COVID‐19 presenting with autoimmune hemolytic anemia in the setting of underlying immune dysregulation

Wahlster, Lara; Weichert-Leahey, Nina; Trissal, Maria; Grace, Rachael F.; Sankaran, Vijay G.

doi:10.1002/pbc.28382

Cited by 35 publications

(35 citation statements)

References 5 publications

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“…A few cases of AIHA have been shown to be associated with SARS-CoV-2 infection ( Table 1 ) including both warm and cold AIHA. In some cases, patients were affected by pre-existent immune thrombocytopenia, suggesting a susceptible background for hematological dysregulations [ [12] , [13] , [14] ]. Molecular mimicry has been proposed to trigger AIHA, with antibodies elicited against viral proteins cross reacting with self-antigens.…”

Section: Resultsmentioning

confidence: 99%

“… Manifestation Patients number Sex Age (years) Treatment Outcome Ref. AIHA 1 warm F 46 IVIg, glucocorticoids and transfusion Recovered [ 12 ] AIHA 7 (4 warm, 3 cold) 4 M 3 F median 62 (range 61–89) Glucocorticoids (5 cases), + rituximab (2 cases), transfusion (2 cases) Partly recovered [ 18 ] AIHA 1 cold F 46 None Death [ 13 ] Evans syndrome 1 M 39 IVIg Recovered [ 16 ] AIHA 1 warm M 17 Glucocorticoids, transfusion Recovered [ 14 ] AIHA 1 cold M 62 Transfusion Recovered [ 100 ] AIHA 2 cold F M 43 63 Transfusion None Recovered Recovered [ …”

Section: Resultsmentioning

confidence: 99%

See 1 more Smart Citation

The JANUS of chronic inflammatory and autoimmune diseases onset during COVID-19 – A systematic review of the literature

Novelli

Motta

Santis

et al. 2021

Journal of Autoimmunity

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Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

The JANUS of chronic inflammatory and autoimmune diseases onset during COVID-19 – A systematic review of the literature

Novelli

Motta

Santis

et al. 2021

Journal of Autoimmunity

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“…Even after receiving methylprednisolone (1 mg/kg every 6 hours) for 1 day, he had a markedly elevated soluble IL-2 receptor level (15,990 pg/mL), which was increased from his previous level of 1313 pg/mL (normal, 45-1105 pg/mL), reflecting widespread T-cell activation. Although his AIHAwas initially thought to be the most prominent feature of his SARS-CoV-2 infection, 7 his history of fever, involvement of the gastrointestinal, hematologic, and respiratory systems, and elevated inflammatory markers were consistent with the subsequently described MIS-C. 8 He was treated with fresh frozen plasma, intravenous immunoglobulin, and methylprednisolone, and he received multiple transfusions, with eventual resolution of his hypoxia and cytopenias. One month after recovery from COVID-19, while he was still maintained on a high-dose corticosteroid wean, patient 2 was found to have a normal soluble IL-2R level (577 pg/mL) and undetectable levels of all cytokines measured (IL-10, IL-12, IFN-g, IL-4, IL-5, IL-13, IL-17, IL-1b, IL-6, IL-8, and TNF-a).…”

Section: Resultsmentioning

confidence: 99%

Immune dysregulation and multisystem inflammatory syndrome in children (MIS-C) in individuals with haploinsufficiency of SOCS1

Lee

Platt

Weeks

et al. 2020

Journal of Allergy and Clinical Immunology

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129

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Background: We studied 2 unrelated patients with immune thrombocytopenia and autoimmune hemolytic anemia in the setting of acute infections. One patient developed multisystem inflammatory syndrome in children in the setting of a severe acute respiratory syndrome coronavirus 2 infection. Objectives: We sought to identify the mechanisms underlying the development of infection-driven autoimmune cytopenias. Methods: Whole-exome sequencing was performed on both patients, and the impact of the identified variants was validated by functional assays using the patients' PBMCs. Results: Each patient was found to have a unique heterozygous truncation variant in suppressor of cytokine signaling 1 (SOCS1). SOCS1 is an essential negative regulator of type I and type II IFN signaling. The patients' PBMCs showed increased levels of signal transducer and activator of transcription 1 phosphorylation and a transcriptional signature characterized by increased expression of type I and type II IFN-stimulated genes and proapoptotic genes. The enhanced IFN signature exhibited by the patients' unstimulated PBMCs parallels the hyperinflammatory state associated with multisystem inflammatory syndrome in children, suggesting the contributions of SOCS1 in regulating the inflammatory response characteristic of multisystem inflammatory syndrome in children. Conclusions: Heterozygous loss-of-function SOCS1 mutations are associated with enhanced IFN signaling and increased immune cell activation, thereby predisposing to infectionassociated autoimmune cytopenias. (J Allergy Clin Immunol 2020;nnn:nnn-nnn.)

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“…39 One case report described a 17-year-old male with a history of refractory chronic immune thrombocytopenia that manifested as autoimmune hemolytic anemia during infection with SARS-CoV-2. 40 TA B L E 1 Studies on hematological laboratory findings in children with COVID-19, December 2019 to April 2020…”

Section: Abnormalities In the Red Blood Cell Count In Children With Cmentioning

confidence: 99%

Hematological manifestations of SARS‐CoV‐2 in children

Kosmeri

Koumpis

Tsabouri

et al. 2020

Pediatric Blood & Cancer

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Infection from severe acute respiratory syndrome coronavirus-2 (SARS-CoV-2), though mainly a respiratory disease, can impair many systems, including causing hematological complications. Lymphopenia and hypercoagulability have been reported in adults with coronavirus disease 2019 (COVID-19) and are considered markers of poor prognosis. This review summarizes the hematological findings in children with SARS-CoV-2 infection. The majority of infected children had a normal leukocyte count, while the most common white blood cell abnormality was leukopenia. Lymphopenia, which may be a marker of severe disease, was rarer in children than in adults, possibly due to their immature immune system or due to the less severe manifestation of COVID-19 in this age group. Age may have an impact, and in neonates and infants the most common abnormality was lymphocytosis. Abnormalities of red blood cells and platelets were uncommon. Anemia and hypercoagulability were reported mainly in children presenting the novel multisystem inflammatory syndrome (MIS) associated with SARS-CoV-2.

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COVID‐19 presenting with autoimmune hemolytic anemia in the setting of underlying immune dysregulation

Cited by 35 publications

References 5 publications

The JANUS of chronic inflammatory and autoimmune diseases onset during COVID-19 – A systematic review of the literature

The JANUS of chronic inflammatory and autoimmune diseases onset during COVID-19 – A systematic review of the literature

Immune dysregulation and multisystem inflammatory syndrome in children (MIS-C) in individuals with haploinsufficiency of SOCS1

Hematological manifestations of SARS‐CoV‐2 in children

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