Validation of Nijmegen-Bethesda assay modifications to allow inhibitor measurement during replacement therapy and facilitate inhibitor surveillance

J Thromb Haemost. 2012 Jun;10(6):1055-61. doi: 10.1111/j.1538-7836.2012.04705.x.

Abstract

Background: As part of a pilot U.S. inhibitor surveillance project initiated at the Centers for Disease Control and Prevention (CDC) in 2006, a centralized inhibitor measurement was instituted.

Objective: To validate a modified method for inhibitor measurement suitable for surveillance of treated and untreated patients.

Methods/results: In all, 710 subjects with hemophilia A were enrolled; 122 had a history of inhibitor (HI). Nijmegen-Bethesda assay (NBA) results on 50 split specimens shipped on cold packs and frozen were equivalent (r=0.998). Because 55% of 228 initial specimens had factor (F)VIII activity (VIII:C) present, a heat treatment step was added. Heating specimens to 56°C for 30 min and centrifuging removed FVIII, as demonstrated by a reduction of VIII:C and FVIII antigen to <1 U dL(-1) in recently treated patients. Among specimens inhibitor-negative before heating, one of 159 with negative HI and five of 30 with positive HI rose to ≥ 0.5 Nijmegen-Bethesda units (NBU) after heating. Correlation of heated and unheated inhibitor-positive specimens was 0.94 (P=0.0001). The modified method had a coefficient of variation (CV) for a 1 NBU positive control of 10.3% and for the negative control of 9.8%. Based on results on 710 enrollment specimens, a positive CDC inhibitor was defined as ≥ 0.5 NBU. Results were similar when 643 post-enrollment specimens were included. Of 160 enrolled hemophilia B patients, two had HI. All others had NBU ≤ 0.2 at enrollment.

Conclusion: The CDC experience demonstrates that this modified NBA can be standardized to be within acceptable limits for clinical tests and can be used for national surveillance.

Publication types

  • Multicenter Study
  • Research Support, U.S. Gov't, P.H.S.
  • Validation Study

MeSH terms

  • Antibodies / blood*
  • Antigen-Antibody Reactions
  • Artifacts
  • Blood Coagulation Tests* / standards
  • Calibration
  • Coagulants / immunology*
  • Coagulants / therapeutic use*
  • Drug Monitoring / methods*
  • Drug Monitoring / standards
  • Enzyme-Linked Immunosorbent Assay
  • Factor VIII / immunology*
  • Factor VIII / therapeutic use*
  • Hemophilia A / blood
  • Hemophilia A / diagnosis
  • Hemophilia A / drug therapy*
  • Hemophilia A / immunology
  • Hemophilia B / blood
  • Hemophilia B / diagnosis
  • Hemophilia B / drug therapy
  • Hemophilia B / immunology
  • Hot Temperature
  • Humans
  • Predictive Value of Tests
  • Prospective Studies
  • Protein Denaturation
  • Reference Standards
  • Reproducibility of Results
  • Severity of Illness Index
  • Time Factors
  • Treatment Outcome
  • United States

Substances

  • Antibodies
  • Coagulants
  • Factor VIII