Dramatic Recovery after Etoposide Phosphate Infusion for Hemophagocytic Lymphohistiocytosis/Macrophage Activation Syndrome following Treatment with Tisagenlecleucel in a Young Patient with Relapsed Acute Lymphoblastic Leukemia: A Case Report

Abstract

The occurrence of a secondary hemophagocytic lymphohistiocytosis/macrophage activation syndrome (HLH/MAS) after CAR-T cells infusion is very rare and mostly fatal. Treatment recommendations for such a complication are not yet established. Here we report the dramatic recovery of a HLH/MAS following tisagenlecleucel infusion in a young patient with relapsed acute lymphoblastic leukemia using etoposide phosphate (EP). We propose that monitoring for the occurrence of HLH/MAS should be part of surveillance after CAR T-cells infusion and that EP treatment appears to be useful to control this severe and rare complication.

Risk factors and outcome of Chimeric Antigen Receptor T-Cell patients admitted to Pediatric Intensive Care Unit: CART-PICU study

“…Nevertheless, type of relapse was different: the majority of relapses in patients admitted to PICU admission were CD19- (82%). These might probably relate to high disease burden, which has been associated with higher risk of CD19- relapse ( 47 ). However, it is important to take into consideration that other factors, which are not included in this analysis, have been related to CD19- relapses, such as prior bilatumomab nonresponse or older age ( 48 – 50 ).…”

Treatment strategies for progressive immune effector cell-associated hemophagocytic lymphohistiocytosis-like syndrome: case series

“…Ultimately, low-dose etoposide (50 mg/m 2 )-a topoisomerase II inhibitor, was given on D+25 because of its proven efficacy in the treatment of primary 7,8 and secondary 9 HLH to specifically target the lymphocytosis by inducing T-cell apoptosis and decreasing the inflammatory response. 10 With a single dose, the absolute lymphocyte count (ALC) decreased from 38,870/mcL to 1,190/mcL and the ferritin levels rapidly decreased from over 70,000 ng/mL to less than 20,000 ng/mL, with concurrent improvement in inflammatory markers, and steroids and anakinra were weaned. D+30 restaging reveled a CR, which was maintained at 3 months.…”

Hemophagocytic lymphohistiocytosis secondary to CAR‐T cells: Update from the FDA and Vizient databases

“…3 Except for two case reports of patients developing HLH after the infusion of CAR-T cells, no additional data on this potentially lethal toxicity were published. 6,7 As far as experimental, non-FDA approved CAR T cells are concerned, a significantly higher incidence was recently observed with a CD22 product. 8 Therefore, we decided to access the FDA and Vizient databases to get further insight into trends, incidence, risk factors, and potential outcomes of CAR-HLH.The FDA Adverse Events Reporting System (FAERS) was last accessed on March 3, 2022, to collect data on the reported cases of CAR-HLH.…”