M412 a Balancing Act: Treatment of Covid-19 and Cytomegalovirus in a Patient With Primary Immunodeficiency

Szafron, Vibha; Pereira, Marcel de Paula; Vogel, Teri; Leung, Doris; Forbes-Satter, Lisa R.

doi:10.1016/j.anai.2020.08.359

Cited by 4 publications

(11 citation statements)

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“…A total of 459 people with PI and COVID-19 are described from 54 of the 68 studies [ 12 , 13 , 16 , [18] , [19] , [20] , [21] , 24 , 26 , 27 , [29] , [30] , [31] , [32] , [33] , [34] , [35] , [36] , [37] , [38] , [39] , [40] , [41] , [42] , [43] , [44] , [45] , [46] , [47] , [48] , [49] , [50] , [51] , [52] , [53] , [54] , [55] , [56] , [57] , [58] , [59] , [60] , [61] , [62] , [63] , [64] , [65] , [66] , [67] , [68] , [69] , [70] , [71] ] (Table 2). These cases came from case reports as well as cohort studies with information available for individual patients.…”

Section: Resultsmentioning

confidence: 99%

COVID-19-related health outcomes in people with primary immunodeficiency: A systematic review

Drzymalla

Green

Knuth

et al. 2022

Clinical Immunology

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Section: Resultsmentioning

confidence: 99%

COVID-19-related health outcomes in people with primary immunodeficiency: A systematic review

Drzymalla

Green

Knuth

et al. 2022

Clinical Immunology

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“…Predominantly antibody deficiencies were the first most-common IEIs in children who experienced COVID-19 (n = 197, 27.7%) [ 8 , 19 , 27 , 28 , 34 , 36 , 40 , 42 , 45 , 46 , 49 , 50 , 52 , 55 , 62 – 66 , 71 , 73 , 74 , 80 , 81 , 83 , 85 , 91 , 92 , 95 , 97 , 105 , 108 , 114 , 117 , 120 , 122 , 124 , 125 , 129 , 130 ] (see Additional file 2 : Table S3). Among them, 53 have common variable immunodeficiency (CVID, 26.9% of all predominantly antibody deficiencies) [ 19 , 27 , 28 , 34 , 36 , 40 , 46 , 50 , 62 – 66 , 73 , 74 , 80 , 95 , 97 , 114 , 122 , 124 , 125 ], 45 have X-linked agammaglobulinemia (XLA, 22.8%) [ 8 , 19 , 34 , 40 , 42 , 46 , 49 , 50 , ...…”

Section: Resultsmentioning

confidence: 99%

“…The remaining 18 patients have other isotype, light chain, or functional deficiencies with generally normal numbers of B cells [including IgG subclass deficiency with IgA and/or IgM deficiency (n = 2) [ 108 ], IgG, IgA and IgM deficiencies (n = 2) [ 108 ], partial IgA deficiency (n = 1) [ 124 ], and low IgM level (n = 1) [ 124 ]]; selective IgM deficiencies (n = 3) [ 81 , 108 ]; specific antibody deficiency with normal immunoglobulin and B cells levels (n = 4) [ 40 , 65 ], unspecified predominantly antibody deficiency (n = 3) [ 65 , 95 ]; UNG deficiency (n = 1) [ 19 ] and APRIL deficiency (n = 1) [ 40 ]. The most frequent main genetic causes of predominantly antibody deficiencies in children infected with SARS-CoV-2 were BTK (n = 41) [ 8 , 19 , 34 , 40 , 46 , 49 , 50 , 52 , 63 , 65 , 71 , 73 , 74 , 85 , 91 , 92 , 95 , 97 , 105 , 117 , 120 , 129 ], NFKB2 (n = 4) [ 27 , 36 , 95 , 122 ], TNFRSF13B (n = 3) [ 62 , 114 , 125 ], PIK3CD (n = 3) [ 19 , 65 , 97 ], and PIK3R1 (n = 2) [ 45 , 63 ]. For patients with predominately antibody deficiencies who acquired SARS-CoV-2, the median interquartile range (IQR) age was 120 months [83–175], with a male predominance [n = 94, 47.7%] [ 8 , 19 , 27 , 28 , 34 , 40 , 42 , 45 , 46 <...…”

Section: Resultsmentioning

confidence: 99%

“…The most frequent main genetic causes of predominantly antibody deficiencies in children infected with SARS-CoV-2 were BTK (n = 41) [ 8 , 19 , 34 , 40 , 46 , 49 , 50 , 52 , 63 , 65 , 71 , 73 , 74 , 85 , 91 , 92 , 95 , 97 , 105 , 117 , 120 , 129 ], NFKB2 (n = 4) [ 27 , 36 , 95 , 122 ], TNFRSF13B (n = 3) [ 62 , 114 , 125 ], PIK3CD (n = 3) [ 19 , 65 , 97 ], and PIK3R1 (n = 2) [ 45 , 63 ]. For patients with predominately antibody deficiencies who acquired SARS-CoV-2, the median interquartile range (IQR) age was 120 months [83–175], with a male predominance [n = 94, 47.7%] [ 8 , 19 , 27 , 28 , 34 , 40 , 42 , 45 , 46 , 49 , 50 , 52 , 55 , 62 , 63 , 65 , 66 , 71 , 73 , 74 , 80 , 85 , 91 , 92 , 95 , 105 , 108 , 117 , 120 , 125 ,…”

