European Archives of Oto-Rhino-Laryngology

, Volume 265, Issue 3, pp 377–380

Parotid localized Castleman’s disease and HHV-8 infection: a case report

  • Elisabetta Caselli
  • Davide Padovani
  • Roberto Di Carlo
  • Enrico Grandi
  • Monica Galvan
  • Enzo Cassai
  • Antonio Pastore
Case Report


Castleman’s disease (CD) is an unusual massive proliferation of lymphoid tissue distinct in two clinical forms, localized and multicentric. The multicentric form has been related to human herpesvirus 8 (HHV-8), especially in HIV-infected patients, whereas the localized form of CD is still unrelated to viral pathogens. We report a case of a HIV-negative 16-year-old male referred to our hospital with a 12-month history of a painless swelling in his right parotid region. A parotidectomy was performed, and histological analysis evidenced a localized CD. The search for HHV-8 revealed an active virus infection. The patient was commenced on corticosteroid therapy and a follow-up was performed every 6 months. The patient was commenced on corticosteroid therapy and there has been no recurrence after 24 months. The authors report a case of localized parotid CD in a patient with evidence of an active HHV-8 infection. The results of this study does for the first time suggest an association between HHV-8 and localized CD in HIV-negative subjects.


Castleman’s disease Parotid gland Human herpesvirus-8 Angiofollicular lymph node hyperplasia Viral infection 


