Induction of Immunotolerance in Hemophilia for High Titre Inhibitor Eradication: A Long-Term Follow-Up

G Mariani; S Solinas; D Pasqualetti; A Ghirardini; P Verani; S Buttó; M Lopez; T Moretti

doi:10.1055/s-0038-1651013

Thrombosis and Haemostasis, Table of Contents

Thromb Haemost 1989; 62(03): 835-839
DOI: 10.1055/s-0038-1651013

Original Article

Schattauer GmbH Stuttgart

Induction of Immunotolerance in Hemophilia for High Titre Inhibitor Eradication: A Long-Term Follow-Up

Authors

G Mariani

Dipartimento di Biopatologia Umana, Sezione di Ematologia Universita' di Roma “La Sapienza”, Roma, Italy
S Solinas

Dipartimento di Biopatologia Umana, Sezione di Ematologia Universita' di Roma “La Sapienza”, Roma, Italy
D Pasqualetti

Dipartimento di Biopatologia Umana, Sezione di Ematologia Universita' di Roma “La Sapienza”, Roma, Italy
A Ghirardini

Dipartimento di Biopatologia Umana, Sezione di Ematologia Universita' di Roma “La Sapienza”, Roma, Italy
P Verani

¹Laboratorio di Virologia, Instituto Superiore di Sanita' Roma, Italy
S Buttó

¹Laboratorio di Virologia, Instituto Superiore di Sanita' Roma, Italy
M Lopez

Dipartimento di Biopatologia Umana, Sezione di Ematologia Universita' di Roma “La Sapienza”, Roma, Italy
T Moretti

Dipartimento di Biopatologia Umana, Sezione di Ematologia Universita' di Roma “La Sapienza”, Roma, Italy

Abstract

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Summary

Three hemophiliacs with high titre inhibitor were treated with a medium-high FVIII dose schedule (100 IU/kg bw daily) with the aim of inducing the immunotolerance. These patients were followed-up extensively concerning their immunological status and HIV serology. In all of them the inhibitor disappeared and normal FVIII kinetics were obtained after 22, 15 and 29 months. After eradication of the inhibitor, no recurrence took place in any of the patients. All the patients were HIV Ab positive before the beginning of the treatment. In one of them CD4+ cells fell progessively 32 months after the treatment was started, a fullblown AIDS showed up, and the patient died 5½ years after the beginning of the treatment. In the second and third patient the CD4+ cells varied widely but remained >400/μl during the whole immunotolerance treatment. The latter two patients are AIDS and ARC free so far, but patient No. 2 developed a mild-to-severe thrombocytopenia.

Considering the high cost of the treatment and the possibility that such an intensive administration of FVIII concentrates might worsen the immunological status of patients, this therapeutic procedure should only be applied with caution.

Keywords

FVIII inhibitors - High responders - HIV-infection - AIDS - Immunotolerance

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References

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