The PRO-ACT database: design, initial analyses, and predictive features

To pool data from completed amyotrophic lateral sclerosis (ALS) clinical trials and create an open-access resource that enables greater understanding of the phenotype and biology of ALS. Clinical trials data were pooled from 16 completed phase II/III ALS clinical trials and one observational study....

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Veröffentlicht in:Neurology Jg. 83; H. 19; S. 1719
Hauptverfasser: Atassi, Nazem, Berry, James, Shui, Amy, Zach, Neta, Sherman, Alexander, Sinani, Ervin, Walker, Jason, Katsovskiy, Igor, Schoenfeld, David, Cudkowicz, Merit, Leitner, Melanie
Format: Journal Article
Sprache:Englisch
Veröffentlicht: United States 04.11.2014
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ISSN:1526-632X, 1526-632X
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Abstract To pool data from completed amyotrophic lateral sclerosis (ALS) clinical trials and create an open-access resource that enables greater understanding of the phenotype and biology of ALS. Clinical trials data were pooled from 16 completed phase II/III ALS clinical trials and one observational study. Over 8 million de-identified longitudinally collected data points from over 8,600 individuals with ALS were standardized across trials and merged to create the Pooled Resource Open-Access ALS Clinical Trials (PRO-ACT) database. This database includes demographics, family histories, and longitudinal clinical and laboratory data. Mixed effects models were used to describe the rate of disease progression measured by the Revised ALS Functional Rating Scale (ALSFRS-R) and vital capacity (VC). Cox regression models were used to describe survival data. Implementing Bonferroni correction, the critical p value for 15 different tests was p = 0.003. The ALSFRS-R rate of decline was 1.02 (±2.3) points per month and the VC rate of decline was 2.24% of predicted (±6.9) per month. Higher levels of uric acid at trial entry were predictive of a slower drop in ALSFRS-R (p = 0.01) and VC (p < 0.0001), and longer survival (p = 0.02). Higher levels of creatinine at baseline were predictive of a slower drop in ALSFRS-R (p = 0.01) and VC (p < 0.0001), and longer survival (p = 0.01). Finally, higher body mass index (BMI) at baseline was associated with longer survival (p < 0.0001). The PRO-ACT database is the largest publicly available repository of merged ALS clinical trials data. We report that baseline levels of creatinine and uric acid, as well as baseline BMI, are strong predictors of disease progression and survival.
AbstractList To pool data from completed amyotrophic lateral sclerosis (ALS) clinical trials and create an open-access resource that enables greater understanding of the phenotype and biology of ALS. Clinical trials data were pooled from 16 completed phase II/III ALS clinical trials and one observational study. Over 8 million de-identified longitudinally collected data points from over 8,600 individuals with ALS were standardized across trials and merged to create the Pooled Resource Open-Access ALS Clinical Trials (PRO-ACT) database. This database includes demographics, family histories, and longitudinal clinical and laboratory data. Mixed effects models were used to describe the rate of disease progression measured by the Revised ALS Functional Rating Scale (ALSFRS-R) and vital capacity (VC). Cox regression models were used to describe survival data. Implementing Bonferroni correction, the critical p value for 15 different tests was p = 0.003. The ALSFRS-R rate of decline was 1.02 (±2.3) points per month and the VC rate of decline was 2.24% of predicted (±6.9) per month. Higher levels of uric acid at trial entry were predictive of a slower drop in ALSFRS-R (p = 0.01) and VC (p < 0.0001), and longer survival (p = 0.02). Higher levels of creatinine at baseline were predictive of a slower drop in ALSFRS-R (p = 0.01) and VC (p < 0.0001), and longer survival (p = 0.01). Finally, higher body mass index (BMI) at baseline was associated with longer survival (p < 0.0001). The PRO-ACT database is the largest publicly available repository of merged ALS clinical trials data. We report that baseline levels of creatinine and uric acid, as well as baseline BMI, are strong predictors of disease progression and survival.
