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  1. Article ; Online: Developing a Broader Understanding of Pediatric PAH Worldwide.

    Sullivan, Rachel T / Austin, Eric D

    Chest

    2023  Volume 163, Issue 6, Page(s) 1353–1355

    MeSH term(s) Child ; Humans ; Hypertension, Pulmonary ; Familial Primary Pulmonary Hypertension
    Language English
    Publishing date 2023-06-09
    Publishing country United States
    Document type Editorial ; Comment
    ZDB-ID 1032552-9
    ISSN 1931-3543 ; 0012-3692
    ISSN (online) 1931-3543
    ISSN 0012-3692
    DOI 10.1016/j.chest.2023.03.027
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    In stock of ZB MED Cologne/Königswinter

    Ui III Zs.45: Show issues Location:
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    bis Jg. 2021: Bestellungen von Artikeln über das Online-Bestellformular
    ab Jg. 2022: Lesesaal (EG)
    Zs.MO 349: Show issues

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  2. Article ; Online: Recent Advances in Pediatric Pulmonary Hypertension: Implications for Diagnosis and Treatment.

    Sullivan, Rachel T / Raj, J Usha / Austin, Eric D

    Clinical therapeutics

    2023  Volume 45, Issue 9, Page(s) 901–912

    Abstract: Purpose: Pediatric pulmonary hypertension (PH) is a condition characterized by elevated pulmonary arterial pressure, which has the potential to be life-limiting. The etiology of pediatric PH varies. When compared with adult cohorts, the etiology is ... ...

    Abstract Purpose: Pediatric pulmonary hypertension (PH) is a condition characterized by elevated pulmonary arterial pressure, which has the potential to be life-limiting. The etiology of pediatric PH varies. When compared with adult cohorts, the etiology is often multifactorial, with contributions from prenatal, genetic, and developmental factors. This review aims to provide an up-to-date overview of the causes and classification of pediatric PH, describe current therapeutics in pediatric PH, and discuss upcoming and necessary research in pediatric PH.
    Methods: PubMed was searched for articles relating to pediatric pulmonary hypertension, with a particular focus on articles published within the past 10 years. Literature was reviewed for pertinent areas related to this topic.
    Findings: The evaluation and approach to pediatric PH are unique when compared with that of adults, in large part because of the different, often multifactorial, causes of the disease in children. Collaborative registry studies have found that the most common disease causes include developmental lung disease and subsets of pulmonary arterial hypertension, which includes genetic variants and PH associated with congenital heart disease. Treatment with PH-targeted therapies in pediatrics is often guided by extrapolation of adult data, small clinical studies in pediatrics, and/or expert consensus opinion. We review diagnostic considerations and treatment in some of the more common pediatric subpopulations of patients with PH, including developmental lung diseases, congenital heart disease, and trisomy 21.
    Implications: The care of pediatric patients with PH requires consideration of unique pediatric-specific factors. With significant variability in disease etiology, ongoing efforts are needed to optimize treatment strategies based on disease phenotype and guide evidence-based practices.
    MeSH term(s) Pregnancy ; Adult ; Female ; Child ; Humans ; Hypertension, Pulmonary/diagnosis ; Hypertension, Pulmonary/etiology ; Hypertension, Pulmonary/therapy ; Pulmonary Arterial Hypertension ; Heart Defects, Congenital/complications ; Heart Defects, Congenital/diagnosis ; Phenotype
    Language English
    Publishing date 2023-07-28
    Publishing country United States
    Document type Journal Article ; Review
    ZDB-ID 603113-4
    ISSN 1879-114X ; 0149-2918
    ISSN (online) 1879-114X
    ISSN 0149-2918
    DOI 10.1016/j.clinthera.2023.07.001
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    Zs.MO 404: Show issues

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  3. Article: Diazoxide-associated pulmonary hypertension in a patient with noncompaction cardiomyopathy.

    Sullivan, Rachel T / Tillman, Kathryn A / Kindel, Steven J / Handler, Stephanie S

    Pulmonary circulation

    2021  Volume 11, Issue 1, Page(s) 2045894020987117

    Abstract: Development of pulmonary hypertension after initiation of diazoxide for the treatment of neonatal hyperinsulinemic hypoglycemia is a rare, but previously described association. Risk factors for development of diazoxide-associated pulmonary hypertension ... ...

