Post-SARS-CoV-2 Atypical Inflammatory Syndrome in a Toddler with X-Linked Inhibitor of Apoptosis Deficiency After Stem Cell Transplant

J Clin Immunol. 2022 Nov;42(8):1600-1603. doi: 10.1007/s10875-022-01316-3. Epub 2022 Jul 11.

Abstract

A 10-month-old boy was diagnosed with X-linked lymphoproliferative syndrome type 2 due to X-linked inhibitor of apoptosis deficiency after presenting with failure to thrive and refractory inflammatory bowel disease. He underwent a matched unrelated donor stem cell transplant with reduced intensity conditioning at 16 months. At 27 months, he presented with an atypical inflammatory syndrome in the setting of recent COVID-19 infection, Epstein-Barr viremia, and low chimerism (7.3%). He recovered after treatment with intravenous immunoglobulin and steroids.

Keywords: Atypical inflammatory syndrome; COVID-19; SARS-CoV-2; X-linked inhibitor of apoptosis deficiency (XIAP); X-linked lymphoproliferative (XLP); bone marrow transplant; multisystem inflammatory syndrome in children (MIS-C); primary immunodeficiency; stem cell transplant.

Publication types

  • Letter

MeSH terms

  • Apoptosis
  • COVID-19* / diagnosis
  • Child, Preschool
  • Hematopoietic Stem Cell Transplantation* / adverse effects
  • Humans
  • Infant
  • Lymphoproliferative Disorders* / diagnosis
  • Male
  • SARS-CoV-2
  • X-Linked Inhibitor of Apoptosis Protein

Substances

  • X-Linked Inhibitor of Apoptosis Protein

Supplementary concepts

  • Lymphoproliferative Syndrome, X-Linked, 2