Literature DB >> 22786587

Patient-reported impact of symptoms in myotonic dystrophy type 1 (PRISM-1).

Chad Heatwole1, Rita Bode, Nicholas Johnson, Christine Quinn, William Martens, Michael P McDermott, Nan Rothrock, Charles Thornton, Barbara Vickrey, David Victorson, Richard Moxley.   

Abstract

OBJECTIVE: To determine the most critical symptoms in a national myotonic dystrophy type 1 (DM1) population and to identify the modifying factors that have the greatest effect on the severity of these symptoms.
METHODS: We performed a cross-sectional study of 278 adult patients with DM1 from the national registry of patients with DM1 between April and August 2010. We assessed the prevalence and relative significance of 221 critical DM1 symptoms and 14 disease themes. These symptoms and themes were chosen for evaluation based on prior interviews with patients with DM1. Responses were categorized by age, CTG repeat length, gender, and duration of symptoms.
RESULTS: Participants with DM1 provided symptom rating survey responses to address the relative frequency and importance of each DM1 symptom. The symptomatic themes with the highest prevalence in DM1 were problems with hands or arms (93.5%), fatigue (90.8%), myotonia (90.3%), and impaired sleep or daytime sleepiness (87.9%). Participants identified fatigue and limitations in mobility as the symptomatic themes that have the greatest effect on their lives. We found an association between age and the average prevalence of all themes (p < 0.01) and between CTG repeat length and the average effect of all symptomatic themes on participant lives (p < 0.01).
CONCLUSIONS: There are a wide range of symptoms that significantly affect the lives of patients with DM1. These symptoms, some previously underrecognized, have varying levels of importance in the DM1 population and are nonlinearly dependent on patient age and CTG repeat length.

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Year:  2012        PMID: 22786587      PMCID: PMC3400095          DOI: 10.1212/WNL.0b013e318260cbe6

Source DB:  PubMed          Journal:  Neurology        ISSN: 0028-3878            Impact factor:   9.910


  24 in total

1.  Unstable DNA sequence in myotonic dystrophy.

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Journal:  Lancet       Date:  1992-05-09       Impact factor: 79.321

2.  Outcomes measurement: a report from the front.

Authors:  R Geigle; S B Jones
Journal:  Inquiry       Date:  1990       Impact factor: 1.730

3.  New nomenclature and DNA testing guidelines for myotonic dystrophy type 1 (DM1). The International Myotonic Dystrophy Consortium (IDMC).

Authors: 
Journal:  Neurology       Date:  2000-03-28       Impact factor: 9.910

4.  Criteria for establishing the validity of genetic recombination in myotonic dystrophy.

Authors:  R C Griggs; D S Wood
Journal:  Neurology       Date:  1989-03       Impact factor: 9.910

5.  Myotonic dystrophy mutation: an unstable CTG repeat in the 3' untranslated region of the gene.

Authors:  M Mahadevan; C Tsilfidis; L Sabourin; G Shutler; C Amemiya; G Jansen; C Neville; M Narang; J Barceló; K O'Hoy
Journal:  Science       Date:  1992-03-06       Impact factor: 47.728

6.  An unstable triplet repeat in a gene related to myotonic muscular dystrophy.

Authors:  Y H Fu; A Pizzuti; R G Fenwick; J King; S Rajnarayan; P W Dunne; J Dubel; G A Nasser; T Ashizawa; P de Jong
Journal:  Science       Date:  1992-03-06       Impact factor: 47.728

7.  Prevalence of irritable bowel syndrome: a community survey.

Authors:  Sue Wilson; Lesley Roberts; Andrea Roalfe; Pam Bridge; Sukhdev Singh
Journal:  Br J Gen Pract       Date:  2004-07       Impact factor: 5.386

8.  Molecular basis of myotonic dystrophy: expansion of a trinucleotide (CTG) repeat at the 3' end of a transcript encoding a protein kinase family member.

Authors:  J D Brook; M E McCurrach; H G Harley; A J Buckler; D Church; H Aburatani; K Hunter; V P Stanton; J P Thirion; T Hudson
Journal:  Cell       Date:  1992-02-21       Impact factor: 41.582

9.  Incorporating the patient's perspective into drug development and communication: an ad hoc task force report of the Patient-Reported Outcomes (PRO) Harmonization Group meeting at the Food and Drug Administration, February 16, 2001.

