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Multiple primary enoral soft tissue manifestations of a Hodgkin lymphoma—case report and literature review

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Abstract

Background

Hodgkin lymphoma (HL) are lymphoproliferative neoplasms, histologically comprising of mononuclear and multinucleated Hodgkin and Reed Sternberg cells (HRS). About 4 % of all lymphatic malignancies of the head and neck are HL. The typical disease presents itself as a nodal lesion. Extranodal, enoral soft tissue involvement by HL is very rare.

Case report

A 73-year-old man with a suspect, ulcerating lesion in the left retromolar region of the mandible was assigned to our hospital. Prior anti-inflammatory therapy has been without success. Subsequently, three biopsies were taken which could only show inflammation. Finally, two biopsies from the left retromolar region and the left inner cheek showed HRS cells with positive expressions of CD15 and CD30 corresponding to a Hodgkin lymphoma. No lymphatic node or bone involvement could be detected. The patient was designated to receive radio-chemotherapy, but died 3 weeks after diagnosis of multiple organ failure. In a literature review, together with this report, nine cases were found concerning primary HL of the oral mucosa. Accordingly, this is the first case of primary multiple extranodal HL in the oral mucosa in absence of lymphatic node involvement.

Discussion

Neither clinical features nor radiological appearances of HL presenting as primary enoral lesions are pathognomonic. Especially when only small biopsy specimens are available, histological diagnosis remains challenging, may lead to a delay in therapy and may result in a significant worse prognosis.

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References

  1. DePena CA, Van Tassel P, Lee YY (1990) Lymphoma of the head and neck. Radiol Clin North Am 28(4):723–743

    PubMed  CAS  Google Scholar 

  2. Urquhart A, Berg R (2001) Hodgkin’s and non-Hodgkin’s lymphoma of the head and neck. Laryngoscope 111(9):1565–1569

    Article  PubMed  CAS  Google Scholar 

  3. Kuppers R, Rajewsky K (1998) The origin of Hodgkin and Reed/Sternberg cells in Hodgkin’s disease. Annu Rev Immunol 16:471–493

    Article  PubMed  CAS  Google Scholar 

  4. Harris NL (1999) Hodgkin’s disease: classification and differential diagnosis. Mod Pathol 12(2):159–175

    PubMed  CAS  Google Scholar 

  5. Brauninger A, Schmitz R, Bechtel D, Renne C, Hansmann ML, Kuppers R (2006) Molecular biology of Hodgkin’s and Reed/Sternberg cells in Hodgkin’s lymphoma. Int J Cancer 118(8):1853–1861. doi:10.1002/ijc.21716

    Article  PubMed  Google Scholar 

  6. Kuppers R, Zhao M, Hansmann ML, Rajewsky K (1993) Tracing B cell development in human germinal centres by molecular analysis of single cells picked from histological sections. Embo J 12(13):4955–4967

    PubMed  CAS  Google Scholar 

  7. Ansell SM, Armitage JO (2006) Management of Hodgkin lymphoma. Mayo Clin Proc 81(3):419–426

    Article  PubMed  Google Scholar 

  8. Knight JS, Sharma N, Robertson ES (2005) Epstein–Barr virus latent antigen 3C can mediate the degradation of the retinoblastoma protein through an SCF cellular ubiquitin ligase. Proc Natl Acad Sci U S A 102(51):18562–18566. doi:10.1073/pnas.0503886102

    Article  PubMed  CAS  Google Scholar 

  9. Harris NL, Stein H, Coupland SE, Hummel M, Favera RD, Pasqualucci L, Chan WC (2001) New approaches to lymphoma diagnosis. Hematology Am Soc Hematol Educ Program:194–220

  10. Harris NL, Jaffe ES, Diebold J, Flandrin G, Muller-Hermelink HK, Vardiman J, Lister TA, Bloomfield CD (2000) The World Health Organization classification of hematological malignancies report of the Clinical Advisory Committee Meeting, Airlie House, Virginia, November 1997. Mod Pathol 13(2):193–207

    Article  PubMed  CAS  Google Scholar 

  11. Quinones-Avila Mdel P, Gonzalez-Longoria AA, Admirand JH, Medeiros LJ (2005) Hodgkin lymphoma involving Waldeyer ring: a clinicopathologic study of 22 cases. Am J Clin Pathol 123(5):651–656

    Article  PubMed  Google Scholar 

  12. Cionini L, Bastiani P, Biti GP, Mungai V, Ponticelli P, Di Lollo S (1985) Waldeyer’s ring (WR) involvement in Hodgkin’s disease. Radiother Oncol 3(4):299–302

    Article  PubMed  CAS  Google Scholar 

  13. Baden E, Al Saati T, Caveriviere P, Gorguet B, Delsol G (1987) Hodgkin’s lymphoma of the oropharyngeal region: report of four cases and diagnostic value of monoclonal antibodies in detecting antigens associated with Reed-Sternberg cells. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 64(1):88–94

    CAS  Google Scholar 

  14. Whitt JC, Dunlap CL, Martin KF (2007) Oral Hodgkin lymphoma: a wolf in wolf’s clothing. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 104(5):e45–e51

    Article  PubMed  Google Scholar 

  15. Pileri SA, Ascani S, Leoncini L, Sabattini E, Zinzani PL, Piccaluga PP, Pileri A Jr, Giunti M, Falini B, Bolis GB, Stein H (2002) Hodgkin’s lymphoma: the pathologist’s viewpoint. J Clin Pathol 55(3):162–176

    Article  PubMed  CAS  Google Scholar 

  16. DeVita VTJ, Hubbard SM (1993) Hodgkin’s disease. N Engl J Med 328(8):560–565

    Article  PubMed  Google Scholar 

  17. Quinones-Avila Mdel P, Gonzalez-Longoria AA, Admirand JH, Medeiros LJ (2005) Hodgkin lymphoma involving Waldeyer ring: a clinicopathologic study of 22 cases. Am J Clin Pathol 123(5):651–656

