Alemtuzumab-induced thyroid eye disease successfully treated with a single low dose of rituximab

Eur Thyroid J. 2024 Apr 11;13(2):e230236. doi: 10.1530/ETJ-23-0236. Print 2024 Apr 1.

Abstract

Introduction: Secondary thyroid autoimmunity, especially Graves' disease (GD), frequently develops in patients with multiple sclerosis (MS) following alemtuzumab treatment (ALTZ; anti-CD52). Thyroid eye disease (TED) can also develop, and rituximab (RTX; anti-CD20) is a suitable treatment.

Case presentation: A 37-year-old woman with MS developed steroid-resistant active moderate-to-severe TED 3 years after ALTZ, that successfully responded to a single 500 mg dose of i.v. RTX. Before RTX peripheral B-cells were low, and were totally depleted immediately after therapy. Follow-up analysis 4 years post ALTZ and 1 year post RTX showed persistent depletion of B cells, and reduction of T regulatory cells in both peripheral blood and thyroid tissue obtained at thyroidectomy.

Conclusion: RTX therapy successfully inactivated TED in a patient with low B-cell count derived from previous ALTZ treatment. B-cell depletion in both thyroid and peripheral blood was still present 1 year after RTX, indicating a likely cumulative effect of both treatments.

Keywords: Graves’ orbitopathy; alemtuzumab; immune reconstitution therapy; lymphocyte depletion; rituximab; thyroid eye disease.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Alemtuzumab / adverse effects
  • Antibodies, Monoclonal, Murine-Derived / therapeutic use
  • Female
  • Graves Disease* / drug therapy
  • Graves Ophthalmopathy* / chemically induced
  • Humans
  • Multiple Sclerosis* / drug therapy
  • Rituximab / adverse effects

Substances

  • Rituximab
  • Alemtuzumab
  • Antibodies, Monoclonal, Murine-Derived