Patient-reported study of the impact of pediatric-onset myotonic dystrophy

Muscle Nerve. 2019 Oct;60(4):392-399. doi: 10.1002/mus.26632. Epub 2019 Jul 26.

Abstract

Introduction: The prevalence and impact of symptoms affecting individuals with pediatric forms of myotonic dystrophy type-1 (DM1) are not well understood.

Methods: Patients from the United States, Canada, and Sweden completed a survey that investigated 20 themes associated with pediatric-onset DM1. Participants reported the prevalence and importance of each theme affecting their lives. Surveys from participants were matched with surveys from their caregivers for additional analysis.

Results: The most prevalent symptomatic themes included problems with hands or fingers (79%) and gastrointestinal issues (75%). Problems with urinary/bowel control and gastrointestinal issues were reported to have the greatest impact on patients' lives. Responses from participants and their caregivers had varying levels of agreement among symptomatic themes.

Discussion: Many symptoms have meaningful impact on disease burden. The highest levels of agreement between caregivers and individuals with pediatric forms of myotonic dystrophy were found for physical activity themes.

Keywords: caregivers; health status indicators; myotonic dystrophy; neuromuscular disease; pediatric onset; symptom assessment.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Activities of Daily Living
  • Adolescent
  • Adult
  • Caregivers
  • Child
  • Child, Preschool
  • Communication
  • Cost of Illness
  • Deglutition Disorders / etiology
  • Deglutition Disorders / physiopathology
  • Female
  • Fingers / physiopathology
  • Gastrointestinal Diseases / etiology
  • Gastrointestinal Diseases / physiopathology
  • Hand / physiopathology
  • Humans
  • Male
  • Mobility Limitation
  • Muscle Weakness / etiology
  • Muscle Weakness / physiopathology
  • Myotonia / etiology
  • Myotonia / physiopathology
  • Myotonic Dystrophy / complications
  • Myotonic Dystrophy / physiopathology*
  • Myotonic Dystrophy / psychology*
  • Patient Reported Outcome Measures
  • Young Adult