To pool data from completed amyotrophic lateral sclerosis (ALS) clinical trials and create an open-access resource that enables greater understanding of the phenotype and biology of ALS.OBJECTIVETo pool data from completed amyotrophic lateral sclerosis (ALS) clinical trials and create an open-access resource that enables greater understanding of the phenotype and biology of ALS.Clinical trials data were pooled from 16 completed phase II/III ALS clinical trials and one observational study. Over 8 million de-identified longitudinally collected data points from over 8,600 individuals with ALS were standardized across trials and merged to create the Pooled Resource Open-Access ALS Clinical Trials (PRO-ACT) database. This database includes demographics, family histories, and longitudinal clinical and laboratory data. Mixed effects models were used to describe the rate of disease progression measured by the Revised ALS Functional Rating Scale (ALSFRS-R) and vital capacity (VC). Cox regression models were used to describe survival data. Implementing Bonferroni correction, the critical p value for 15 different tests was p = 0.003.METHODSClinical trials data were pooled from 16 completed phase II/III ALS clinical trials and one observational study. Over 8 million de-identified longitudinally collected data points from over 8,600 individuals with ALS were standardized across trials and merged to create the Pooled Resource Open-Access ALS Clinical Trials (PRO-ACT) database. This database includes demographics, family histories, and longitudinal clinical and laboratory data. Mixed effects models were used to describe the rate of disease progression measured by the Revised ALS Functional Rating Scale (ALSFRS-R) and vital capacity (VC). Cox regression models were used to describe survival data. Implementing Bonferroni correction, the critical p value for 15 different tests was p = 0.003.The ALSFRS-R rate of decline was 1.02 (±2.3) points per month and the VC rate of decline was 2.24% of predicted (±6.9) per month. Higher levels of uric acid at trial entry were predictive of a slower drop in ALSFRS-R (p = 0.01) and VC (p < 0.0001), and longer survival (p = 0.02). Higher levels of creatinine at baseline were predictive of a slower drop in ALSFRS-R (p = 0.01) and VC (p < 0.0001), and longer survival (p = 0.01). Finally, higher body mass index (BMI) at baseline was associated with longer survival (p < 0.0001).RESULTSThe ALSFRS-R rate of decline was 1.02 (±2.3) points per month and the VC rate of decline was 2.24% of predicted (±6.9) per month. Higher levels of uric acid at trial entry were predictive of a slower drop in ALSFRS-R (p = 0.01) and VC (p < 0.0001), and longer survival (p = 0.02). Higher levels of creatinine at baseline were predictive of a slower drop in ALSFRS-R (p = 0.01) and VC (p < 0.0001), and longer survival (p = 0.01). Finally, higher body mass index (BMI) at baseline was associated with longer survival (p < 0.0001).The PRO-ACT database is the largest publicly available repository of merged ALS clinical trials data. We report that baseline levels of creatinine and uric acid, as well as baseline BMI, are strong predictors of disease progression and survival.CONCLUSIONThe PRO-ACT database is the largest publicly available repository of merged ALS clinical trials data. We report that baseline levels of creatinine and uric acid, as well as baseline BMI, are strong predictors of disease progression and survival.
