Literature DB >> 19327618

Calcium upregulation by percutaneous administration of gene therapy in cardiac disease (CUPID Trial), a first-in-human phase 1/2 clinical trial.

Brian E Jaski1, Mariell L Jessup, Donna M Mancini, Thomas P Cappola, Daniel F Pauly, Barry Greenberg, Kenneth Borow, Howard Dittrich, Krisztina M Zsebo, Roger J Hajjar.   

Abstract

BACKGROUND: SERCA2a deficiency is commonly seen in advanced heart failure (HF). This study is designed to investigate safety and biological effects of enzyme replacement using gene transfer in patients with advanced HF. METHODS AND
RESULTS: A total of 9 patients with advanced HF (New York Heart Association [NYHA] Class III/IV, ejection fraction [EF] < or = 30%, maximal oxygen uptake [VO2 max] <16 mL.kg.min, with maximal pharmacological and device therapy) received a single intracoronary infusion of AAV1/SERCA2a in the open-label portion of this ongoing study. Doses administered ranged from 1.4 x 10(11) to 3 x 10(12) DNase resistant particles per patient. We present 6- to 12-month follow-up data for these patients. AAV1/SERCA2a demonstrated an acceptable safety profile in this advanced HF population. Of the 9 patients treated, several demonstrated improvements from baseline to month 6 across a number of parameters important in HF, including symptomatic (NYHA and Minnesota Living with Heart Failure Questionnaire, 5 patients), functional (6-minute walk test and VO2 max, 4 patients), biomarker (NT-ProBNP, 2 patients), and LV function/remodeling (EF and end-systolic volume, 5 patients). Of note, 2 patients who failed to improve had preexisting anti-AAV1 neutralizing antibodies.
CONCLUSIONS: Quantitative evidence of biological activity across a number of parameters important for assessing HF status could be detected in several patients without preexisting neutralizing antibodies in this open-label study, although the number of patients in each cohort is too small to conduct statistical analyses. These findings support the initiation of the Phase 2 double-blind, placebo-controlled portion of this study.

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Year:  2009        PMID: 19327618      PMCID: PMC2752875          DOI: 10.1016/j.cardfail.2009.01.013

Source DB:  PubMed          Journal:  J Card Fail        ISSN: 1071-9164            Impact factor:   5.712


  44 in total

1.  Further evidence supporting an SEM-based criterion for identifying meaningful intra-individual changes in health-related quality of life.

Authors:  K W Wyrwich; W M Tierney; F D Wolinsky
Journal:  J Clin Epidemiol       Date:  1999-09       Impact factor: 6.437

2.  Linking clinical relevance and statistical significance in evaluating intra-individual changes in health-related quality of life.

Authors:  K W Wyrwich; N A Nienaber; W M Tierney; F D Wolinsky
Journal:  Med Care       Date:  1999-05       Impact factor: 2.983

3.  Clinical recovery from end-stage heart failure using left-ventricular assist device and pharmacological therapy correlates with increased sarcoplasmic reticulum calcium content but not with regression of cellular hypertrophy.

Authors:  C M N Terracciano; J Hardy; E J Birks; A Khaghani; N R Banner; M H Yacoub
Journal:  Circulation       Date:  2004-05-10       Impact factor: 29.690

4.  Echocardiographic predictors of morbidity and mortality in patients with advanced heart failure: the Beta-blocker Evaluation of Survival Trial (BEST).

Authors:  Paul A Grayburn; Christopher P Appleton; Anthony N DeMaria; Barry Greenberg; Brian Lowes; Jae Oh; Jonathan F Plehn; Peter Rahko; Martin St John Sutton; Eric J Eichhorn
Journal:  J Am Coll Cardiol       Date:  2005-04-05       Impact factor: 24.094

Review 5.  Adeno-associated virus vectors in clinical trials.

Authors:  Barrie J Carter
Journal:  Hum Gene Ther       Date:  2005-05       Impact factor: 5.695

Review 6.  Successes and failures of current treatment of heart failure.

Authors:  J G Cleland; K Swedberg; P A Poole-Wilson
Journal:  Lancet       Date:  1998-08       Impact factor: 79.321

Review 7.  Implantable left ventricular assist devices.