    Abstract Development of pulmonary hypertension after initiation of diazoxide for the treatment of neonatal hyperinsulinemic hypoglycemia is a rare, but previously described association. Risk factors for development of diazoxide-associated pulmonary hypertension include lower gestational age and congenital heart disease. This novel case report describes an infant with noncompaction cardiomyopathy who developed pulmonary hypertension shortly after initiation of diazoxide for hyperinsulinemic hypoglycemia which resolved upon cessation of the drug. This case highlights the benefit of having pre-treatment knowledge of underlying cardiac anatomy and makes a case for routine echocardiographic screening for neonates initiating diazoxide treatment.
    Language English
    Publishing date 2021-02-03
    Publishing country United States
    Document type Case Reports
    ZDB-ID 2638089-4
    ISSN 2045-8940 ; 2045-8932
    ISSN (online) 2045-8940
    ISSN 2045-8932
    DOI 10.1177/2045894020987117
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  4. Article ; Online: Congenital Left Coronary Atresia: Diagnostic Pearls and Management Challenges in Two Children Presenting with Syncope.

    Sullivan, Rachel T / Gerardin, Jennifer F / Frommelt, Peter C / Gudausky, Todd M

    CASE (Philadelphia, Pa.)

    2021  Volume 5, Issue 5, Page(s) 280–285

    Language English
    Publishing date 2021-06-25
    Publishing country United States
    Document type Case Reports
    ISSN 2468-6441
    ISSN (online) 2468-6441
    DOI 10.1016/j.case.2021.05.003
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  5. Article: A case of acquired von Willebrand disease in severe pediatric pulmonary hypertension contributing to bleeding following reverse Potts shunt.

    Sullivan, Rachel T / Lo, Clara / Martin, Elisabeth / Kameny, Rebecca J / Hopper, Rachel K

    Pulmonary circulation

    2022  Volume 12, Issue 1, Page(s) e12042

    Abstract: The reverse Potts shunt is increasingly used as a palliative measure for end-stage pulmonary arterial hypertension (PAH) as a means to offload the right ventricle and improve functional status. This case report describes a child who developed significant ...

    Abstract The reverse Potts shunt is increasingly used as a palliative measure for end-stage pulmonary arterial hypertension (PAH) as a means to offload the right ventricle and improve functional status. This case report describes a child who developed significant hemothorax after reverse Potts shunt that required surgical exploration, blood product administration, and prolonged intensive care hospitalization. Despite lack of preoperative bleeding symptoms, testing revealed acquired von Willebrand disease (aVWD), with subsequent resolution of bleeding. Alterations in von Willebrand factor, including aVWD, have been reported in children with severe PAH but have not previously been associated with bleeding after reverse Potts shunt procedure. As bleeding is a recognized postoperative morbidity in PAH patients undergoing reverse Potts shunt, we highlight a potential role for preoperative testing for aVWD as perioperative factor replacement therapy may improve postoperative outcomes.
    Language English
    Publishing date 2022-02-04
    Publishing country United States
    Document type Case Reports
    ZDB-ID 2638089-4
    ISSN 2045-8940 ; 2045-8932
    ISSN (online) 2045-8940
    ISSN 2045-8932
    DOI 10.1002/pul2.12042
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  6. Article: Role of left atrial hypertension in pulmonary hypertension associated with bronchopulmonary dysplasia.

    Sullivan, Rachel T / Tandel, Megha D / Bhombal, Shazia / Adamson, Gregory T / Boothroyd, Derek B / Tracy, Michael / Moy, Amanda / Hopper, Rachel K

    Frontiers in pediatrics

    2022  Volume 10, Page(s) 1012136

    Abstract: Left atrial hypertension (LAH) may contribute to pulmonary hypertension (PH) in premature infants with bronchopulmonary dysplasia (BPD). Primary causes of LAH in infants with BPD include left ventricular diastolic dysfunction or hemodynamically ... ...

    Abstract Left atrial hypertension (LAH) may contribute to pulmonary hypertension (PH) in premature infants with bronchopulmonary dysplasia (BPD). Primary causes of LAH in infants with BPD include left ventricular diastolic dysfunction or hemodynamically significant left to right shunt. The incidence of LAH, which is definitively diagnosed by cardiac catheterization, and its contribution to PH is unknown in patients with BPD-PH. We report the prevalence of LAH in an institutional cohort with BPD-PH with careful examination of hemodynamic contributors and impact on patient outcomes. This single-center, retrospective cohort study examined children <2 years of age with BPD-PH who underwent cardiac catheterization at Lucile Packard Children's Hospital Stanford. Patients with unrepaired simple shunt congenital heart disease (CHD) and pulmonary vein stenosis (only 1 or 2 vessel disease) were included. Patients with complex CHD were excluded. From April 2010 to December 2021, 34 patients with BPD-PH underwent cardiac catheterization. We define LAH as pulmonary capillary wedge pressure (PCWP) or left atrial pressure (LAP) of at least 10 mmHg. In this cohort, median PCWP was 8 mmHg, with LAH present in 32% (
    Language English
    Publishing date 2022-09-20
    Publishing country Switzerland
    Document type Journal Article
    ZDB-ID 2711999-3
    ISSN 2296-2360
    ISSN 2296-2360
    DOI 10.3389/fped.2022.1012136
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  7. Article ; Online: Earlier Pulmonary Valve Replacement in Down Syndrome Patients Following Tetralogy of Fallot Repair.

