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Blood, 1 April 2004, Vol. 103, No. 7, pp. 2467-2473.
Prepublished online as a Blood First Edition Paper on November 13, 2003; DOI 10.1182/blood-2003-05-1457.

Submitted May 8, 2003
Accepted November 4, 2003
Joint range of motion limitations among young males with hemophilia: prevalence and risk factors
John M Soucie*, Christy Cianfrini, Robert L Janco, Roshni Kulkarni, Julie Hambleton, Bruce L Evatt, Angela Forsyth, Sue Geraghty, Keith Hoots, Tom Abshire, Randall Curtis, Ann Forsberg, Heather Huszti, Margaret Wagner, and Gilbert C White
Hematologic Diseases Branch, Centers for Disease Control and Prevention, Atlanta, GA, USA
Department of Pediatrics, Vanderbilt University School of Medicine, Nashville, TN, USA
Department of Pediatrics and Human Development, Michigan State University, East Lansing, MI, USA
Division of Hematology/Oncology, University of California, San Francisco, CA, USA
Penn Comprehensive Hemophilia Program, Presbyterian Medical Center, Philadelphia, PA, USA
Mountain States Regional Hemophilia Center, Aurora, CO, USA
Department of Pediatrics, University of Texas Health Sciences Center, Houston, TX, USA
Pediatric HemOnc, Emory University School of Medicine, Atlanta, GA, USA
Genetic Disease Branch, Department of Health Services, Berkeley, CA, USA
New England Hemophilia Center, UMass Memorial Health Care, Worcester, MA, USA
Department of Health Psychology, Children's Hospital of Orange County, Orange, CA, USA
Christiana Care Health System, Newark, DE, USA
Division of Hematology-Oncology, University of North Carolina, Chapel Hill, NC, USA
* Corresponding author; email: msoucie{at}cdc.gov.
Chronic joint disease from repeated bleeding into joints is a serious complication of hemophilia. To measure the extent of and to identify risk factors for joint range of motion (ROM) loss, we used cross-sectional data collected from 4,343 males with hemophilia aged 2-19 years who received care at 136 U.S. hemophilia treatment centers (HTCs). Factors examined included age, race/ethnicity, family history, insurance status, age at diagnosis and first HTC visit, frequency of HTC visits, hemophilia type, bleeding frequency, prophylaxis use, inhibitor status, body mass index (BMI), and recent orthopedic procedures. Trained personnel using a standard protocol obtained ROM measurements on 10 joints (hips, knees, shoulders, elbows, and ankles). Analyses used multiple linear regression to model overall ROM loss separately by disease severity. For persons in all severity groups, joint ROM loss was positively associated with older age, non-white race, and increased BMI. For those with severe disease, ROM loss was also positively associated with number of bleeds and was greater for those with inhibitors or recent orthopedic procedures. We conclude that ROM loss begins at an early age, especially for those with severe and moderate disease, and that BMI is an important, potentially modifiable risk factor for joint ROM loss.

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