Rituximab in the Treatment of Refractory Anti-HMGCR Immune-mediated Necrotizing Myopathy

Océane Landon-Cardinal; Yves Allenbach; Antoine Soulages; Aude Rigolet; Baptiste Hervier; Nicolas Champtiaux; Quentin Monzani; Guilhem Solé; Olivier Benveniste

doi:10.3899/jrheum.171495

Rituximab in the Treatment of Refractory Anti-HMGCR Immune-mediated Necrotizing Myopathy

J Rheumatol. 2019 Jun;46(6):623-627. doi: 10.3899/jrheum.171495. Epub 2018 Dec 15.

Authors

Océane Landon-Cardinal^{1

2

3}, Yves Allenbach^{4

5

6}, Antoine Soulages^{4

5

6}, Aude Rigolet^{4

5

6}, Baptiste Hervier^{4

5

6}, Nicolas Champtiaux^{4

5

6}, Quentin Monzani^{4

5

6}, Guilhem Solé^{4

5

6}, Olivier Benveniste^{4

5

6}

Affiliations

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.

PMID: 30554155
DOI: 10.3899/jrheum.171495

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.

Keywords: ANTI-3-HYDROXY-3-METHYLGLUTARYL-COENZYME A REDUCTASE; IMMUNE-MEDIATED NECROTIZING MYOPATHY; MYOSITIS; RITUXIMAB.

MeSH terms

Adult
Autoantibodies
Autoimmune Diseases / drug therapy*
Autoimmune Diseases / immunology
Female
Humans
Hydroxymethylglutaryl CoA Reductases / immunology*
Immunologic Factors / pharmacology
Immunologic Factors / therapeutic use*
Male
Middle Aged
Muscle Strength / drug effects
Muscle, Skeletal / immunology
Myositis / drug therapy*
Myositis / immunology
Retrospective Studies
Rituximab / pharmacology
Rituximab / therapeutic use*
Treatment Outcome
Young Adult

Substances

Autoantibodies
Immunologic Factors
Rituximab
HMGCR protein, human
Hydroxymethylglutaryl CoA Reductases