Section: Resultsmentioning

confidence: 99%

“…For patients with predominately antibody deficiencies who acquired SARS-CoV-2, the median interquartile range (IQR) age was 120 months [83–175], with a male predominance [n = 94, 47.7%] [ 8 , 19 , 27 , 28 , 34 , 40 , 42 , 45 , 46 , 49 , 50 , 52 , 55 , 62 , 63 , 65 , 66 , 71 , 73 , 74 , 80 , 85 , 91 , 92 , 95 , 105 , 108 , 117 , 120 , 125 , 129 , 130 ], and majority of the patients belonged to White (Caucasian) (n = 144, 73.1%) [ 8 , 27 , 36 , 42 , 46 , 50 , 52 , 55 , 62 – 64 , 66 , 71 , 73 , 80 , 81 , 83 , 85 , 95 , 97 , 105 , 108 , 114 , 120 , 122 , 124 , 129 ], Hispanic (n = 30, 15.2%) [ 40 , 65 , 95 , 125 ] and Persian (n = 15, 7.6%) [ 19 , 28 , 34 , ...…”

Section: Resultsmentioning

confidence: 99%

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Severity of SARS-CoV-2 infection in children with inborn errors of immunity (primary immunodeficiencies): a systematic review

Alhumaid,

Al Mutared,

Al Alawi

et al. 2023

Allergy Asthma Clin Immunol

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Background Inborn errors of immunity (IEIs) are considered significant challenges for children with IEIs, their families, and their medical providers. Infections are the most common complication of IEIs and children can acquire coronavirus disease 2019 (COVID-19) even when protective measures are taken. Objectives To estimate the incidence of severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) infection in children with IEIs and analyse the demographic parameters, clinical characteristics and treatment outcomes in children with IEIs with COVID-19 illness. Methods For this systematic review, we searched ProQuest, Medline, Embase, PubMed, CINAHL, Wiley online library, Scopus and Nature through the Preferred Reporting Items for Systematic Reviews and Meta Analyses (PRISMA) guideline for studies on the development of COVID-19 in children with IEIs, published from December 1, 2019 to February 28, 2023, with English language restriction. Results Of the 1095 papers that were identified, 116 articles were included in the systematic review (73 case report, 38 cohort 4 case-series and 1 case–control studies). Studies involving 710 children with IEIs with confirmed COVID-19 were analyzed. Among all 710 IEIs pediatric cases who acquired SARS-CoV-2, some children were documented to be admitted to the intensive care unit (ICU) (n = 119, 16.8%), intubated and placed on mechanical ventilation (n = 87, 12.2%), suffered acute respiratory distress syndrome (n = 98, 13.8%) or died (n = 60, 8.4%). Overall, COVID-19 in children with different IEIs patents resulted in no or low severity of disease in more than 76% of all included cases (COVID-19 severity: asymptomatic = 105, mild = 351, or moderate = 88). The majority of children with IEIs received treatment for COVID-19 (n = 579, 81.5%). Multisystem inflammatory syndrome in children (MIS-C) due to COVID-19 in children with IEIs occurred in 103 (14.5%). Fatality in children with IEIs with COVID-19 was reported in any of the included IEIs categories for cellular and humoral immunodeficiencies (n = 19, 18.6%), immune dysregulatory diseases (n = 17, 17.9%), innate immunodeficiencies (n = 5, 10%), bone marrow failure (n = 1, 14.3%), complement deficiencies (n = 1, 9.1%), combined immunodeficiencies with associated or syndromic features (n = 7, 5.5%), phagocytic diseases (n = 3, 5.5%), autoinflammatory diseases (n = 2, 3%) and predominantly antibody deficiencies (n = 5, 2.5%). Mortality was COVID-19-related in a considerable number of children with IEIs (29/60, 48.3%). The highest ICU admission and fatality rates were observed in cases belonging to cellular and humoral immunodeficiencies (26.5% and 18.6%) and immune dysregulatory diseases (35.8% and 17.9%) groups, especially in children infected with SARS-CoV-2 who suffered severe combined immunodeficiency (28.6% and 23.8%), combined immunodeficiency (25% and 15%), familial hemophagocytic lymphohistiocytosis (40% and 20%), X-linked lymphoproliferative diseases-1 (75% and 75%) and X-linked lymphoproliferative diseases-2 (50% and 50%) compared to the other IEIs cases. Conclusion Children with IEIs infected with SARS-CoV-2 may experience higher rates of ICU admission and mortality in comparison with the immunocompetent pediatric populations. Underlying immune defects does seem to be independent risk factors for severe SARS-CoV-2 infection in children with IEIs, a number of children with SCID and CID were reported to have prolonged infections–though the number of patients is small–but especially immune dysregulation diseases (XLP1 and XLP2) and innate immunodeficiencies impairing type I interferon signalling (IFNAR1, IFNAR2 and TBK1).

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Adalimumab/anakinra/dexamethasone

2020

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M412 a Balancing Act: Treatment of Covid-19 and Cytomegalovirus in a Patient With Primary Immunodeficiency

Cited by 4 publications

References 0 publications

COVID-19-related health outcomes in people with primary immunodeficiency: A systematic review

COVID-19-related health outcomes in people with primary immunodeficiency: A systematic review

Severity of SARS-CoV-2 infection in children with inborn errors of immunity (primary immunodeficiencies): a systematic review

Adalimumab/anakinra/dexamethasone

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