Skip to main content

Advertisement

SpringerLink
Log in

Aseptic meningitis in relapsing polychondritis: a case report and literature review

  • Case Based Review
  • Published:
Clinical Rheumatology Aims and scope Submit manuscript

Abstract

Aseptic meningitis is an extremely rare neurologic complication of relapsing polychondritis (RP). We reported a case of a 58-year-old Chinese female with intractable headache, puffy ears, pleocytosis, and cranial magnetic resonance imaging (MRI) showing thickened and enhanced meninges. She was finally diagnosed of aseptic meningitis due to RP after full exclusion of infectious causes. She gradually developed neurosensory hearing loss, vertigo, and saddle nose while glucocorticosteroid therapy and combined cyclophosphamide could not control her headache. Ultimately, cyclosporin A was tried showing a good response. Only 18 previous cases were found in the literature and the clinical manifestation, cerebrospinal fluid (CSF) characteristics, imaging features, and therapy considerations of RP-related aseptic meningitis were summarized by reviewing the literature. Aseptic meningitis due to RP is a rare condition of undetermined pathoetiology. Its diagnosis is primarily based on clinical manifestations combined with CSF and MRI examinations plus adequate exclusion of possible infections. Corticosteroid is the basic therapy but choice of protocol should be individualized.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1

Similar content being viewed by others

References

  1. Cantarini L, Vitale A, Brizi MG, Caso F, Frediani B, Punzi L et al (2014) Diagnosis and classification of relapsing polychondritis. J Autoimmun 48-49:53–59

    Article  CAS  PubMed  Google Scholar 

  2. Puechal X, Terrier B, Mouthon L, Costedoat-Chalumeau N, Guillevin L, Le Jeunne C (2014) Relapsing polychondritis. Joint Bone Spine 81:118–124

    Article  CAS  PubMed  Google Scholar 

  3. Brod S, Booss J (1988) Idiopathic CSF pleocytosis in relapsing polychondritis. Neurology 38:322–323

    Article  CAS  PubMed  Google Scholar 

  4. Stewart SS, Ashizawa T, Dudley AW Jr, Goldberg JW, Lidsky MD (1988) Cerebral vasculitis in relapsing polychondritis. Neurology 38:150–152

    Article  CAS  PubMed  Google Scholar 

  5. Hanslik T, Wechsler B, Piette JC, Vidailhet M, Robin PM, Godeau P (1994) Central nervous system involvement in relapsing polychondritis. Clin Exp Rheumatol 12:539–541

    CAS  PubMed  Google Scholar 

  6. Ragnaud JM, Tahbaz A, Morlat P, Sire S, Gin H, Aubertin J (1996) Recurrent aseptic purulent meningitis in a patient with relapsing polychondritis. Clin Infect Dis 22:374–375

    Article  CAS  PubMed  Google Scholar 

  7. Berg AM, Kasznica J, Hopkins P, Simms RW (1996) Relapsing polychondritis and aseptic meningitis. J Rheumatol 23:567–569

    CAS  PubMed  Google Scholar 

  8. Kothare SV, Chu CC, VanLandingham K, Richards KC, Hosford DA, Radtke RA (1998) Migratory leptomeningeal inflammation with relapsing polychondritis. Neurology 51:614–617

    Article  CAS  PubMed  Google Scholar 

  9. Ota M, Mizukami K, Hayashi T, Sumida T, Asada T (2005) Brain magnetic resonance imaging and single photon emission computerized tomography findings in a case of relapsing polychondritis showing cognitive impairment and personality changes. Prog Neuro-Psychopharmacol Biol Psychiatry 29:347–349

    Article  Google Scholar 

  10. Hsu KC, Wu YR, Lyu RK, Tang LM (2006) Aseptic meningitis and ischemic stroke in relapsing polychondritis. Clin Rheumatol 25:265–267

    Article  PubMed  Google Scholar 

  11. Kao KT, Potrebic S, Evans JR (2007) Relapsing polychondritis presenting as meningoencephalitis with valvular abnormality: a case report. Clin Rheumatol 26:1985–1988

    Article  PubMed  Google Scholar 

  12. Erten-Lyons D, Oken B, Woltjer RL, Quinn J (2008) Relapsing polychondritis: an uncommon cause of dementia. J Neurol Neurosurg Psychiatry 79:609–610

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  13. Yaguchi H, Tsuzaka K, Niino M, Yabe I, Sasaki H (2009) Aseptic meningitis with relapsing polychondritis mimicking bacterial meningitis. Intern Med 48:1841–1844

    Article  PubMed  Google Scholar 

  14. Wang ZJ, Pu CQ, Wang ZJ, Zhang JT, Wang XQ, Yu SY et al (2011) Meningoencephalitis or meningitis in relapsing polychondritis: four case reports and a literature review. J Clin Neurosci 18:1608–1615

    Article  CAS  PubMed  Google Scholar 

  15. Hatti K, Giuliano V (2014) Central nervous system involvement in relapsing polychondritis. J Clin Rheumatol 20:396–397

    PubMed  Google Scholar 

  16. McAdam LP, O'Hanlan MA, Bluestone R, Pearson CM (1976) Relapsing polychondritis: prospective study of 23 patients and a review of the literature. Medicine (Baltimore) 55:193–215

    Article  CAS  Google Scholar 

  17. Kemta Lekpa F, Kraus VB, Chevalier X (2012) Biologics in relapsing polychondritis: a literature review. Semin Arthritis Rheum 41:712–719

    Article  PubMed  Google Scholar 

Download references

Author information

Author notes

  1. Kai Shen and Geng Yin contributed equally to this work.

Authors and Affiliations

  1. Department of Hematology, West China Hospital, Sichuan University, Chengdu, 610041, China

    Kai Shen & Chenlu Yang

  2. Department of General Practice, West China Hospital, Sichuan University, Chengdu, 610041, China

    Geng Yin

  3. Department of Rheumatology and Immunology, West China Hospital, Sichuan University, Chengdu, 610041, China

    Qibing Xie

Authors
  1. Kai Shen
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Geng Yin
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Chenlu Yang
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Qibing Xie
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Qibing Xie.

Ethics declarations

Disclosures

None.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Shen, K., Yin, G., Yang, C. et al. Aseptic meningitis in relapsing polychondritis: a case report and literature review. Clin Rheumatol 37, 251–255 (2018). https://doi.org/10.1007/s10067-017-3616-7

Download citation

  • Received:

  • Revised:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s10067-017-3616-7

Keywords

Search

Navigation