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Spontaneous regression of multiple solitary plasmacytoma harboring Epstein–Barr virus: a case report and literature review

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Abstract

We report a rare case of spontaneous regression (SR) in an elderly untreated patient with multiple solitary plasmacytoma (MSP). Diagnosis of MSP was confirmed through surgical resection of the left nasal cavity mass and subsequent biopsy of the right humerus. The patient was considered ineligible for chemotherapy due to poor performance status. At 3-month post-diagnosis, the patient’s condition worsened with deteriorating bone lesions and emergence of a new serum monoclonal protein. However, these clinical findings completely disappeared at 6 months, and positron emission tomography–computed tomography at 1 year confirmed complete metabolic remission. Notably, peripheral blood lymphocyte counts were inversely correlated with tumor progression and remission. Pathological re-evaluation of the initial biopsy specimens revealed programmed cell death protein 1 (PD-1) expression in tumor-infiltrating CD8+ T cells. In addition, tumor cells were infected with Epstein–Barr virus (EBV) but were negative for programmed cell death ligand 1 (PD-L1) expression, which is the most potent immune escape mechanism in tumor cells. While the mechanism underlying SR remains unclear, our findings suggest that host immune response as well as EBV infection may contribute to SR. Further studies are needed to elucidate the clinicopathologic mechanisms of tumor regression in plasma cell neoplasms.

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The data are available from the corresponding author upon reasonable request.

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Acknowledgements

The manuscript was edited and proofread by Editage (https://www.editage.jp/).

Author information

Authors and Affiliations

  1. Department of Hematology, National Hospital Organization Iwakuni Clinical Center, 1-1-1, Atago-Cho, Iwakuni, 740-8510, Japan

    Wataru Kitamura

  2. Department of Hematology, Oncology and Respiratory Medicine, Dentistry and Pharmaceutical Sciences, Okayama University Graduate School of Medicine, 2-5-1, Shikata-Cho, Kita-Ku, Okayama, 700-8558, Japan

    Wataru Kitamura, Hiroki Kobayashi & Yoshinobu Maeda

  3. Department of Otorhinolaryngology, National Hospital Organization Iwakuni Clinical Center, 1-1-1, Atago-Cho, Iwakuni, 740-8510, Japan

    Minori Noda

  4. Department of Pathology, National Hospital Organization Iwakuni Clinical Center, 1-1-1, Atago-Cho, Iwakuni, 740-8510, Japan

    Akiko Iseki & Yumi Sato

  5. Department of Respiratory Medicine, National Hospital Organization Iwakuni Clinical Center, 1-1-1, Atago-Cho, Iwakuni, 740-8510, Japan

    Shoichi Kuyama

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  1. Wataru Kitamura
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Contributions

W.K. collected clinical and histological data and wrote the original draft. H.K. and M.N. performed review and editing. A.I. and Y.K. performed histological diagnosis. Y.M. and S.K. performed supervision, review, and editing. All authors approved the final manuscript.

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Correspondence to Wataru Kitamura.

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Kitamura, W., Kobayashi, H., Noda, M. et al. Spontaneous regression of multiple solitary plasmacytoma harboring Epstein–Barr virus: a case report and literature review. Int J Hematol (2024). https://doi.org/10.1007/s12185-024-03765-5

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  • DOI: https://doi.org/10.1007/s12185-024-03765-5

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