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Magnetic resonance imaging in boys with severe hemophilia A: Serial and end‐of‐study findings from the Canadian Hemophilia Primary Prophylaxis Study

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 e...

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Autores principales: Stimec, Jennifer, Dover, Saunya, Pullenayegum, Eleanor, Blanchette, Victor S., Doria, Andrea S., Feldman, Brian M., Carcao, Manuel, Rivard, Georges E., Israels, Sara J., Chan, Anthony K., Steele, MacGregor, Cloutier, Stephanie, Klaassen, Robert J., Price, Victoria E., Sinha, Roona, Laferriere, Nicole, Paradis, Elizabeth, Wu, John K. M., Babyn, Paul
Formato: Online Artículo Texto
Lenguaje:English
Publicado: John Wiley and Sons Inc. 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8520573/
https://www.ncbi.nlm.nih.gov/pubmed/34703973
http://dx.doi.org/10.1002/rth2.12565
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author Stimec, Jennifer
Dover, Saunya
Pullenayegum, Eleanor
Blanchette, Victor S.
Doria, Andrea S.
Feldman, Brian M.
Carcao, Manuel
Rivard, Georges E.
Israels, Sara J.
Chan, Anthony K.
Steele, MacGregor
Cloutier, Stephanie
Klaassen, Robert J.
Price, Victoria E.
Sinha, Roona
Laferriere, Nicole
Paradis, Elizabeth
Wu, John K. M.
Babyn, Paul
author_facet Stimec, Jennifer
Dover, Saunya
Pullenayegum, Eleanor
Blanchette, Victor S.
Doria, Andrea S.
Feldman, Brian M.
Carcao, Manuel
Rivard, Georges E.
Israels, Sara J.
Chan, Anthony K.
Steele, MacGregor
Cloutier, Stephanie
Klaassen, Robert J.
Price, Victoria E.
Sinha, Roona
Laferriere, Nicole
Paradis, Elizabeth
Wu, John K. M.
Babyn, Paul
author_sort Stimec, Jennifer
collection PubMed
description 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. [Image: see text]
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spelling pubmed-85205732021-10-25 Magnetic resonance imaging in boys with severe hemophilia A: Serial and end‐of‐study findings from the Canadian Hemophilia Primary Prophylaxis Study Stimec, Jennifer Dover, Saunya Pullenayegum, Eleanor Blanchette, Victor S. Doria, Andrea S. Feldman, Brian M. Carcao, Manuel Rivard, Georges E. Israels, Sara J. Chan, Anthony K. Steele, MacGregor Cloutier, Stephanie Klaassen, Robert J. Price, Victoria E. Sinha, Roona Laferriere, Nicole Paradis, Elizabeth Wu, John K. M. Babyn, Paul Res Pract Thromb Haemost Original Articles 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. [Image: see text] John Wiley and Sons Inc. 2021-10-16 /pmc/articles/PMC8520573/ /pubmed/34703973 http://dx.doi.org/10.1002/rth2.12565 Text en © 2021 The Authors. Research and Practice in Thrombosis and Haemostasis published by Wiley Periodicals LLC on behalf of International Society on Thrombosis and Haemostasis (ISTH) https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open access article under the terms of the http://creativecommons.org/licenses/by-nc-nd/4.0/ (https://creativecommons.org/licenses/by-nc-nd/4.0/) License, which permits use and distribution in any medium, provided the original work is properly cited, the use is non‐commercial and no modifications or adaptations are made.
spellingShingle Original Articles
Stimec, Jennifer
Dover, Saunya
Pullenayegum, Eleanor
Blanchette, Victor S.
Doria, Andrea S.
Feldman, Brian M.
Carcao, Manuel
Rivard, Georges E.
Israels, Sara J.
Chan, Anthony K.
Steele, MacGregor
Cloutier, Stephanie
Klaassen, Robert J.
Price, Victoria E.
Sinha, Roona
Laferriere, Nicole
Paradis, Elizabeth
Wu, John K. M.
Babyn, Paul
Magnetic resonance imaging in boys with severe hemophilia A: Serial and end‐of‐study findings from the Canadian Hemophilia Primary Prophylaxis Study
title Magnetic resonance imaging in boys with severe hemophilia A: Serial and end‐of‐study findings from the Canadian Hemophilia Primary Prophylaxis Study
title_full Magnetic resonance imaging in boys with severe hemophilia A: Serial and end‐of‐study findings from the Canadian Hemophilia Primary Prophylaxis Study
title_fullStr Magnetic resonance imaging in boys with severe hemophilia A: Serial and end‐of‐study findings from the Canadian Hemophilia Primary Prophylaxis Study
title_full_unstemmed Magnetic resonance imaging in boys with severe hemophilia A: Serial and end‐of‐study findings from the Canadian Hemophilia Primary Prophylaxis Study
title_short Magnetic resonance imaging in boys with severe hemophilia A: Serial and end‐of‐study findings from the Canadian Hemophilia Primary Prophylaxis Study
title_sort magnetic resonance imaging in boys with severe hemophilia a: serial and end‐of‐study findings from the canadian hemophilia primary prophylaxis study
topic Original Articles
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8520573/
https://www.ncbi.nlm.nih.gov/pubmed/34703973
http://dx.doi.org/10.1002/rth2.12565
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