Abstract

Background

This study examined the structural outcomes for joints of boys with severe hemophilia A receiving frequency/dose‐escalated primary prophylaxis using magnetic resonance imaging (MRI), and the importance of interval MRI changes.

Methods

Forty‐six subjects (27 with interval studies) were evaluated by radiographs (X‐rays) and mid‐ and end‐of‐study MRIs (using the International Prophylaxis Study Group scale), as part of the Canadian Hemophilia Prophylaxis Study. The primary outcome was the presence of MRI osteochondral findings.

Results

The median (range) time on study at the end‐of‐study MRI examination was 9.6 (4.8–16.0) years, during which 18 of 46 subjects (39%) had osteochondral changes in at least one joint. An interval change in MRI score of at least 1 point was observed in 44% of joints (43 ankles, 21 elbows, 4 knees); at least one joint showed this change in all 27 subjects. Self‐reported interval hemarthrosis was associated with a higher likelihood of interval osteochondral change (odds ratio [OR], 1.49; 95% confidence interval [CI] = 1.08–2.06). Presence of synovial hypertrophy or hemosiderin on interval MRIs was associated with an OR of 4.71 (95% CI, 1.92–11.57) and 5.25 (95% CI, 2.05–13.40) of later osteochondral changes on MRI.

Discussion

MRI changes were seen in 39% of subjects. Interval index joint bleeding was associated with an increased risk of later MRI changes, and earlier soft‐tissue changes were associated with subsequent osteochondral changes.

Details

Title
Magnetic resonance imaging in boys with severe hemophilia A: Serial and end‐of‐study findings from the Canadian Hemophilia Primary Prophylaxis Study
Author
Stimec, Jennifer 1 ; Dover, Saunya 2   VIAFID ORCID Logo  ; Pullenayegum, Eleanor 3 ; Blanchette, Victor S 4   VIAFID ORCID Logo  ; Doria, Andrea S 1   VIAFID ORCID Logo  ; Feldman, Brian M 5   VIAFID ORCID Logo  ; Carcao, Manuel 4 ; Rivard, Georges E 6   VIAFID ORCID Logo  ; Israels, Sara J 7   VIAFID ORCID Logo  ; Chan, Anthony K 8   VIAFID ORCID Logo  ; Steele, MacGregor 9   VIAFID ORCID Logo  ; Cloutier, Stephanie 10 ; Klaassen, Robert J 11   VIAFID ORCID Logo  ; Price, Victoria E 12 ; Sinha, Roona 13 ; Laferriere, Nicole 14 ; Paradis, Elizabeth 15 ; Wu, John K M 16 ; Babyn, Paul 17 

 Department of Medical Imaging, The Hospital for Sick Children, University of Toronto, Toronto, ON, Canada 
 Child Health Evaluative Sciences, The Hospital for Sick Children Research Institute, Toronto, ON, Canada 
 Child Health Evaluative Sciences, The Hospital for Sick Children Research Institute, Toronto, ON, Canada; Institute of Health Policy, Management & Evaluation, The Dalla Lana School of Public Health, University of Toronto, Toronto, ON, Canada 
 Child Health Evaluative Sciences, The Hospital for Sick Children Research Institute, Toronto, ON, Canada; Department of Pediatrics, University of Toronto, Toronto, ON, Canada; Division of Hematology/Oncology, The Hospital for Sick Children, Toronto, ON, Canada 
 Child Health Evaluative Sciences, The Hospital for Sick Children Research Institute, Toronto, ON, Canada; Institute of Health Policy, Management & Evaluation, The Dalla Lana School of Public Health, University of Toronto, Toronto, ON, Canada; Department of Pediatrics, University of Toronto, Toronto, ON, Canada; Division of Rheumatology, The Hospital for Sick Children, Toronto, ON, Canada 
 Division of Hematology/Oncology, Department of Pediatrics, CHU Sainte‐Justine, Montreal, QC, Canada 
 Department of Pediatrics and Child Health, University of Manitoba, Winnipeg, MB, Canada 
 Department of Pediatrics, McMaster Children's Hospital, McMaster University, Hamilton, ON, Canada 
 Section of Pediatric Hematology, Department of Pediatrics, University of Calgary and Alberta Children's Hospital, Calgary, AB, Canada 
10  Centre de l’hémophilie de l'est du Québec, Hôpital de l'Enfant‐Jésus, Université Laval, Québec, QC, Canada 
11  Division of Hematology Oncology, Children’s Hospital of Eastern Ontario Research Institute, Ottawa, ON, Canada 
12  Division of Pediatric Hematology/Oncology, IWK Health Centre, Halifax, NS, Canada 
13  Saskatchewan Bleeding Disorders Program, Royal University Hospital, Saskatoon, SK, Canada 
14  Division of Hematology/Oncology, Thunder Bay Regional Cancer Care, Thunder Bay, ON, Canada 
15  Health Sciences North, Sudbury, ON, Canada 
16  Division of Hematology/Oncology/BMT, Department of Pediatrics, UBC & BC Children’s Hospital, Vancouver, BC, Canada 
17  Department of Medical Imaging, University of Saskatchewan, Saskatoon, SK, Canada 
Section
ORIGINAL ARTICLES
Publication year
2021
Publication date
Oct 2021
Publisher
Elsevier Limited
e-ISSN
24750379
Source type
Scholarly Journal
Language of publication
English
ProQuest document ID
2597045473
Copyright
© 2021. This work is published under http://creativecommons.org/licenses/by-nc-nd/4.0/ (the “License”). Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License.