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

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)...

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Autores principales:	Jennifer Stimec, Saunya Dover, Eleanor Pullenayegum, Victor S. Blanchette, Andrea S. Doria, Brian M. Feldman, Manuel Carcao, Georges E. Rivard, Sara J. Israels, Anthony K. Chan, MacGregor Steele, Stephanie Cloutier, Robert J. Klaassen, Victoria E. Price, Roona Sinha, Nicole Laferriere, Elizabeth Paradis, John K. M. Wu, Paul Babyn
Formato:	article
Lenguaje:	EN
Publicado:	Wiley 2021
Materias:	hemophilia magnetic resonance imaging musculoskeletal system prophylaxis X‐rays Diseases of the blood and blood-forming organs RC633-647.5
Acceso en línea:	https://doaj.org/article/68fbd9e419e743b5a680bc936b57aeb5
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spelling	oai:doaj.org-article:68fbd9e419e743b5a680bc936b57aeb52021-11-29T09:35:28ZMagnetic resonance imaging in boys with severe hemophilia A: Serial and end‐of‐study findings from the Canadian Hemophilia Primary Prophylaxis Study2475-037910.1002/rth2.12565https://doaj.org/article/68fbd9e419e743b5a680bc936b57aeb52021-10-01T00:00:00Zhttps://doi.org/10.1002/rth2.12565https://doaj.org/toc/2475-0379Abstract 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.Jennifer StimecSaunya DoverEleanor PullenayegumVictor S. BlanchetteAndrea S. DoriaBrian M. FeldmanManuel CarcaoGeorges E. RivardSara J. IsraelsAnthony K. ChanMacGregor SteeleStephanie CloutierRobert J. KlaassenVictoria E. PriceRoona SinhaNicole LaferriereElizabeth ParadisJohn K. M. WuPaul BabynWileyarticlehemophiliamagnetic resonance imagingmusculoskeletal systemprophylaxisX‐raysDiseases of the blood and blood-forming organsRC633-647.5ENResearch and Practice in Thrombosis and Haemostasis, Vol 5, Iss 7, Pp n/a-n/a (2021)
institution	DOAJ
collection	DOAJ
language	EN
topic	hemophilia magnetic resonance imaging musculoskeletal system prophylaxis X‐rays Diseases of the blood and blood-forming organs RC633-647.5
spellingShingle	hemophilia magnetic resonance imaging musculoskeletal system prophylaxis X‐rays Diseases of the blood and blood-forming organs RC633-647.5 Jennifer Stimec Saunya Dover Eleanor Pullenayegum Victor S. Blanchette Andrea S. Doria Brian M. Feldman Manuel Carcao Georges E. Rivard Sara J. Israels Anthony K. Chan MacGregor Steele Stephanie Cloutier Robert J. Klaassen Victoria E. Price Roona Sinha Nicole Laferriere Elizabeth Paradis John K. M. Wu Paul Babyn Magnetic resonance imaging in boys with severe hemophilia A: Serial and end‐of‐study findings from the Canadian Hemophilia Primary Prophylaxis Study
description	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.
format	article
author	Jennifer Stimec Saunya Dover Eleanor Pullenayegum Victor S. Blanchette Andrea S. Doria Brian M. Feldman Manuel Carcao Georges E. Rivard Sara J. Israels Anthony K. Chan MacGregor Steele Stephanie Cloutier Robert J. Klaassen Victoria E. Price Roona Sinha Nicole Laferriere Elizabeth Paradis John K. M. Wu Paul Babyn
author_facet	Jennifer Stimec Saunya Dover Eleanor Pullenayegum Victor S. Blanchette Andrea S. Doria Brian M. Feldman Manuel Carcao Georges E. Rivard Sara J. Israels Anthony K. Chan MacGregor Steele Stephanie Cloutier Robert J. Klaassen Victoria E. Price Roona Sinha Nicole Laferriere Elizabeth Paradis John K. M. Wu Paul Babyn
author_sort	Jennifer Stimec
title	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_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_sort	magnetic resonance imaging in boys with severe hemophilia a: serial and end‐of‐study findings from the canadian hemophilia primary prophylaxis study
publisher	Wiley
publishDate	2021
url	https://doaj.org/article/68fbd9e419e743b5a680bc936b57aeb5
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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

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