A Case of Severe COVID-19 in a Patient with Good's Syndrome

Duarte, Maria; Faria, Luciana; Patronillo, Catarina; Fernandes, Sónia da Costa; Seara, Vera

doi:10.12890/2021_002976

Cited by 6 publications

(3 citation statements)

References 7 publications

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“…At least in 2 cases with relapsing nature of disease has been reported (8). Despite antimicrobial treatment and immunoglobulin replacement mortality remain high (13)(14)(15).…”

Section: Discussionmentioning

confidence: 99%

Clinical and Genomic Evaluations of a persistent fatal SARS-CoV-2 infection in a Goods syndrome patient: A case report

Tabarsi

Maleki

Abtahian

et al. 2023

Preprint

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COVID-19 resulted from an infection by SARS-Cov-2 which is the main cause of ADRS in global population from 2019 on. It may contribute to higher rate of death among the patients with immunodeficiency based on recent reports. In addition, Good syndrome (GS) as a result of thymoma removal might cause in some long-lasting microbial infections. We described clinical aspects and viral mutations on a case of GS suffering from COVID-19. A 46-year-old man with fever, general respiratory signs and positive COVID-19 PCR test, with the history of thymoma removal surgery was admitted to Masih Daneshvari Hospital, Tehran, Iran. Lung radiographs and Oxygen saturation measurement disclosed considerable implication resulted in application of several anti-microbial medication. The delta variant (B.1.617.2 (21J Clade)) was the strain isolated from the patient by sequencing methods done by CNRL while the dominant strain circulated mostly among population was Omicron (B.1.1.529) at the time of sampling. Unfortunately, the patient had passed away a month later by sudden respiratory failure progressed in refractory septic shock. Despite the fact that opportunistic infections may lead the GS patients to a major health problematic condition, unusual persistent of infections such as non-dominant variant of SARS-Cov-2 could be observed through the disease timeline. Therefore, a fully screening of thymoma plus intra-host evolution monitoring of SARS-CoV-2 is highly recommended in immunocompromised patients.

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“…At least in 2 cases with relapsing nature of disease has been reported (8). Despite antimicrobial treatment and immunoglobulin replacement mortality remain high (13)(14)(15).…”

Section: Discussionmentioning

confidence: 99%

Clinical and Genomic Evaluations of a persistent fatal SARS-CoV-2 infection in a Goods syndrome patient: A case report

Tabarsi

Maleki

Abtahian

et al. 2023

Preprint

View full text Add to dashboard Cite

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“…Patients with thymoma, a thymic epithelial tumor, 156 frequently develop T‐cell or Ab‐mediated autoimmune disease (including late‐onset myasthenia gravis, 157 and tissue‐specific auto‐Abs of the APS‐1 spectrum 158–160 ). They harbor auto‐Abs against IL‐17A/F, which are associated with the occurrence of the CMC, 38,158,159,161 and about ∼70% of patients have auto‐Abs against type I IFNs, 87,158,162 leading to a particularly high fatality rate for COVID‐19, especially in patients with Good syndrome (the association of thymoma and immunodeficiency) 163–173 . Interestingly, neoplastic mTECs do not express AIRE, 174,175 suggesting that the breakdown of T‐cell tolerance is a consequence of impaired central tolerance linked to impaired AIRE expression in the neoplastic tissue.…”

Section: Genetic Causes Of Auto‐abs Against Type I Ifnsmentioning

confidence: 99%

“…They harbor auto-Abs against IL-17A/F, which are associated with the occurrence of the CMC, 38,158,159,161 and about ∼70% of patients have auto-Abs against type I IFNs, 87,158,162 leading to a particularly high fatality rate for COVID-19, especially in patients with Good syndrome (the association of thymoma and immunodeficiency). [163][164][165][166][167][168][169][170][171][172][173] Interestingly, neoplastic mTECs do not express AIRE, 174,175 suggesting that the breakdown of T-cell tolerance is a consequence of impaired central tolerance linked to impaired AIRE expression in the neoplastic tissue.…”

Section: G Ene Ti CC Aus E Sofauto -Abs Ag Ain S T T Ype I Ifn Smentioning

confidence: 99%

Human autoantibodies neutralizing type I IFNs: From 1981 to 2023

Bastard,

Gervais,

Le Voyer

et al. 2024

Immunological Reviews

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SummaryHuman autoantibodies (auto‐Abs) neutralizing type I IFNs were first discovered in a woman with disseminated shingles and were described by Ion Gresser from 1981 to 1984. They have since been found in patients with diverse conditions and are even used as a diagnostic criterion in patients with autoimmune polyendocrinopathy syndrome type 1 (APS‐1). However, their apparent lack of association with viral diseases, including shingles, led to wide acceptance of the conclusion that they had no pathological consequences. This perception began to change in 2020, when they were found to underlie about 15% of cases of critical COVID‐19 pneumonia. They have since been shown to underlie other severe viral diseases, including 5%, 20%, and 40% of cases of critical influenza pneumonia, critical MERS pneumonia, and West Nile virus encephalitis, respectively. They also seem to be associated with shingles in various settings. These auto‐Abs are present in all age groups of the general population, but their frequency increases with age to reach at least 5% in the elderly. We estimate that at least 100 million people worldwide carry auto‐Abs neutralizing type I IFNs. Here, we briefly review the history of the study of these auto‐Abs, focusing particularly on their known causes and consequences.

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COVID-19 and Inborn Errors of Immunity

2022

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Inborn errors of immunity (IEI) are a heterogeneous group of disorders affecting immune host defense and immunoregulation. Considering the predisposition to develop severe and chronic infections, it is crucial to understand the clinical evolution of COVID-19 in IEI patients. This review analyzes clinical outcomes following SARS-CoV-2 infection, as well as response to COVID-19 vaccines in patients with IEI.

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A Case of Severe COVID-19 in a Patient with Good's Syndrome

Cited by 6 publications

References 7 publications

Clinical and Genomic Evaluations of a persistent fatal SARS-CoV-2 infection in a Goods syndrome patient: A case report

Clinical and Genomic Evaluations of a persistent fatal SARS-CoV-2 infection in a Goods syndrome patient: A case report

Human autoantibodies neutralizing type I IFNs: From 1981 to 2023

COVID-19 and Inborn Errors of Immunity

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