Authors:  D J Goldstein; M C Oz; E A Rose
Journal:  N Engl J Med       Date:  1998-11-19       Impact factor: 91.245

8.  Modulation of ventricular function through gene transfer in vivo.

Authors:  R J Hajjar; U Schmidt; T Matsui; J L Guerrero; K H Lee; J K Gwathmey; G W Dec; M J Semigran; A Rosenzweig
Journal:  Proc Natl Acad Sci U S A       Date:  1998-04-28       Impact factor: 11.205

9.  High intraindividual variation of B-type natriuretic peptide (BNP) and amino-terminal proBNP in patients with stable chronic heart failure.

Authors:  Sanne Bruins; M Rebecca Fokkema; Jeroen W P Römer; Mike J L Dejongste; Fey P L van der Dijs; Jody M W van den Ouweland; Frits A J Muskiet
Journal:  Clin Chem       Date:  2004-09-02       Impact factor: 8.327

10.  Unchanged protein levels of SERCA II and phospholamban but reduced Ca2+ uptake and Ca(2+)-ATPase activity of cardiac sarcoplasmic reticulum from dilated cardiomyopathy patients compared with patients with nonfailing hearts.

Authors:  R H Schwinger; M Böhm; U Schmidt; P Karczewski; U Bavendiek; M Flesch; E G Krause; E Erdmann
Journal:  Circulation       Date:  1995-12-01       Impact factor: 29.690

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  176 in total

1.  Gene transfer for ischemic heart failure in a preclinical model.

Authors:  Kiyotake Ishikawa; Dennis Ladage; Lisa Tilemann; Kenneth Fish; Yoshiaki Kawase; Roger J Hajjar
Journal:  J Vis Exp       Date:  2011-05-15       Impact factor: 1.355

2.  AAV6-βARKct gene delivery mediated by molecular cardiac surgery with recirculating delivery (MCARD) in sheep results in robust gene expression and increased adrenergic reserve.

Authors:  Michael G Katz; Anthony S Fargnoli; JaBaris D Swain; Catherine E Tomasulo; Michele Ciccarelli; Z Maggie Huang; Joseph E Rabinowitz; Charles R Bridges
Journal:  J Thorac Cardiovasc Surg       Date:  2011-12-03       Impact factor: 5.209

Review 3.  Cardiac gene therapy: from concept to reality.

Authors:  Razmig Garo Kratlian; Roger J Hajjar
Journal:  Curr Heart Fail Rep       Date:  2012-03

Review 4.  Reverse remodeling in heart failure--mechanisms and therapeutic opportunities.

Authors:  Norimichi Koitabashi; David A Kass
Journal:  Nat Rev Cardiol       Date:  2011-12-06       Impact factor: 32.419

Review 5.  Endogenous migration modulators as parent compounds for the development of novel cardiovascular and anti-inflammatory drugs.

Authors:  Wolfgang Poller; Madlen Rother; Carsten Skurk; Carmen Scheibenbogen
Journal:  Br J Pharmacol       Date:  2012-04       Impact factor: 8.739

6.  General overview of the Sixth International Symposium on Stem Cell Therapy and Cardiovascular Innovations.

Authors:  Ma Eugenia Vázquez-Alvarez; Ricardo Sanz-Ruiz; Enrique Gutiérrez; Adolfo Villa; Ma Eugenia Fernández; Sandra Vázquez; Ma José Lorenzo; Lucía Fernández; Isaac Pascual; Pedro L Sánchez; Francisco Fernández-Avilés
Journal:  J Cardiovasc Transl Res       Date:  2009-12-10       Impact factor: 4.132

Review 7.  Regenerative therapies in electrophysiology and pacing: introducing the next steps.

Authors:  Gerard J J Boink; Michael R Rosen
Journal:  J Interv Card Electrophysiol       Date:  2010-12-16       Impact factor: 1.900

Review 8.  Update on gene therapy for myocardial ischaemia and left ventricular systolic dysfunction or heart failure.

Authors:  Jerome Roncalli; Jörn Tongers; Douglas W Losordo
Journal:  Arch Cardiovasc Dis       Date:  2010-06-23       Impact factor: 2.340

Review 9.  Cardiovascular gene therapy for myocardial infarction.

Authors:  Maria C Scimia; Anna M Gumpert; Walter J Koch
Journal:  Expert Opin Biol Ther       Date:  2013-12-16       Impact factor: 4.388

Review 10.  Gene Therapy Strategies to Restore ER Proteostasis in Disease.

Authors:  Vicente Valenzuela; Kasey L Jackson; Sergio P Sardi; Claudio Hetz
Journal:  Mol Ther       Date:  2018-04-07       Impact factor: 11.454

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