Literature DB >> 26173644

Potential trade-offs in treatment of premanifest Huntington's disease.

Roger L Albin1,2,3,4, James F Burke1,2.   

Abstract

The potential long-term consequences of treatments delaying manifestations of neurodegenerative diseases have not been explored. Using Huntington disease (HD) data and Markov chain Monte Carlo methods, we simulated the effects of therapies with equivalent effects on time to onset of HD and survival with HD. Our results suggest substantial potential trade-offs in effects of these therapies; significant delays in time to onset of HD were accompanied by significant prolongations of survival after onset of HD. Under a variety of assumptions, treatments delaying onset of HD result in some patients likely to have a greater increase in survival with manifest HD compared to delays in time to onset of HD. Our results suggest that future work in HD should be sensitive to the potential existence of such trade-offs and that understanding the preferences of HD patients and the broader HD community will be increasingly important. Future research, trial design, and treatment strategies in HD and other mid-life-onset neurodegenerative disorders should consider the possibility of trade-offs in long-term consequences of disease-modifying treatments.
© 2015 International Parkinson and Movement Disorder Society.

Entities:  

Keywords:  Huntington's disease; Monte Carlo simulation; outcomes analysis; presymptomatic treatment; simulation

Mesh:

Year:  2015        PMID: 26173644      PMCID: PMC4873710          DOI: 10.1002/mds.26318

Source DB:  PubMed          Journal:  Mov Disord        ISSN: 0885-3185            Impact factor:   10.338


  9 in total

1.  Reversal of neuropathology and motor dysfunction in a conditional model of Huntington's disease.

Authors:  A Yamamoto; J J Lucas; R Hen
Journal:  Cell       Date:  2000-03-31       Impact factor: 41.582

2.  Shared decision making--pinnacle of patient-centered care.

Authors:  Michael J Barry; Susan Edgman-Levitan
Journal:  N Engl J Med       Date:  2012-03-01       Impact factor: 91.245

3.  Sustained therapeutic reversal of Huntington's disease by transient repression of huntingtin synthesis.

Authors:  Holly B Kordasiewicz; Lisa M Stanek; Edward V Wancewicz; Curt Mazur; Melissa M McAlonis; Kimberly A Pytel; Jonathan W Artates; Andreas Weiss; Seng H Cheng; Lamya S Shihabuddin; Gene Hung; C Frank Bennett; Don W Cleveland
Journal:  Neuron       Date:  2012-06-21       Impact factor: 17.173

4.  Differences in duration of Huntington's disease based on age at onset.

Authors:  T Foroud; J Gray; J Ivashina; P M Conneally
Journal:  J Neurol Neurosurg Psychiatry       Date:  1999-01       Impact factor: 10.154

5.  Designing a pilot sequential multiple assignment randomized trial for developing an adaptive treatment strategy.

Authors:  Daniel Almirall; Scott N Compton; Meredith Gunlicks-Stoessel; Naihua Duan; Susan A Murphy
Journal:  Stat Med       Date:  2012-03-22       Impact factor: 2.373

6.  Preclinical safety of RNAi-mediated HTT suppression in the rhesus macaque as a potential therapy for Huntington's disease.

Authors:  Jodi L McBride; Mark R Pitzer; Ryan L Boudreau; Brett Dufour; Theodore Hobbs; Sergio R Ojeda; Beverly L Davidson
Journal:  Mol Ther       Date:  2011-10-25       Impact factor: 11.454

7.  Preparing for preventive clinical trials: the Predict-HD study.

Authors:  Jane S Paulsen; Michael Hayden; Julie C Stout; Douglas R Langbehn; Elizabeth Aylward; Christopher A Ross; Mark Guttman; Martha Nance; Karl Kieburtz; David Oakes; Ira Shoulson; Elise Kayson; Shannon Johnson; Elizabeth Penziner
Journal:  Arch Neurol       Date:  2006-06

8.  The relationship between CAG repeat length and clinical progression in Huntington's disease.

Authors:  Bernard Ravina; Megan Romer; Radu Constantinescu; Kevin Biglan; Alicia Brocht; Karl Kieburtz; Ira Shoulson; Michael P McDermott
Journal:  Mov Disord       Date:  2008-07-15       Impact factor: 10.338

9.  A new model for prediction of the age of onset and penetrance for Huntington's disease based on CAG length.

Authors:  D R Langbehn; R R Brinkman; D Falush; J S Paulsen; M R Hayden
Journal:  Clin Genet       Date:  2004-04       Impact factor: 4.438

  9 in total
  2 in total

Review 1.  Polyglutamine Repeats in Neurodegenerative Diseases.

Authors:  Andrew P Lieberman; Vikram G Shakkottai; Roger L Albin
Journal:  Annu Rev Pathol       Date:  2018-08-08       Impact factor: 23.472

2.  Huntington's Disease Community Perspectives on Desired Characteristics of Disease Modifying Therapies.

Authors:  Michele C Gornick; Kerry A Ryan; Praveen Dayalu; Noelle E Carlozzi; Roger L Albin; Darin B Zahuranec
Journal:  Tremor Other Hyperkinet Mov (N Y)       Date:  2021-01-20
  2 in total

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