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February 08, 2022; 98 (6) Resident & Fellow Section

Teaching NeuroImage: Seizures as the Initial Symptom of Relapsing Polychondritis

Yingying Xu, Yujie Meng, Ping Wang, Lin Sun, Shunliang Xu
First published December 3, 2021, DOI: https://doi.org/10.1212/WNL.0000000000013144
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A 41-year-old man presented with generalized tonic-clonic seizures. MRI revealed left subinsular and right frontal cortex lesions (Figure 1). CSF analysis showed 17 white cells/mm3 (lymphocytes 92%), normal protein, and glucose. The infection and autoimmune screening were negative. On day 3, bilateral auricles swelling and episcleritis emerged (Figure 2). Biopsy of left auricle revealed perivascular lymphocytic infiltration (Figure 2), consistent with relapsing polychondritis (RP). The patient's symptoms improved after oral prednisolone.

Figure 1
Figure 1 Brain Magnetic Resonance Imaging (MRI)

(A-D) MRI fluid-attenuated inversion recovery (FLAIR) showed hyperintensities, and postcontrast T1-weighted showed partial enhancement in left subinsular and right frontal cortices (white arrows). (E and F) Two months later, MRI FLAIR revealed a size reduction of lesions.

Figure 2
Figure 2 Clinical Manifestations and Auricle Biopsy

(A-C) Bilateral auricular chondritis and episcleritis. (D) Biopsy of left auricle revealed perivascular lymphocytic infiltration (black arrows; Hematoxylin/eosin).

Only 3%–13.81% of the patients with RP exhibited CNS involvement, and CNS manifestations were heterogeneous.1 RP with onset of seizures is rare. A previous autopsy report showed extensive cerebral and systemic vasculitis in RP.2

Study Funding

This work was financially supported by Shandong Provincial Natural Science Foundation, China (Grant Nos. ZR2015HM024 and 2019GSF108066 to SLX); IIFDU and SFR for ROCS, SEM.

Disclosure

The authors report no disclosures relevant to the manuscript. Go to Neurology.org/N for full disclosures.

Acknowledgment

We thank Dr. Anting Xu and Dr. Zhe Wang (Department of Otolaryngology, The Second Hospital, Cheeloo College of Medicine, Shandong University) for patient's auricle biopsy. We also thank Dr. Haitao Wang (Department of Pathology, The Second Hospital, Cheeloo College of Medicine, Shandong University) for the pathologic diagnosis.

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Footnotes

  • Go to Neurology.org/N for full disclosures. Funding information and disclosures deemed relevant by the authors, if any, are provided at the end of the article.

  • Teaching slides links.lww.com/WNL/B685

References

  1. 1.↵
    1. Cao X,
    2. Zhu L,
    3. Li H, et al.
    Comparison of relapsing polychondritis patients with and without central nervous system involvement: a retrospective study of 181 patients. Int J immunopathology Pharmacol. 2021;35:20587384211000547. doi: 10.1177/20587384211000547
    OpenUrlCrossRef
  2. 2.↵
    1. Stewart SS,
    2. Ashizawa T,
    3. Dudley AW,
    4. Goldberg JW,
    5. Lidsky MD
    . Cerebral vasculitis in relapsing polychondritis. Neurology. 1988;38(1):150-152.
    OpenUrlAbstract/FREE Full Text

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