB Id: 1466401-X
    Standort Signatur Einschränkungen Verfügbarkeit
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  • 7
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    Abstract 15610: Long Term Health Related Quality of Life Outcomes Across the Therapeutic Spectrum in Patients With Congenitally Corrected Transposition of Great Arteries
    Ovid Technologies (Wolters Kluwer Health) ; 2022
    In:  Circulation Vol. 146, No. Suppl_1 ( 2022-11-08)
    Details
    In: Circulation, Ovid Technologies (Wolters Kluwer Health), Vol. 146, No. Suppl_1 ( 2022-11-08)
    Kurzfassung: Introduction: Congenitally corrected transposition of the great arteries (ccTGA) encompasses a diverse morphologic cohort with multiple treatment pathways. We characterized long term health related quality of life (HR-QoL) outcomes in adult patients with ccTGA across the therapeutic spectrum. Hypothesis: Methods: Between 1995-2020, 240 ccTGA patients were treated at the Cleveland Clinic. Of these, 59 adults were prospectively assessed with a hybrid PROMIS-10 and MacNew HR-QoL questionnaire at median follow-up of 10 years. Mental and physical scores were captured utilizing PROMIS 10 questionnaire, and social score from MacNew instrument. One-sample non-parametric sign tests evaluated deviations from referenced normative values. Differences among anatomic (AR) and physiologic repairs (PR) were assessed with Wilcoxon rank-sum test. Confidence intervals (95%) around measured means were generated from 1000 bootstrap samples. Results: Thirty one (18%) patients were female with median presentation age 26.3 years. Sixteen (27%) patients had PR, 12 (20%) had AR, 12 (20%) were medically managed, 5 (8.5%) had heart transplant and 3 (5.1%) underwent Fontan. Social scores were higher than the population norms (median= 6, P =.0002) for the overall cohort, but physical scores were lower than normative values (median = 44, P =.0183).Mental functioning was equivalent to normative values (median = 50, P =.9). AR and PR scores across domains were similar (Table 1). Conclusions: Adult patients living with ccTGA across the treatment spectrum have lower physical functioning than population norms. Despite these perceived physical impediments, social and mental functioning are unaffected. PR and AR had equivalent HR-QoL, providing important data to inform patient selection and triage.
    Materialart: Online-Ressource
    ISSN: 0009-7322 , 1524-4539
    URL: Article
    DOI: 10.1161/circ.146.suppl_1.15610
    Sprache: Englisch
    Verlag: Ovid Technologies (Wolters Kluwer Health)
    Publikationsdatum: 2022
    ZDB Id: 1466401-X
    Standort Signatur Einschränkungen Verfügbarkeit
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