Author Sherman, Alexander
Cudkowicz, Merit
Sinani, Ervin
Katsovskiy, Igor
Atassi, Nazem
Walker, Jason
Leitner, Melanie
Berry, James
Shui, Amy
Zach, Neta
Schoenfeld, David
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  email: natassi@partners.org
  organization: From the Neurological Clinical Research Institute (NCRI), Department of Neurology (N.A., J.B., A. Sherman, E.S., J.W., I.K., M.C.), and the Biostatistics Center (A. Shui, D.S.), Massachusetts General Hospital, Boston; and Prize4Life (N.Z., M.L.), Cambridge, MA. natassi@partners.org
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  surname: Berry
  fullname: Berry, James
  organization: From the Neurological Clinical Research Institute (NCRI), Department of Neurology (N.A., J.B., A. Sherman, E.S., J.W., I.K., M.C.), and the Biostatistics Center (A. Shui, D.S.), Massachusetts General Hospital, Boston; and Prize4Life (N.Z., M.L.), Cambridge, MA
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  surname: Shui
  fullname: Shui, Amy
  organization: From the Neurological Clinical Research Institute (NCRI), Department of Neurology (N.A., J.B., A. Sherman, E.S., J.W., I.K., M.C.), and the Biostatistics Center (A. Shui, D.S.), Massachusetts General Hospital, Boston; and Prize4Life (N.Z., M.L.), Cambridge, MA
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  organization: From the Neurological Clinical Research Institute (NCRI), Department of Neurology (N.A., J.B., A. Sherman, E.S., J.W., I.K., M.C.), and the Biostatistics Center (A. Shui, D.S.), Massachusetts General Hospital, Boston; and Prize4Life (N.Z., M.L.), Cambridge, MA
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  fullname: Sherman, Alexander
  organization: From the Neurological Clinical Research Institute (NCRI), Department of Neurology (N.A., J.B., A. Sherman, E.S., J.W., I.K., M.C.), and the Biostatistics Center (A. Shui, D.S.), Massachusetts General Hospital, Boston; and Prize4Life (N.Z., M.L.), Cambridge, MA
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  surname: Sinani
  fullname: Sinani, Ervin
  organization: From the Neurological Clinical Research Institute (NCRI), Department of Neurology (N.A., J.B., A. Sherman, E.S., J.W., I.K., M.C.), and the Biostatistics Center (A. Shui, D.S.), Massachusetts General Hospital, Boston; and Prize4Life (N.Z., M.L.), Cambridge, MA
– sequence: 7
  givenname: Jason
  surname: Walker
  fullname: Walker, Jason
  organization: From the Neurological Clinical Research Institute (NCRI), Department of Neurology (N.A., J.B., A. Sherman, E.S., J.W., I.K., M.C.), and the Biostatistics Center (A. Shui, D.S.), Massachusetts General Hospital, Boston; and Prize4Life (N.Z., M.L.), Cambridge, MA
– sequence: 8
  givenname: Igor
  surname: Katsovskiy
  fullname: Katsovskiy, Igor
  organization: From the Neurological Clinical Research Institute (NCRI), Department of Neurology (N.A., J.B., A. Sherman, E.S., J.W., I.K., M.C.), and the Biostatistics Center (A. Shui, D.S.), Massachusetts General Hospital, Boston; and Prize4Life (N.Z., M.L.), Cambridge, MA
– sequence: 9
  givenname: David
  surname: Schoenfeld
  fullname: Schoenfeld, David
  organization: From the Neurological Clinical Research Institute (NCRI), Department of Neurology (N.A., J.B., A. Sherman, E.S., J.W., I.K., M.C.), and the Biostatistics Center (A. Shui, D.S.), Massachusetts General Hospital, Boston; and Prize4Life (N.Z., M.L.), Cambridge, MA
– sequence: 10
  givenname: Merit
  surname: Cudkowicz
  fullname: Cudkowicz, Merit
  organization: From the Neurological Clinical Research Institute (NCRI), Department of Neurology (N.A., J.B., A. Sherman, E.S., J.W., I.K., M.C.), and the Biostatistics Center (A. Shui, D.S.), Massachusetts General Hospital, Boston; and Prize4Life (N.Z., M.L.), Cambridge, MA
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  givenname: Melanie
  surname: Leitner
  fullname: Leitner, Melanie
  organization: From the Neurological Clinical Research Institute (NCRI), Department of Neurology (N.A., J.B., A. Sherman, E.S., J.W., I.K., M.C.), and the Biostatistics Center (A. Shui, D.S.), Massachusetts General Hospital, Boston; and Prize4Life (N.Z., M.L.), Cambridge, MA
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ContentType Journal Article
Copyright 2014 American Academy of Neurology.
