%0 Case Reports %T Pregnancy-associated atypical hemolytic uremic syndrome. Case report. %A Barrera-Hoffmann C %A Mariaca-Ortíz Y %A Ruiz-Villa JG %A Cuevas-Cruz LE %A López-Mendoza MDR %A Briones-Garduño JC %J J Obstet Gynaecol Res %V 50 %N 7 %D 2024 Jul 9 %M 38720638 %F 1.697 %R 10.1111/jog.15958 %X Pregnancy-associated atypical hemolytic uremic syndrome (P-aHUS) is a rare disease. There are only few reports in the literature, and most are in the puerperium period. It is a thrombotic microangiopathy (TMA) characterized for microangiopathic hemolytic anemia, thrombocytopenia, and renal dysfunction. We report the case of a pregnant patient at 26.3 gestation weeks, who developed clinical features of TMA, neurological alterations, and septic shock; then after fetus and placental delivery, no clinical improvement was observed; a diagnostic protocol was performed due to suspicion of P-aHUS, showing improvement after the plasma exchange sessions and eculizumab. We present here a brief review of the case since it is an entity that needs to be suspected during pregnancy when TMA features and requires an immediate diagnosis to provide timely treatment.