Two cases of angioimmunoblastic T-cell lymphoma with concomitant positive serology for acute Epstein-Barr virus infection

Facchinelli, Davide; Polino, A.; Dima, Francesco; Parisi, Alice; Ambrosetti, Achille; Veneri, Dino

doi:10.4081/hr.2017.7088

Cited by 6 publications

(9 citation statements)

References 16 publications

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“…6,11 Few data are available regarding the anti-EBV serology status and clinical outcome. 9,10 Here, although in a small cohort of patients, we show that a triple-positive anti-EBV serology is very frequent (77.7%) at the diagnosis of AITL. This finding does not allow us to differentiate a late primary infection from a viral reactivation; serology prior to the development of lymphoma is not available.…”

Section: Discussionmentioning

confidence: 53%

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Is triple-positive serology for Epstein-Barr virus (VCA-IgG, VCA-IgM, EBNA-IgG) a specific feature of angioimmunoblastic T-cell lymphoma?

Facchinelli

Parisi

Krampera

et al. 2019

Tumori

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Purpose: We assessed the frequency of triple-positive serology (viral capsid antigen [VCA]–immunoglobulin G [IgG], VCA–immunoglobulin M, Epstein-Barr nuclear antigen–IgG) for Epstein-Barr virus (EBV) in a small number of patients with angioimmunoblastic T-cell lymphoma (AITL) at disease onset. Methods: Nine patients with newly diagnosed AITL were retrospectively enrolled in the present study. For all of them, EBV serology data were available. Results: Of 9 patients, 7 (77.7%) had a triple-positive serology (VCA-IgG, VCA-IgM, EBNA-IgG ) for EBV. These patients were characterized by bone marrow involvement, high incidence of thrombocytopenia, and poor prognosis according to Revised International Prognostic Index and Prognostic Index for Angioimmunoblastic T-cell Lymphoma scores. Conclusion: Assessment of both viremia and serology for EBV could be useful in patients with clinical and laboratory data suggesting lymphoma diagnosis; furthermore, although our data need to be validated in a larger cohort of patients, triple positivity for EBV serology might help to direct the diagnosis toward AITL.

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Section: Discussionmentioning

confidence: 53%

“…In the literature, only 3 AITL cases with positive serology for anti-EBV IgM at disease onset are described. 9,10 In this study, we analyze EBV serology retrospectively in a series of patients with AITL at disease onset.…”

Section: Introductionmentioning

confidence: 99%

Is triple-positive serology for Epstein-Barr virus (VCA-IgG, VCA-IgM, EBNA-IgG) a specific feature of angioimmunoblastic T-cell lymphoma?

Facchinelli

Parisi

Krampera

et al. 2019

Tumori

Self Cite

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“…This study was carried out on an EBV-seropositive patient who, after chemotherapy and allo-SCT to treat a CD4 + AITL, suffered from relapse and simultaneous increase of EBV load, probably connected to reemergence of malignancy. High EBV viremia is associated with AITL progression, which suggests a possible role in its pathogenesis [2] and increased aggressiveness [4]. Despite DLI and Rituximab treatment, adoptive transfer of T cells enriched for EBV epitopes was provided due to increasing levels of EBV viral DNA in blood and lack of EBV peptide-MHC multimer-binding T cells for six known HLArelevant epitopes.…”

Section: Discussionmentioning

confidence: 99%

Reconstitution of T cell immunity against EBV in the immunocompromised host by adoptive transfer of peptide-stimulated T cells after allogeneic stem cell transplantation

