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Introduction <p>Autoimmune encephalitis (AE) is a spectrum of autoimmune disorders manifesting by epilepsy, loss of consciousness, and neuropsychiatric symptoms. Anti-recoverin antibodies are intracellular antibodies and a well-known marker of paraneoplastic retinopathy. However, they have rarely been associated with neurological and psychiatric diseases with or without cancer. To date, there have been only a few identified cases of anti-recoverin antibody-associated limbic AE.</p> Case presentation <p>A 33-year-old North-African female with a family history of cancer and no relevant medical history presented to the neurology department of the Military Hospital of Tunis, Tunisia, with a 3-month history of temporal epilepsy, insomnia, and mild cognitive impairment. The episodes lasted 3–4 minutes. Scalp electroencephalogram showed bilateral epileptiform spikes and spike waves in the temporal regions. Brain magnetic resonance imaging showed bilateral hippocampal and thalamic hyperintense lesions on T2-weighted imaging with hypointensities in the same regions on T1-weighted imaging and no contrast enhancement. Anti-recoverin antibodies were strongly positive (3+) in blood and cerebrospinal fluid (CSF). The patient met the criteria for definite limbic AE after exclusion of alternative etiologies. The extended cancer work-up was unremarkable. The patient received 1 g daily of intravenous methylprednisolone for 5 days, along with 1000 mg/day of oral levetiracetam followed by 0.4 g/kg/day of intravenous immunoglobulin (IVIG) for 5 days. Levetiracetam was replaced by lamotrigine (100 mg/day) orally. During the 2-year follow-up, the patient’s condition improved clinically and radiologically. Anti-recoverin antibody positivity persisted (1+) to date with minimal symptoms.</p> Conclusion <p>Our case illustrated the value of testing anti-recoverin antibodies in CSF and provides insights into management and prognosis through a longitudinal 2-year follow-up. Anti-recoverin antibody-related AE is a rare but treatable cause of adult subacute encephalopathy and epilepsy. Prompt and optimized management associating appropriate anti-epileptic drugs and immunotherapies is essential to alleviate sequelae.</p>

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Autoimmune encephalitis associated with anti-recoverin antibodies

  • 期刊论文

  • 出版模式：

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  • 发表日期：

    2026年2月9日

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  • Mohamed Slim Majoul,
  • Meriam Messelmani,
  • Hajer Derbali,
  • Yasmine Amor,
  • Nejiba Fekih-Mrissa,
  • Jamel Zaouali

摘要

Introduction

Autoimmune encephalitis (AE) is a spectrum of autoimmune disorders manifesting by epilepsy, loss of consciousness, and neuropsychiatric symptoms. Anti-recoverin antibodies are intracellular antibodies and a well-known marker of paraneoplastic retinopathy. However, they have rarely been associated with neurological and psychiatric diseases with or without cancer. To date, there have been only a few identified cases of anti-recoverin antibody-associated limbic AE.

Case presentation

A 33-year-old North-African female with a family history of cancer and no relevant medical history presented to the neurology department of the Military Hospital of Tunis, Tunisia, with a 3-month history of temporal epilepsy, insomnia, and mild cognitive impairment. The episodes lasted 3–4 minutes. Scalp electroencephalogram showed bilateral epileptiform spikes and spike waves in the temporal regions. Brain magnetic resonance imaging showed bilateral hippocampal and thalamic hyperintense lesions on T2-weighted imaging with hypointensities in the same regions on T1-weighted imaging and no contrast enhancement. Anti-recoverin antibodies were strongly positive (3+) in blood and cerebrospinal fluid (CSF). The patient met the criteria for definite limbic AE after exclusion of alternative etiologies. The extended cancer work-up was unremarkable. The patient received 1 g daily of intravenous methylprednisolone for 5 days, along with 1000 mg/day of oral levetiracetam followed by 0.4 g/kg/day of intravenous immunoglobulin (IVIG) for 5 days. Levetiracetam was replaced by lamotrigine (100 mg/day) orally. During the 2-year follow-up, the patient’s condition improved clinically and radiologically. Anti-recoverin antibody positivity persisted (1+) to date with minimal symptoms.

Conclusion

Our case illustrated the value of testing anti-recoverin antibodies in CSF and provides insights into management and prognosis through a longitudinal 2-year follow-up. Anti-recoverin antibody-related AE is a rare but treatable cause of adult subacute encephalopathy and epilepsy. Prompt and optimized management associating appropriate anti-epileptic drugs and immunotherapies is essential to alleviate sequelae.