Measuring the impact of hemophilia on families: Development of the Hemophilia Family Impact Tool (H-FIT)

Saunya Dover; Nancy L. Young; Victor S. Blanchette; Robert J. Klaassen; Anthony K. Chan; Cindy Wakefield; Vanessa Bouskill; Manuel Carcao; Mark Belletrutti; Aisha A.K. Bruce; Victoria E. Price

doi:10.1002/rth2.12519

Measuring the impact of hemophilia on families: Development of the Hemophilia Family Impact Tool (H-FIT)

Saunya Dover, Nancy L. Young, Victor S. Blanchette, Robert J. Klaassen, Anthony K. Chan, Cindy Wakefield, Vanessa Bouskill, Manuel Carcao, Mark Belletrutti, Aisha A.K. Bruce, Victoria E. Price

Medicine

Résultat de recherche: Article › examen par les pairs

3 Citations (Scopus)

Résumé

Introduction: This study aimed to assess the impact of hemophilia on families, in the context of current and emerging hemostatic therapies, and explore the need for a hemophilia-specific tool targeted at parents of boys aged <4 years. A secondary aim was to develop and validate the new tool. Methods: Focus groups were conducted with parents of boys with hemophilia and hemophilia health care providers at Canadian hemophilia treatment centers (HTCs) to review the relevance of the Pediatric Quality of Life Family Impact Module (PedsQL-FIM); a novel questionnaire was developed by identifying core themes expressed. This questionnaire, the Hemophilia Family Impact Tool (H-FIT) was validated in a sample of parents of boys with hemophilia relative to the PedsQL-FIM. Results: Seven focus groups were conducted at four HTCs, generating themes specific to hemophilia not covered by the PedsQL-FIM, suggesting that a new tool be developed (the H-FIT). In the validation phase, 54 parents completed the H-FIT and PedsQL-FIM. The H-FIT had a strong correlation with the PedsQL-FIM across all ages (r = 0.79; P <.0001) and a moderate correlation for parents of boys aged <7 years (r = 0.64; P =.0007). There was a significant difference between the mean H-FIT scores for parents of boys using extended half-life factor (68.1; standard deviation [SD]=14.2) compared to standard half-life factor (54.7; SD=18.4; P =.04). Conclusion: A novel, disease-specific tool, the H-FIT, has been developed to measure the impact of hemophilia on families. The H-FIT has good preliminary measurement properties and may be responsive to changes in therapy associated with a decreased burden of administration.

Langue d'origine	English
Numéro d'article	e12519
Journal	Research and Practice in Thrombosis and Haemostasis
Volume	5
Numéro de publication	4
DOI	https://doi.org/10.1002/rth2.12519
Statut de publication	Published - mai 2021

Note bibliographique

Funding Information:
Funding for this study was provided by Sanofi-Genzyme. The funder did not have any role in study design, data collection, analysis, or preparation of the manuscript. The Hemophilia Family Impact Tool (H-FIT) is copyrighted. The tool is available by way of an academic or commercial license. For details regarding licensure to allow use of the H-FIT and its scoring instructions please contact the Industry Partnership and Commercialization Office at The Hospital for Sick Children (555 University Avenue, Toronto, Ontario M5G 1X8, Canada; e-mail, ipc.requests@sickkids.ca; telephone, +1-416-813-6635).

Funding Information:
Funding for this study was provided by Sanofi‐Genzyme. The funder did not have any role in study design, data collection, analysis, or preparation of the manuscript.

