10.5061/DRYAD.6R19M91
Touat, Mehdi
Harvard Medical School
Maisonobe, Thierry
OncoNeuroTox Group, Center for Patients With Neurological Complications
of Oncologic Treatments, Hôpitaux Universitaires
Pitié-Salpetrière-Charles Foix et Hôpital Percy, Paris, France
Knauss, Samuel
Charité
Salem, Omar Ben Hadj
Hôpital Cochin
Hervier, Baptiste
Centre d'Immunologie et des Maladies Infectieuses
Auré, Karine
CHU Ambroise Paré
Szwebel, Tali-Anne
Hôpital Cochin
Kramkimel, Nora
Hôpital Cochin
Lethrosne, Claire
Service de Pneumologie and Service d’Anatomie Pathologique, Centre
Hospitalier Chartres-Louis-Pasteur, Le-Coudray
Bruch, Jean-Frédéric
Service de Pneumologie and Service d’Anatomie Pathologique, Centre
Hospitalier Chartres-Louis-Pasteur, Le-Coudray
Laly, Pauline
Hôpital Saint-Louis
Cadranel, Jacques
Tenon Hospital
Weiss, Nicolas
Sorbonne University
Béhin, Anthony
Service de Neuropathologie Raymond Escourolle, Départment de
Neurophysiologie Clinique, Département de Médecine Interne et
Immunologie Clinique, and Centre de Référence de Pathologie
Neuromusculaire Paris-Est, APHP, Hôpitaux Universitaires La Pitié
Salpêtrière-Charles Foix, Paris, France
Allenbach, Yves
Sorbonne University
Benveniste, Olivier
Sorbonne University
Lenglet, Timothée
OncoNeuroTox Group, Center for Patients With Neurological Complications
of Oncologic Treatments, Hôpitaux Universitaires
Pitié-Salpetrière-Charles Foix et Hôpital Percy, Paris, France
Psimaras, Dimitri
Inserm
Stenzel, Werner
Berlin Institute of Health
Léonard-Louis, Sarah
Service de Neuropathologie Raymond Escourolle, Départment de
Neurophysiologie Clinique, Département de Médecine Interne et
Immunologie Clinique, and Centre de Référence de Pathologie
Neuromusculaire Paris-Est, APHP, Hôpitaux Universitaires La Pitié
Salpêtrière-Charles Foix, Paris, France
Data from: Immune checkpoint inhibitor–related myositis and myocarditis in
patients with cancer
Dryad
dataset
2019
immune related adverse events
PD-L1
Muscle disease
CTLA-4
PD-1
2019-06-08T00:00:00Z
2019-06-08T00:00:00Z
en
https://doi.org/10.1212/wnl.0000000000006124
76540 bytes
1
CC0 1.0 Universal (CC0 1.0) Public Domain Dedication
Objective: To report the clinicopathological features and outcome of
myositis in patients treated with immune checkpoint inhibitors
(irMyositis). Methods: We retrospectively analyzed patients diagnosed with
irMyositis in tertiary centers in Paris, France, and Berlin, Germany, from
January 2015, to July 2017. Main outcomes were clinical manifestations and
muscle histology, which included MHC-I, C5b-9, CD3, CD4, CD8, CD20, CD68,
PD-1, PD-L1 and PD-L2 immunohistochemical stains. Results: Ten patients
with metastatic cancer were included; median age was 73 years (range,
56-87). Median follow-up duration was 48 weeks (range 16-88). Six patients
developed myositis during nivolumab therapy, one during pembrolizumab, one
during durvalumab, and two during combined nivolumab and ipilimumab.
Median delay between immune checkpoint inhibitor (ICI) initiation and
myositis onset was 25 days (range, 5-87). Clinical manifestations were
dominated by acute or subacute myalgia (8 patients), limb-girdle (7),
axial (7) and oculomotor (7) weakness. Four patients had evidence of
myocarditis. In all patients, CK levels were elevated (median 2668 U/L,
range 1059-16620), while anti-acetylcholine receptor and
myositis-associated antibodies were negative. Electrodiagnostic studies
showed myopathic process without decrement in all patients. Muscle biopsy
constantly showed multifocal necrotic myofibers, sarcolemmal MHC-I and
endomysial inflammation, mainly consisting of CD68+ cells expressing PD-L1
and CD8+ cells expressing PD-1. ICI treatment was withdrawn in all
patients; nine patients received immunosuppressive therapy, which
consistently led to marked clinical improvement. Conclusions: irMyositis
presents with remarkably homogeneous and unique clinicopathological
features, expanding the nosological spectrum of inflammatory myopathies in
patients with cancer. ICI withdrawal and treatment with corticosteroids
improve outcome.
Supplementary Table 1. Detailed auto-antibodies work-up in patients with
irMyositis.Touat et al_irMyositis_R1_Suppl Table 1.pdf