TY - JOUR
T1 - New Inhibitors in the Ageing Population
T2 - A Retrospective, Observational, Cohort Study of New Inhibitors in Older People with Hemophilia
AU - Astermark, Jan
AU - Ay, Cihan
AU - Carvalho, Manuela
AU - D'Oiron, Roseline
AU - Moerloose, Philippe De
AU - Dolan, Gerard
AU - Fontana, Pierre
AU - Hermans, Cedric
AU - Holme, Pål Andre
AU - Katsarou, Olga
AU - Kenet, Gili
AU - Klamroth, Robert
AU - Mancuso, Maria Elisa
AU - Marquardt, Natascha
AU - Núñez, Ramiro
AU - Pabinger, Ingrid
AU - Tait, Robert
AU - Valk, Paul van der
N1 - Funding Information:
Support with data analysis and writing was provided by Gabrielle Hepworth and Karen Smith from Atrium Healthcare Communications Ltd., United Kingdom, and funded by Bayer.
Funding Information:
J.A. reports research grants from Bayer, CSL Behring, Shire/Takeda, SOBI; consulting fees from Bayer, CSL Behring, Novo Nordisk, Octapharma, Pfizer, Roche, Shire/Takeda, SOBI; honoraria for lectures, presentations, manuscript writing from Bayer, CSL Behring, Novo Nordisk, Octa-pharma, Pfizer, Roche, Shire/Takeda, SOBI; participation on a DSMB or Advisory Board from Bayer, CSL Behring, Novo Nordisk, Octapharma, Pfizer, Roche, Shire/Takeda, SOBI. C. A. reports honoraria for lectures, presentations, manuscript writing from Bayer, CSL Behring, Novo Nordisk, Shire/Takeda, SOBI; support for attending meetings from SOBI. M.C. reports consulting fees from Bayer, CSL Behring, Novo Nordisk, Roche, SOBI; honoraria for lectures, presentations, manuscript writing from Bayer, CSL Behring, Novo Nordisk, Roche, SOBI; support for attending meetings from Bayer, CSL Behring, Novo Nordisk, Pfizer, Roche, SOBI; participation on a DSMB or Advisory Board from Bayer, CSL Behring, Novo Nordisk, Roche, SOBI. RdO reports consulting fees from Bayer, CSL Behring, LFB, Novo Nordisk, Octapharma, Pfizer, Roche, Shire/Takeda, SOBI, Spark; honoraria for lectures, presentations, manuscript writing from Bayer, CSL Behring, LFB, Novo Nordisk, Octapharma, Pfizer, Roche, Shire/Takeda, SOBI, Spark; support for attending meetings from Bayer, CSL Behring, LFB, Novo Nordisk, Octapharma, Pfizer, Roche, Shire/Takeda, SOBI, Spark. GD reports consulting fees from Novo Nordisk, Roche; honoraria for lectures, presentations, manuscript writing from Novo Nordisk, Pfizer, Roche, Shire/Takeda. P.F. reports support for attending meetings from SOBI. C.H. reports honoraria for lectures, presentations, manuscript writing from Bayer. P.A.H. reports honoraria for lectures, presentations, manuscript writing from Bayer, Octapharma, Pfizer, Roche, Shire/Takeda, SOBI; support for attending meetings from Bayer, Octapharma. G.K. reports research grants from Bayer, BioMarin, Novo Nordisk, Pfizer, Roche, Sanofi-Aventis; consulting fees from ASC Therapeutics, Bayer, Pfizer, Roche, Sanofi-Aventis; honoraria for lectures, presentations, manuscript writing from Bayer, BioMarin, CSL Behring, Novo Nordisk, Pfizer, Roche, Sanofi-Aventis, Shire/Takeda, Spark; participation on a DSMB or Advisory Board from Bayer, Novo Nordisk, Pfizer, Roche, Sanofi-Aventis, Shire/Takeda, UniQure. R.K. reports research grants from Bayer, Leo; consulting fees from Bayer, Biomarin, Biotest, CSL Behring, Novo Nordisk, Octapharma, Pfizer, Roche, Shire/Takeda, SOBI; honoraria for lectures, presentations, manuscript writing from Bayer, Biomarin, Biotest, CSL Behring, Novo Nordisk, Octapharma, Pfizer, Roche, Shire/Takeda, SOBI. M. E.M. reports consulting fees from Bayer, Biomarin, Catalyst, CSL Behring, Grifols, Kedrion, LFB, Novo Nordisk, Octa-pharma, Pfizer, Roche, Shire/Takeda, Sobi; honoraria for lectures, presentations, manuscript writing from Bayer, Biomarin, CSL Behring, Grifols, Kedrion, Novo Nordisk, Octapharma, Roche, Shire/Takeda, SOBI, Spark; support for attending meetings from Bayer, Novo Nordisk, Roche, SOBI. NM reports honoraria for lectures, presentations, manuscript writing from Bayer, Chugai, CSL Behring, Novo Nordisk, Octapharma, Pfizer, Roche, SOBI; support for attending meetings from Novo Nordisk, Octapharma, Shire/Takeda, SOBI; participation on a DSMB or Advisory Board from Bayer, Novo Nordisk, SOBI. R.N. reports honoraria for lectures, presentations, manuscript writing from
Publisher Copyright:
© 2022 Georg Thieme Verlag. All rights reserved.
