Thrombotic microangiopathy after long lasting treatment by Gemcitabine: description, evolution and treatment of a rare case report

Abstract Background : Thrombotic microangiopathy is an uncommon but severe complication that may occur in cancer patients under Gemcitabine chemotherapy. Gemcitabine induced thrombotic microangiopathy can clinically and biologically present as atypical hemolytic uremic syndrome, with activation of the complement pathway asking the question of the use of Eculizumab. Case presentation : We describe here the case of a patient suffering from metastatic cholangiocarcinoma treated by Gemcitabine for 4 years leading to remission of the underlying neoplasia. Despite an impressive response to therapy, she developed thrombopenia, regenerative anemia and acute kidney injury leading to the suspicion then diagnosis based on renal biopsy of a very late Gemcitabine associated thrombotic microangiopathy. Spontaneous evolution after treatment interruption was favorable without dialysis requirement. However, in this case where Gemcitabine is the only chemotherapy remaining for a mortal underlying condition, we discussed re-initiation of Gemcitabine under Eculizumab treatment. Conclusions : This atypical case of thrombotic microangiopathy illustrates the importance of recognizing, even belatedly, this rare but serious complication of chemotherapy. It asks the question of resumption of discontinued chemotherapy notably under Eculizumab cover, in this population with high risk of cancer progression.