Abstract
Thrombotic microangiopathy (TMA) is an uncommon but severe complication that may occur in cancer patients under gemcitabine chemotherapy. Gemcitabine-induced thrombotic microangiopathy (G-TMA) can clinically and biologically present as atypical hemolytic uremic syndrome, with activation of the complement pathway asking the question of the use of eculizumab. We describe here the case of a patient suffering from metastatic cholangiocarcinoma treated by gemcitabine for 4 years leading to the 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 the renal biopsy of a very late G-TMA. 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 the re-initiation of gemcitabine under eculizumab treatment. This atypical case of TMA illustrates the importance of recognizing, even belatedly, this rare but serious complication of chemotherapy. It asks the question of rechallenging discontinued chemotherapy notably under eculizumab cover, in this population with a high risk of cancer progression.