TY - JOUR
T1 - Inherited IFNAR1 deficiency in otherwise healthy patients with adverse reaction to measles and yellow fever live vaccines
AU - Hernandez, Nicholas
AU - Bucciol, Giorgia
AU - Moens, Leen
AU - Le Pen, Jérémie
AU - Shahrooei, Mohammad
AU - Goudouris, Ekaterini
AU - Shirkani, Afshin
AU - Changi-Ashtiani, Majid
AU - Rokni-Zadeh, Hassan
AU - Sayar, Esra Hazar
AU - Reisli, Ismail
AU - Lefevre-Utile, Alain
AU - Zijlmans, Dick
AU - Jurado, Andrea
AU - Pholien, Ruben
AU - Drutman, Scott
AU - Belkaya, Serkan
AU - Cobat, Aurelie
AU - Boudewijns, Robbert
AU - Jochmans, Dirk
AU - Neyts, Johan
AU - Seeleuthner, Yoann
AU - Lorenzo-Diaz, Lazaro
AU - Enemchukwu, Chibuzo
AU - Tietjen, Ian
AU - Hoffmann, Hans Heinrich
AU - Momenilandi, Mana
AU - Pöyhönen, Laura
AU - Siqueira, Marilda M.
AU - de Lima, Sheila M.Barbosa
AU - de Souza Matos, Denise C.
AU - Homma, Akira
AU - Maia, Maria de Lourdes S.
AU - da Costa Barros, Tamiris Azamor
AU - de Oliveira, Patricia Mouta Nunes
AU - Mesquita, Emersom Ciclini
AU - Gijsbers, Rik
AU - Zhang, Shen Ying
AU - Seligman, Stephen J.
AU - Abel, Laurent
AU - Hertzog, Paul
AU - Marr, Nico
AU - Martins, Reinaldo de Menezes
AU - Meyts, Isabelle
AU - Zhang, Qian
AU - MacDonald, Margaret R.
AU - Rice, Charles M.
AU - Casanova, Jean Laurent
AU - Jouanguy, Emmanuelle
AU - Bossuyt, Xavier
PY - 2019/9/2
Y1 - 2019/9/2
N2 - 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.
AB - 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.
UR - http://www.scopus.com/inward/record.url?scp=85071786268&partnerID=8YFLogxK
U2 - 10.1084/jem.20182295
DO - 10.1084/jem.20182295
M3 - Article
C2 - 31270247
AN - SCOPUS:85071786268
SN - 0022-1007
VL - 216
SP - 2057
EP - 2070
JO - Journal of Experimental Medicine
JF - Journal of Experimental Medicine
IS - 9
ER -
