M C Rolfes1, P Sriaroon2, B J Dávila Saldaña3, C C Dvorak4, H Chapdelaine5, R M Ferdman6, K Chen7, S Jolles8, N C Patel9, Y J Kim10, T K Tarrant11, T Martelius12, M Seppanen13, A Y Joshi14. 1. Mayo Clinic School of Medicine, Rochester, MN. 2. USF/All Children's Hospital Allergy/Immunology, St. Petersburg, FL. 3. Division of Blood and Marrow Transplantation, Children's National Health System, Washington, DC. 4. Division of Pediatric Allergy, Immunology, and Bone Marrow Transplantation, University of California, San Francisco, CA. 5. Department of Allergy and Immunology, Centre Hospitalier de l'Université de Montréal, Montreal, Quebec, Canada. 6. Division of Clinical Immunology and Allergy, Children's Hospital Los Angeles, Department of Pediatrics, Keck School of Medicine, University of Southern California, Los Angeles, CA. 7. Department of Pediatrics, Division of Allergy and Immunology, University of Utah School of Medicine, Salt Lake City, UT. 8. Immunodeficiency Centre for Wales, University Hospital of Wales, Cardiff, UK. 9. Division of Pediatric Infectious Disease and Immunology, Levine Children's Hospital, Carolinas Medical Center, Charlotte, NC. 10. Division of Infectious Diseases and Immunodeficiency, Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Republic of Korea. 11. Division of Rheumatology and Immunology, Department of Medicine, Duke University, Durham, NC. 12. Adult Immunodeficiency Unit, Infectious Diseases, Inflammation Center, University of Helsinki and Helsinki University Hospital, Helsinki, Finland. 13. Adult Immunodeficiency Unit, Infectious Diseases, Inflammation Center, University of Helsinki and Helsinki University Hospital, Helsinki, Finland; Rare Disease Center, Children's Hospital, University of Helsinki and Helsinki University Hospital, Helsinki, Finland. 14. Mayo Clinic School of Medicine, Rochester, MN; Division of Pediatric Allergy and Immunology, Department of Pediatric and Adolescent Medicine, Mayo Clinic Children's Center, Rochester, MN. Electronic address: joshi.avni@mayo.edu.
Abstract
OBJECTIVE: Predictive factors associated with clinical outcomes of chronic norovirus infection (CNI) in primary immunodeficiency diseases (PIDD) are lacking. METHOD: We sought to characterize CNI using a multi-institutional cohort of patients with PIDD and CNI using the Clinical Immunology Society's CIS-PIDD Listserv e-mail group. RESULTS: Thirty-four subjects (21 males and 13 females) were reported from centers across North America, Europe, and Asia. All subjects were receiving high doses (median IgG dose: 1200 mg/kg/month) of supplemental immunoglobulin therapy. Fifty-three percent had a complete absence of B cells (median B-cell count 0; range 0-139 cells/μL). Common Variable Immune Deficiency (CVID) subjects manifested a unique phenotype with B-cell lymphopenia, non O+ blood type, and villous atrophy (logistic regression model, P = 0.01). Five subjects died, all of whom had no evidence of villous atrophy. CONCLUSION: While Norovirus (NoV) is thought to replicate in B cells, in this PIDD cohort of CNI, B-cell lymphopenia was common, indicating that the presence of B lymphocytes is not essential for CNI.
OBJECTIVE: Predictive factors associated with clinical outcomes of chronic norovirus infection (CNI) in primary immunodeficiency diseases (PIDD) are lacking. METHOD: We sought to characterize CNI using a multi-institutional cohort of patients with PIDD and CNI using the Clinical Immunology Society's CIS-PIDD Listserv e-mail group. RESULTS: Thirty-four subjects (21 males and 13 females) were reported from centers across North America, Europe, and Asia. All subjects were receiving high doses (median IgG dose: 1200 mg/kg/month) of supplemental immunoglobulin therapy. Fifty-three percent had a complete absence of B cells (median B-cell count 0; range 0-139 cells/μL). Common Variable Immune Deficiency (CVID) subjects manifested a unique phenotype with B-cell lymphopenia, non O+ blood type, and villous atrophy (logistic regression model, P = 0.01). Five subjects died, all of whom had no evidence of villous atrophy. CONCLUSION: While Norovirus (NoV) is thought to replicate in B cells, in this PIDD cohort of CNI, B-cell lymphopenia was common, indicating that the presence of B lymphocytes is not essential for CNI.
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