    Article  PubMed  Google Scholar 

  18. Kapadia SB, Roman LN, Kingma DW, Jaffe ES, Frizzera G (1995) Hodgkin’s disease of Waldeyer’s ring. Clinical and histoimmunophenotypic findings and association with Epstein–Barr virus in 16 cases. Am J Surg Pathol 19(12):1431–1439

    Article  PubMed  CAS  Google Scholar 

  19. Papo T, Oksenhendler E, Bouvet E, Marche C, Monteil JP, Clauvel JP (1991) Hodgkin’s disease of the tongue in a homosexual HIV-infected patient. Am J Hematol 37(2):143

    Article  PubMed  CAS  Google Scholar 

  20. Jamshed A, Allard WF, Mourad WA, Rostom AY (1997) Primary Hodgkin’s disease of the mandible: a case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 83(6):680–684

    Article  PubMed  CAS  Google Scholar 

  21. Rosenberg SA, Kaplan HS (1966) Evidence for an orderly progression in the spread of Hodgkin’s disease. Cancer Res 26(6):1225–1231

    PubMed  CAS  Google Scholar 

  22. Pileri SA, Dirnhofer S, Went P, Ascani S, Sabattini E, Marafioti T, Tzankov A, Leoncini L, Falini B, Zinzani PL (2002) Diffuse large B-cell lymphoma: one or more entities? Present controversies and possible tools for its subclassification. Histopathology 41(6):482–509

    Article  PubMed  CAS  Google Scholar 

  23. Pileri SA, Ascani S, Leoncini L, Sabattini E, Zinzani PL, Piccaluga PP, Pileri A Jr, Giunti M, Falini B, Bolis GB, Stein H (2002) Hodgkin’s lymphoma: the pathologist’s viewpoint. Journal of clinical pathology 55(3):162–176

    Article  PubMed  CAS  Google Scholar 

  24. Rudiger T, Jaffe ES, Delsol G, deWolf-Peeters C, Gascoyne RD, Georgii A, Harris NL, Kadin ME, MacLennan KA, Poppema S, Stein H, Weiss LE, Muller-Hermelink HK (1998) Workshop report on Hodgkin’s disease and related diseases (‘grey zone’ lymphoma). Ann Oncol 9(Suppl 5):S31–S38

    Article  PubMed  Google Scholar 

  25. Kapatai G, Murray P (2007) Contribution of the Epstein Barr virus to the molecular pathogenesis of Hodgkin lymphoma. J Clin Pathol 60(12):1342–1349

    Article  PubMed  CAS  Google Scholar 

  26. Hjalgrim H, Smedby KE, Rostgaard K, Molin D, Hamilton-Dutoit S, Chang ET, Ralfkiaer E, Sundstrom C, Adami HO, Glimelius B, Melbye M (2007) Infectious mononucleosis, childhood social environment, and risk of Hodgkin lymphoma. Cancer Res 67(5):2382–2388

    Article  PubMed  CAS  Google Scholar 

  27. Kuppers R, Schwering I, Brauninger A, Rajewsky K, Hansmann ML (2002) Biology of Hodgkin’s lymphoma. Ann Oncol 13(Suppl 1):11–18

    Article  PubMed  Google Scholar 

  28. Weinreb M, Day PJ, Niggli F, Green EK, Nyong’o AO, Othieno-Abinya NA, Riyat MS, Raafat F, Mann JR (1996) The consistent association between Epstein-Barr virus and Hodgkin’s disease in children in Kenya. Blood 87(9):3828–3836

    PubMed  CAS  Google Scholar 

  29. Advani RH, Horning SJ (1999) Treatment of early-stage Hodgkin’s disease. Semin Hematol 36(3):270–281

    PubMed  CAS  Google Scholar 

  30. Rowley H, McRae RD, Cook JA, Helliwell TR, Husband D, Jones AS (1995) Lymphoma presenting to a head and neck clinic. Clin Otolaryngol Allied Sci 20(2):139–144

    Article  PubMed  CAS  Google Scholar 

  31. Diehl V, Josting A (2000) Hodgkin’s disease. Cancer J 6(Suppl 2):S150–S158

    PubMed  Google Scholar 

  32. Halbsguth TV, Böll B, Borchmann P, Diehl V (2011) The unique characteristics and management of patients over 60 years of age with classic Hodgkin lymphoma. Curr Hematol Malig Rep 6(3):164–171

    Article  PubMed  Google Scholar 

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Conflict of Interest

The authors declare that they have no conflict of interest.

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Authors and Affiliations

  1. Department of Oral and Maxillofacial and Plastic Surgery, University Medical Centre of the Johannes Gutenberg-University, Augustusplatz 2, Mainz, 55131, Germany

    P. W. Kämmerer, E. Schiegnitz, G. F. Draenert, H. D. Kuffner & M. O. Klein

  2. Department of Pathology, University Medical Centre of the Johannes Gutenberg-University, Mainz, Germany

    T. Hansen

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  1. P. W. Kämmerer
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  2. E. Schiegnitz
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  3. T. Hansen
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  4. G. F. Draenert
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  5. H. D. Kuffner
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  6. M. O. Klein
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Correspondence to P. W. Kämmerer.

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Kämmerer, P.W., Schiegnitz, E., Hansen, T. et al. Multiple primary enoral soft tissue manifestations of a Hodgkin lymphoma—case report and literature review. Oral Maxillofac Surg 17, 53–57 (2013). https://doi.org/10.1007/s10006-012-0321-1

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  • DOI: https://doi.org/10.1007/s10006-012-0321-1

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