    Sullivan, Rachel T / Frommelt, Peter C / Hill, Garick D

    Pediatric cardiology

    2017  Volume 38, Issue 6, Page(s) 1251–1256

    Abstract: The association between Down syndrome and pulmonary hypertension could contribute to more severe pulmonary regurgitation after tetralogy of Fallot repair and possibly earlier pulmonary valve replacement. We compared cardiac magnetic resonance measures of ...

    Abstract The association between Down syndrome and pulmonary hypertension could contribute to more severe pulmonary regurgitation after tetralogy of Fallot repair and possibly earlier pulmonary valve replacement. We compared cardiac magnetic resonance measures of pulmonary regurgitation and right ventricular dilation as well as timing of pulmonary valve replacement between those with and without Down syndrome after tetralogy of Fallot repair. Review of our surgical database from 2000 to 2015 identified patients with tetralogy of Fallot with pulmonary stenosis. Those with Down syndrome were compared to those without. The primary outcome of interest was time from repair to pulmonary valve replacement. Secondary outcomes included pulmonary regurgitation and indexed right ventricular volume on cardiac magnetic resonance imaging. The cohort of 284 patients included 35 (12%) with Down syndrome. Transannular patch repair was performed in 210 (74%). Down syndrome showed greater degree of pulmonary regurgitation (55 ± 14 vs. 37 ± 16%, p = 0.01) without a significantly greater rate of right ventricular dilation (p = 0.09). In multivariable analysis, Down syndrome (HR 2.3, 95% CI 1.2-4.5, p = 0.02) and transannular patch repair (HR 5.5, 95% CI 1.7-17.6, p = 0.004) were significant risk factors for valve replacement. Those with Down syndrome had significantly lower freedom from valve replacement (p = 0.03). Down syndrome is associated with an increased degree of pulmonary regurgitation and earlier pulmonary valve replacement after tetralogy of Fallot repair. These patients require earlier assessment by cardiac magnetic resonance imaging to determine timing of pulmonary valve replacement and evaluation for and treatment of preventable causes of pulmonary hypertension.
    MeSH term(s) Cardiac Surgical Procedures/adverse effects ; Child ; Child, Preschool ; Down Syndrome/complications ; Female ; Heart Valve Prosthesis Implantation ; Humans ; Infant ; Infant, Newborn ; Male ; Pulmonary Valve/surgery ; Pulmonary Valve Insufficiency/etiology ; Pulmonary Valve Insufficiency/surgery ; Pulmonary Valve Stenosis/surgery ; Retrospective Studies ; Tetralogy of Fallot/complications ; Tetralogy of Fallot/surgery ; Time Factors
    Language English
    Publishing date 2017-08
    Publishing country United States
    Document type Journal Article
    ZDB-ID 800857-7
    ISSN 1432-1971 ; 0172-0643
    ISSN (online) 1432-1971
    ISSN 0172-0643
    DOI 10.1007/s00246-017-1653-2
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    Zs.A 1528: Show issues Location:
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  8. Article ; Online: Cardioprotective effect of nicorandil on isoproterenol induced cardiomyopathy in the Mdx mouse model.

    Sullivan, Rachel T / Lam, Ngoc T / Haberman, Margaret / Beatka, Margaret J / Afzal, Muhammad Z / Lawlor, Michael W / Strande, Jennifer L

    BMC cardiovascular disorders

    2021  Volume 21, Issue 1, Page(s) 302

    Abstract: Background: Duchenne muscular dystrophy (DMD) associated cardiomyopathy is a major cause of morbidity and mortality. In an in vitro DMD cardiomyocyte model, nicorandil reversed stress-induced cell injury through multiple pathways implicated in DMD. We ... ...

