Obinutuzumab is a humanized anti-CD20 monoclonal antibody commonly used in the treatment of non-Hodgkin lymphoma and chronic lymphocytic leukaemia (CLL). In follicular lymphoma (FL) and CLL, obinutuzumab demonstrated superior efficacy compared to rituximab, however at the cost of increased toxicity. 1,2 Among the side effects of obinutuzumab, the Summary of Product Characteristics (SPC) mentions 'severe and lifethreatening thrombocytopenia including acute thrombocytopenia (occurring within 24 h after the infusion)'. 3 However, obinutuzumab-induced acute thrombocytopenia (OIAT) has been rarely reported. Furthermore, the management of OIAT is challenging because of the lack of specific guidelines.Here, we report the case of two patients who experienced acute and severe thrombocytopenia after obinutuzumab infusion. We also performed a review of the literature about this rare but life-threatening side effect. Using PubMed®, we searched for articles published between 2015 and 2023 containing the words 'obinutuzumab', 'thrombocytopenia', 'severe', 'acute', 'lymphoma' or 'leukemia'.