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Little discrepancy between one-stage and chromogenic factor VIII (FVIII)/IX assays in a large international cohort of persons with nonsevere hemophilia A and B.
Zwagemaker, Anne-Fleur; Kloosterman, Fabienne R; Gouw, Samantha C; Boyce, Sara; Brons, Paul; Cnossen, Marjon H; Collins, Peter W; Eikenboom, Jeroen; Hay, Charles; Hengeveld, Rutger C C; Jackson, Shannon; Klopper-Tol, Caroline A M; Kruip, Marieke J H A; Gorkom, Britta Laros-van; Male, Christoph; Nieuwenhuizen, Laurens; Shapiro, Susan; Fijnvandraat, Karin; Coppens, Michiel.
Afiliación
  • Zwagemaker AF; Amsterdam UMC, University of Amsterdam, Emma Children's Hospital, Pediatric Hematology, Meibergdreef, Amsterdam, The Netherlands.
  • Kloosterman FR; Amsterdam UMC, University of Amsterdam, Emma Children's Hospital, Pediatric Hematology, Meibergdreef, Amsterdam, The Netherlands.
  • Gouw SC; Amsterdam UMC, University of Amsterdam, Emma Children's Hospital, Pediatric Hematology, Meibergdreef, Amsterdam, The Netherlands.
  • Boyce S; Department of Haematology, University Hospital Southampton, Southampton, United Kingdom.
  • Brons P; Department of Pediatric Hemato-Oncology, Radboud University Medical Center, Nijmegen, The Netherlands.
  • Cnossen MH; Department of Pediatric Hematology, Erasmus MC Sophia Children's Hospital, Erasmus University Medical Center, Rotterdam, The Netherlands.
  • Collins PW; Cardiff Haemophilia Centre, School of Medicine, Cardiff University, Cardiff, United Kingdom.
  • Eikenboom J; Department of Internal Medicine, Division of Thrombosis and Hemostasis, Leiden University Medical Center, Leiden, The Netherlands.
  • Hay C; Manchester University Department of Haematology, Manchester Royal Infirmary, Manchester, United Kingdom.
  • Hengeveld RCC; Amsterdam UMC, University of Amsterdam, Clinical Chemistry, Meibergdreef, Amsterdam, The Netherlands.
  • Jackson S; Adult Bleeding Disorders Program of BC - Adult Division St. Paul's Hospital, Vancouver, British Columbia, Canada.
  • Klopper-Tol CAM; Amsterdam UMC, University of Amsterdam, Clinical Chemistry, Meibergdreef, Amsterdam, The Netherlands.
  • Kruip MJHA; Department of Hematology, Erasmus MC, Erasmus University Medical Center, Rotterdam, The Netherlands.
  • Gorkom BL; Department of Hematology, Radboud University Medical Center, Nijmegen, The Netherlands.
  • Male C; Department of Pediatrics, Medical University of Vienna, Vienna, Austria.
  • Nieuwenhuizen L; Department of Hematology, Maxima Medical Center, Veldhoven, The Netherlands.
  • Shapiro S; Department of Haematology, Oxford University Hospitals NHS Foundation, Oxford NIHR Biomedical Research Centre, Oxford, United Kingdom; Radcliffe Department of Medicine, Oxford University, Oxford, United Kingdom.
  • Fijnvandraat K; Amsterdam UMC, University of Amsterdam, Emma Children's Hospital, Pediatric Hematology, Meibergdreef, Amsterdam, The Netherlands; Department of Molecular Cellular Hemostasis, Sanquin Research and Landsteiner Laboratory, Amsterdam, The Netherlands.
  • Coppens M; Amsterdam UMC, University of Amsterdam, Vascular Medicine, Meibergdreef, Amsterdam, The Netherlands; Amsterdam Cardiovascular Sciences, Pulmonary Hypertension & Thrombosis, Amsterdam, The Netherlands. Electronic address: m.coppens@amsterdamumc.nl.
J Thromb Haemost ; 21(4): 850-861, 2023 04.
Article en En | MEDLINE | ID: mdl-36696222
BACKGROUND: Accurate measurements of coagulation factor activity form an essential part of hemophilia management and are performed by the one-stage or chromogenic assay. Current literature suggests that approximately one-third of persons with nonsevere hemophilia A exhibit assay discrepancy, albeit with a high variability between studies. Such data are scarce in nonsevere hemophilia B. OBJECTIVES: To investigate the extent of factor VIII/IX one-stage and chromogenic assay discrepancy in moderate and mild hemophilia A and B. METHODS: Persons with previously diagnosed nonsevere hemophilia A and B with a factor level of 2 to 35 IU/dL were included from the international DYNAMO cohort study. Central measurements of the factor VIII and IX activity levels were performed by the one-stage and chromogenic assay. Relative and absolute discrepancy definitions were used, with the International Society on Thrombosis and Haemostasis-Scientific and Standardization Committee proposed ratio of >2.0 or <0.5 being the primary outcome. Discrepancy was also evaluated in a subgroup of 13 persons with mutations previously associated with discrepancy (≥3 cases reported in literature). RESULTS: A total of 220 persons were included, of whom 3 (1%) showed assay discrepancy: 2/175 hemophilia A and 1/45 hemophilia B. Six persons (3%) exhibited an absolute difference >10 IU/dL between the assay results. In addition, with more lenient definitions, over 90% of participants (n = 197) had no discrepant results. Only 1 out of 13 persons with a mutation previously associated with discrepancy had significant assay discrepancy. CONCLUSION: Little assay discrepancy was observed despite the presence of mutations previously associated with discrepancy, suggesting that the presence and magnitude of assay discrepancy are largely determined by laboratory variables.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Hemostáticos / Hemofilia B / Hemofilia A Tipo de estudio: Diagnostic_studies / Etiology_studies / Incidence_studies / Observational_studies / Risk_factors_studies Límite: Humans Idioma: En Revista: J Thromb Haemost Asunto de la revista: HEMATOLOGIA Año: 2023 Tipo del documento: Article País de afiliación: Países Bajos Pais de publicación: Reino Unido

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Hemostáticos / Hemofilia B / Hemofilia A Tipo de estudio: Diagnostic_studies / Etiology_studies / Incidence_studies / Observational_studies / Risk_factors_studies Límite: Humans Idioma: En Revista: J Thromb Haemost Asunto de la revista: HEMATOLOGIA Año: 2023 Tipo del documento: Article País de afiliación: Países Bajos Pais de publicación: Reino Unido