Funding Information:
SD, NLY, VSB, and VEP have a patent on the H‐FIT, with royalties paid to The Hospital for Sick Children, NLY, and VEP. VSB reports that he is chair of the International Prophylaxis Study Group, a cooperative study group that is funded by education grants from Bayer Healthcare, Bioverativ/Sanofi, Novo Nordisk, Pfizer, Shire/Takeda, and Spark Therapeutics to The Hospital for Sick Children’s “SickKids” Foundation. He has received fees for participation in advisory boards/education events supported by Amgen, Bayer, Novo Nordisk, Pfizer, Roche, and Shire/Takeda and for participation in data safety monitoring boards for Octapharma and Shire/Takeda. He has received investigator‐initiated, industry‐supported research grants from Novo Nordisk, Bioverativ/Sanofi, and Shire/Takeda. RJK reports receiving speaker and/or consultant fees from Agios Pharmaceuticals Inc., Amgen, Hoffman‐LaRoche LTD, Shire Pharma Canada ULC, Novo Nordisk Canada Inc, Octapharma AG, Takeda, and Sanofi‐Genzyme. MB has received fees for participation in advisory boards from Roche, Novo Nordisk, and Takeda, and has received grants from Octapharma AG. MC reports having received research support from Bayer, Bioverativ/Sanofi, CSL‐Behring, Novo Nordisk, Octapharma, Pfizer, and Shire. He has also received honoraria for speaking/participating in advisory boards from Bayer, Bioverativ/Sanofi, Biotest, CSL Behring, Grifols, LFB, Novo Nordisk, Octapharma, Pfizer, Roche, and Shire. The remaining authors have no disclosures.

Publisher Copyright:
© 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).

ASJC Scopus Subject Areas

Hematology

PubMed: MeSH publication types

Journal Article

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10.1002/rth2.12519

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Dover, S., Young, N. L., Blanchette, V. S., Klaassen, R. J., Chan, A. K., Wakefield, C., Bouskill, V., Carcao, M., Belletrutti, M., Bruce, A. A. K., & Price, V. E. (2021). Measuring the impact of hemophilia on families: Development of the Hemophilia Family Impact Tool (H-FIT). Research and Practice in Thrombosis and Haemostasis, 5(4), Article e12519. https://doi.org/10.1002/rth2.12519

@article{041d14ebffda4dfda2b55d84e3fd1443,

title = "Measuring the impact of hemophilia on families: Development of the Hemophilia Family Impact Tool (H-FIT)",

abstract = "Introduction: This study aimed to assess the impact of hemophilia on families, in the context of current and emerging hemostatic therapies, and explore the need for a hemophilia-specific tool targeted at parents of boys aged <4 years. A secondary aim was to develop and validate the new tool. Methods: Focus groups were conducted with parents of boys with hemophilia and hemophilia health care providers at Canadian hemophilia treatment centers (HTCs) to review the relevance of the Pediatric Quality of Life Family Impact Module (PedsQL-FIM); a novel questionnaire was developed by identifying core themes expressed. This questionnaire, the Hemophilia Family Impact Tool (H-FIT) was validated in a sample of parents of boys with hemophilia relative to the PedsQL-FIM. Results: Seven focus groups were conducted at four HTCs, generating themes specific to hemophilia not covered by the PedsQL-FIM, suggesting that a new tool be developed (the H-FIT). In the validation phase, 54 parents completed the H-FIT and PedsQL-FIM. The H-FIT had a strong correlation with the PedsQL-FIM across all ages (r = 0.79; P <.0001) and a moderate correlation for parents of boys aged <7 years (r = 0.64; P =.0007). There was a significant difference between the mean H-FIT scores for parents of boys using extended half-life factor (68.1; standard deviation [SD]=14.2) compared to standard half-life factor (54.7; SD=18.4; P =.04). Conclusion: A novel, disease-specific tool, the H-FIT, has been developed to measure the impact of hemophilia on families. The H-FIT has good preliminary measurement properties and may be responsive to changes in therapy associated with a decreased burden of administration.",

author = "Saunya Dover and Young, {Nancy L.} and Blanchette, {Victor S.} and Klaassen, {Robert J.} and Chan, {Anthony K.} and Cindy Wakefield and Vanessa Bouskill and Manuel Carcao and Mark Belletrutti and Bruce, {Aisha A.K.} and Price, {Victoria E.}",

