| Title: |
Inherited IFNAR1 deficiency in otherwise healthy patients with adverse reaction to measles and yellow fever live vaccines |
| Authors: |
Hernandez, N; Bucciol, G; Moens, L; Le Pen, J; Shahrooei, M; Goudouris, E; Shirkani, A; Changi-Ashtiani, M; Rokni-Zadeh, H; Sayar, EH; Reisli, I; Lefevre-Utile, A; Zijlmans, D; Jurado, A; Pholien, R; Drutman, S; Belkaya, S; Cobat, A; Boudewijns, R; Jochmans, D; Neyts, J; Seeleuthner, Y; Lorenzo-Diaz, L; Enemchukwu, C; Tietjen, I; Hoffmann, HH; Momenilandi, M; Pöyhönen, L; Siqueira, MM; De Lima, SMB; De Souza Matos, DC; Homma, A; De Maia, MLS; Da Costa Barros, TA; De Oliveira, PMN; Mesquita, EC; Gijsbers, R; Zhang, SY; Seligman, SJ; Abel, L; Hertzog, P; Marr, N; De Menezes Martins, R; Meyts, I; Zhang, Q; MacDonald, MR; Rice, CM; Casanova, JL; Jouanguy, E; Bossuyt, X |
| Publisher Information: |
Rockefeller University Press |
| Publication Year: |
2019 |
| Collection: |
The University of Melbourne: Digital Repository |
| Description: |
Vaccination against measles, mumps, and rubella (MMR) and yellow fever (YF) with live attenuated viruses can rarely cause life-threatening disease. Severe illness by MMR vaccines can be caused by inborn errors of type I and/or III interferon (IFN) immunity (mutations in IFNAR2, STAT1, or STAT2). Adverse reactions to the YF vaccine have remained unexplained. We report two otherwise healthy patients, a 9-yr-old boy in Iran with severe measles vaccine disease at 1 yr and a 14-yr-old girl in Brazil with viscerotropic disease caused by the YF vaccine at 12 yr. The Iranian patient is homozygous and the Brazilian patient compound heterozygous for loss-of-function IFNAR1 variations. Patient-derived fibroblasts are susceptible to viruses, including the YF and measles virus vaccine strains, in the absence or presence of exogenous type I IFN. The patients' fibroblast phenotypes are rescued with WT IFNAR1 Autosomal recessive, complete IFNAR1 deficiency can result in life-threatening complications of vaccination with live attenuated measles and YF viruses in previously healthy individuals. |
| Document Type: |
article in journal/newspaper |
| Language: |
English |
| ISSN: |
0022-1007 |
| Relation: |
pii: jem.20182295; https://hdl.handle.net/11343/272010 |
| Availability: |
https://hdl.handle.net/11343/272010 |
| Rights: |
https://creativecommons.org/licenses/by-nc-sa/4.0 ; CC BY-NC-SA |
| Accession Number: |
edsbas.6F522293 |
| Database: |
BASE |