Article ; Online: Characterization of Plasmacytoid Dendritic Cells, Microbial Sequences, and Identification of a Candidate Public T-Cell Clone in Kikuchi-Fujimoto Disease.
2021 Volume 24, Issue 3, Page(s) 193–205
Abstract: Objectives: Kikuchi-Fujimoto disease (KFD) is a self-limited lymphadenitis of unclear etiology. We aimed to further characterize this disease in pediatric patients, including evaluation of the CD123 immunohistochemical (IHC) staining and investigation ... ...
Abstract | Objectives: Kikuchi-Fujimoto disease (KFD) is a self-limited lymphadenitis of unclear etiology. We aimed to further characterize this disease in pediatric patients, including evaluation of the CD123 immunohistochemical (IHC) staining and investigation of potential immunologic and infectious causes. Methods: Seventeen KFD cases and 12 controls were retrospectively identified, and the histologic and clinical features were evaluated. CD123 IHC staining was quantified by digital image analysis. Next generation sequencing was employed for comparative microbial analysis via RNAseq (5 KFD cases) and to evaluate the immune repertoire (9 KFD cases). Results: In cases of lymphadenitis with necrosis, >0.85% CD123+ cells by IHC was found to be six times more likely in cases with a final diagnosis of KFD (sensitivity 75%, specificity 87.5%). RNAseq based comparative microbial analysis did not detect novel or known pathogen sequences in KFD. A shared complementarity determining region 3 (CDR3) sequence and use of the same T-cell receptor beta variable region family was identified in KFD LNs but not controls, and was not identified in available databases. Conclusions: Digital quantification of CD123 IHC can distinguish KFD from other necrotizing lymphadenitides. The presence of a unique shared CDR3 sequence suggests that a shared antigen underlies KFD pathogenesis. |
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MeSH term(s) | Adolescent ; Biomarkers/analysis ; Child ; Child, Preschool ; Clone Cells ; Complementarity Determining Regions/immunology ; Dendritic Cells/immunology ; Diagnosis, Differential ; Female ; Histiocytic Necrotizing Lymphadenitis/diagnosis ; Histiocytic Necrotizing Lymphadenitis/immunology ; Humans ; Interleukin-3 Receptor alpha Subunit/analysis ; Interleukin-3 Receptor alpha Subunit/immunology ; Male ; T-Lymphocytes/immunology |
Chemical Substances | Biomarkers ; Complementarity Determining Regions ; IL3RA protein, human ; Interleukin-3 Receptor alpha Subunit |
Language | English |
Publishing date | 2021-02-02 |
Publishing country | United States |
Document type | Journal Article |
ZDB-ID | 1463498-3 |
ISSN | 1615-5742 ; 1093-5266 |
ISSN (online) | 1615-5742 |
ISSN | 1093-5266 |
DOI | 10.1177/1093526620987961 |
Database | MEDical Literature Analysis and Retrieval System OnLINE |
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