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Blood, 1 June 2007, Vol. 109, No. 11, pp. 4648-4654. Prepublished online as a Blood First Edition Paper on February 8, 2007; DOI 10.1182/blood-2006-11-056291. This Article Full Text Full Text (PDF) Supplemental Appendix All Versions of this Article: blood-2006-11-056291v1 109/11/4648    most recent Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Download to citation manager Rights and Permissions Citing Articles Citing Articles via HighWire Citing Articles via CrossRef Citing Articles via Google Scholar Google Scholar Articles by Gouw, S. C. Articles by Marijke van den Berg, H. Search for Related Content PubMed PubMed Citation Articles by Gouw, S. C. Articles by Marijke van den Berg, H. Related Collections Hemostasis, Thrombosis, and Vascular Biology Clinical Trials and Observations Related Article in Blood Online Social Bookmarking                   What's this?  Previous Article  |  Table of Contents  |  Next Article  CLINICAL TRIALS AND OBSERVATIONS Treatment-related risk factors of inhibitor development in previously untreated patients with hemophilia A: the CANAL cohort study Samantha C. Gouw1,2, Johanna G. van der Bom3, H. Marijke van den Berg, for the CANAL Study group1 1 Van Creveldkliniek, University Medical Center Utrecht, Utrecht, the Netherlands; 2 Department of Pediatrics, Wilhelmina Children's Hospital, University Medical Center Utrecht, Utrecht, the Netherlands; 3 Department of Clinical Epidemiology, Leiden University Medical Center, Leiden, the Netherlands The CANAL Study (Concerted Action on Neutralizing Antibodies in severe hemophilia A) was designed to describe the relationship between treatment characteristics and inhibitor development in previously untreated patients with severe hemophilia A. This multicenter retrospective cohort study investigated 366 consecutive patients born between 1990 and 2000. The outcome was clinically relevant inhibitor development, defined as the occurrence of at least 2 positive inhibitor titers combined with a decreased recovery. Eighty-seven (24%) patients developed inhibitors (69 high titer [19%]). The incidence of inhibitors appeared to be associated with age at first treatment, decreasing from 41% for those treated within the first month of age to 18% in those treated after 18 months; after adjustment for treatment intensity, this association largely disappeared. Surgical procedures and peak treatment moments at start of treatment increased inhibitor risk (relative risk [RR], 3.7; 95% confidence interval [CI], 2.0-7.1; and RR, 3.3; CI, 2.1-5.3, respectively). Regular prophylaxis was associated with a 60% lower risk than on-demand treatment (RR, 0.4; CI, 0.2-0.8). Our findings suggest that the previously reported associated between an early age at first exposure and the risk of inhibitor development is largely explained by early, intensive treatment. The latter appears to be an independent risk factor for inhibitor development. In addition, early, regular prophylaxis may protect patients with hemophilia against the development of inhibitors. CiteULike    Connotea    Del.icio.us    Digg    Reddit    Technorati    What's this? Related Article in Blood Online: Inhibitors in hemophilia: nature or nurture? Thomas Abshire Blood 2007 109: 4596. [Full Text] [PDF] This article has been cited by other articles: J. G. van der Bom, S. C. Gouw, and H. M. van den Berg Response: Plasma-derived or recombinant factor VIII products and inhibitors in previously untreated patients with severe hemophilia Blood, August 1, 2007; 110(3): 1074 - 1075. [Full Text] [PDF]

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