Clinical pitfalls and serological diagnostics of MuSK myasthenia gravis

J Neurol. 2023 Mar;270(3):1478-1486. doi: 10.1007/s00415-022-11458-4. Epub 2022 Nov 17.

Abstract

Background: We aimed to evaluate the diagnostic accuracy of enzyme-linked immunosorbent assay (ELISA) for anti-muscle specific tyrosine kinase (MuSK) antibody (Ab) in a large cohort of anti-acetylcholine receptor (AChR) Ab-negative generalized myasthenia gravis (MG), and also to investigate clinical contexts for the diagnosis of MuSK MG.

Methods: A retrospective study of 160 patients with a clinical suspicion of AChR Ab-negative generalized MG was performed. The serum samples were tested for anti-clustered AChR Ab by cell-based assay (CBA), anti-MuSK Ab by ELISA, CBA and/or radioimmunoprecipitation assay (RIPA). Clinical data were compared between anti-MuSK Ab-positive MG and double seronegative (AChR and MuSK) MG groups.

Results: After excluding non-MG and clustered AChR Ab-positive patients, we identified 89 patients as a cohort of AChR Ab-negative generalized MG. Anti-MuSK Ab was positive by ELISA in 22 (24.7%) patients. While CBA identified five additional anti-MuSK Ab-positive patients, the results of ELISA were mostly consistent with CBA and RIPA with Cohen's kappa of 0.80 and 0.90, respectively (p < 0.001). The most frequent differential diagnosis was motor neuron disease particularly of bulbar onset which showed remarkably overlapping clinical and electrophysiological features with MuSK MG at presentation.

Conclusion: While confirming the highest sensitivity of CBA for detecting anti-MuSK Ab, our results highlight the clinical pitfalls in making a diagnosis of MuSK MG and may support a diagnostic utility of MuSK-ELISA in clinical practice.

Keywords: Anti-MuSK antibody; Cell-based assay; ELISA; Radioimmunoprecipitation assay; Seronegative myasthenia gravis.

MeSH terms

  • Autoantibodies
  • Enzyme-Linked Immunosorbent Assay
  • Humans
  • Myasthenia Gravis*
  • Receptor Protein-Tyrosine Kinases*
  • Receptors, Cholinergic
  • Retrospective Studies

Substances

  • Receptor Protein-Tyrosine Kinases
  • Receptors, Cholinergic
  • Autoantibodies
  • MUSK protein, human