Cobo

Ritter

Gary

et al. 2021

Preprint

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Reconstitution of T cell repertoire after allogeneic stem cell transplantation is a long and often incomplete process. As a result, reactivation of Epstein-Barr virus (EBV) is a frequent complication that may be treated by adoptive transfer of donor-derived EBV-specific T cells. We generated donor-derived EBV-specific T cells by peptide stimulation and adoptively transferred them to a patient with angioimmunoblastic T-cell lymphoma (AITL), who had developed persisting high titers of EBV concomitant to relapse after transplantation. T cell receptor beta (TCRβ) deep sequencing showed that the T cell repertoire of the patient early after transplantation (day 60) was strongly reduced and only very low numbers of EBV-specific T cells were detectable. Manufacturing and in vitro expansion of donor-derived EBV-specific T cells resulted in enrichment of EBV epitope-specific, HLA-restricted T cells. Monitoring after adoptive transfer revealed that the dominant TCR sequences from peptide-stimulated T cells persisted long-term and established an EBV-specific TCR clonotype repertoire in the host, with many of the EBV-specific TCRs present in the donor. This reconstituted repertoire was associated with immunological control of EBV and with lack of further AITL relapse.Author summaryA characteristic feature of all herpesviruses is their persistence in the host’s body after primary infection. Hence, the host’s immune system is confronted with the problem to control these viruses life-long. Well-known representative of the herpesvirus group are the classic Herpes-Simplex Virus (HSV-1) and Varicella Zoster Virus (VZV, causing chicken pox); a less known representative is Epstein Barr Virus (EBV, causing mononucleosis). When the immune system is severely compromised, for example after stem cell transplantation from a foreign (allogeneic) donor, these viruses can reappear, as they are already in the host’s body. Especially EBV cause life-threatening complications after stem cell transplantation and only reinforcement of the host’s immune system can reestablish viral control. Here we show that ex vivo manufactured EBV-specific T cells can reestablish long-term control of EBV and that these cells persist in the host’s body over months. These results give us a better understanding of viral immune reconstitution post-transplant and of clinically-relevant T cell populations against EBV.

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“…Our patient is the fourth reported, and as far as we can tell, the first reported presenting with a gastric mass. Several theories exist regarding the role of EBV in the pathogenesis of AITL 1,3,4 . A large prospective study showed that circulating EBV DNA is tied to circulating AITL cells.…”

Section: Discussion/conclusionmentioning

confidence: 99%

Hiding in plain sight: Gastric mass as a herald of angioimmunoblastic T‐cell lymphoma

et al. 2020

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Background/study aims Angioimmunoblastic T‐cell Lymphoma (AITL) is a rare T‐cell lymphoma often presenting with constitutional symptoms and lymphadenopathy. It can have an association with Epstein‐Barr Virus (EBV) infection and auto‐immune phenomena such as haemolytic anaemia. This case details a Gastric Mass as the initial findings of AITL, a rare occurrence. Patients and methods Case report detailing a 64‐year‐old female presenting with a suspicious gastric mass and auto‐immune haemolytic anaemia. Hospital workup was notable for serology consistent with acute EBV infection and positive anti‐smooth muscle antibody. Tissue biopsy and lymph node resection yielded histologic diagnosis of AITL. Results Once the patient was diagnosed with AITL, they were placed on Gemcitabine, Cisplatin, Prednisone and Thalidomide with subsequent improvement in haemolytic anaemia and gastric mass size. Conclusions This case demonstrates a unique presentation of Angioimmunoblastic T‐cell Lymphoma in the context of a Gastric Mass and associated Auto‐immune Haemolytic Anaemia. This case also continues to show evidence of a strong relationship between Epstein‐Barr Virus and Angioimmunoblastic T‐cell Lymphoma.

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Two cases of angioimmunoblastic T-cell lymphoma with concomitant positive serology for acute Epstein-Barr virus infection

Cited by 6 publications

References 16 publications

Is triple-positive serology for Epstein-Barr virus (VCA-IgG, VCA-IgM, EBNA-IgG) a specific feature of angioimmunoblastic T-cell lymphoma?

Is triple-positive serology for Epstein-Barr virus (VCA-IgG, VCA-IgM, EBNA-IgG) a specific feature of angioimmunoblastic T-cell lymphoma?

Reconstitution of T cell immunity against EBV in the immunocompromised host by adoptive transfer of peptide-stimulated T cells after allogeneic stem cell transplantation

Hiding in plain sight: Gastric mass as a herald of angioimmunoblastic T‐cell lymphoma

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