Systemic Capillary Leak Syndrome (Clarkson’s Disease) as a Complication of Anti–Programmed Death 1 Immunotherapy

Lescure, Céline; Lescoat, Alain; Salé, A.; Bazin, Yann; Duvergé, L.; Desrues, B.

doi:10.1016/j.jtho.2019.02.003

Cited by 17 publications

(20 citation statements)

References 5 publications

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“…She had no notable response to high-dose intravenous corticosteroids, as seen in our patient, which led to her death. 8 In our case, CLS was confirmed after extensive unremarkable work-up for a cardiac, nephrological, and gastrointestinal tract etiology. Angiographic imaging (lymphangiography and lymphoscintigraphy) confirmed our suspicion.…”

Section: Discussionsupporting

confidence: 57%

Successful Treatment of Pembrolizumab-Induced Severe Capillary Leak Syndrome and Lymphatic Capillary Dysfunction

Qin

Vlaminck

Owoyemi

et al. 2021

Mayo Clinic Proceedings: Innovations, Quality & Outcomes

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Although capillary leak syndrome has a high mortality rate, its trigger, diagnosis, and treatment remain a challenge to clinicians because of the poor understanding of its mechanism and lack of treatment guidelines. With the extended use of immune checkpoint inhibitors in modern oncology, immune checkpoint inhibitoreassociated immune-related adverse events have also expanded. We present a case of pembrolizumab-induced capillary leak syndrome and lymphatic capillary dysfunction in which the patient had an excellent clinical response to a tailored treatment strategy.

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Section: Discussionsupporting

confidence: 57%

Successful Treatment of Pembrolizumab-Induced Severe Capillary Leak Syndrome and Lymphatic Capillary Dysfunction

Qin

Vlaminck

Owoyemi

et al. 2021

Mayo Clinic Proceedings: Innovations, Quality & Outcomes

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“…This further rules out an idiopathic cause as those tend to have recurrent attacks [11]. There has been one other case report showing nivolumab-induced SCLS who also responded to IVIG therapy with no improvement on high-dose steroids; however, no follow-up was reported [15]. Currently, known drug-induced causes of SCLS are treated with high-dose steroids and show a good response [13].…”

Section: Discussionmentioning

confidence: 99%

Nivolumab-Induced Systemic Capillary Leak Syndrome as an Ultra Rare Life-Threatening Phenomenon of Late Toxicity and Intravenous Immunoglobulin Efficacy

et al. 2021

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Systemic capillary leak syndrome (SCLS) is a life-threatening disease. It is characterized by severe capillary hyperpermeability to proteins resulting in hemoconcentration, hypoalbuminemia and hypovolemic shock. Its treatment remains supportive, and the prognosis is generally poor. We report on a 51-year old male with melanoma treated with nivolumab for 1 year. 1 month following the completion of the treatment, the patient presented with signs of hypovolemic shock, anasarca, hemoconcentration and hypoalbuminemia. After excluding other diseases, a diagnosis of nivolumab-induced systemic capillary leak syndrome was made. A high dose of intravenous steroid therapy was promptly initiated without any significant clinical improvement. Intravenous immunoglobulin therapy was then administered with normalization of blood pressure, hemoconcentration and complete resolution of anasarca. Intravenous immunoglobulin should be considered a first-line treatment option for this rare phenomenon.

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“…Cases of CLS have recently been reported to be associated with the administration of immune checkpoint inhibitors (ICIs), including Nivolumab 4 and during infection with SARS-CoV-2. 5 These experiences suggest a probable pathogenesis due to a dysregulation of the immune response with cytokine alterations, in the absence of a cellular inflammatory response, impacting on endothelial permeability.…”

Section: Discussionmentioning

confidence: 99%

Capillary leak syndrome induced by neoadjuvant cisplatin and gemcitabine in a patient with bladder cancer

Massafra¹,

Passalacqua²,

Lupo³

et al. 2021

Urology Case Reports

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Capillary leak syndrome (CLS) is a rare disorder associated with an increased capillar permeability due to an endothelial damage, causing leakage of plasma and proteins into the interstitial compartment. CLS is characterized by rapidly developing edema, hypotension and hypoproteinemia. We observed CLS in a 54-year-old man affected by muscle-invasive bladder cancer who received neoadjuvant treatment with CISPLATIN and GEMCITABINE. Treatment with infusion of albumin and increasing corticosteroid doses and diuretics led to a complete regression of all signs and symptoms related to the disorder. Of note, the patient showed an objective complete response to chemotherapy and underwent radical surgery on schedule.

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Systemic Capillary Leak Syndrome (Clarkson’s Disease) as a Complication of Anti–Programmed Death 1 Immunotherapy

Cited by 17 publications

References 5 publications

Successful Treatment of Pembrolizumab-Induced Severe Capillary Leak Syndrome and Lymphatic Capillary Dysfunction

Successful Treatment of Pembrolizumab-Induced Severe Capillary Leak Syndrome and Lymphatic Capillary Dysfunction

Nivolumab-Induced Systemic Capillary Leak Syndrome as an Ultra Rare Life-Threatening Phenomenon of Late Toxicity and Intravenous Immunoglobulin Efficacy

Capillary leak syndrome induced by neoadjuvant cisplatin and gemcitabine in a patient with bladder cancer

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