Case Report: Rubella Virus-Induced Cutaneous Granulomas in Two Pediatric Patients With DNA Double Strand Breakage Repair Disorders – Outcome After Hematopoietic Stem Cell Transplantation

We report two patients with DNA repair disorders (Artemis deficiency, Ataxia telangiectasia) with destructive skin granulomas, presumably triggered by live-attenuated rubella vaccinations. Both patients showed reduced naïve T cells. Rapid resolution of skin lesions was observed following hematopoiet...

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Veröffentlicht in:Frontiers in immunology 2022-06, Vol.13
Hauptverfasser: Baumann, Ulrich, Schulte, Johannes H., Groß, Jonathan P., Beier, Rita, Ludwig, Marius, Wahn, Volker, Hofmann, Jörg, Maecker-Kolhoff, Britta, Sauer, Martin, Kaiser-Labusch, Petra, Karimian, Negin, Blume-Peytavi, Ulrike, Ghoreschi, Franziska, Ott, Hagen, Perelygina, Ludmila, Klemann, Christian, Blankenstein, Oliver, von Bernuth, Horst, Krüger, Renate
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Sprache:eng
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Zusammenfassung:We report two patients with DNA repair disorders (Artemis deficiency, Ataxia telangiectasia) with destructive skin granulomas, presumably triggered by live-attenuated rubella vaccinations. Both patients showed reduced naïve T cells. Rapid resolution of skin lesions was observed following hematopoietic stem cell transplantation. However, the patient with AT died due to complications of severe hepatic veno-occlusive disease 6 month after HSCT. Dried blood spots obtained after birth were available from this patient and showed absent T-cell receptor excision circles (TRECs). Therefore, newborn screening may help to prevent patients with moderate T-cell deficiency from receiving live-attenuated rubella vaccine potentially causing granulomas.
ISSN:1664-3224
1664-3224
DOI:10.3389/fimmu.2022.886540