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Treatment of Disseminated Mycobacterial Infection with High-Dose IFN-γ in a Patient with IL-12Rβ1 Deficiency
IFN-γ has been used in the treatment of IL-12Rβ1 deficiency patients with disseminated BCG infection (BCGosis), but the optimal dose to reach efficacy is not clear. We used IFN-γ in the treatment of a 2.7-year-old patient with IL-12Rβ1 deficiency and refractory BCG-osis. IFNγ was started at a dose o...
Autores principales: | , , , , , , , |
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Formato: | Texto |
Lenguaje: | English |
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Hindawi Publishing Corporation
2011
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Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3014684/ https://www.ncbi.nlm.nih.gov/pubmed/21234109 http://dx.doi.org/10.1155/2011/691956 |
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author | Alangari, Abdullah A. Al-Zamil, Fahad Al-Mazrou, Abdulrahman Al-Muhsen, Saleh Boisson-Dupuis, Stéphanie Awadallah, Sitalbanat Kambal, Abdelmageed Casanova, Jean-Laurent |
author_facet | Alangari, Abdullah A. Al-Zamil, Fahad Al-Mazrou, Abdulrahman Al-Muhsen, Saleh Boisson-Dupuis, Stéphanie Awadallah, Sitalbanat Kambal, Abdelmageed Casanova, Jean-Laurent |
author_sort | Alangari, Abdullah A. |
collection | PubMed |
description | IFN-γ has been used in the treatment of IL-12Rβ1 deficiency patients with disseminated BCG infection (BCGosis), but the optimal dose to reach efficacy is not clear. We used IFN-γ in the treatment of a 2.7-year-old patient with IL-12Rβ1 deficiency and refractory BCG-osis. IFNγ was started at a dose of 50 μg/m(2) 3 times per week. The dose was upgraded to 100 mcg/m(2) after 3 months, then to 200 mcg/m(2) 6 months afterwards. Serum mycobactericidal activity and lymphocytes number and function were evaluated throughout the study. There was no clinical response to IFN-γ with 50 or 100 μg/m(2) doses. However, there was some response to the 200 μg/m(2) 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-12Rβ1 deficiency, IFN-γ at a dose of 200 μg/m(2), but not at lower dosages, was found to have a noticeable clinical effect with no additional adverse effects. |
format | Text |
id | pubmed-3014684 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2011 |
publisher | Hindawi Publishing Corporation |
record_format | MEDLINE/PubMed |
spelling | pubmed-30146842011-01-13 Treatment of Disseminated Mycobacterial Infection with High-Dose IFN-γ in a Patient with IL-12Rβ1 Deficiency Alangari, Abdullah A. Al-Zamil, Fahad Al-Mazrou, Abdulrahman Al-Muhsen, Saleh Boisson-Dupuis, Stéphanie Awadallah, Sitalbanat Kambal, Abdelmageed Casanova, Jean-Laurent Clin Dev Immunol Research Article IFN-γ has been used in the treatment of IL-12Rβ1 deficiency patients with disseminated BCG infection (BCGosis), but the optimal dose to reach efficacy is not clear. We used IFN-γ in the treatment of a 2.7-year-old patient with IL-12Rβ1 deficiency and refractory BCG-osis. IFNγ was started at a dose of 50 μg/m(2) 3 times per week. The dose was upgraded to 100 mcg/m(2) after 3 months, then to 200 mcg/m(2) 6 months afterwards. Serum mycobactericidal activity and lymphocytes number and function were evaluated throughout the study. There was no clinical response to IFN-γ with 50 or 100 μg/m(2) doses. However, there was some response to the 200 μg/m(2) 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-12Rβ1 deficiency, IFN-γ at a dose of 200 μg/m(2), but not at lower dosages, was found to have a noticeable clinical effect with no additional adverse effects. Hindawi Publishing Corporation 2011 2010-12-22 /pmc/articles/PMC3014684/ /pubmed/21234109 http://dx.doi.org/10.1155/2011/691956 Text en Copyright © 2011 Abdullah A. Alangari et al. https://creativecommons.org/licenses/by/3.0/ This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. |
spellingShingle | Research Article Alangari, Abdullah A. Al-Zamil, Fahad Al-Mazrou, Abdulrahman Al-Muhsen, Saleh Boisson-Dupuis, Stéphanie Awadallah, Sitalbanat Kambal, Abdelmageed Casanova, Jean-Laurent Treatment of Disseminated Mycobacterial Infection with High-Dose IFN-γ in a Patient with IL-12Rβ1 Deficiency |
title | Treatment of Disseminated Mycobacterial Infection with High-Dose IFN-γ in a Patient with IL-12Rβ1 Deficiency |
title_full | Treatment of Disseminated Mycobacterial Infection with High-Dose IFN-γ in a Patient with IL-12Rβ1 Deficiency |
title_fullStr | Treatment of Disseminated Mycobacterial Infection with High-Dose IFN-γ in a Patient with IL-12Rβ1 Deficiency |
title_full_unstemmed | Treatment of Disseminated Mycobacterial Infection with High-Dose IFN-γ in a Patient with IL-12Rβ1 Deficiency |
title_short | Treatment of Disseminated Mycobacterial Infection with High-Dose IFN-γ in a Patient with IL-12Rβ1 Deficiency |
title_sort | treatment of disseminated mycobacterial infection with high-dose ifn-γ in a patient with il-12rβ1 deficiency |
topic | Research Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3014684/ https://www.ncbi.nlm.nih.gov/pubmed/21234109 http://dx.doi.org/10.1155/2011/691956 |
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