  1. 1.
    Sanchez-Cuellar A, de Pedro M, Martin-Granizo R, Berguer A (2001) Castleman disease (giant lymph node hyperplasia) in the maxillofacial region: a report of 3 cases. J Oral Maxillofac Surg 59:228–231PubMedCrossRefGoogle Scholar
  2. 2.
    Soulier J, Grollet L, Oksenhendler E et al (1995) Kaposi’s sarcoma-associated herpesvirus-like DNA sequences in multicentric Castleman’s disease. Blood 86:1276–1280PubMedGoogle Scholar
  3. 3.
    Sarid R, Klepfish A, Schattner A (2002) Virology, pathogenetic mechanisms, and associated diseases of Kaposi sarcoma-associated herpesvirus (human herpesvirus 8). Mayo Clin Proc 77:941–949PubMedCrossRefGoogle Scholar
  4. 4.
    Chang Y CE, Pessin MS, Lee F, Culpepper J, Knowles DM, Moore PS (1994) Identification of herpesvirus-like DNA sequences in AIDS-associated Kaposi’s sarcoma. Science 266:1865–1869PubMedCrossRefGoogle Scholar
  5. 5.
    Cesarman E, Chang Y, Moore PS, Said JW, Knowles DM (1995) Kaposi’s sarcoma-associated herpesvirus-like DNA sequences in AIDS-related body-cavity-based lymphomas. N Engl J Med 332:1186–1191PubMedCrossRefGoogle Scholar
  6. 6.
    Gao SJ, Kingsley L, Li M et al (1996) KSHV antibodies among Americans, Italians and Ugandans with and without Kaposi’s sarcoma. Nat Med 2:925–928PubMedCrossRefGoogle Scholar
  7. 7.
    Kedes DH, Operskalski E, Busch M, Kohn R, Flood J, Ganem D (1996) The seroepidemiology of human herpesvirus 8 (Kaposi’s sarcoma-associated herpesvirus): distribution of infection in KS risk groups and evidence for sexual transmission. Nat Med 2:918–924PubMedCrossRefGoogle Scholar
  8. 8.
    Whitby D, Luppi M, Barozzi P, Boshoff C, Weiss RA, Torelli G (1998) Human herpesvirus 8 seroprevalence in blood donors and lymphoma patients from different regions of Italy. J Natl Cancer Inst 90:395–397PubMedCrossRefGoogle Scholar
  9. 9.
    Suchankova A, Stankova M, Roubalova K, Vandasova J, Bruckova M (2003) Seroprevalence of HHV 8 antibodies among the general population and HIV positive persons in the Czech Republic. J Clin Virol 28:70–76PubMedCrossRefGoogle Scholar
  10. 10.
    Gambus G, Bourboulia D, Esteve A et al (2001) Prevalence and distribution of HHV-8 in different subpopulations, with and without HIV infection, in Spain. AIDS 15:1167–1174PubMedCrossRefGoogle Scholar
  11. 11.
    Osmond DH, Buchbinder S, Cheng A et al (2002) Prevalence of Kaposi sarcoma-associated herpesvirus infection in homosexual men at beginning of and during the HIV epidemic. JAMA 287:221–225PubMedCrossRefGoogle Scholar
  12. 12.
    Dupin N, Fisher C, Kellam P et al (1999) Distribution of human herpesvirus-8 latently infected cells in Kaposi’s sarcoma, multicentric Castleman’s disease, and primary effusion lymphoma. Proc Natl Acad Sci USA 96:4546–4551PubMedCrossRefGoogle Scholar
  13. 13.
    Caselli E, Galvan M, Cassai E, Caruso A, Sighinolfi L, Di Luca D (2005) Human herpesvirus 8 enhances human immunodeficiency virus replication in acutely infected cells and induces reactivation in latently infected cells. Blood 106:2790–2797PubMedCrossRefGoogle Scholar
  14. 14.
    Caselli E, Galvan M, Santoni F et al (2003) Human herpesvirus-8 (Kaposi’s sarcoma-associated virus) ORF50 increases in vitro cell susceptibility to human immunodeficiency virus type 1 infection. J Gen Virol 84:1123–1131PubMedCrossRefGoogle Scholar
  15. 15.
    Caselli E, Fiorentini S, Amici C, Di Luca D, Caruso A, Santoro MG (2007) Human Herpesvirus-8 acute infection of endothelial cells induces monocyte chemoattractant protein 1-dependent capillary-like structure formation: role of the IKK/NF-kB pathway. Blood 109:2718–2726PubMedGoogle Scholar
  16. 16.
    Goodisson DW, Carr RJ, Stirling RW (1997) Parotid presentation of Castleman’s disease: report of a case. J Oral Maxillofac Surg 55:515–517PubMedCrossRefGoogle Scholar
  17. 17.
    Martin DF, Kuppermann BD, Wolitz RA, Palestine AG, Li H, Robinson CA (1999) Oral ganciclovir for patients with cytomegalovirus retinitis treated with a ganciclovir implant. Roche Ganciclovir study group. N Engl J Med 340:1063–1070PubMedCrossRefGoogle Scholar
  18. 18.
    Casper C, Nichols WG, Huang ML, Corey L, Wald A (2004) Remission of HHV-8 and HIV-associated multicentric Castleman disease with ganciclovir treatment. Blood 103:1632–1634PubMedCrossRefGoogle Scholar
  19. 19.
    Atula T, Grenman R, Klemi P, Syrjanen S (1998) Human papillomavirus, Epstein-Barr virus, human herpesvirus 8 and human cytomegalovirus involvement in salivary gland tumours. Oral Oncol 34:391–395PubMedCrossRefGoogle Scholar
  20. 20.
    Klussmann JP, Muller A, Wagner M et al (2000) Human herpesvirus type 8 in salivary gland tumors. J Clin Virol 16:239–246PubMedCrossRefGoogle Scholar
  21. 21.
    Yen TL, Murr AH, Rabin J, Mhatre AN, Lalwani AK (2004) Role of cytomegalovirus, Epstein-Barr virus, and human herpes virus-8 in benign lymphoepithelial cysts of the parotid gland. Laryngoscope 114:1500–1505PubMedCrossRefGoogle Scholar

Copyright information

© Springer-Verlag 2007

Authors and Affiliations

  • Elisabetta Caselli
    • 1
  • Davide Padovani
    • 2
    • 5
  • Roberto Di Carlo
    • 3
  • Enrico Grandi
    • 4
  • Monica Galvan
    • 1
  • Enzo Cassai
    • 1
  • Antonio Pastore
    • 2
  1. 1.Section of Microbiology, Department of Experimental and Diagnostic MedicineUniversity of FerraraFerraraItaly
  2. 2.Otorhinolaryngology ClinicsUniversity of FerraraFerraraItaly
  3. 3.Otorhinolaryngology ClinicsSt. Anna HospitalFerraraItaly
  4. 4.Section of Anatomy Histology and Pathological CitologyUniversity of FerraraFerraraItaly
  5. 5. BondenoItaly

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