SARS-CoV-2 infection associated with hepatitis in an infant with X-linked severe combined immunodeficiency
X-linked severe combined immunodeficiency (X-SCID) is a disorder of adaptive immunity caused by mutations in the IL-2 receptor common gamma chain gene resulting in deficiencies of T and natural killer cells, coupled with severe dysfunction in B cells. X-SCID is lethal without allogeneic stem cell tr...
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| Published in: | Clinical immunology (Orlando, Fla.) Vol. 224; p. 108662 |
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| Main Authors: | , , , , , , |
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01-03-2021
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| Abstract | X-linked severe combined immunodeficiency (X-SCID) is a disorder of adaptive immunity caused by mutations in the IL-2 receptor common gamma chain gene resulting in deficiencies of T and natural killer cells, coupled with severe dysfunction in B cells. X-SCID is lethal without allogeneic stem cell transplant or gene therapy due to opportunistic infections. An infant with X-SCID became infected with SARS-CoV-2 while awaiting transplant. The patient developed severe hepatitis without the respiratory symptoms typical of COVID-19. He was treated with convalescent plasma, and thereafter was confirmed to have SARS-CoV-2 specific antibodies, as detected with a microfluidic antigen array. After resolution of the hepatitis, he received a haploidentical CD34 selected stem cell transplant, without conditioning, from his father who had recovered from COVID-19. SARS CoV-2 was detected via RT-PCR on nasopharyngeal swabs until 61 days post transplantation. He successfully engrafted donor T and NK cells, and continues to do well clinically.
•The presence of uncorrected X-linked SCID in an infant who became infected with SARS-CoV-2 lead to a mild COVID-19 disease course.•COVID-19 disease in this X-SCID infant was characterized by hepatitis, and prolonged viral shedding.•The prolonged period of viral shedding continued post-transplant, with viral clearance correlating with donor T and NK cell engraftment. |
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| AbstractList | X-linked severe combined immunodeficiency (X-SCID) is a disorder of adaptive immunity caused by mutations in the IL-2 receptor common gamma chain gene resulting in deficiencies of T and natural killer cells, coupled with severe dysfunction in B cells. X-SCID is lethal without allogeneic stem cell transplant or gene therapy due to opportunistic infections. An infant with X-SCID became infected with SARS-CoV-2 while awaiting transplant. The patient developed severe hepatitis without the respiratory symptoms typical of COVID-19. He was treated with convalescent plasma, and thereafter was confirmed to have SARS-CoV-2 specific antibodies, as detected with a microfluidic antigen array. After resolution of the hepatitis, he received a haploidentical CD34 selected stem cell transplant, without conditioning, from his father who had recovered from COVID-19. SARS CoV-2 was detected
via
RT-PCR on nasopharyngeal swabs until 61 days post transplantation. He successfully engrafted donor T and NK cells, and continues to do well clinically. X-linked severe combined immunodeficiency (X-SCID) is a disorder of adaptive immunity caused by mutations in the IL-2 receptor common gamma chain gene resulting in deficiencies of T and natural killer cells, coupled with severe dysfunction in B cells. X-SCID is lethal without allogeneic stem cell transplant or gene therapy due to opportunistic infections. An infant with X-SCID became infected with SARS-CoV-2 while awaiting transplant. The patient developed severe hepatitis without the respiratory symptoms typical of COVID-19. He was treated with convalescent plasma, and thereafter was confirmed to have SARS-CoV-2 specific antibodies, as detected with a microfluidic antigen array. After resolution of the hepatitis, he received a haploidentical CD34 selected stem cell transplant, without conditioning, from his father who had recovered from COVID-19. SARS CoV-2 was detected via RT-PCR on nasopharyngeal swabs until 61 days post transplantation. He successfully engrafted donor T and NK cells, and continues to do well clinically. X-linked severe combined immunodeficiency (X-SCID) is a disorder of adaptive immunity caused by mutations in the IL-2 receptor common gamma chain gene resulting in deficiencies of T and natural killer cells, coupled with severe dysfunction in B cells. X-SCID is lethal without allogeneic stem cell transplant or gene therapy due to opportunistic infections. An infant with X-SCID became infected with SARS-CoV-2 while awaiting transplant. The patient developed severe hepatitis without the respiratory symptoms typical of COVID-19. He was treated with convalescent plasma, and thereafter was confirmed to have SARS-CoV-2 specific antibodies, as detected with a microfluidic antigen array. After resolution of the hepatitis, he received a haploidentical CD34 selected stem cell transplant, without conditioning, from his father who had recovered from COVID-19. SARS CoV-2 was detected via RT-PCR on nasopharyngeal swabs until 61 days post transplantation. He successfully engrafted donor T and NK cells, and continues to do well clinically. •The presence of uncorrected X-linked SCID in an infant who became infected with SARS-CoV-2 lead to a mild COVID-19 disease course.•COVID-19 disease in this X-SCID infant was characterized by hepatitis, and prolonged viral shedding.•The prolonged period of viral shedding continued post-transplant, with viral clearance correlating with donor T and NK cell engraftment. |