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References_xml – reference: 22922503 - Neurodegener Dis. 2013;12(2):81-90
– reference: 15534251 - Neurology. 2004 Nov 9;63(9):1656-61
– reference: 17709710 - Neurology. 2007 Aug 21;69(8):776-84
– reference: 19961264 - Amyotroph Lateral Scler. 2010 May 3;11(3):266-71
– reference: 21956723 - Neurology. 2011 Oct 11;77(15):1432-7
– reference: 22728481 - Intern Med. 2012;51(12):1501-8
– reference: 18574760 - Amyotroph Lateral Scler. 2008 Jun;9(3):163-7
– reference: 8628460 - Neurology. 1996 May;46(5):1244-9
– reference: 8960715 - Neurology. 1996 Dec;47(6):1383-8
– reference: 22580741 - Curr Neurol Neurosci Rep. 2012 Aug;12(4):367-75
– reference: 11465936 - Amyotroph Lateral Scler Other Motor Neuron Disord. 2001 Mar;2(1):9-18
– reference: 20063429 - Mov Disord. 2010 Jan 30;25(2):224-8
– reference: 20363190 - Lancet Neurol. 2010 May;9(5):481-8
– reference: 11386269 - N Engl J Med. 2001 May 31;344(22):1688-700
– reference: 12939417 - Neurology. 2003 Aug 26;61(4):456-64
– reference: 8302340 - N Engl J Med. 1994 Mar 3;330(9):585-91
– reference: 19969067 - Free Radic Biol Med. 2010 Mar 1;48(5):629-41
– reference: 23042862 - Science. 2012 Oct 5;338(6103):29
– reference: 19073945 - Neurology. 2009 Feb 24;72(8):705-11
– reference: 19961263 - Amyotroph Lateral Scler. 2010 May 3;11(3):259-65
– reference: 16802291 - Ann Neurol. 2006 Jul;60(1):22-31
– reference: 11033417 - Free Radic Biol Med. 2000 Oct 1;29(7):652-8
– reference: 12021952 - J Neurol. 2002 May;249(5):609-15
– reference: 8757041 - Neurology. 1996 Aug;47(2):571-3
– reference: 22323210 - J Neurol. 2012 Sep;259(9):1923-8
– reference: 24070404 - Amyotroph Lateral Scler Frontotemporal Degener. 2014 Mar;15(1-2):119-29
– reference: 10467383 - Mult Scler. 1999 Aug;5(4):244-50
– reference: 10227630 - Neurology. 1999 Apr 22;52(7):1427-33
– reference: 19822770 - Arch Neurol. 2009 Dec;66(12):1460-8
– reference: 15204012 - Amyotroph Lateral Scler Other Motor Neuron Disord. 2004 Jun;5(2):107-17
– reference: 18413464 - Arch Neurol. 2008 Jun;65(6):716-23
– reference: 10636125 - Neurology. 2000 Jan 11;54(1):53-7
– reference: 21607987 - Muscle Nerve. 2011 Jul;44(1):20-4
– reference: 23678880 - Amyotroph Lateral Scler Frontotemporal Degener. 2013 May;14 Suppl 1:53-61
– reference: 16036424 - Amyotroph Lateral Scler Other Motor Neuron Disord. 2005 Mar;6(1):37-44
– reference: 18551622 - Muscle Nerve. 2008 Jul;38(1):837-44
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Snippet To pool data from completed amyotrophic lateral sclerosis (ALS) clinical trials and create an open-access resource that enables greater understanding of the...
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SubjectTerms Amyotrophic Lateral Sclerosis - diagnosis
Amyotrophic Lateral Sclerosis - physiopathology
Amyotrophic Lateral Sclerosis - therapy
Clinical Trials as Topic - statistics & numerical data
Database Management Systems
Datasets as Topic - statistics & numerical data
Disease Progression
Humans
Longitudinal Studies
Observational Studies as Topic - statistics & numerical data
Predictive Value of Tests
Research Design
Title The PRO-ACT database: design, initial analyses, and predictive features
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