TY - JOUR
T1 - Case report
T2 - Persistent shedding of a live vaccine-derived rubella virus in a young man with severe combined immunodeficiency and cutaneous granuloma
AU - Bonner, Kimberly E.
AU - Sukerman, Ellie
AU - Liko, Juventila
AU - Lanzieri, Tatiana M.
AU - Sutton, Melissa
AU - DeBess, Emilio
AU - Leesman, Christopher
AU - Icenogle, Joseph
AU - Hao, Li Juan
AU - Chen, Min Hsin
AU - Faisthalab, Raeesa
AU - Leman, Richard F.
AU - Cieslak, Paul R.
AU - DeRavin, Suk See
AU - Perelygina, Ludmila
N1 - Publisher Copyright:
Copyright © 2022 Bonner, Sukerman, Liko, Lanzieri, Sutton, DeBess, Leesman, Icenogle, Hao, Chen, Faisthalab, Leman, Cieslak, DeRavin and Perelygina.
PY - 2022/12/8
Y1 - 2022/12/8
N2 - A young man with X-linked severe combined immunodeficiency developed a persistent vaccine-derived rubella virus (VDRV) infection, with the emergence of cutaneous granulomas more than fifteen years after receipt of two doses of measles-mumps-rubella (MMR) vaccine. Following nasopharyngeal swab (NP) collection, VDRV was detected by real-time polymerase chain reaction (RT-qPCR) and sequencing, and live, replication-competent VDRV was isolated in cell culture. To assess duration and intensity of viral shedding, sequential respiratory samples, one cerebrospinal fluid sample, and two urine samples were collected over 15 months, and VDRV RNA was detected in all samples by RT-qPCR. Live VDRV was cultured from nine of the eleven respiratory specimens and from one urine specimen. To our knowledge, this was the first reported instance of VDRV cultured from respiratory specimens or from urine. To assess potential transmission to close contacts, NP specimens and sera were collected from all household contacts, all of whom were immunocompetent and previously vaccinated with MMR. VDRV RNA was not detected in any NP swabs from the contacts, nor did serologic investigations suggest VDRV transmission to any contacts. This report highlights the need to understand the prevalence and duration of VDRV shedding in granuloma patients and to estimate the risk of VDRV transmission to immune and non-immune contacts.
AB - A young man with X-linked severe combined immunodeficiency developed a persistent vaccine-derived rubella virus (VDRV) infection, with the emergence of cutaneous granulomas more than fifteen years after receipt of two doses of measles-mumps-rubella (MMR) vaccine. Following nasopharyngeal swab (NP) collection, VDRV was detected by real-time polymerase chain reaction (RT-qPCR) and sequencing, and live, replication-competent VDRV was isolated in cell culture. To assess duration and intensity of viral shedding, sequential respiratory samples, one cerebrospinal fluid sample, and two urine samples were collected over 15 months, and VDRV RNA was detected in all samples by RT-qPCR. Live VDRV was cultured from nine of the eleven respiratory specimens and from one urine specimen. To our knowledge, this was the first reported instance of VDRV cultured from respiratory specimens or from urine. To assess potential transmission to close contacts, NP specimens and sera were collected from all household contacts, all of whom were immunocompetent and previously vaccinated with MMR. VDRV RNA was not detected in any NP swabs from the contacts, nor did serologic investigations suggest VDRV transmission to any contacts. This report highlights the need to understand the prevalence and duration of VDRV shedding in granuloma patients and to estimate the risk of VDRV transmission to immune and non-immune contacts.
KW - gene therapy
KW - inborn errors of immunity
KW - live virus shedding
KW - skin granuloma
KW - vaccine-derived rubella virus
KW - viral persistence
UR - http://www.scopus.com/inward/record.url?scp=85144575908&partnerID=8YFLogxK
UR - http://www.scopus.com/inward/citedby.url?scp=85144575908&partnerID=8YFLogxK
U2 - 10.3389/fimmu.2022.1075351
DO - 10.3389/fimmu.2022.1075351
M3 - Article
C2 - 36569925
AN - SCOPUS:85144575908
SN - 1664-3224
VL - 13
JO - Frontiers in immunology
JF - Frontiers in immunology
M1 - 1075351
ER -