A severe case of neuro-Sjögren’s syndrome induced by pembrolizumab

Ghosn, Jaqueline; Vicino, Alex; Michielin, Olivier; Coukos, George; Küntzer, Thierry; Obéid, Michel

doi:10.1186/s40425-018-0429-4

Cited by 40 publications

(14 citation statements)

References 20 publications

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“…Clinicians are progressively confronted with new types of irAEs with increasingly pleomorphic presentation [11]. Herein, we report the case of a patient displaying a severe and atypical upper GI tract irAE related to nivolumab immunotherapy.…”

Section: Discussionmentioning

confidence: 99%

A severe case of refractory esophageal stenosis induced by nivolumab and responding to tocilizumab therapy

Horisberger¹,

Rosa²,

Zurcher³

et al. 2018

j. immunotherapy cancer

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BackgroundThe prevalence of esophageal stenosis caused by immune checkpoint inhibitors in the context of induced immune mucositis and esophagitis is extremely rare.Case presentationWe report the case of a patient with stage IV pulmonary adenocarcinoma treated for 6 months with nivolumab who developed bilateral sterile conjunctivitis followed by oropharyngeal mucositis and esophagitis complicated by a severe esophageal stenosis. The laryngeal margin and hypopharyngeal mucosa appeared highly inflammatory with fibrinous deposits. Esophagogastroduodenoscopy revealed mucositis with a scar-like structure immediately below the upper esophageal sphincter with nonulcerative mucosa and an inflammatory aspect of the entire esophagus. No involvement of the stomach was observed. Oropharynx biopsies displayed marked lymphocytic T cell-infiltration with several foci of monocellular necrosis in the squamous epithelium. No morphologic evidence of adenocarcinoma and no signs of mycotic, bacterial or viral infection were noted. A blood sample revealed a discrete increase in the erythrocyte sedimentation rate (ESR) with no eosinophilia or leukocytosis. Liver and kidney function panel tests were normal. A thoracoabdominal CT scan reported no evidence of disease recurrence. Despite multiple boluses of methylprednisolone and high doses of prednisone continued for several months, the patient experienced very rapid symptomatological reappearance during three steroid tapering attempts and aggravation of his esophageal stenosis to an aphagic stage, requiring a nasogastric tube. This long course of high-dose corticosteroid treatment was complicated with osteoporosis-induced fractures with several spontaneous compressions of thoracolumbar vertebrae requiring an enlarged T10 to L5 cementoplasty. Anti-IL-6 blockade therapy with tocilizumab resulted in excellent clinical response, allowing the total resolution of the immune-related adverse events (irAEs) and leading to successful steroid tapering.ConclusionsHerein, we describe the first case of a patient who developed autoimmune mucositis and esophagitis complicated by a severe refractory esophageal stenosis induced during treatment by nivolumab, which completely resolved after personalized treatment with tocilizumab, suggesting a role of IL-6 blockade in the management of severe steroid refractory esophageal stenosis and more broadly in refractory immune-related adverse events.

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Section: Discussionmentioning

confidence: 99%

A severe case of refractory esophageal stenosis induced by nivolumab and responding to tocilizumab therapy

Horisberger¹,

Rosa²,

Zurcher³

et al. 2018

j. immunotherapy cancer

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“…Collectively, the neurological and neuromuscular complications of ICI treatment include myopathies, neuromuscular junction disorders, peripheral neuropathies (PNPs; including axonal and demyelinating polyradiculoneuropathies), length-dependent and non-length-dependent neuropathies, asymmetric mononeuritis multiplex, cerebellar ataxia, retinopathy, bilateral internuclear ophthalmoplegia and headache 93 . We emphasize, however, that a growing body of literature on neurological irAEs with pleomorphic presentations continues to accumulate 94 .…”

Section: Neurological Iraesmentioning

confidence: 92%

Adverse effects of immune-checkpoint inhibitors: epidemiology, management and surveillance

