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Three coexisting lymphomas in a single patient: composite lymphoma derived from a common germinal center B-cell precursor and unrelated discordant lymphoma

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Abstract

Composite lymphoma (CL) is a rare disorder defined as the coexistence of two or more distinct lymphoma subtypes at a single anatomic site. Discordant lymphoma (DL), which is the simultaneous occurrence of two or more distinct lymphoma subtypes at different sites, is also rare. CL complicated with DL involving three distinct subtypes of lymphoma in the same patient is an extremely rare disease. Clonal relationships in CL and DL are commonly investigated by molecular analysis using mutational status with t(14;18)BCL2/IgH translocation and immunoglobulin heavy chain variable-region (IgVH) gene rearrangement. A 73-year-old woman was admitted to our hospital with systemic lymphadenopathy and was initially diagnosed with diffuse large B-cell lymphoma based on pathological features of the biopsied esophageal tumor. However, the results of inguinal lymph node biopsy led to a revised pathological diagnosis CL consisting of Hodgkin lymphoma and follicular lymphoma. Three distinct coexisting lymphomas were identified in this individual patient. Molecular analysis revealed CL derived from common germinal center B-cell precursors, while clonal relationship between CL and DL was not clarified. This case suggests a mechanism underlying B-cell lymphoma pathogenesis involving two pivotal somatic mutations, t(14;18)BCL2/IgH translocation and IgVH rearrangement.

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References

  1. Küppers R, Dührsen U, Hansmann M-L. Pathogenesis, diagnosis, and treatment of composite lymphomas. Lancet Oncol. 2014;15:e435–46.

    Article  PubMed  CAS  Google Scholar 

  2. Kim H, Dorfman RF. Morphological studies of 84 untreated patients subjected to laparotomy for the staging of non-Hodgkin’s lymphomas. Cancer. 1974;33:657–74.

    Article  PubMed  CAS  Google Scholar 

  3. Bräuninger A, Hansmann ML, Strickler JG, Dummer R, Burg G, Rajewsky K, et al. Identification of common germinal-center B-cell precursors in two patients with both Hodgkin’s disease and non-Hodgkin’s lymphoma. N Engl J Med. 1999;340:1239–47.

    Article  PubMed  Google Scholar 

  4. Schneider S, Crescenzi B, Schneider M, Ascani S, Hartmann S, Hansmann ML, et al. Subclonal evolution of a classical Hodgkin lymphoma from a germinal center B-cell-derived mantle cell lymphoma. Int J Cancer. 2014;134:832–43.

    Article  PubMed  CAS  Google Scholar 

  5. Schmitz R, Renné C, Rosenquist R, Tinguely M, Distler V, Menestrina F, et al. Insights into the multistep transformation process of lymphomas: IgH-associated translocations and tumor suppressor gene mutations in clonally related composite Hodgkin’s and non-Hodgkin’s lymphomas. Leukemia. 2005;19:1452–8.

    Article  PubMed  CAS  Google Scholar 

  6. Ramasamy I, Brisco M, Morley A. Improved PCR method for detecting monoclonal immunoglobulin heavy chain rearrangement in B cell neoplasms. J Clin Pathol. 1992;45:770–5.

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  7. Reed TJ, Reid A, Wallberg K, O’Leary TJ, Frizzera G. Determination of B-cell clonality in paraffin-embedded lymph nodes using the polymerase chain reaction. Diagn Mol Pathol. 1993;2:42–9.

    Article  PubMed  CAS  Google Scholar 

  8. Delabie J, Tierens A, Wu G, Weisenburger DD, Chan WC. Lymphocyte predominance Hodgkin’s disease: lineage and clonality determination using a single-cell assay. Blood. 1994;84:3291–8.

    PubMed  CAS  Google Scholar 

  9. Nakamura N, Ohshima K, Abe M, Osamura Y. Demonstration of chimeric DNA of bcl-2 and immunoglobulin heavy chain in follicular lymphoma and subsequent Hodgkin lymphoma from the same patient. J Clin Exp Hematopathol. 2007;47:9–13.

    Article  CAS  Google Scholar 

  10. Marafioti T, Hummel M, Anagnostopoulos I, Foss HD, Huhn D, Stein H. Classical Hodgkin’s disease and follicular lymphoma originating from the same germinal center B cell. J Clin Oncol. 1999;17:3804–9.

    Article  PubMed  CAS  Google Scholar 

  11. Hirt C, Dölken G, Janz S, Rabkin CS. Distribution of t(14;18)-positive, putative lymphoma precursor cells among B-cell subsets in healthy individuals1. Br J Haematol. 2007;138:349–53.

    Article  PubMed  Google Scholar 

  12. Galteland E, Sivertsen EA, Svendsrud DH, Smedshammer L, Kresse SH, Meza-Zepeda LA, et al. Translocation t(14;18) and gain of chromosome 18/BCL2: effects on BCL2 expression and apoptosis in B-cell non-Hodgkin’s lymphomas. Leukemia. 2005;19:2313–23.

    Article  PubMed  CAS  Google Scholar 

  13. Schüler F, Hirt C, Dölken G. Chromosomal translocation t(14;18) in healthy individuals. Semin Cancer Biol. 2003;13:203–9.

