Ca2+ signalling is critical for autoantibody-induced blistering of human
epidermis in pemphigus vulgaris
Abstract
Background: Pemphigus vulgaris (PV) is a severe autoimmune skin disease.
Autoantibodies in PV (PV-IgG) targeting desmoglein (DSG)1 are required
for loss of desmosome adhesion and skin blistering. Objectives: The aim
was to connect the single steps of a Ca2+ flux dependent pathway and
show it’s importance for the pathogenesis of pemphigus vulgaris.
Methods: Applied methods include immunoprecipitation, Ca2+-flux analysis
with FURA2-AM, analysis of protein phosphorylation with
Western-blotting, Immunofluorescence, investigation of loss of adhesion
in dissociation assays and human skin ex-vivo blistering model. Results:
PV-IgG but not a DSG3-specific monoclonal antibody (AK23) caused Ca2+
influx in primary human keratinocytes. Phosphatidyl inositol 4 kinase-α
(PI4K) interacts with DSG1 but not DSG3. Its downstream targets,
phospholipase-C-γ1 (PLC) and protein-kinase-C α, similar to p38MAPK
interacted with both. PLC was activated by PV-IgG but not by AK23. PLC
regulates the release of Inositol-1,4,5-trisphosphate (IP3) causing
IP3-receptor (IP3R) activation and Ca2+ flux from the endoplasmic
reticulum into the cytosol. This stimulates
Ca2+-release-activated-channels (CRAC) causing Ca2+ influx. Inhibitors
against PI4K, PLC, IP3R or CRAC effectively blocked PV-IgG-induced Ca2+
influx and ameliorated alterations of DSG1 and 3 localization, keratin
filament retraction, actin disruption and loss of cell adhesion in
vitro. Moreover, inhibiting PLC was protective against blister
formation, redistribution of DSG1 and 3 and actin disruption in human
skin ex vivo. Conclusions: These results demonstrate that Ca2+-mediated
signalling is important for pemphigus pathology and dependent on the
autoantibody profile, with different roles for the Dsg1 and Dsg3
complexes. PLC could be an interesting pharmaceutical target.