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Case Reports in Immunology publishes case reports and case series related to allergies, immunodeficiencies, autoimmune diseases, immune disorders, cancer immunology and transplantation immunology.
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Case Reports in Immunology maintains an Editorial Board of practicing researchers from around the world, to ensure manuscripts are handled by editors who are experts in the field of study.
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More articlesAutoreactive Antibodies Associated with Castleman Disease Triad
The Castleman triad has been described in a select few patients presenting with a retroperitoneal mass, mucocutaneous pemphigus vulgaris, and bronchiolitis obliterans. Here, we describe the Castleman triad in a 19-year-old male with unicentric hyaline vascular type Castleman disease (HV-CD). This patient presented with an array of positive antibodies, including anti-cyclic citrullinated peptide, anti-double-stranded DNA, and Sjogren’s IgG. Interestingly, the patient’s rheumatologic symptoms resolved after tumor resection, while his antibody profile remained relatively unchanged. HV-CD, with a triad presentation, was thought to be from a paraneoplastic syndrome secondary to an underlying lymphoproliferative disorder. The findings presented here identify multiple autoantibodies potentially contributing to this patient’s presentation with HV-CD.
Multiple Abscess Collections: Antibiotics or Steroids?
Aseptic abscess syndrome (AAS) is a medical rarity. The combination of multiple abscess collections in different organs, negative microbiological studies, and the association with an inflammatory bowel disease is highly suggestive for an AAS. The AAS is an acute neutrophilic dermatosis, so “generalized pyoderma gangraenosum” or “generalized bullous sweet syndrome” might be used synonymously. It is important to note that the diagnosis of an AAS can be made only after careful exclusion of an infectious disease. Of interest, despite the severity of the inflammation, patients with AAS are commonly hemodynamically stable. To date, no studies have investigated the optimal regimen, dose, and duration of therapy. Corticosteroids are the cornerstone of immunosuppression during the acute phase. After the induction phase, therapy might be switched to anakinra or infliximab.
Omeprazole Induced Rapid Drug Reaction with Eosinophilia, Systemic Symptoms, and Cross-Reactivity in Delayed-Type Hypersensitivity Associated with Proton-Pump Inhibitors: A Case Report and Literature Review
Background. Omeprazole, a proton pump inhibitor (PPI), is a widely used and generally safe agent for treating acid-related gastrointestinal conditions. However, drug reaction with eosinophilia and systemic symptoms (DRESSs) syndrome has been reported. Objectives. To report a case of omeprazole-induced rapid DRESS syndrome and to review the literature. Methods. Descriptive analysis of one new case and a case series from literature review. Results. We report a case of 82-year-old woman presenting with rapid-onset of DRESS syndrome. The condition was initially suspected to be caused by antibiotic, but the definite diagnosis was eventually omeprazole-induced DRESS syndrome as suggested by the enzyme-linked immune absorbent spot (ELISpot) assay along with the clinical picture. Previous literatures regarding cases of PPI-induced DRESS syndrome were pooled for descriptive analysis. Among 21 PPI cases pooled, esomeprazole was the most commonly implicated PPI (52.4%), followed by pantoprazole (19.1%), and omeprazole along with lansoprazole (both 14.3%). The issue of cross-reactivities amongst PPIs remains uncertain. Nonetheless, in situations in which a PPIs are deemed necessary, a prudent approach could be considering a switch to an alternative agent with distinct chemical structure. Conclusion. PPI is commonly used safely as an agent for acid-related gastrointestinal conditions. However, PPI-induced rapid DRESS syndrome can occur, particularly with prior exposure history. ELISpot is an in vitro test, useful in identifying the culprit agent in patients with delayed-type hypersensitivity reaction.
Novel BTK Mutation in Patient with Late Diagnosis of X-Linked Agammaglobulinemia
X-linked agammaglobulinemia (XLA) is a genetic disorder with mutation in Bruton’s tyrosine kinase (BTK). Defects in B cell development and immunoglobulin production lead to recurrent infections following loss of maternal IgG at 6 months of age. A 55-year-old male with a longstanding common variable immunodeficiency diagnosis on infusion therapy presented to the clinic with cutaneous T-cell lymphoma, which inspired overall repeat evaluation. Immunoglobulin levels and lymphocyte markers, family history, and genetic testing prompted a true diagnosis of XLA and novel mutation in the BTK gene. Disease-associated mutations have been noted in all five domains of BTK, with missense variants most commonly cited among the 100s of reported genetic alterations. The BTK protein is expressed in hematopoietic lineages and plasma cells, with the exception of T lymphocytes. Disruption in the protein function or absence of BTK halts normal B cell development at the pre-B transitional cell stage and induces premature apoptosis. We present the first reported case of a novel hemizygous BTK c.1492C > G mutation in a patient causing XLA.
Toxic Epidermal Necrolysis after COVID-19 mRNA-1237 Vaccination
This letter illustrates a case of toxic epidermal necrolysis (TEN) after COVID-19 mRNA-1273 vaccination, which corresponds with the existing published data and contributes detailed knowledge of TEN reaction after vaccination. Interestingly, the reaction started at the site of vaccination and the patient went on to tolerate a major excipient of the vaccine suggesting the reaction may be associated with the mRNA itself or is triggered by the immunostimulatory action of the vaccine.
Pernicious Anemia in an Adult with Trisomy 21
Pernicious anemia is an autoimmune disease caused by the malabsorption of vitamin B12. It usually appears in the elderly. People with trisomy 21 are susceptible to autoimmune diseases. This susceptibility is thought to be due to altered expression of the AIRE gene, which is located in the 21q22.3 region. Although pernicious anemia is not common in people with trisomy 21, AIRE is pointed out as a susceptibility gene of pernicious anemia in a genome-wide association study. Here, we report a man with trisomy 21, who suffered from the pernicious anemia. When he was in his 30 s, he visited our hospital because of diarrhea and poor oral intake. He showed thrombocytopenic purpura-like features, and was diagnosed as pernicious anemia. After supplementation of vitamin B12, he recovered from the illness. The reason for his early onset may be because of trisomy 21. Altered expression of AIRE might trigger the disease.