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10.1016/j.jaci.2020.09.020 http://scihub22266oqcxt.onion/10.1016/j.jaci.2020.09.020 33007327!7525247!33007327     free     free     free Deprecated: Implicit conversion from float 233.6 to int loses precision in C:\Inetpub\vhosts\kidney.de\httpdocs\pget.php on line 534 Deprecated: Implicit conversion from float 233.6 to int loses precision in C:\Inetpub\vhosts\kidney.de\httpdocs\pget.php on line 534 Deprecated: Implicit conversion from float 233.6 to int loses precision in C:\Inetpub\vhosts\kidney.de\httpdocs\pget.php on line 534 Deprecated: Implicit conversion from float 233.6 to int loses precision in C:\Inetpub\vhosts\kidney.de\httpdocs\pget.php on line 534       J+Allergy+Clin+Immunol 2021 ; 147 (2): 532-544.e1 Nephropedia Template TP {{tp|p=33007327|t=2021. Severe SARS-CoV-2 disease in the context of a NF-kappaB2 loss-of-function pathogenic variant |pdf=|usr=}}{{33007327}} gab.com Text Severe SARS-CoV-2 disease in the context of a NF-kappaB2 loss-of-function pathogenic variant JO: J Allergy Clin Immunol http://www.kidney.de/pget.php?&tw=1&pmid=33007327 #FOAvip BACKGROUND: Severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) is a novel coronavirus that emerged recently and has created a global pandemic. Symptomatic SARS-CoV-2 infection, termed coronavirus disease 2019 (COVID-19), has been associated with a host of symptoms affecting numerous organ systems, including the lungs, cardiovascular system, kidney, central nervous system, gastrointestinal tract, and skin, among others. OBJECTIVE: Although several risk factors have been identified as related to complications from and severity of COVID-19, much about the virus remains unknown. The host immune response appears to affect the outcome of disease. It is not surprising that patients with intrinsic or secondary immune compromise might be particularly susceptible to complications from SARS-CoV-2 infection. Pathogenic loss-of-function or gain-of-function heterozygous variants in nuclear factor-kappaB2 have been reported to be associated with either a combined immunodeficiency or common variable immunodeficiency phenotype. METHODS: We evaluated the functional consequence and immunologic phenotype of a novel NFKB2 loss of function variant in a 17-year-old male patient and describe the clinical management of SARS-CoV-2 infection in this context. RESULTS: This patient required a 2-week hospitalization for SARS-CoV-2 infection, including 7 days of mechanical ventilation. We used biologic therapies to avert potentially fatal acute respiratory distress syndrome and treat hyperinflammatory responses. The patient had an immunologic phenotype of B-cell dysregulation with decreased switched memory B cells. Despite the underlying immune dysfunction, he recovered from the infection with intense management. CONCLUSIONS: This clinical case exemplifies some of the practical challenges in management of patients with SARS-CoV-2 infection, especially in the context of underlying immune dysregulation. Twit Text FOAVip Severe SARS-CoV-2 disease in the context of a NF-kappaB2 loss-of-function pathogenic variant ????J Allergy Clin Immunol http://www.kidney.de/pget.php?&tw=1&pmid=33007327 #FOAvip Twit Text # Free Paper on # # # # # LINK http://www.kidney.de/pget.php?&tw=1&pmid=33007327 #FOAvip English Wikipedia <ref>{{Cite pmid|33007327}}</ref> Severe SARS-CoV-2 disease in the context of a NF-kappaB2 loss-of-function pathogenic variant #MMPMID33007327 Abraham RS; Marshall JM; Kuehn HS; Rueda CM; Gibbs A; Guider W; Stewart C; Rosenzweig SD; Wang H; Jean S; Peeples M; King T; Hunt WG; Honegger JR; Ramilo O; Mustillo PJ; Mejias A; Ardura MI; Shimamura M J Allergy Clin Immunol 2021[Feb]; 147 (2): 532-544.e1 PMID33007327show ga BACKGROUND: Severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) is a novel coronavirus that emerged recently and has created a global pandemic. Symptomatic SARS-CoV-2 infection, termed coronavirus disease 2019 (COVID-19), has been associated with a host of symptoms affecting numerous organ systems, including the lungs, cardiovascular system, kidney, central nervous system, gastrointestinal tract, and skin, among others. OBJECTIVE: Although several risk factors have been identified as related to complications from and severity of COVID-19, much about the virus remains unknown. The host immune response appears to affect the outcome of disease. It is not surprising that patients with intrinsic or secondary immune compromise might be particularly susceptible to complications from SARS-CoV-2 infection. Pathogenic loss-of-function or gain-of-function heterozygous variants in nuclear factor-kappaB2 have been reported to be associated with either a combined immunodeficiency or common variable immunodeficiency phenotype. METHODS: We evaluated the functional consequence and immunologic phenotype of a novel NFKB2 loss of function variant in a 17-year-old male patient and describe the clinical management of SARS-CoV-2 infection in this context. RESULTS: This patient required a 2-week hospitalization for SARS-CoV-2 infection, including 7 days of mechanical ventilation. We used biologic therapies to avert potentially fatal acute respiratory distress syndrome and treat hyperinflammatory responses. The patient had an immunologic phenotype of B-cell dysregulation with decreased switched memory B cells. Despite the underlying immune dysfunction, he recovered from the infection with intense management. CONCLUSIONS: This clinical case exemplifies some of the practical challenges in management of patients with SARS-CoV-2 infection, especially in the context of underlying immune dysregulation. |*SARS-CoV-2/immunology[MESH] |Adenosine Monophosphate/analogs & derivatives/therapeutic use[MESH] |Adolescent[MESH] |Alanine/analogs & derivatives/therapeutic use[MESH] |Antibodies, Viral/blood[MESH] |Antiviral Agents/therapeutic use[MESH] |B-Lymphocytes/immunology[MESH] |COVID-19/diagnosis/*genetics/immunology/therapy[MESH] |Hospitalization[MESH] |Humans[MESH] |Interleukin-6/blood[MESH] |Male[MESH] |NF-kappa B p52 Subunit/*genetics[MESH] |Respiration, Artificial[MESH]Severe SARS-CoV-2 disease in the context of a NF-kappaB2 loss-of-funct(epmc).pdf DeepDyve Pubget Overpricing