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Susac’s syndrome as an immune-related adverse event after pembrolizumab: a case report

  • M. De GrootEmail author
  • A. Compter
  • A. J. De Langen
  • D. Brandsma
Letter to the Editors
  • 31 Downloads

Dear Sirs,

Immune checkpoint inhibitors are increasingly used in cancer treatment and have a significant impact on survival [1]. Pembrolizumab is a recombinant monoclonal antibody that blocks the programmed death-1 (PD-1) receptor, which is an inhibitor of T-cell activation, and thereby can promote the immune response against the tumor [2]. The increased immune response can also induce auto-immune side effects [3]. For the first time, we report a case of Susac’s syndrome induced by pembrolizumab.

A 55-year-old woman was diagnosed with stage IV non-small cell lung cancer (NSCLC) with metastases in the adrenal gland, bones and brain. The primary tumor was thyroid transcription factor-1 (TTF-1) positive and tested negative for oncogenic drivers, including epidermal growth factor receptor (EGFR), B-Raf proto-oncogene serine/threonine kinase (BRAF), anaplastic lymphoma kinase (ALK), ROS proto-oncogene 1 receptor tyrosine kinase (ROS1) and KRAS proto-oncogene GTPase (KRAS). Oncogenic drivers...

Notes

Compliance with ethical standards

Conflicts of interest

The authors declare that they have no competing interests.

Ethical standards

The patient provided informed oral and written consent in accordance with specific national laws and the ethics standards laid down in the 1964 Declaration of Helsinki and its later amendments.

References

  1. 1.
    Assi HI, Kamphorst AO, Moukalled NM, Ramalingam SS (2018) Immune checkpoint inhibitors in advanced non-small cell lung cancer. Cancer 124(2):248–261.  https://doi.org/10.1002/cncr.31105 CrossRefPubMedGoogle Scholar
  2. 2.
    Pardoll D (2015) Cancer and the immune system: basic concepts and targets for intervention. Semin Oncol 42(4):523–538.  https://doi.org/10.1053/j.seminoncol.2015.05.003 CrossRefPubMedPubMedCentralGoogle Scholar
  3. 3.
    Haanen J, Carbonnel F, Robert C, Kerr KM, Peters S, Larkin J, Jordan K (2017) Management of toxicities from immunotherapy: ESMO Clinical Practice Guidelines for diagnosis, treatment and follow-up. Ann Oncol 28(suppl_4):iv119–iv142.  https://doi.org/10.1093/annonc/mdx225 CrossRefPubMedGoogle Scholar
  4. 4.
    Patel SP, Kurzrock R (2015) PD-L1 expression as a predictive biomarker in cancer immunotherapy. Mol Cancer Ther 14(4):847–856.  https://doi.org/10.1158/1535-7163.Mct-14-0983 CrossRefPubMedGoogle Scholar
  5. 5.
    Kao JC, Liao B, Markovic SN, Klein CJ, Naddaf E, Staff NP, Liewluck T, Hammack JE, Sandroni P, Finnes H, Mauermann ML (2017) Neurological complications associated with anti-programmed death 1 (PD-1) antibodies. JAMA Neurol 74(10):1216–1222.  https://doi.org/10.1001/jamaneurol.2017.1912 CrossRefPubMedPubMedCentralGoogle Scholar
  6. 6.
    Mancone S, Lycan T, Ahmed T, Topaloglu U, Dothard A, Petty WJ, Strowd RE (2018) Severe neurologic complications of immune checkpoint inhibitors: a single-center review. J Neurol.  https://doi.org/10.1007/s00415-018-8890-z CrossRefPubMedGoogle Scholar
  7. 7.
    Laubli H, Hench J, Stanczak M, Heijnen I, Papachristofilou A, Frank S, Zippelius A, Stenner-Liewen F (2017) Cerebral vasculitis mimicking intracranial metastatic progression of lung cancer during PD-1 blockade. J Immunother Cancer 5:46.  https://doi.org/10.1186/s40425-017-0249-y CrossRefPubMedPubMedCentralGoogle Scholar
  8. 8.
    Weber JS, Hodi FS, Wolchok JD, Topalian SL, Schadendorf D, Larkin J, Sznol M, Long GV, Li H, Waxman IM, Jiang J, Robert C (2017) Safety profile of nivolumab monotherapy: a pooled analysis of patients with advanced melanoma. J Clin Oncol 35(7):785–792.  https://doi.org/10.1200/jco.2015.66.1389 CrossRefPubMedGoogle Scholar
  9. 9.
    Dorr J, Krautwald S, Wildemann B, Jarius S, Ringelstein M, Duning T, Aktas O, Ringelstein EB, Paul F, Kleffner I (2013) Characteristics of Susac syndrome: a review of all reported cases. Nat Rev Neurol 9(6):307–316.  https://doi.org/10.1038/nrneurol.2013.82 CrossRefPubMedGoogle Scholar
  10. 10.
    Susac JO, Murtagh FR, Egan RA, Berger JR, Bakshi R, Lincoff N, Gean AD, Galetta SL, Fox RJ, Costello FE, Lee AG, Clark J, Layzer RB, Daroff RB (2003) MRI findings in Susac's syndrome. Neurology 61(12):1783–1787CrossRefGoogle Scholar
  11. 11.
    Kleffner I, Dorr J, Ringelstein M, Gross CC, Bockenfeld Y, Schwindt W, Sundermann B, Lohmann H, Wersching H, Promesberger J, von Konigsmarck N, Alex A, Guthoff R, Frijns CJ, Kappelle LJ, Jarius S, Wildemann B, Aktas O, Paul F, Wiendl H, Duning T (2016) Diagnostic criteria for Susac syndrome. J Neurol Neurosurg Psychiatry 87(12):1287–1295.  https://doi.org/10.1136/jnnp-2016-314295 CrossRefPubMedGoogle Scholar
  12. 12.
    Greco A, De Virgilio A, Gallo A, Fusconi M, Turchetta R, Tombolini M, Rizzo MI, de Vincentiis M (2014) Susac's syndrome—pathogenesis, clinical variants and treatment approaches. Autoimmun Rev 13(8):814–821.  https://doi.org/10.1016/j.autrev.2014.04.004 CrossRefPubMedGoogle Scholar
  13. 13.
    van der Kooij SM, van Buchem MA, Overbeek OM, Dijkman G, Huizinga TW (2015) Susac syndrome: a report of four cases and a review of the literature. Neth J Med 73(1):10–16PubMedGoogle Scholar

Copyright information

© Springer-Verlag GmbH Germany, part of Springer Nature 2019

Authors and Affiliations

  • M. De Groot
    • 1
    Email author
  • A. Compter
    • 1
  • A. J. De Langen
    • 2
  • D. Brandsma
    • 1
  1. 1.Department of Neuro-OncologyNetherlands Cancer Institute-Antoni Van LeeuwenhoekAmsterdamThe Netherlands
  2. 2.Department of Thoracic OncologyNetherlands Cancer Institute-Antoni Van LeeuwenhoekAmsterdamThe Netherlands

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