    Abstract Background: Duchenne muscular dystrophy (DMD) associated cardiomyopathy is a major cause of morbidity and mortality. In an in vitro DMD cardiomyocyte model, nicorandil reversed stress-induced cell injury through multiple pathways implicated in DMD. We aimed to test the efficacy of nicorandil on the progression of cardiomyopathy in mdx mice following a 10-day treatment protocol.
    Methods: A subset of mdx mice was subjected to low-dose isoproterenol injections over 5 days to induce a cardiac phenotype and treated with vehicle or nicorandil for 10 days. Baseline and day 10 echocardiograms were obtained to assess cardiac function. At 10 days, cardiac tissue was harvested for further analysis, which included histologic analysis and assessment of oxidative stress. Paired student's t test was used for in group comparison, and ANOVA was used for multiple group comparisons.
    Results: Compared to vehicle treated mice, isoproterenol decreased ejection fraction and fractional shortening on echocardiogram. Nicorandil prevented isoproterenol induced cardiac dysfunction. Isoproterenol increased cardiac fibrosis, which nicorandil prevented. Isoproterenol increased gene expression of NADPH oxidase, which decreased to baseline with nicorandil treatment. Superoxide dismutase 2 protein expression increased in those treated with nicorandil, and xanthine oxidase activity decreased in mice treated with nicorandil during isoproterenol stress compared to all other groups.
    Conclusions: In conclusion, nicorandil is cardioprotective in mdx mice and warrants continued investigation as a therapy for DMD associated cardiomyopathy.
    MeSH term(s) Animals ; Cardiomyopathies/chemically induced ; Cardiomyopathies/metabolism ; Cardiomyopathies/physiopathology ; Cardiomyopathies/prevention & control ; Disease Models, Animal ; Female ; Fibrosis ; Isoproterenol ; Mice, Inbred mdx ; Muscular Dystrophy, Duchenne/complications ; Myocytes, Cardiac/drug effects ; Myocytes, Cardiac/metabolism ; Myocytes, Cardiac/pathology ; NADPH Oxidases/genetics ; NADPH Oxidases/metabolism ; Nicorandil/pharmacology ; Reactive Oxygen Species/metabolism ; Stroke Volume/drug effects ; Superoxide Dismutase/genetics ; Superoxide Dismutase/metabolism ; Ventricular Function, Left/drug effects ; Xanthine Oxidase/metabolism ; Mice
    Chemical Substances Reactive Oxygen Species ; Nicorandil (260456HAM0) ; Superoxide Dismutase (EC 1.15.1.1) ; superoxide dismutase 2 (EC 1.15.1.1) ; Xanthine Oxidase (EC 1.17.3.2) ; NADPH Oxidases (EC 1.6.3.-) ; Isoproterenol (L628TT009W)
    Language English
    Publishing date 2021-06-15
    Publishing country England
    Document type Journal Article ; Research Support, N.I.H., Extramural ; Research Support, Non-U.S. Gov't
    ZDB-ID 2059859-2
    ISSN 1471-2261 ; 1471-2261
    ISSN (online) 1471-2261
    ISSN 1471-2261
    DOI 10.1186/s12872-021-02112-4
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  9. Article ; Online: Subcutaneous Treprostinil Improves Surgical Candidacy for Next Stage Palliation in Single Ventricle Patients With High-Risk Hemodynamics.

    Sullivan, Rachel T / Handler, Stephanie S / Feinstein, Jeffrey A / Ogawa, Michelle / Liu, Esther / Ma, Michael / Hopper, Rachel K / Norris, Jana / Hollander, Seth A / Chen, Sharon

    Seminars in thoracic and cardiovascular surgery

    2022  Volume 35, Issue 4, Page(s) 733–743

    Abstract: Single ventricle (SV) patients with pulmonary vascular disease (SV-PVD) are considered poor surgical candidates for Glenn or Fontan palliation. Given limited options for Stage 1 (S1) and Stage 2 (S2) SV patients with SV-PVD, we report on the use of ... ...

    Abstract Single ventricle (SV) patients with pulmonary vascular disease (SV-PVD) are considered poor surgical candidates for Glenn or Fontan palliation. Given limited options for Stage 1 (S1) and Stage 2 (S2) SV patients with SV-PVD, we report on the use of subcutaneous treprostinil (TRE) to treat SV-PVD in this population. This single-center, retrospective cohort study examined SV patients who were not candidates for subsequent surgical palliation due to SV-PVD and were treated with TRE. The primary outcome was ability to progress to the next surgical stage; secondary outcomes included changes in hemodynamics after TRE initiation. Between 3/2014 and 8/2021, 17 SV patients received TRE for SV-PVD: 11 after S1 and 6 after S2 (median PVR 4.1 [IQR 3.2-4.8] WU*m
    MeSH term(s) Humans ; Retrospective Studies ; Treatment Outcome ; Heart Ventricles/surgery ; Fontan Procedure/adverse effects ; Hemodynamics ; Heart Defects, Congenital/diagnostic imaging ; Heart Defects, Congenital/surgery
    Chemical Substances treprostinil (RUM6K67ESG)
    Language English
    Publishing date 2022-08-02
    Publishing country United States
    Document type Journal Article
    ZDB-ID 1038278-1
    ISSN 1532-9488 ; 1043-0679
    ISSN (online) 1532-9488
    ISSN 1043-0679
    DOI 10.1053/j.semtcvs.2022.07.011
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