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Sicca Syndrome and Dementia in a Patient with Hepatitis C Infection: A Case Report with Unusual Bifocal Extrahepatic Manifestations

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Abstract

Introduction

Hepatitis C virus (HCV) infections are associated with extrahepatic manifestations in 40–75 % of cases. Sialitis and secondary Sjögren syndrome are well characterized complications of chronic HCV infections but the mechanisms (primary or secondary) leading to xerostomia are not understood. Similarly, brain lesions due to HCV can be primary or secondary but the pathology of primary HCV-related brain lesions is not well described.

Case report

We report the postmortem case of a 60-year old patient initially presenting with sicca syndrome and dementia. HCV was identified in the brain but not in the salivary glands using transcription-mediated amplification (TMA). Focal sialitis was found in submandibular glands. Neuropathological examination revealed the presence of multiple dot-sized demyelination foci.

Conclusion

Sicca syndrome is a common concern in chronic HCV infections and may be due to secondary immune mechanisms (we could not isolate HCV in salivary gland tissues). TMA had never been applied to the detection of viruses in salivary glands and neural tissues and proves to be a promising technique. Neuropathological reports in HCV infections are rare and the lesions we report may be the first characterization of the direct effect of HCV on brain cells. More cases are needed to define the full spectrum of lesions potentially caused by the direct action of the HCV on salivary glands and neural tissues.

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References

  1. Ferreiro MC, Prieto MH, Rodríguez SB, Vázquez RL, Iglesias AC, Dios PD (2002) Whole stimulated salivate flow in patients with chronic hepatitis C virus infection. J Oral Pathol Med 31:117–120

    Article  PubMed  Google Scholar 

  2. Grossmann SDM, Teixeira R, Oliveira GC, Gleber-Netto FO, Araújo FM, Araújo FM, Carmo MA (2010) Xerostomia, hyposalivation and sialadenitis in patients with chronic hepatitis C are not associated with the detection of HCV RNA in saliva or salivary glands. J Clin Pathol 63:1002–1007

    Article  CAS  Google Scholar 

  3. Hofmann WP, Dries V, Herrmann E, Gartner B, Zeuzem S, Sarrazin C (2005) Comparison of transcription mediated amplification (TMA) and reverse transcription polymerase chain reaction (RT-PCR) for detection of hepatitis C virus RNA in liver tissue. J Clin Virol 32:289–293

    Article  CAS  PubMed  Google Scholar 

  4. Tedeschi R, Pivetta E, Zanussi S, Bidoli E, Ros M, di Gennaro G, Nasti G, De Paoli P (2003) Quantification of hepatitis C virus (HCV) in liver specimens and sera from patients with human immunodeficiency virus coinfection by using the Versant HCV RNA 3.0 bDNA (branched DNA-based) assay. J Clin Microbiol 41:3046–3050

    Article  PubMed Central  PubMed  Google Scholar 

  5. Sarrazin C, Teuber G, Kokka R, Rabenau H, Zeuzem S (2000) Detection of residual hepatitis C virus RNA by transcription-mediated amplification in patients with complete virologic response according to polymerase chain reaction-based assays. Hepatology 32:818–823

    Article  CAS  PubMed  Google Scholar 

  6. Haddad J, Deny P, Munz-Gotheil C, Ambrosini JC, Trinchet JC, Pateron D, Mal F, Callard P, Beaugrand M (1992) Lymphocytic sialadenitis of Sjögren’s syndrome associated with chronic hepatitis C virus liver disease. Lancet 339:321–323

    Article  CAS  PubMed  Google Scholar 

  7. Loustaud-Ratti V, Riche A, Liozon E, Labrousse F, Soria P, Rogez S, Babany G, Delaire L, Denis F, Vidal E (2001) Prevalence and characteristics of Sjögren’s syndrome or sicca syndrome in chronic hepatitis C virus infection: a prospective study. J Rheumatol 28:2245–2251

    CAS  PubMed  Google Scholar 

  8. Ramos-Casals M, Loustaud-Ratti V, De Vita S, Zeher M, Bosch JA, Toussirot E, Medina F, Rosas J, Anaya JM, Font J, SS-HCV Study Group (2005) Sjögren syndrome associated with hepatitis C virus: a multicenter analysis of 137 cases. Medicine (Baltimore) 84:81–89

    Article  Google Scholar 

  9. Ramos-Casals M, Muñoz S, Medina F, Jara LJ, Rosas J, Calvo-Alen J, Brito-Zerón P, Forns X, Sánchez-Tapias JM, HISPAMEC Study Group (2009) Systemic autoimmune diseases in patients with hepatitis C virus infection: characterization of 1020 cases (The HISPAMEC Registry). J Rheumatol 36:1442–1448