| ArticleNumber | 108662 |
| Author | Aquino, Victor Schoggins, John W. Wysocki, Christian A. Li, Quan-Zhen van Oers, Nicolai S.C. Hanners, Natasha W. Sue, Paul K. |
| Author_xml | – sequence: 1 givenname: Nicolai S.C. surname: van Oers fullname: van Oers, Nicolai S.C. organization: Department of Immunology, The University of Texas Southwestern Medical Center, 6000 Harry Hines Boulevard, Dallas, TX 75390-9093, United States of America – sequence: 2 givenname: Natasha W. surname: Hanners fullname: Hanners, Natasha W. organization: Department of Pediatrics, The University of Texas Southwestern Medical Center, 6000 Harry Hines Boulevard, Dallas, TX 75390-9093, United States of America – sequence: 3 givenname: Paul K. surname: Sue fullname: Sue, Paul K. organization: Children's Health, 1935 Medical District Drive, Dallas, TX 75235, United States of America – sequence: 4 givenname: Victor surname: Aquino fullname: Aquino, Victor organization: Children's Health, 1935 Medical District Drive, Dallas, TX 75235, United States of America – sequence: 5 givenname: Quan-Zhen surname: Li fullname: Li, Quan-Zhen organization: Department of Immunology, The University of Texas Southwestern Medical Center, 6000 Harry Hines Boulevard, Dallas, TX 75390-9093, United States of America – sequence: 6 givenname: John W. surname: Schoggins fullname: Schoggins, John W. organization: Department of Microbiology, The University of Texas Southwestern Medical Center, 6000 Harry Hines Boulevard, Dallas, TX 75390-9093, United States of America – sequence: 7 givenname: Christian A. surname: Wysocki fullname: Wysocki, Christian A. email: Christian.Wysocki@UTSouthwestern.edu organization: Department of Pediatrics, The University of Texas Southwestern Medical Center, 6000 Harry Hines Boulevard, Dallas, TX 75390-9093, United States of America |
| BackLink | https://www.ncbi.nlm.nih.gov/pubmed/33412294$$D View this record in MEDLINE/PubMed |
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| CitedBy_id | crossref_primary_10_1007_s10875_021_01093_5 crossref_primary_10_1016_j_clim_2022_109097 crossref_primary_10_1152_physiol_00016_2022 crossref_primary_10_3389_fimmu_2021_721738 crossref_primary_10_1161_CIRCULATIONAHA_121_056038 crossref_primary_10_1007_s10875_021_01063_x crossref_primary_10_1016_j_jaci_2022_11_010 crossref_primary_10_3389_fimmu_2022_1020165 crossref_primary_10_1186_s12879_022_07219_3 crossref_primary_10_1016_j_mayocp_2021_02_008 crossref_primary_10_1186_s40001_022_00824_7 crossref_primary_10_1016_j_jaci_2023_02_003 crossref_primary_10_1001_jamanetworkopen_2022_50647 crossref_primary_10_1080_14760584_2021_1932475 crossref_primary_10_1186_s13223_023_00831_1 crossref_primary_10_1097_ACI_0000000000000786 crossref_primary_10_1097_MOP_0000000000001062 crossref_primary_10_1007_s10875_021_00993_w crossref_primary_10_1097_INF_0000000000003938 |
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| Copyright | 2020 Elsevier Inc. Copyright © 2020 Elsevier Inc. All rights reserved. 2020 Elsevier Inc. All rights reserved. 2020 Elsevier Inc. |
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| Keywords | COVID-19 SARS-CoV-2 Adaptive immunity Severe combined immunodeficiency Inborn errors of immunity |
| Language | English |
| License | Copyright © 2020 Elsevier Inc. All rights reserved. Since January 2020 Elsevier has created a COVID-19 resource centre with free information in English and Mandarin on the novel coronavirus COVID-19. The COVID-19 resource centre is hosted on Elsevier Connect, the company's public news and information website. Elsevier hereby grants permission to make all its COVID-19-related research that is available on the COVID-19 resource centre - including this research content - immediately available in PubMed Central and other publicly funded repositories, such as the WHO COVID database with rights for unrestricted research re-use and analyses in any form or by any means with acknowledgement of the original source. These permissions are granted for free by Elsevier for as long as the COVID-19 resource centre remains active. |
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| SubjectTerms | Adaptive immunity COVID-19 COVID-19 - complications COVID-19 - therapy COVID-19 Serotherapy Hepatitis - virology Humans Immunization, Passive - methods Inborn errors of immunity Infant Male SARS-CoV-2 Severe combined immunodeficiency Severe Combined Immunodeficiency - complications |
| Title | SARS-CoV-2 infection associated with hepatitis in an infant with X-linked severe combined immunodeficiency |
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