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Leukemic and meningeal relapse of CD5+ intravascular large B-cell lymphoma with down-modulation of CD20 after rituximab therapy

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Abstract

Although CD20- relapses of B-cell lymphoma following rituximab therapy have increasingly been reported recently, coexistence of both the original and selected clones on relapse in a single patient have not been described. We experienced such a case with rare CD5+ intravascular lymphomatosis (IVL). A 46-year-old woman was admitted because of IVL complicated with cauda equina syndrome and pulmonary infarction. Complete remission was successfully achieved with multidrug chemotherapy in combination with rituximab. However, the disease recurred after 8 months with leukemic progression and meningeal involvement. The phenotype of the abnormal lymphocytes in the peripheral blood was fundamentally the same (CD20+CD5+CD10-CD19+CD23-sIgλ+) as that of the cells in the cerebrospinal fluid (CSF). However, CD20 expression was decreased remarkably compared with that in the CSF and that in the bone marrow before therapy. The targeting of CD20 molecules on the tumor cell surface by rituximab may have provided a selective pressure on lymphoma cells. The escape phenomenon of the lymphoma cells from rituximab was observed by simultaneously comparing the CD20 expression of cells in the peripheral blood and in a site of sanctuary from rituximab, the CSF.

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References

  1. Wick MR, Mills SE. Intravascular lymphomatosis: clinicopathologic features and differential diagnosis. Semin Diagn Pathol. 1991;8:91–101.

    CAS  PubMed  Google Scholar 

  2. Glass J, Hochberg FH, Miller DC. Intravascular lymphomatosis: a systemic disease with neurologic manifestations. Cancer. 1993;71:3156–3164.

    Article  CAS  PubMed  Google Scholar 

  3. DiGiuseppe JA, Nelson WG, Seifter EJ, Boitnott JK, Mann RB. Intravascular lymphomatosis: a clinicopathologic study of 10 cases and assessment of response to chemotherapy. J Clin Oncol. 1994;12:2573–2579.

    CAS  PubMed  Google Scholar 

  4. Yamaguchi M, Ohno T, Oka K, et al. De novo CD5+ diffuse large B-cell lymphoma: clinicopathologic study of 109 patients. Blood. 2002;99:815–821.

    Article  CAS  PubMed  Google Scholar 

  5. Murase T, Nakamura S, Kawaguchi, et al. An Asian variant of intravascular large B-cell lymphoma: clinical, pathological and cytogenetic approaches to diffuse large B-cell lymphoma associated with hemophagocytic syndrome. Br J Haematol. 2000;111:826–834.

    CAS  PubMed  Google Scholar 

  6. Kennedy GA, Tey SK, Cobcroft R, et al. Incidence and nature of CD20-negative relapses following rituximab therapy in aggressive B-cell non-Hodgkin’s lymphoma: a retrospective review. Br J Haematol. 2002;119:412–416.

    Article  CAS  PubMed  Google Scholar 

  7. Chu PG, Chen YY, Molina A, Arber DA, Weiss LM. Recurrent B-cell neoplasms after rituximab therapy: an immunophenotypic and genotypic study. Leuk Lymphoma. 2002;43:2335–2341.

    Article  CAS  PubMed  Google Scholar 

  8. Diss TC, Peng H, Wotherspoon AC, Isaacson PG, Pan L. Detection of monoclonality in low-grade B-cell lymphomas using the polymerase chain reaction is dependent on primer selection and lymphoma type. J Pathol. 1993;169:291–295.

    Article  CAS  PubMed  Google Scholar 

  9. Ohno T, Miyake N, Hada S, et al. Hemophagocytic syndrome in five patients with Epstein-Barr virus-negative B-cell lymphoma. Cancer. 1998;82:1963–1972.

    Article  CAS  PubMed  Google Scholar 

  10. Ito M, Kim Y, Choi JW, Ozawa H, Fujino M. Prevalence of intravascular B-cell lymphoma with bone marrow involvement at initial presentation. Int J Hematol. 2003;77:159–163.

    Article  PubMed  Google Scholar 

  11. Ohno T, Ueda Y, Nagai K, et al. The serum cytokine profiles of lymphoma-associated hemophagocytic syndrome: a comparative analysis of B-cell and T-cell/natural killer cell lymphomas. Int J Hematol. 2003;77:286–294.

    Article  CAS  PubMed  Google Scholar 

  12. Khoury H, Dalal BI, Nantel SH. Intravascular lymphoma presenting with bone marrow involvement and leukemic phase. Leuk Lymphoma. 2003;44:1043–1047.

    Article  CAS  PubMed  Google Scholar 

  13. Ponzoni M, Arrigoni G, Gould VE, et al. Lack of CD29 (β1 integrin) and CD54 (ICAM-1) adhesion molecules in intravascular lymphomatosis. Hum Pathol. 2000;31:220–226.

    Article  CAS  PubMed  Google Scholar 

  14. Kobayashi T, Yamaguchi T, Kim S, et al. Microarray reveals differences in both tumors and vascular specific gene expression in de novo CD5+ and CD5-diffuse large B-cell lymphomas. Cancer Res. 2003;63:60–66.

    CAS  PubMed  Google Scholar 

  15. Carter DK, Batts KP, de Groen PC, Kurtin PJ. Angiotropic large cell lymphoma (intravascular lymphomatosis) occurring after follicular small cleaved cell lymphoma. Mayo Clin Proc. 1996;71:869–873.

    Article  CAS  PubMed  Google Scholar 

  16. Berinstein NL, Grillo-López AJ, White CA, et al. Association of serum rituximab (IDEC-C2B8) concentration and anti-tumor response in the treatment of recurrent low-grade or follicular non-Hodgkin’s lymphoma. Ann Oncol. 1998;9:995–1001.

    Article  CAS  PubMed  Google Scholar 

  17. Igarashi T, Kobayashi Y, Ogura M, et al. Factors affecting toxicity, response, and progression-free survival in relapsed patients with indolent B-cell lymphoma and mantle cell lymphoma treated with rituximab: a Japanese phase II study. Ann Oncol. 2002;13:928–943.

    Article  CAS  PubMed  Google Scholar 

  18. Hariunpaa A, Wiklund T, Collan J, et al. Complement activation in circulation and central nervous system after rituximab (anti-CD20) treatment of B-cell lymphoma. Leuk Lymphoma. 2001;42:731–738.

    Article  Google Scholar 

  19. McLaughlin P. Rituximab: perspective on single agent experience, and future directions in combination trials. Crit Rev Oncol Hematol. 2001;40:3–16.

    Article  CAS  PubMed  Google Scholar 

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Correspondence to Tatsuharu Ohno.

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Ohno, T., Sakamoto, T., Mizumoto, C. et al. Leukemic and meningeal relapse of CD5+ intravascular large B-cell lymphoma with down-modulation of CD20 after rituximab therapy. Int J Hematol 84, 74–78 (2006). https://doi.org/10.1532/IJH97.06033

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  • DOI: https://doi.org/10.1532/IJH97.06033

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