Authors:  Catherine Acquadro; Rick Berzon; Dominique Dubois; Nancy Kline Leidy; Patrick Marquis; Dennis Revicki; Margaret Rothman
Journal:  Value Health       Date:  2003 Sep-Oct       Impact factor: 5.725

10.  Prediction of myotonic dystrophy clinical severity based on the number of intragenic [CTG]n trinucleotide repeats.

Authors:  M Gennarelli; G Novelli; F Andreasi Bassi; L Martorell; M Cornet; E Menegazzo; M L Mostacciuolo; J M Martinez; C Angelini; A Pizzuti; M Baiget; B Dallapiccola
Journal:  Am J Med Genet       Date:  1996-11-11
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  53 in total

1.  Diagnostic odyssey of patients with myotonic dystrophy.

Authors:  James E Hilbert; Tetsuo Ashizawa; John W Day; Elizabeth A Luebbe; William B Martens; Michael P McDermott; Rabi Tawil; Charles A Thornton; Richard T Moxley
Journal:  J Neurol       Date:  2013-06-27       Impact factor: 4.849

Review 2.  Daytime sleepiness and myotonic dystrophy.

Authors:  Luc Laberge; Cynthia Gagnon; Yves Dauvilliers
Journal:  Curr Neurol Neurosci Rep       Date:  2013-04       Impact factor: 5.081

3.  Myotonic dystrophy health index: Correlations with clinical tests and patient function.

Authors:  Chad Heatwole; Rita Bode; Nicholas E Johnson; Jeanne Dekdebrun; Nuran Dilek; Katy Eichinger; James E Hilbert; Eric Logigian; Elizabeth Luebbe; William Martens; Michael P Mcdermott; Shree Pandya; Araya Puwanant; Nan Rothrock; Charles Thornton; Barbara G Vickrey; David Victorson; Richard T Moxley
Journal:  Muscle Nerve       Date:  2015-12-29       Impact factor: 3.217

4.  Medication adherence in patients with myotonic dystrophy and facioscapulohumeral muscular dystrophy.

Authors:  Bryan P Fitzgerald; Kelly M Conn; Joanne Smith; Andrew Walker; Amy L Parkhill; James E Hilbert; Elizabeth A Luebbe; Richard T Moxley
Journal:  J Neurol       Date:  2016-10-12       Impact factor: 4.849

5.  Parent-reported multi-national study of the impact of congenital and childhood onset myotonic dystrophy.

Authors:  Nicholas E Johnson; Anne-Berit Ekstrom; Craig Campbell; Man Hung; Heather R Adams; Wei Chen; Elizabeth Luebbe; James Hilbert; Richard T Moxley; Chad R Heatwole
Journal:  Dev Med Child Neurol       Date:  2015-10-28       Impact factor: 5.449

Review 6.  Myotonic dystrophy.

Authors:  Charles A Thornton
Journal:  Neurol Clin       Date:  2014-06-06       Impact factor: 3.806

7.  The Mef2 transcription network is disrupted in myotonic dystrophy heart tissue, dramatically altering miRNA and mRNA expression.

Authors:  Auinash Kalsotra; Ravi K Singh; Priyatansh Gurha; Amanda J Ward; Chad J Creighton; Thomas A Cooper
Journal:  Cell Rep       Date:  2014-01-09       Impact factor: 9.423

8.  Increased EEG Theta Spectral Power in Sleep in Myotonic Dystrophy Type 1.

Authors:  Joseph Cheung; Chad Ruoff; Hyatt Moore; Katharine A Hagerman; Jennifer Perez; Sarada Sakamuri; Simon C Warby; Emmanuel Mignot; John Day; Jacinda Sampson
Journal:  J Clin Sleep Med       Date:  2018-02-15       Impact factor: 4.062

9.  Patient-identified disease burden in facioscapulohumeral muscular dystrophy.

Authors:  Nicholas E Johnson; Christine Quinn; Eileen Eastwood; Rabi Tawil; Chad R Heatwole
Journal:  Muscle Nerve       Date:  2012-12       Impact factor: 3.217

10.  Quality-of-life in Charcot-Marie-Tooth disease: the patient's perspective.

Authors:  Nicholas E Johnson; Chad R Heatwole; Nuran Dilek; Janet Sowden; Callyn A Kirk; Denise Shereff; Michael E Shy; David N Herrmann
Journal:  Neuromuscul Disord       Date:  2014-06-27       Impact factor: 4.296

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