    Article  PubMed  Google Scholar 

  10. Scott CA, Avellini C, Desinan L, Pirisi M, Ferraccioli GF, Bardus P, Fabris C, Casatta L, Bartoli E, Beltrami CA (1997) Chronic lymphocytic sialoadenitis in HCV-related chronic liver disease: comparison of Sjögren’s syndrome. Histopathology 30:41–48

    Article  CAS  PubMed  Google Scholar 

  11. Grossmann Sde M, Teixeira R, Oliveira GC, Carmo MA (2010) Detection of HCV RNA in saliva does not correlate with salivary flow or xerostomia in patients with chronic hepatitis C. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 109:851–856

    Article  Google Scholar 

  12. Origgi L, Vanoli M, Carbone A, Grasso M, Scorza R (1998) Central nervous system involvement in patients with HCV-related cryoglobulinemia. Am J Med Sci 315:208–210

    Article  CAS  PubMed  Google Scholar 

  13. Bolay H, Soylemezoglu F, Nurlu G, Tuncer S, Varli K (1996) PCR detected hepatitis C virus genome in the brain of a case with progressive encephalomyelitis with rigidity. Clin Neurol Neurosurg 98:305–308

    Article  CAS  PubMed  Google Scholar 

  14. Buccoliero R, Gambelli S, Sicurelli F, Malandrini A, Palmeri S, De Santis M, Stromillo ML, De Stefano N, Sperduto A, Musumeci SA, Federico A (2006) Leukoencephalopathy as a rare complication of hepatitis C infection. Neurol Sci 27:360–363

    Article  CAS  PubMed  Google Scholar 

  15. Mestre TA, Correia de Sa J, Pimentel J (2007) Multifocal central and peripheral demyelination associated with hepatitis C virus infection. J Neurol 254:1754–1756

    Article  CAS  PubMed  Google Scholar 

  16. Wilkinson J, Radkowski M, Eschbacher JM, Laskus T (2010) Activation of brain macrophages/microglia cells in hepatitis C infection. Gut 59:1394–1400

    Article  CAS  PubMed  Google Scholar 

  17. Radkowski M, Wilkinson J, Nowicki M, Adair D, Vargas H, Ingui C, Rakela J, Laskus T (2002) Search for hepatitis C virus negative-strand RNA sequences and analysis of viral sequences in the central nervous system: evidence of replication. J Virol 76:600–608

    Article  PubMed Central  CAS  PubMed  Google Scholar 

  18. Vargas HE, Laskus T, Radkowski M, Wilkinson J, Balan V, Douglas DD, Harrison ME, Mulligan DC, Olden K, Adair D, Rakela J (2002) Detection of hepatitis C virus sequences in brain tissue obtained in recurrent hepatits C after liver tranplantation. Liver Transpl 8:1014–1019

    Article  PubMed  Google Scholar 

  19. Stohlman SA, Hinton DR (2001) Viral induced demyelination. Brain Pathol 11:92–106

    Article  CAS  PubMed  Google Scholar 

  20. Letendre S, Paulino AD, Rockenstein E, Adame A, Crews L, Cherner M, Heaton R, Ellis R, Everall IP, Grant I, Masliah E, HIV Neurobehavioral Research Center Group (2007) Pathogenesis of hepatitis C virus coinfection in the brains of patients infected with HIV. J Infect Dis 196:361–370

    Article  CAS  PubMed  Google Scholar 

  21. Wilkinson J, Radkowski M, Laskus T (2009) Hepatits C virus neuroinvasion: identification of infected cells. J Virol 83:1312–1319

    Article  PubMed Central  CAS  PubMed  Google Scholar 

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Acknowledgments

Authors would like to thank Pr Pierre Bedossa for his useful advices and Pr Patrick Marcellin for having allowed two of them (SM and MMP) to lead the virus screening in his department.

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Correspondence to R. H. Khonsari.

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Khonsari, R.H., Maylin, S., Nicol, P. et al. Sicca Syndrome and Dementia in a Patient with Hepatitis C Infection: A Case Report with Unusual Bifocal Extrahepatic Manifestations. J. Maxillofac. Oral Surg. 14 (Suppl 1), 388–392 (2015). https://doi.org/10.1007/s12663-014-0632-x

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  • DOI: https://doi.org/10.1007/s12663-014-0632-x

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