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CASE REPORT article

Front. Immunol.
Sec. Autoimmune and Autoinflammatory Disorders : Autoimmune Disorders
Volume 15 - 2024 | doi: 10.3389/fimmu.2024.1484798
This article is part of the Research Topic Next Generation Therapeutic Modality to Cure Autoimmune Diseases View all 8 articles

Hematopoietic Stem Cell Transplantation as Rescue Therapy for Refractory Autoimmune Retinopathy: A Case Report

Provisionally accepted
WENDY MEIHUA WONG WENDY MEIHUA WONG 1,2Yvonne Loh Yvonne Loh 3Hwei Wuen Chan Hwei Wuen Chan 1,2Warren Fong Warren Fong 4Soon Phaik Chee Soon Phaik Chee 5Adrian Koh Adrian Koh 5Graham E Holder Graham E Holder 1,2*
  • 1 National University Health System (Singapore), Singapore, Singapore
  • 2 National University of Singapore, Singapore, Singapore
  • 3 Curie Oncology, Mount Elizabeth Novena Specialist Centre, Singapore, Singapore
  • 4 Singapore General Hospital, Singapore, Singapore
  • 5 Camden Medical Centre, Singapore, Singapore

The final, formatted version of the article will be published soon.

    Autoimmune retinopathy (AIR) is a rare, potentially blinding retinal disease that remains a challenging condition to manage when resistant to conventional immune-modulatory approaches. We report clinical and electrophysiological improvement in a 49-year-old patient who underwent an autologous hematopoietic stem cell transplant (aHSCT) for thymoma-associated AIR after experiencing progressive disease despite receiving periocular and systemic steroids, mycophenolate mofetil, baricitinib, tacrolimus, bortezomib, rituximab, plasmapheresis and intravenous immunoglobulin. The aHSCT was two-staged: (i) peripheral blood stem cell harvest following mobilisation with cyclophosphamide and granulocyte colony stimulating factor, and (ii) conditioning regimen with plasmapheresis, rituximab, cyclophosphamide and anti-thymocyte globulin high dose therapy, followed by autologous hematopoietic cell infusion of 5.74 million cells. Symptoms of photopsia rapidly abated after undergoing aHSCT, and objective investigations of structure and function similarly demonstrated improvement. At 22-months follow-up, she continued to demonstrate durability of the clinical response. The present report suggests that in judiciously selected patients, HSCT may provide a rescue option for refractory AIR. Further cases are needed to confirm these results.

    Keywords: Antiretinal antibodies, Autoimmune retinopathy, Electroretinogram, hematopoietic stem cell transplant, Plasmapheresis, Thymoma, Retinal Vasculitis

    Received: 22 Aug 2024; Accepted: 26 Nov 2024.

    Copyright: © 2024 WONG, Loh, Chan, Fong, Chee, Koh and Holder. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) or licensor are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

    * Correspondence: Graham E Holder, National University of Singapore, Singapore, 119077, Singapore

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