note = "Funding Information: Funding for this study was provided by Sanofi-Genzyme. The funder did not have any role in study design, data collection, analysis, or preparation of the manuscript. The Hemophilia Family Impact Tool (H-FIT) is copyrighted. The tool is available by way of an academic or commercial license. For details regarding licensure to allow use of the H-FIT and its scoring instructions please contact the Industry Partnership and Commercialization Office at The Hospital for Sick Children (555 University Avenue, Toronto, Ontario M5G 1X8, Canada; e-mail, ipc.requests@sickkids.ca; telephone, +1-416-813-6635). Funding Information: Funding for this study was provided by Sanofi‐Genzyme. The funder did not have any role in study design, data collection, analysis, or preparation of the manuscript. Funding Information: SD, NLY, VSB, and VEP have a patent on the H‐FIT, with royalties paid to The Hospital for Sick Children, NLY, and VEP. VSB reports that he is chair of the International Prophylaxis Study Group, a cooperative study group that is funded by education grants from Bayer Healthcare, Bioverativ/Sanofi, Novo Nordisk, Pfizer, Shire/Takeda, and Spark Therapeutics to The Hospital for Sick Children{\textquoteright}s “SickKids” Foundation. He has received fees for participation in advisory boards/education events supported by Amgen, Bayer, Novo Nordisk, Pfizer, Roche, and Shire/Takeda and for participation in data safety monitoring boards for Octapharma and Shire/Takeda. He has received investigator‐initiated, industry‐supported research grants from Novo Nordisk, Bioverativ/Sanofi, and Shire/Takeda. RJK reports receiving speaker and/or consultant fees from Agios Pharmaceuticals Inc., Amgen, Hoffman‐LaRoche LTD, Shire Pharma Canada ULC, Novo Nordisk Canada Inc, Octapharma AG, Takeda, and Sanofi‐Genzyme. MB has received fees for participation in advisory boards from Roche, Novo Nordisk, and Takeda, and has received grants from Octapharma AG. MC reports having received research support from Bayer, Bioverativ/Sanofi, CSL‐Behring, Novo Nordisk, Octapharma, Pfizer, and Shire. He has also received honoraria for speaking/participating in advisory boards from Bayer, Bioverativ/Sanofi, Biotest, CSL Behring, Grifols, LFB, Novo Nordisk, Octapharma, Pfizer, Roche, and Shire. The remaining authors have no disclosures. Publisher Copyright: {\textcopyright} 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).",

year = "2021",

month = may,

doi = "10.1002/rth2.12519",

language = "English",

volume = "5",

journal = "Research and Practice in Thrombosis and Haemostasis",

issn = "2475-0379",

publisher = "Wiley-Blackwell Publishing Ltd",

number = "4",

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TY - JOUR

T1 - Measuring the impact of hemophilia on families

T2 - Development of the Hemophilia Family Impact Tool (H-FIT)

AU - Dover, Saunya

AU - Young, Nancy L.

AU - Blanchette, Victor S.

AU - Klaassen, Robert J.

AU - Chan, Anthony K.

AU - Wakefield, Cindy

AU - Bouskill, Vanessa

AU - Carcao, Manuel

AU - Belletrutti, Mark

AU - Bruce, Aisha A.K.

AU - Price, Victoria E.

N1 - Funding Information: Funding for this study was provided by Sanofi-Genzyme. The funder did not have any role in study design, data collection, analysis, or preparation of the manuscript. The Hemophilia Family Impact Tool (H-FIT) is copyrighted. The tool is available by way of an academic or commercial license. For details regarding licensure to allow use of the H-FIT and its scoring instructions please contact the Industry Partnership and Commercialization Office at The Hospital for Sick Children (555 University Avenue, Toronto, Ontario M5G 1X8, Canada; e-mail, ipc.requests@sickkids.ca; telephone, +1-416-813-6635). Funding Information: Funding for this study was provided by Sanofi‐Genzyme. The funder did not have any role in study design, data collection, analysis, or preparation of the manuscript. Funding Information: SD, NLY, VSB, and VEP have a patent on the H‐FIT, with royalties paid to The Hospital for Sick Children, NLY, and VEP. VSB reports that he is chair of the International Prophylaxis Study Group, a cooperative study group that is funded by education grants from Bayer Healthcare, Bioverativ/Sanofi, Novo Nordisk, Pfizer, Shire/Takeda, and Spark Therapeutics to The Hospital for Sick Children’s “SickKids” Foundation. He has received fees for participation in advisory boards/education events supported by Amgen, Bayer, Novo Nordisk, Pfizer, Roche, and Shire/Takeda and for participation in data safety monitoring boards for Octapharma and Shire/Takeda. He has received investigator‐initiated, industry‐supported research grants from Novo Nordisk, Bioverativ/Sanofi, and Shire/Takeda. RJK reports receiving speaker and/or consultant fees from Agios Pharmaceuticals Inc., Amgen, Hoffman‐LaRoche LTD, Shire Pharma Canada ULC, Novo Nordisk Canada Inc, Octapharma AG, Takeda, and Sanofi‐Genzyme. MB has received fees for participation in advisory boards from Roche, Novo Nordisk, and Takeda, and has received grants from Octapharma AG. MC reports having received research support from Bayer, Bioverativ/Sanofi, CSL‐Behring, Novo Nordisk, Octapharma, Pfizer, and Shire. He has also received honoraria for speaking/participating in advisory boards from Bayer, Bioverativ/Sanofi, Biotest, CSL Behring, Grifols, LFB, Novo Nordisk, Octapharma, Pfizer, Roche, and Shire. The remaining authors have no disclosures. Publisher Copyright: © 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).

