Opsoclonus-myoclonus syndrome associated with anti Kelch-like protein-11 antibodies in a young female patient without cancer

J Neuroimmunol. 2021 Jun 15:355:577570. doi: 10.1016/j.jneuroim.2021.577570. Epub 2021 Apr 7.

Abstract

Opsoclonus-myoclonus syndrome (OMS) is a rare neurological disorder. The pathogenesis is thought to be immune-mediated. In adults, it may be idiopathic or paraneoplastic in origin. However, most cases of paraneoplastic OMS in adults are not associated with well-characterized antibodies, except for a small subgroup who have anti-Ri antibodies. Herein, we provide the first detailed description of a case of OMS associated with a Kelch-like protein-11 antibody, a newly discovered biomarker for paraneoplastic neurological syndromes associated with germ-cell tumors. This was a young female patient in whom no tumor was ever detected and who had an excellent response to rituximab.

Keywords: Autoimmune encephalitis; Kelch-like protein-11 antibodies; Opsoclonus-myoclonus syndrome; Paraneoplastic neurological syndromes; Rituximab.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Autoantibodies / blood*
  • Carrier Proteins / blood*
  • Female
  • HEK293 Cells
  • Humans
  • Neoplasms*
  • Opsoclonus-Myoclonus Syndrome / blood*
  • Opsoclonus-Myoclonus Syndrome / diagnostic imaging*

Substances

  • Autoantibodies
  • Carrier Proteins
  • KLHL11 protein, human