    Article  PubMed  CAS  Google Scholar 

  14. Green MR, Gentles AJ, Nair RV, Irish JM, Kihira S, Liu CL, et al. Hierarchy in somatic mutations arising during genomic evolution and progression of follicular lymphoma. Blood. 2013;121:1604–11.

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  15. Lohr JG, Stojanov P, Lawrence MS, Auclair D, Chapuy B, Sougnez C, et al. Discovery and prioritization of somatic mutations in diffuse large B-cell lymphoma (DLBCL) by whole-exome sequencing. Proc Natl Acad Sci USA. 2012;109:3879–84.

    Article  PubMed  PubMed Central  Google Scholar 

  16. Mani H, Jaffe ES. Hodgkin lymphoma: an update on its biology with new insights into classification. Clin Lymphoma Myeloma. 2009;9:206–16.

    Article  PubMed  PubMed Central  Google Scholar 

  17. Nishikori M, Uchiyama T. Molecular pathogenesis of Hodgkin lymphoma. Int J Hematol. 2006;83:398–403.

    Article  PubMed  CAS  Google Scholar 

  18. Brudno J, Tadmor T, Pittaluga S, Nicolae A, Polliack A, Dunleavy K. Discordant bone marrow involvement in non-Hodgkin lymphoma. Blood. 2016;127:965–70.

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  19. Park J, Lee JE, Kim M, Lim J, Kim Y, Han K, et al. Discordant lymphocyte-depleted classical Hodgkin’s and peripheral T-cell lymphoma arising in a patient 11 years after diagnosis of multicentric Castleman’s disease. Int J Hematol. 2013;98:114–21.

    Article  PubMed  Google Scholar 

  20. Brown JR, Weng AP, Freedman AS. Hodgkin disease associated with T-cell non-Hodgkin lymphomas: case reports and review of the literature. Am J Clin Pathol. 2004;121:701–8.

    Article  PubMed  Google Scholar 

  21. Zhang C, Yi Y, Chen C, Wang J, Liu Z. Discordant lymphoma consisting of mediastinal large B-cell lymphoma and nodular sclerosis Hodgkin lymphoma in the right supraclavicular lymph nodes: a case report. Diagn Pathol. 2015;10:215.

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  22. Kremer M, Spitzer M, Mandl-Weber S, Stecker K, Schmidt B, Höfler H, et al. Discordant bone marrow involvement in diffuse large B-cell lymphoma: comparative molecular analysis reveals a heterogeneous group of disorders. Lab Investig. 2003;83:107–14.

    Article  PubMed  CAS  Google Scholar 

  23. Huang JZ, Sanger WG, Greiner TC, Staudt LM, Weisenburger DD, Pickering DL, et al. The t(14;18) defines a unique subset of diffuse large B-cell lymphoma with a germinal center B-cell gene expression profile. Blood. 2002;99:2285–90.

    Article  PubMed  CAS  Google Scholar 

  24. Chan WJC. Pathogenesis of diffuse large B cell lymphoma. Int J Hematol. 2010;92:219–30.

    Article  PubMed  CAS  Google Scholar 

  25. Barrans SL, Evans PAS, O’Connor SJM, Kendall SJ, Owen RG, Haynes AP, et al. The t(14;18) is associated with germinal center-derived diffuse large B-cell lymphoma and is a strong predictor of outcome. Clin Cancer Res. 2003;9:2133–9.

    PubMed  CAS  Google Scholar 

  26. Casulo C, Burack WR, Friedberg JW. Transformed follicular non-Hodgkin lymphoma. Blood. 2015;125:40–7.

    Article  PubMed  CAS  Google Scholar 

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Acknowledgements

The authors greatly thank the members of the Department of Pathology, Kurume University, School of Medicine, for pathological analyses.

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Authors and Affiliations

  1. Department of Hematology, Community Health Care Organization (JCHO) Saga Central Hospital, Saga, Japan

    Atsujiro Nishioka

  2. Division of Hematology, Respiratory Medicine and Oncology, Department of Internal Medicine, Faculty of Medicine, Saga University, 5-1-1 Nabeshima, Saga, 849-8501, Japan

    Atsujiro Nishioka, Hiroshi Ureshino, Toshihiko Ando, Haruna Kizuka, Kana Kusaba, Haruhiko Sano, Hidekazu Itamura, Yasushi Kubota, Kensuke Kojima & Shinya Kimura

  3. Department of Pathology, School of Medicine, Kurume University, Kurume, Japan

    Koichi Ohshima

Authors
  1. Atsujiro Nishioka
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  2. Hiroshi Ureshino
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Corresponding author

Correspondence to Hiroshi Ureshino.

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The authors declare no potential conflicts of interest (COI).

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All authors contributed to patient care and wrote the report. Informed consent was obtained from the patient for publication of this case report.

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12185_2017_2370_MOESM1_ESM.pdf

Computed tomographic findings. No enlarged lymph node was detected. A, Neck; B, Chest; C, Abdomen; D, Inguinal (PDF 277 kb)

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Nishioka, A., Ureshino, H., Ando, T. et al. Three coexisting lymphomas in a single patient: composite lymphoma derived from a common germinal center B-cell precursor and unrelated discordant lymphoma. Int J Hematol 107, 703–708 (2018). https://doi.org/10.1007/s12185-017-2370-6

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  • DOI: https://doi.org/10.1007/s12185-017-2370-6

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