PY - 2021/5

Y1 - 2021/5

N2 - Introduction: This study aimed to assess the impact of hemophilia on families, in the context of current and emerging hemostatic therapies, and explore the need for a hemophilia-specific tool targeted at parents of boys aged <4 years. A secondary aim was to develop and validate the new tool. Methods: Focus groups were conducted with parents of boys with hemophilia and hemophilia health care providers at Canadian hemophilia treatment centers (HTCs) to review the relevance of the Pediatric Quality of Life Family Impact Module (PedsQL-FIM); a novel questionnaire was developed by identifying core themes expressed. This questionnaire, the Hemophilia Family Impact Tool (H-FIT) was validated in a sample of parents of boys with hemophilia relative to the PedsQL-FIM. Results: Seven focus groups were conducted at four HTCs, generating themes specific to hemophilia not covered by the PedsQL-FIM, suggesting that a new tool be developed (the H-FIT). In the validation phase, 54 parents completed the H-FIT and PedsQL-FIM. The H-FIT had a strong correlation with the PedsQL-FIM across all ages (r = 0.79; P <.0001) and a moderate correlation for parents of boys aged <7 years (r = 0.64; P =.0007). There was a significant difference between the mean H-FIT scores for parents of boys using extended half-life factor (68.1; standard deviation [SD]=14.2) compared to standard half-life factor (54.7; SD=18.4; P =.04). Conclusion: A novel, disease-specific tool, the H-FIT, has been developed to measure the impact of hemophilia on families. The H-FIT has good preliminary measurement properties and may be responsive to changes in therapy associated with a decreased burden of administration.

AB - Introduction: This study aimed to assess the impact of hemophilia on families, in the context of current and emerging hemostatic therapies, and explore the need for a hemophilia-specific tool targeted at parents of boys aged <4 years. A secondary aim was to develop and validate the new tool. Methods: Focus groups were conducted with parents of boys with hemophilia and hemophilia health care providers at Canadian hemophilia treatment centers (HTCs) to review the relevance of the Pediatric Quality of Life Family Impact Module (PedsQL-FIM); a novel questionnaire was developed by identifying core themes expressed. This questionnaire, the Hemophilia Family Impact Tool (H-FIT) was validated in a sample of parents of boys with hemophilia relative to the PedsQL-FIM. Results: Seven focus groups were conducted at four HTCs, generating themes specific to hemophilia not covered by the PedsQL-FIM, suggesting that a new tool be developed (the H-FIT). In the validation phase, 54 parents completed the H-FIT and PedsQL-FIM. The H-FIT had a strong correlation with the PedsQL-FIM across all ages (r = 0.79; P <.0001) and a moderate correlation for parents of boys aged <7 years (r = 0.64; P =.0007). There was a significant difference between the mean H-FIT scores for parents of boys using extended half-life factor (68.1; standard deviation [SD]=14.2) compared to standard half-life factor (54.7; SD=18.4; P =.04). Conclusion: A novel, disease-specific tool, the H-FIT, has been developed to measure the impact of hemophilia on families. The H-FIT has good preliminary measurement properties and may be responsive to changes in therapy associated with a decreased burden of administration.

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Measuring the impact of hemophilia on families: Development of the Hemophilia Family Impact Tool (H-FIT)

Résumé

Note bibliographique

ASJC Scopus Subject Areas

PubMed: MeSH publication types

Accès au document

Autres fichiers et liens

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