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Thrombin Generation Assay (TGA) as Predictive Test for Haemostatic. Effectiveness of FVIII Concentrates in Haemophiliac A With Inhibitors

Conditions
Severe Hemophilia A With Inhibitor
Registration Number
NCT01505946
Lead Sponsor
Grifols Italia S.p.A
Brief Summary

This is an observational, prospective, longitudinal, multicenter, cohort study designed with the scope to verify whether or not TGA may predict effectiveness of different FVIII concentrates class (devoid or rich of VWF) in patient affected by severe or moderately severe inherited haemophilia A and inhibitors.

Detailed Description

Rationale:

Hemophilia A is a serious and common hereditary bleeding disorder caused by deficiency of coagulation factor VIII (FVIII). Patients with this disease are treated with recombinant factor VIII or factor VIII concentrates derived from plasma.

Administration of exogenous FVIII in 15-35% of cases, cause the formation of antibodies to FVIII (inhibitors) that neutralize the activity of factor VIII, making the treatment ineffective.The development of inhibitors of factor VIII (FVIII) is the most serious and challenging complication of the treatment of hemophilia A and represents the highest economic burden for a chronic disease. Therefore, research is making great efforts to optimize the best therapeutic approach for the disease.

It has been observed that FVIII inhibitors display a wide range of immunoreactivity when tested against different classes of FVIII concentrates (with/without von Willebrand factor -VWF). It has been demonstrated that the different inhibitors reactivity may correlate with different ability of inhibitors to impair thrombin generation, as tested by Thrombin Generation Assay (TGA). In these patients TGA assay might be a tool to predict which FVIII concentrate has the greater haemostatic effectiveness.

It is also uncertain if the different classes of FVIII used in ITI protocols may have a different effectiveness in reducing the occurrence of BT bleedings and if this may correlate to lower reactivity, epitope specificity, VWF content and may be predicted by TGA. It would be very helpful to be able to give an evidence based diagnostic and prognostic instrument, the TGA, to aid physician to optimize the therapy for all inhibitors patients.

Recruitment & Eligibility

Status
UNKNOWN
Sex
Male
Target Recruitment
25
Inclusion Criteria
  • Diagnosis of inherited, severe (FVIII:C < 1%) or moderately severe haemophilia A (FVIII ≤ 2%)
  • Any age
  • Ability to comply with study methods and willingness to participate to the study
  • Written informed consent.

FOR THE LOW RESPONDERS COHORT

  • Documented low anamnestic response after FVIII exposure (FVIII inhibitors titre >0.6 and < 5 BU/ml tested by Bethesda assay, Nijmegen modification). It will be included in this study those patients who have never been submitted to ITI and also those patients who have completed ITI with partial success (defined as inhibitors titre >0.6 and < 5 BU/ml and no increase in the INH titer > 5 BU over treatment with FVIII)

INCLUSION CRITERIA FOR THE HIGH RESPONDERS COHORT

  • Documented high response after FVIII exposure (FVIII inhibitors titre > 5 BU/ml tested by Bethesda assay, Nijmegen modification). It will be included in this study those patients who are potential candidates to a first or rescue ITI.
  • Any historical peak ≥ 5 BU
Exclusion Criteria
  • Diagnosis of acquired haemophilia
  • Diagnosis of inherited mild haemophilia A (FVIII > 2%)
  • Life expectancy lower than 1 year
  • Psychiatric illness and any other conditions may impair ability to comply with study methods

Study & Design

Study Type
OBSERVATIONAL
Study Design
Not specified
Primary Outcome Measures
NameTimeMethod
Thrombin generation result12 months

Thrombin generation results of the TGA applied on plasma patients whith inhibitor matched with different class of FVIII concentrate

Secondary Outcome Measures
NameTimeMethod
Incidence of all breakthrough (BT) bleedings12 months

events/month

Total FVIII dose required to treat the patients12 months

(IU/year)

Use of bypassing agents12 months

incidence of BT bleedings who require bypassing agents (events/months) average dose of bypassing agents (or days of treatment) needed to treat BT bleedings total dose of bypassing agent (and days of treatment) required overall (IU/year) and (days of treatment/year)

ITI outcome only for patient under this kind of treatment3 years

% of Success (total, partial success or failure will be defined as in ITI study protocol) Time to tolerance (months), defined as the time to ITI success (total/partial)

Epitope mapping results12 months

epitope mapping test on the plasma patient during the therapy for a follow-up period of 12 month

the inhibitor titre course12 months

Trial Locations

Locations (15)

Ospedale Civile dell' Annunziata

🇮🇹

Cosenza, Calabria, Italy

Az. Universitaria Policlinico "Federico II" Dip. Assist. di Clinica Medica

🇮🇹

Napoli, Campania, Italy

UO Angiologia e Malattie della Coagulazione "Marino Golinelli" Az Osp. Policlinico S. Orsola Malpighi

🇮🇹

Bologna, Emilia Romagna, Italy

Azienda Ospedaliera "Santa Maria della Misericordia"

🇮🇹

Udine, Friuli Venezia Giulia, Italy

Ematologia Dipartimento di Biotecnologie Cellulari Università La Sapienza - Policlinico Umberto I

🇮🇹

Rome, Lazio, Italy

Ospedale Pediatrico Bambino Gesù di Roma

🇮🇹

Rome, Lazio, Italy

Università Cattolica - Policlinico A. Gemelli

🇮🇹

Rome, Lazio, Italy

Azienda Ospedialiera Ospedale Infantile Regina Margherita - S.Anna

🇮🇹

Turin, Piemonte, Italy

Ospedale Le Molinette "S. G. Battista"

🇮🇹

Turin, Piemonte, Italy

Azienda Ospedaliero Universitaria Consorziale Policlinico di Bari

🇮🇹

BAri, Puglia, Italy

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Ospedale Civile dell' Annunziata
🇮🇹Cosenza, Calabria, Italy
Filomena Daniele, MD
Principal Investigator

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