Yet, it would be important to further validate our results in additional ROSAH patients to understand if I ALPK i is involved in the regulation of B cell homeostasis or whether the observed defects in B cells were caused pharmacologically by one of the multiple pre-treatments of our patient (Table S3). Peripheral blood mononuclear cells (PBMCs) of the ROSAH patient and HDs were ex vivo stimulated with phorbol 12-myristate 13-acetate (PMA)/ionomycin (Iono) or lipopolysaccharide (LPS) for 4 h followed by fixation and mass cytometry staining and acquisition. However, the production of pro-inflammatory cytokines of ex vivo activated T cells obtained from patients treated with tofacitinib is only slightly reduced [[4]], arguing that the pronounced reduction of IFN and TNF expression in T cells of the ROSAH patient might not be caused by tofacitinib treatment alone. Accordingly, a literature review of Jamilloux et al. of 21 reported patients with I APLK1 i -associated diseases [[1]] revealed a resolution of auto-inflammation-mediated symptoms in two out of four patients treated with TNF blockers. [Extracted from the article]