Hideo Nishizawa

Cardiogenic shock due to takotsubo cardiomyopathy associated with serotonin syndrome

A 65-year-old woman, with valvular heart disease, atrial fibrillation, and depression, presented to the emergency room due to dyspnea with shock state accompanied by agitation. An electrocardiogram showed ST segment elevation in leads II, III, aVF, I, aVL, and V4-6. An echocardiography revealed extensive akinesis in the apex, but hyperkinesis in the base, with apical ballooning appearance. An emergent coronary angiography showed no obstructive disease. The patient required intubation under mechanical ventilator, and an intra-aortic balloon pump to recover from shock state. She had been taking maprotiline, a tetracyclic antidepressant, and had added dextromethorphan, a cough suppressant, just before admission. The patient was diagnosed with takotsubo cardiomyopathy associated with serotonin syndrome due to serotonergic drug interactions. After discontinuation of these drugs and administration of serotonin antagonist under mechanical supportive care, she became hemodynamically stable. Apical ballooning was completely resolved 2 weeks later, and she was discharged well. We diagnosed serotonin syndrome manifesting as excessive serotonin toxicity that resulted in a hyperserotonergic and hyperadrenergic state, causing takotsubo cardiomyopathy. Here, we report a case of takotsubo cardiomyopathy associated with serotonin syndrome. This case suggests that serotonin syndrome should be recognized promptly and complications, including takotsubo cardiomyopathy, need to be treated appropriately. <Learning objective: We must closely monitor serotonergic agents because serotonin syndrome can occur from a combination some serotonergic drugs, even when each is used at a therapeutic dose. Serotonin syndrome can lead to not only serotonergic but also hyperadrenergic state that may be a trigger of takotsubo cardiomyopathy. Serotonin syndrome should be diagnosed surely to ensure the prompt initiation of the treatment included discontinuation of the precipitating drugs because the disease occasionally progresses rapidly to fatal condition.>.

Successful treatment of fatal macrophage activation syndrome and haemophagocytic lymphohistiocytosis by combination therapy including continuous haemodiafiltration with a cytokine-adsorbing haemofilter (AN69ST) in a patient with systemic lupus erythematosus

Abstract Continuous haemodiafiltration (CHDF) with a cytokine-adsorbing haemofilter (AN69ST–CHDF) was successfully used to treat systemic lupus erythematosus (SLE) complicated with macrophage activation syndrome (MAS) and haemophagocytic lymphohistiocytosis (HLH) in a 35-year-old male. Five months before admission, serositis, proteinuria, and neuropsychiatric SLE recurred. He was treated with glucocorticoid, rituximab, and intravenous cyclophosphamide. He was subsequently admitted for the examination of hepatic disorder and leukopenia. Following admission, he was diagnosed with MAS/HLH, and glucocorticoid pulse therapy was initiated. Although leukopenia temporarily improved, his pancytopenia and hepatic dysfunction worsened, leading to multiple organ dysfunction syndrome (MODS) on day 22. Because hypercytokinemia contributed to MAS/HLH exacerbation, we implemented a combination therapy with cyclosporine A and plasma exchange. This strategy failed to improve MODS; thus, AN69ST–CHDF was initiated and added rituximab which led to improved hypercytokinemia and eventual recovery from MODS. AN69ST–CHDF might be an effective therapeutic option for MAS/HLH.