ArticleFrontiers in immunology2026
Anti-IgLON5 disease presenting with myokymia and favourable response to FcRN-antagonist combined with oral glucocorticoids: case report.
Article in Frontiers in immunology, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.
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Abstract
Anti-IgLON5 disease is a rare autoimmune neurological disorder characterized by anti- neuronal surface autoantibodies targeting the Immunoglobulin-Like Cell Adhesion Molecule 5 (IgLON5) protein. We report a 53-year-old male patient diagnosed with anti-IgLON5 disease who presented with the uncommon manifestation of generalized multi-site myokymia. Laboratory tests showed positive anti-IgLON5 IgG antibodies in both serum (titer 1:100) and cerebrospinal fluid (CSF) (titer 1:1), with no specific abnormalities detected on brain magnetic resonance imaging (MRI) and whole-exome sequencing. Considering patients' refusal to use high-dose glucocorticoids due to concerns about drug side effects, as well as the uncertainty surrounding the efficacy of intravenous immunoglobulin in treating IgLON5 disease, we selected a combined immunosuppressive therapy regimen consisting of intravenous neonatal fragment crystallizable receptor (FcRn) antagonist (efgartigimod) and sequential oral prednisone. The patient's myokymia were rapidly alleviated after combined treatment. During an 18-month follow-up, symptoms completely abated within the first 6 months, and antibody titers declined gradually to negative. Mild symptom recurred at 7 months post-initial treatment. Serum anti-IgLON5 IgG antibodies became positive (titer 1:10) at 14 months, however, these antibodies had been negative at 3 months after the start of treatment. Symptomatology was well-controlled after adjusting the oral prednisone dose. This case expands the clinical spectrum of anti-IgLON5 disease, highlights the necessity of screening for neuronal autoantibodies in patients with myokymia, and suggests that the combination of FcRn antagonists and glucocorticoids may emerge as a new immunotherapeutic option for this condition.
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