Risk factors for first fractures among males with duchenne or becker muscular dystrophy

Katherine A. James, Christopher M Cunniff, Susan D. Apkon, Katherine Mathews, Zhenqiang Lu, Caleb Holtzer, Shree Pandya, Emma Ciafaloni, Lisa Miller

Research output: Contribution to journalArticle

10 Citations (Scopus)

Abstract

Background: Fractures are a significant concern for individuals with Duchenne/Becker muscular dystrophy with 21% to 44% of males experiencing a fracture. Factors that increase or decrease the risk for fracture have been suggested in past research, although statistical risk has not been determined. Methods: In this retrospective cohort study, we used the Muscular Dystrophy Surveillance, Tracking and Research Network cohort, a large, population-based sample to identify risk factors associated with first fractures in patients with Duchenne or Becker muscular dystrophy. Our study cohort included males with Duchenne or Becker muscular dystrophy born between 1982 and 2006 who resided in Arizona, Colorado, Georgia, Iowa, and Western New York, retrospectively identified and followed through 2010. We utilized a multivariate Cox proportional hazard model to determine hazard ratios for relevant factors associated with first fracture risk including race/ethnicity, surveillance site, ambulation status, calcium/vitamin D use and duration, bisphosphonate use and duration, and corticosteroid use and duration. Results: Of 747 cases, 249 had at least 1 fracture (33.3%). Fulltime wheelchair use increased the risk of first fracture by 75% for every 3 months of use (hazard ratio=1.75, 95% confidence interval, 1.14, 2.68), but corticosteroid use, bisphosphonate use, and calcium/vitamin D use did not significantly affect risk in the final adjusted model. Conclusions: In this cohort, first fractures were common and full-time wheelchair use, but not corticosteroid use, was identified as a risk factor. The impact of prevention measures should be more thoroughly assessed. Clinical Relevance: Fractures are a significant concern for individuals with dystrophinopathies, but the contribution of various risk factors has not been consistently demonstrated.

Original languageEnglish (US)
Pages (from-to)640-644
Number of pages5
JournalJournal of Pediatric Orthopaedics
Volume35
Issue number6
StatePublished - 2015

Fingerprint

Duchenne Muscular Dystrophy
Adrenal Cortex Hormones
Wheelchairs
Diphosphonates
Vitamin D
Cohort Studies
Calcium
Muscular Dystrophies
Proportional Hazards Models
Research
Walking
Retrospective Studies
Confidence Intervals
Population

Keywords

  • Ambulation
  • Boys
  • Corticosteroid
  • Fracture
  • Muscular dystrophy

ASJC Scopus subject areas

  • Pediatrics, Perinatology, and Child Health
  • Orthopedics and Sports Medicine

Cite this

James, K. A., Cunniff, C. M., Apkon, S. D., Mathews, K., Lu, Z., Holtzer, C., ... Miller, L. (2015). Risk factors for first fractures among males with duchenne or becker muscular dystrophy. Journal of Pediatric Orthopaedics, 35(6), 640-644.

Risk factors for first fractures among males with duchenne or becker muscular dystrophy. / James, Katherine A.; Cunniff, Christopher M; Apkon, Susan D.; Mathews, Katherine; Lu, Zhenqiang; Holtzer, Caleb; Pandya, Shree; Ciafaloni, Emma; Miller, Lisa.

In: Journal of Pediatric Orthopaedics, Vol. 35, No. 6, 2015, p. 640-644.

Research output: Contribution to journalArticle

James, KA, Cunniff, CM, Apkon, SD, Mathews, K, Lu, Z, Holtzer, C, Pandya, S, Ciafaloni, E & Miller, L 2015, 'Risk factors for first fractures among males with duchenne or becker muscular dystrophy', Journal of Pediatric Orthopaedics, vol. 35, no. 6, pp. 640-644.
James, Katherine A. ; Cunniff, Christopher M ; Apkon, Susan D. ; Mathews, Katherine ; Lu, Zhenqiang ; Holtzer, Caleb ; Pandya, Shree ; Ciafaloni, Emma ; Miller, Lisa. / Risk factors for first fractures among males with duchenne or becker muscular dystrophy. In: Journal of Pediatric Orthopaedics. 2015 ; Vol. 35, No. 6. pp. 640-644.
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abstract = "Background: Fractures are a significant concern for individuals with Duchenne/Becker muscular dystrophy with 21{\%} to 44{\%} of males experiencing a fracture. Factors that increase or decrease the risk for fracture have been suggested in past research, although statistical risk has not been determined. Methods: In this retrospective cohort study, we used the Muscular Dystrophy Surveillance, Tracking and Research Network cohort, a large, population-based sample to identify risk factors associated with first fractures in patients with Duchenne or Becker muscular dystrophy. Our study cohort included males with Duchenne or Becker muscular dystrophy born between 1982 and 2006 who resided in Arizona, Colorado, Georgia, Iowa, and Western New York, retrospectively identified and followed through 2010. We utilized a multivariate Cox proportional hazard model to determine hazard ratios for relevant factors associated with first fracture risk including race/ethnicity, surveillance site, ambulation status, calcium/vitamin D use and duration, bisphosphonate use and duration, and corticosteroid use and duration. Results: Of 747 cases, 249 had at least 1 fracture (33.3{\%}). Fulltime wheelchair use increased the risk of first fracture by 75{\%} for every 3 months of use (hazard ratio=1.75, 95{\%} confidence interval, 1.14, 2.68), but corticosteroid use, bisphosphonate use, and calcium/vitamin D use did not significantly affect risk in the final adjusted model. Conclusions: In this cohort, first fractures were common and full-time wheelchair use, but not corticosteroid use, was identified as a risk factor. The impact of prevention measures should be more thoroughly assessed. Clinical Relevance: Fractures are a significant concern for individuals with dystrophinopathies, but the contribution of various risk factors has not been consistently demonstrated.",
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AU - James, Katherine A.

