| Literature DB >> 33424831 |
Shelly Pathak1, Dorota Rowczenio2, Samuel Lara-Reyna1, Mark Kacar1, Roger Owen3, Gina Doody4, Karoline Krause5, Helen Lachmann2, Rainer Doffinger6, Darren Newton4, Sinisa Savic1.
Abstract
The Schnitzler Syndrome (SchS) is an acquired, autoinflammatory condition successfully treated with IL-1 inhibition. The two main defining features of this late-onset condition are neutrophilic urticarial dermatoses (NUD) and the presence of an IgM monoclonal component. While the former aspect has been extensively studied in this disease setting, the enigmatic paraproteinaemia and its potential consequential effects within SchS, has not previously been thoroughly addressed. Previous studies analyzing clonal B cell repertoires have largely focused on autoimmune disorders such as Systemic Lupus Erythematous (SLE) and hematological malignancies such as Chronic Lymphocytic Leukaemia (CLL), where B-cell clonality is central to disease pathology. The present study uses next-generation sequencing to provide detailed insight into aspects of B cell VDJ recombination and properties of the resulting immunoglobulin chains. An overview of IgH regional dynamics in 10 SchS patients, with a particular focus on CDR3 sequences and VDJ gene usage is reported, highlighting the presence of specific B cell expansions. Protein microarray detected a substantial proportion of autoreactive IgM to nuclear target proteins, though a single universal target was not identified. Together, these genetic and functional findings impart new understanding into this rare disorder.Entities:
Keywords: B cell repertoire; IgM; Schnitzler Syndrome; autoinflammatory diseases; paraprotein
Year: 2020 PMID: 33424831 PMCID: PMC7793813 DOI: 10.3389/fimmu.2020.569006
Source DB: PubMed Journal: Front Immunol ISSN: 1664-3224 Impact factor: 7.561