Abstract
BackgroundCapillary leak syndrome (CLS) is a rare condition characterized by recurrent episodes of generalized edema and severe hypotension associated with hypoproteinemia. Interleukin-11 (IL-11) is a promising therapeutic agent for thrombocytopenia. A direct correlation between IL-11 and CLS has never been reported previously, particularly in patients with hepatic carcinoma.Case presentationWe describe two cases of CLS after IL-11 administration in two males with thrombocytopenia. Case 1 was a 46-year-old man with recurrence of hepatic carcinoma who was treated with IL-11 (3 mg per day). After four days of therapy, hypotension and hypoproteinemia were detected. The chest X-ray and B ultrasound of the abdomen showed pleural effusion and ascites. IL-11 was then discontinued, fluid resuscitation was performed, and fresh frozen plasma and packed red blood cells were transfused into this patient. The patient had recovered after 19 days of treatment.Case 2 was a 66-year-old man who had undergone radiofrequency ablation (RFA) for hepatic carcinoma. He was treated with IL-11 (3 mg per day) for thrombocytopenia. After two days of therapy, this patient complained of dyspnea with bilateral edema of the hands. Laboratory values showed hypoproteinemia. IL-11 was stopped and human albumin was transfused at a rate of 10 g per day. On the 4th day, fluid resuscitation was performed. The patient had recovered after treatment for two weeks.ConclusionsThe detection of IL-11-induced CLS supports the hypothesis that CLS could be a severe side effect of IL-11 treatment in some patients. These two case reports also demonstrate that patients with hepatic carcinoma who experience this rare form of CLS after treatment with IL-11 seem to respond to a therapeutic regimen that involves hydroxyethyl starch, albumin, and diuretic therapy. Liver cancer patients might be more susceptible to CLS because of poor liver function and hypersplenia. In addition, bleeding after RFA might be a further inducer of CLS.
Highlights
Capillary leak syndrome (CLS) is a rare condition characterized by recurrent episodes of generalized edema and severe hypotension associated with hypoproteinemia
The detection of IL-11-induced CLS supports the hypothesis that CLS could be a severe side effect of IL-11 treatment in some patients
These two case reports demonstrate that patients with hepatic carcinoma who experience this rare form of CLS after treatment with IL-11 seem to respond to a therapeutic regimen that involves hydroxyethyl starch, albumin, and diuretic therapy
Summary
IL-11 is a cytokine derived from stromal cells that has multiple effects on thrombocytopenia induced by chemotherapy or bone marrow transplantation (BMT). According to some clinical trials, the proportion of hemorrhagic complications after RFA is approximately 1.5% [18] Specific treatments, such as blood transfusion and drainage, are required in high-risk patients. Liver pathologies have a negative influence on numerous conditions, including those that affect the endothelial system [23] In this report, both patients had chronic bleeding after RFA, which indicated that injury of the liver and hypersplenia might be a precipitating factor for CLS. Further studies are necessary with the objective of collecting sufficient patients with CLS to observe the natural history of the disease and evaluate the efficacy of empiric treatments These two cases should help to improve the understanding and treatment of the potentially deadly CLS that is related to IL-11 and has been recognized in this study. A copy of the written consent is available for review by the Editor-in-Chief of this journal
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