PY - 2022/6/1
Y1 - 2022/6/1
N2 - INTRODUCTION: A second peak of inhibitors has been reported in patients with severe hemophilia A (HA) aged >50 years in the United Kingdom. The reason for this suggested breakdown of tolerance in the aging population is unclear, as is the potential impact of regular exposure to the deficient factor by prophylaxis at higher age. No data on hemophilia B (HB) have ever been reported.AIM: The ADVANCE Working Group investigated the incidence of late-onset inhibitors and the use of prophylaxis in patients with HA and HB aged ≥40 years.METHODS: A retrospective, observational, cohort, survey-based study of all patients aged ≥40 years with HA or HB treated at an ADVANCE hemophilia treatment center.RESULTS: Information on 3,095 people aged ≥40 years with HA or HB was collected. Of the 2,562 patients with severe HA, the majority (73% across all age groups) received prophylaxis. In patients with severe HA, the inhibitor incidence per 1,000 treatment years was 2.37 (age 40-49), 1.25 (age 50-59), and 1.45 (age 60 + ). Overall, the inhibitor incidence was greatest in those with moderate HA (5.77 [age 40-49], 6.59 [age 50-59], and 4.69 [age 60 + ]) and the majority of inhibitor cases were preceded by a potential immune system challenge. No inhibitors in patients with HB were reported.CONCLUSION: Our data do not identify a second peak of inhibitor development in older patients with hemophilia. Prophylaxis may be beneficial in older patients with severe, and possibly moderate HA, to retain a tolerant state at a higher age.
AB - INTRODUCTION: A second peak of inhibitors has been reported in patients with severe hemophilia A (HA) aged >50 years in the United Kingdom. The reason for this suggested breakdown of tolerance in the aging population is unclear, as is the potential impact of regular exposure to the deficient factor by prophylaxis at higher age. No data on hemophilia B (HB) have ever been reported.AIM: The ADVANCE Working Group investigated the incidence of late-onset inhibitors and the use of prophylaxis in patients with HA and HB aged ≥40 years.METHODS: A retrospective, observational, cohort, survey-based study of all patients aged ≥40 years with HA or HB treated at an ADVANCE hemophilia treatment center.RESULTS: Information on 3,095 people aged ≥40 years with HA or HB was collected. Of the 2,562 patients with severe HA, the majority (73% across all age groups) received prophylaxis. In patients with severe HA, the inhibitor incidence per 1,000 treatment years was 2.37 (age 40-49), 1.25 (age 50-59), and 1.45 (age 60 + ). Overall, the inhibitor incidence was greatest in those with moderate HA (5.77 [age 40-49], 6.59 [age 50-59], and 4.69 [age 60 + ]) and the majority of inhibitor cases were preceded by a potential immune system challenge. No inhibitors in patients with HB were reported.CONCLUSION: Our data do not identify a second peak of inhibitor development in older patients with hemophilia. Prophylaxis may be beneficial in older patients with severe, and possibly moderate HA, to retain a tolerant state at a higher age.
KW - Adult
KW - Aging
KW - Cohort Studies
KW - Factor VIII/therapeutic use
KW - Hemophilia A/drug therapy
KW - Hemophilia B/drug therapy
KW - Humans
KW - Middle Aged
KW - Retrospective Studies
KW - inhibitors
KW - prophylaxis
KW - hemophilia A/B
KW - aging
UR - http://www.scopus.com/inward/record.url?scp=85119113861&partnerID=8YFLogxK
U2 - 10.1055/a-1642-4067
DO - 10.1055/a-1642-4067
M3 - Article
C2 - 34507368
SN - 0340-6245
VL - 122
SP - 905
EP - 912
JO - Thrombosis and Haemostasis
JF - Thrombosis and Haemostasis
IS - 6
ER -