Martins

Latifyan

Sykiotis³

et al. 2019

Nat Rev Clin Oncol

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1,183

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Immune-checkpoint inhibitors (ICIs), including anti-cytotoxic T lymphocyte antigen 4 (CTL A-4), anti-programmed cell death 1 (PD-1) and anti-programmed cell death 1 ligand 1 (PD-L1) antibodies, are arguably the most important development in cancer therapy over the past decade. The indications for these agents continue to expand across malignancies and disease settings, thus reshaping many of the previous standard-of-care approaches and bringing new hope to patients. One of the costs of these advances is the emergence of a new spectrum of immune-related adverse events (irAEs), which are often distinctly different from the classical chemotherapy-related toxicities. Owing to the growing use of ICIs in oncology, clinicians will increasingly be confronted with common but also rare irAEs; hence, awareness needs to be raised regarding the clinical presentation, diagnosis and management of these toxicities. In this Review, we provide an overview of the various types of irAEs that have emerged to date. We discuss the epidemiology of these events and their kinetics, risk factors, subtypes and pathophysiology, as well as new insights regarding screening and surveillance strategies. We also highlight the most important aspects of the management of irAEs.

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“…One patient with neuro-Sjögren's syndrome was successfully treated with rituximab after intravenous pulses of methylprednisolone, immunoglobulins and one dose of cyclophosphamide. 87 Rituximab was also used in one patient with acral vasculitis without improvement and the need of surgical amputation. 105 7.…”

Section: Recommendationmentioning

confidence: 99%

EULAR points to consider for the diagnosis and management of rheumatic immune-related adverse events due to cancer immunotherapy with checkpoint inhibitors

et al. 2020

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BackgroundRheumatic and musculoskeletal immune-related adverse events (irAEs) are observed in about 10% of patients with cancer receiving checkpoint inhibitors (CPIs). Given the recent emergence of these events and the lack of guidance for rheumatologists addressing them, a European League Against Rheumatism task force was convened to harmonise expert opinion regarding their identification and management.MethodsFirst, the group formulated research questions for a systematic literature review. Then, based on literature and using a consensus procedure, 4 overarching principles and 10 points to consider were developed.ResultsThe overarching principles defined the role of rheumatologists in the management of irAEs, highlighting the shared decision-making process between patients, oncologists and rheumatologists. The points to consider inform rheumatologists on the wide spectrum of musculoskeletal irAEs, not fulfilling usual classification criteria of rheumatic diseases, and their differential diagnoses. Early referral and facilitated access to rheumatologist are recommended, to document the target organ inflammation. Regarding therapeutic, three treatment escalations were defined: (1) local/systemic glucocorticoids if symptoms are not controlled by symptomatic treatment, then tapered to the lowest efficient dose, (2) conventional synthetic disease-modifying antirheumatic drugs, in case of inadequate response to glucocorticoids or for steroid sparing and (3) biological disease-modifying antirheumatic drugs, for severe or refractory irAEs. A warning has been made on severe myositis, a life-threatening situation, requiring high dose of glucocorticoids and close monitoring. For patients with pre-existing rheumatic disease, baseline immunosuppressive regimen should be kept at the lowest efficient dose before starting immunotherapies.ConclusionThese statements provide guidance on diagnosis and management of rheumatic irAEs and aim to support future international collaborations.

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A severe case of neuro-Sjögren’s syndrome induced by pembrolizumab

Cited by 40 publications

References 20 publications

A severe case of refractory esophageal stenosis induced by nivolumab and responding to tocilizumab therapy

A severe case of refractory esophageal stenosis induced by nivolumab and responding to tocilizumab therapy

Adverse effects of immune-checkpoint inhibitors: epidemiology, management and surveillance

EULAR points to consider for the diagnosis and management of rheumatic immune-related adverse events due to cancer immunotherapy with checkpoint inhibitors

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