Interleukin‐12/‐23 receptor beta 1 deficiency in an infant with draining BCG lymphadenitis

Asilsoy, Suna; Bilgili, Gökmen; Turul, Tugba; Dizdarer, Ceyhun; Kalkan, Sema; Yaslı, Hüseyin; Can, Demet; Genel, Ferah; Sanal, Özden

doi:10.1111/j.1442-200x.2009.02818.x

Cited by 9 publications

(3 citation statements)

References 9 publications

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“… 19 A diagnosis of IL-12/IL-23Rb1 deficiency or other defects of MSMD should be considered in patients with BCG infection even in patients only with marked BCG adenitis (although not disseminated), because early treatment and genetic counseling have great importance in these patients. 25 The other two vaccine reactions in this study occurred with axillary lymphadenitis with severe systemic multiorgan infections, diagnosing the presence of an innate immune deficiency, and presumably severe secondary multiorgan infections as the direct cause of death. No autopsy was performed in any of the three BCG post-vaccination deaths to determine the immediate cause of death, and according to the National Guideline for the Surveillance of AEFI, the children were not examined for immune function prior to vaccination, and none of the parties involved were at fault, and were therefore classified as severe vaccine reactions.…”

Section: Discussionmentioning

confidence: 75%

Analysis of death cases in Shenyang City, China, for immunization adverse event surveillance, 2009-2021

Gao,

Dong,

Zhu

et al. 2023

Human Vaccines & Immunotherapeutics

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Through the Chinese National Immunization Adverse Event Surveillance System (CNAEFIS), we collected reports of Adverse Event Following Immunization (AEFI) deaths in Shenyang from 2009 to 2021 with the aim of analyzing AEFI-related deaths and assessing the safety of vaccination. From 2009 to 2021, a total of 12 AEFI-related deaths were reported in Shenyang City, and autopsies were performed in 6 deaths. According to the assessment of the Expert Committee on Investigation and Diagnosis of AEFI 3 (25.0%) deaths were classified as severe vaccine reactions, 9 (75.0%) deaths were classified as coincidental events, and there were no immunization errors or psychological reactions. The overall estimated AEFI-related mortality rate was 0.12 per 100,000 vaccination doses. Spearman’s rank correlation analysis showed no correlation between AEFI, severe vaccine reactions, and suspected vaccination-related deaths. Coincidental events are the most common type of death following vaccination, meaning that the risk of death following immunization is low, and ongoing AEFI surveillance and scientific causality assessment are essential to ensure the vaccine confidence. Detailed pre-vaccination health status questioning is also key to avoiding and reducing adverse events.

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Section: Discussionmentioning

confidence: 75%

Analysis of death cases in Shenyang City, China, for immunization adverse event surveillance, 2009-2021

Gao,

Dong,

Zhu

et al. 2023

Human Vaccines & Immunotherapeutics

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“…Some examples include interleukin (IL)-12 defi ciency (BCG, Salmonella enteritidis adenitis) [43]; IL-12/-23 receptor defi ciency (BCG adenitis) [44]; interferon-γ receptor defi ciency (BCG and Mycobacterium abscessus adenitis) [45]; and IL-1 receptor-activated kinase 4 (IRAK-4) defi ciency (S. aureus adenitis with paratracheal abscess) [46].…”

Section: Differential Diagnosismentioning

confidence: 99%

Suppurative lymphadenitis

Fraser

2009

Curr Infect Dis Rep

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Suppurative lymphadenitis is an important and common form of soft tissue infection. Most acute cases of suppurative lymphadenitis are caused by Staphylococcus aureus or by Streptococcus pyogenes. Empiric antibiotic therapy is frequently successful in the early stages of the disease process, but increasing prevalence of methicillin-resistant S. aureus in particular has necessitated a shift in antibiotic choice that is dictated primarily by specific local resistance patterns. Several other organisms and noninfectious inflammatory processes may give rise to a clinical syndrome suggestive of suppurative lymphadenitis. Failure to respond to empiric antibiotics should trigger a diagnostic re-evaluation to determine the need for surgical intervention and/or the possibility of alternative microbiologic diagnoses.

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“…IL-12 receptor beta 1 (IL-12Rβ1) deficiency is the most common form of MSMD and disease spectrum varies between local infections to systemic BCG infection. 1 Biotin is a vitamin that works as a cofactor of carboxylases in the organism. Biotinidase deficiency (BD) is an autosomal recessively inherited metabolic disease caused by disturbed recycling of biotin, leading to metabolic acidosis, cutaneous and neurological manifestations together with specific laboratory signs.…”

mentioning

confidence: 99%

Coexistence of 2 rare autosomal recessively inherited disorders manifesting with immune deficiency; IL-12 receptor β1 and biotinidase deficiencies

et al. 2018

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Interleukin‐12/‐23 receptor beta 1 deficiency in an infant with draining BCG lymphadenitis

Cited by 9 publications

References 9 publications

Analysis of death cases in Shenyang City, China, for immunization adverse event surveillance, 2009-2021

Analysis of death cases in Shenyang City, China, for immunization adverse event surveillance, 2009-2021

Suppurative lymphadenitis

Coexistence of 2 rare autosomal recessively inherited disorders manifesting with immune deficiency; IL-12 receptor β1 and biotinidase deficiencies

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