Kikuchi-Fujimoto disease associated with hemophagocytic lymphohistiocytosis following the BNT162b2 mRNA COVID-19 vaccination

Caocci, Giovanni; Fanni, Daniela; Porru, Mariagrazia; Greco, Marianna; Nemolato, Sonia; Firinu, Davide; Faa, Gavino; Scuteri, Angelo; Nasa, Giorgio La

doi:10.3324/haematol.2021.280239

Cited by 21 publications

(31 citation statements)

References 12 publications

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“…To our knowledge, there are no data concerning the management of HLH in patients with thalassemia, and this is the first case report of a patient with HLH occurring after anti-SARS-CoV-2 booster dose. Other authors have recently described similar cases associated with the first doses of m-RNA or virus-vectored vaccines: some of them were successfully treated with corticosteroid therapy alone, obtaining a rapid remission of symptoms and improvement in blood counts [5][6][7][8]. Some recovered with support therapy alone [9], while others required etoposide or stronger combined therapies with steroids, IVIG and IL-1 receptor antagonists [8,[10][11][12].…”

Section: Discussionmentioning

confidence: 94%

The First Case of Haemophagocytic Lymphohistiocytosis Triggered by the Booster Dose of Anti-SARS-CoV-2 Vaccine in a Patient with β-Thalassemia

Calvaruso

Chiavetta

Renda

et al. 2022

Thalassemia Reports

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Background: Haemophagocytic lymphohistiocytosis (HLH) is a rare and potentially life-threatening systemic hyperinflammatory disease, which can have several aetiologies. Clinical case: a 48-year-old woman affected by a transfusion-dependent β-thalassemia was hospitalized in our haematology unit presenting with intermittent fever, haepatosplenomegaly and pancytopenia, which developed a few days after the booster dose of anti-SARS-CoV-2 mRNA vaccine. The investigations performed during hospitalization led to a diagnosis of HLH and steroid therapy where IV dexamethasone was initiated and provided benefits. Conclusions: the severity of HLH mandates early treatment, but the management of patients with post-vaccine HLH is still challenging and requires further study. No cases of HLH in patients with thalassemia were previously described.

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Section: Discussionmentioning

confidence: 94%

The First Case of Haemophagocytic Lymphohistiocytosis Triggered by the Booster Dose of Anti-SARS-CoV-2 Vaccine in a Patient with β-Thalassemia

Calvaruso

Chiavetta

Renda

et al. 2022

Thalassemia Reports

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“…Three of the patients developed KFD after receiving only the first dose of the vaccine, while the other developed KFD after the second dose. All of the patients had a good prognosis with resolution of symptoms [ 13 – 15 ] . There have been sporadic reports of cases of KFD following administration of the influenza vaccine, the human papilloma virus vaccine and the Japanese encephalitis vaccine [ 16 , 17 ] , although none of these are mRNA vaccines.…”

Section: Discussionmentioning

confidence: 99%

A Transient Inflammatory Reaction with a B-Cell Lymphoid Infiltrate and Dysplastic Bone Marrow Changes Associated with the BNT162b2 COVID-19 Vaccine

Vronsky

Hershkowitz

Hershko

et al. 2022

European Journal of Case Reports in Internal Medicine

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COVID-19 vaccines were introduced soon after the COVID-19 pandemic emerged in 2020. Various side effects were reported worldwide, including several types of common systemic side effects such as fever and general fatigue. Reports of other rare manifestations also emerged. We report the case of an adult male with a rare systemic syndrome mimicking lymphoma after he had received the first dose of an mRNA-based COVID-19 vaccine. After nearly 6 months of investigation with suspicion for an infection or malignancy, all symptoms resolved, laboratory tests normalized, and imaging showed no sign of active disease.

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“…To our knowledge, this is a rare case reported in the literature, and only a few studies have reported HLH after receiving the COVID-19 vaccine. [5][6][7][9][10][11][12][13] It, therefore, it is important to document this condition in order to reiterate the occurrence of such events. Looking at the same scenario from a different angle, it has to be pointed out that rare vaccination events are important, as they help to identify and treat a small number of cases that react in this manner.…”

Section: Discussionmentioning

confidence: 99%

A case of hemophagocytic lymphohistiocytosis after BNT162b2 COVID-19 (Comirnaty®) vaccination

et al. 2022

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Rationale: Coronavirus disease (COVID-19), an infectious disease caused by the severe acute respiratory syndrome coronavirus 2 virus, was reported in Wuhan of China in December 2019. The world is still in a state of pandemic owing to COVID-19. COVID-19 vaccines help our bodies develop immunity against the virus that causes COVID-19 without having to get the illness. Herein, we describe a rare case of a critical disorder, hemophagocytic lymphohistiocytosis (HLH), in a patient with nephritic sclerosis associated with hypertension, following mRNA COVID-19 vaccination. HLH is a life-threatening hyperinflammatory syndrome caused by aberrantly activated macrophages and cytotoxic T cells that may rapidly progress to terminal multiple organ failure.Patient concerns: An 85-year-old Japanese woman with chronic renal failure and hypertension was included in this study. Routine laboratory investigations provided the following results: white blood cell (WBC) count, 4.6 × 10 9 /L; hemoglobin (Hb), 8.1 g/ dL; platelet count, 27 × 10 9 /L; blood urea nitrogen 48.9 mg/dL, and serum creatinine 3.95 mg/dL. The patient developed malaise, vomiting, and persistent high fever (up to 39.7°C) on the 12 th day after receiving the second dose of the vaccine. Initial evaluation revealed neutropenia. The total WBC count was 0.40 × 10 9 /L (Neutrophils 0, Lymphocytes 240/μ, blast 0%); Hb 9.0 g/dL, platelet count 27 × 10 9 /L; and, C Reactive Protein 9.64 mg/dL.Diagnosis: Further tests showed hyperferritinemia (serum ferritin 2284.4 μg/L). Bone marrow examination revealed haemophagocytosis. A provisional diagnosis of HLH associated with the Comirnaty ® vaccination was made based on the HLH-2004 diagnostic criteria. Interventions:The patient was treated with granulocyte colony-stimulating factor and 500 mg methylprednisolone.Outcomes: A significant improvement was observed in the patient's condition; the abnormal laboratory results resolved gradually, and the patient was discharged.Lessons: This case serves to create awareness among clinicians that HLH is a rare complication of COVID-19 vaccination and should be considered, especially in patients with a history of chronic renal failure and hypertension.

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Kikuchi-Fujimoto disease associated with hemophagocytic lymphohistiocytosis following the BNT162b2 mRNA COVID-19 vaccination

Abstract: Not available.

Cited by 21 publications

References 12 publications

The First Case of Haemophagocytic Lymphohistiocytosis Triggered by the Booster Dose of Anti-SARS-CoV-2 Vaccine in a Patient with β-Thalassemia

The First Case of Haemophagocytic Lymphohistiocytosis Triggered by the Booster Dose of Anti-SARS-CoV-2 Vaccine in a Patient with β-Thalassemia

A Transient Inflammatory Reaction with a B-Cell Lymphoid Infiltrate and Dysplastic Bone Marrow Changes Associated with the BNT162b2 COVID-19 Vaccine

A case of hemophagocytic lymphohistiocytosis after BNT162b2 COVID-19 (Comirnaty®) vaccination

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