AU - Cunniff, Christopher M

AU - Apkon, Susan D.

AU - Mathews, Katherine

AU - Lu, Zhenqiang

AU - Holtzer, Caleb

AU - Pandya, Shree

AU - Ciafaloni, Emma

AU - Miller, Lisa

PY - 2015

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N2 - Background: Fractures are a significant concern for individuals with Duchenne/Becker muscular dystrophy with 21% to 44% of males experiencing a fracture. Factors that increase or decrease the risk for fracture have been suggested in past research, although statistical risk has not been determined. Methods: In this retrospective cohort study, we used the Muscular Dystrophy Surveillance, Tracking and Research Network cohort, a large, population-based sample to identify risk factors associated with first fractures in patients with Duchenne or Becker muscular dystrophy. Our study cohort included males with Duchenne or Becker muscular dystrophy born between 1982 and 2006 who resided in Arizona, Colorado, Georgia, Iowa, and Western New York, retrospectively identified and followed through 2010. We utilized a multivariate Cox proportional hazard model to determine hazard ratios for relevant factors associated with first fracture risk including race/ethnicity, surveillance site, ambulation status, calcium/vitamin D use and duration, bisphosphonate use and duration, and corticosteroid use and duration. Results: Of 747 cases, 249 had at least 1 fracture (33.3%). Fulltime wheelchair use increased the risk of first fracture by 75% for every 3 months of use (hazard ratio=1.75, 95% confidence interval, 1.14, 2.68), but corticosteroid use, bisphosphonate use, and calcium/vitamin D use did not significantly affect risk in the final adjusted model. Conclusions: In this cohort, first fractures were common and full-time wheelchair use, but not corticosteroid use, was identified as a risk factor. The impact of prevention measures should be more thoroughly assessed. Clinical Relevance: Fractures are a significant concern for individuals with dystrophinopathies, but the contribution of various risk factors has not been consistently demonstrated.

AB - Background: Fractures are a significant concern for individuals with Duchenne/Becker muscular dystrophy with 21% to 44% of males experiencing a fracture. Factors that increase or decrease the risk for fracture have been suggested in past research, although statistical risk has not been determined. Methods: In this retrospective cohort study, we used the Muscular Dystrophy Surveillance, Tracking and Research Network cohort, a large, population-based sample to identify risk factors associated with first fractures in patients with Duchenne or Becker muscular dystrophy. Our study cohort included males with Duchenne or Becker muscular dystrophy born between 1982 and 2006 who resided in Arizona, Colorado, Georgia, Iowa, and Western New York, retrospectively identified and followed through 2010. We utilized a multivariate Cox proportional hazard model to determine hazard ratios for relevant factors associated with first fracture risk including race/ethnicity, surveillance site, ambulation status, calcium/vitamin D use and duration, bisphosphonate use and duration, and corticosteroid use and duration. Results: Of 747 cases, 249 had at least 1 fracture (33.3%). Fulltime wheelchair use increased the risk of first fracture by 75% for every 3 months of use (hazard ratio=1.75, 95% confidence interval, 1.14, 2.68), but corticosteroid use, bisphosphonate use, and calcium/vitamin D use did not significantly affect risk in the final adjusted model. Conclusions: In this cohort, first fractures were common and full-time wheelchair use, but not corticosteroid use, was identified as a risk factor. The impact of prevention measures should be more thoroughly assessed. Clinical Relevance: Fractures are a significant concern for individuals with dystrophinopathies, but the contribution of various risk factors has not been consistently demonstrated.

KW - Ambulation

KW - Boys

KW - Corticosteroid

KW - Fracture

KW - Muscular dystrophy

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