Treatment of disseminated mycobacterial infection with high-dose IFN-? in a patient with IL-12R?1 deficiency.

ABSTRACT: IFN-? has been used in the treatment of IL-12R?1 deficiency patients with disseminated BCG infection (BCGosis), but the optimal dose to reach efficacy is not clear. We used IFN-? in the treatment of a 2.7-year-old patient with IL-12R?1 deficiency and refractory BCG-osis. IFN? was started at a dose of 50 ?g/m² 3 times per week. The dose was upgraded to 100 mcg/m² after 3 months, then to 200 mcg/m² 6 months afterwards. Serum mycobactericidal activity and lymphocytes number and function were evaluated throughout the study. There was no clinical response to IFN-? with 50 or 100 ?g/m² doses. However, there was some response to the 200 ?g/m² dose with no additional adverse effects. The serum mycobactericidal activity was not significantly different during the whole treatment period. Lymphocytes proliferation in response to PHA was significantly higher after 3 months of using the highest dose as compared to the lowest dose. The tuberculin skin test reaction remained persistently negative. We conclude that in a patient with IL-12R?1 deficiency, IFN-? at a dose of 200 ?g/m², but not at lower dosages, was found to have a noticeable clinical effect with no additional adverse effects.

SUBMITTER: Alangari AA 

PROVIDER: S-EPMC3014684 | biostudies-literature | 2011

REPOSITORIES: biostudies-literature