Océane Landon-Cardinal1,2,3, Yves Allenbach4,5,6, Antoine Soulages4,5,6, Aude Rigolet4,5,6, Baptiste Hervier4,5,6, Nicolas Champtiaux4,5,6, Quentin Monzani4,5,6, Guilhem Solé4,5,6, Olivier Benveniste4,5,6. 1. From the Department of Internal Medicine and Clinical Immunology and Inflammation-Immunopathology-Biotherapy Department (I2B), Pitié-Salpêtrière University Hospital, AP-HP, East Paris Neuromuscular Diseases Reference Center, Inserm U974, Sorbonne Université, Paris, France; Center of Reference for Neuromuscular Disorders AOC, Department of Neurology, Bordeaux University Hospital, Bordeaux, France; Polyvalent and Oncologic Radiology Department, Musculoskeletal Unit, Pitié-Salpêtrière University Hospital, AP-HP, Paris, France. o.landoncardinal@gmail.com. 2. Dr. Landon-Cardinal is the recipient of Clinical Fellowship awards from the Université de Montréal Rheumatology Program, Abbvie Educational Grant and the Association des médecins rhumatologues du Québec, Visithan-Khy Educational Grant. o.landoncardinal@gmail.com. 3. O. Landon-Cardinal, MD, Myositis post-doctoral Fellowship, Department of Internal Medicine and Clinical Immunology, AP-HP, Hôpital Pitié-Salpêtrière; Y. Allenbach, MD, PhD, Assistant Professor of Medicine, Department of Internal Medicine and Clinical Immunology, AP-HP, Hôpital Pitié-Salpêtrière; A. Soulages, MD, Clinical Associate of Medicine, Center of Reference for Neuromuscular Disorders AOC, Department of Neurology, Bordeaux University Hospital; A. Rigolet, MD, Clinical Associate of Medicine, Department of Internal Medicine and Clinical Immunology, AP-HP, Hôpital Pitié-Salpêtrière; B. Hervier, MD, PhD, Clinical Associate of Medicine, Department of Internal Medicine and Clinical Immunology, AP-HP, Hôpital Pitié-Salpêtrière; N. Champtiaux, MD, PhD, Clinical Associate of Medicine, Department of Internal Medicine and Clinical Immunology, AP-HP, Hôpital Pitié-Salpêtrière; Q. Monzani, MD, Polyvalent and Oncologic Radiology Department, Musculoskeletal Unit, AP-HP, Hôpital Pitié-Salpêtrière; G. Solé, MD, Professor of Medicine, Center of Reference for Neuromuscular Disorders AOC, Department of Neurology, Bordeaux University Hospital; O. Benveniste, MD, PhD, Professor of Medicine, Department of Internal Medicine and Clinical Immunology, AP-HP, Hôpital Pitié-Salpêtrière. o.landoncardinal@gmail.com. 4. From the Department of Internal Medicine and Clinical Immunology and Inflammation-Immunopathology-Biotherapy Department (I2B), Pitié-Salpêtrière University Hospital, AP-HP, East Paris Neuromuscular Diseases Reference Center, Inserm U974, Sorbonne Université, Paris, France; Center of Reference for Neuromuscular Disorders AOC, Department of Neurology, Bordeaux University Hospital, Bordeaux, France; Polyvalent and Oncologic Radiology Department, Musculoskeletal Unit, Pitié-Salpêtrière University Hospital, AP-HP, Paris, France. 5. Dr. Landon-Cardinal is the recipient of Clinical Fellowship awards from the Université de Montréal Rheumatology Program, Abbvie Educational Grant and the Association des médecins rhumatologues du Québec, Visithan-Khy Educational Grant. 6. O. Landon-Cardinal, MD, Myositis post-doctoral Fellowship, Department of Internal Medicine and Clinical Immunology, AP-HP, Hôpital Pitié-Salpêtrière; Y. Allenbach, MD, PhD, Assistant Professor of Medicine, Department of Internal Medicine and Clinical Immunology, AP-HP, Hôpital Pitié-Salpêtrière; A. Soulages, MD, Clinical Associate of Medicine, Center of Reference for Neuromuscular Disorders AOC, Department of Neurology, Bordeaux University Hospital; A. Rigolet, MD, Clinical Associate of Medicine, Department of Internal Medicine and Clinical Immunology, AP-HP, Hôpital Pitié-Salpêtrière; B. Hervier, MD, PhD, Clinical Associate of Medicine, Department of Internal Medicine and Clinical Immunology, AP-HP, Hôpital Pitié-Salpêtrière; N. Champtiaux, MD, PhD, Clinical Associate of Medicine, Department of Internal Medicine and Clinical Immunology, AP-HP, Hôpital Pitié-Salpêtrière; Q. Monzani, MD, Polyvalent and Oncologic Radiology Department, Musculoskeletal Unit, AP-HP, Hôpital Pitié-Salpêtrière; G. Solé, MD, Professor of Medicine, Center of Reference for Neuromuscular Disorders AOC, Department of Neurology, Bordeaux University Hospital; O. Benveniste, MD, PhD, Professor of Medicine, Department of Internal Medicine and Clinical Immunology, AP-HP, Hôpital Pitié-Salpêtrière.
Abstract
OBJECTIVE: A pathogenic role of anti-3-hydroxy-3-methylglutaryl-coenzyme A reductase (anti-HMGCR) antibodies has been proposed. Our objective was to assess efficacy of rituximab (RTX) in anti-HMGCR immune-mediated necrotizing myopathy. METHODS: All patients who had been treated with RTX were retrospectively reviewed to assess features and outcome. RESULTS: Three of 9 patients demonstrated stable or improved muscle strength ± decline in creatine kinase levels, or T2/short-tau inversion recovery hypersignal decrease on magnetic resonance imaging following RTX treatment. RTX permitted intravenous immunoglobulin discontinuation and corticosteroid reduction to low dose in 2 patients. CONCLUSION: One-third of patients with refractory anti-HMGCR had improved strength or other evidence of improved disease activity following RTX treatment.
OBJECTIVE: A pathogenic role of anti-3-hydroxy-3-methylglutaryl-coenzyme A reductase (anti-HMGCR) antibodies has been proposed. Our objective was to assess efficacy of rituximab (RTX) in anti-HMGCR immune-mediated necrotizing myopathy. METHODS: All patients who had been treated with RTX were retrospectively reviewed to assess features and outcome. RESULTS: Three of 9 patients demonstrated stable or improved muscle strength ± decline in creatine kinase levels, or T2/short-tau inversion recovery hypersignal decrease on magnetic resonance imaging following RTX treatment. RTX permitted intravenous immunoglobulin discontinuation and corticosteroid reduction to low dose in 2 patients. CONCLUSION: One-third of patients with refractory anti-HMGCR had improved strength or other evidence of improved disease activity following RTX treatment.
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Keywords:
ANTI-3-HYDROXY-3-METHYLGLUTARYL-COENZYME A REDUCTASE; IMMUNE-MEDIATED NECROTIZING MYOPATHY; MYOSITIS; RITUXIMAB
Authors: Chong Hsien Yeo; Aziman Yaakub; Margaret Choon Lee Wang; Sylvester Andrew Shim; Pui L Chong; Muhammad Abdul Mabood Khalil; Pemasiri U Telisinghe; Kian C Lim; Jackson Tan; Vui H Chong Journal: Cureus Date: 2022-05-06