Inotuzumab ozogamicin is effective in relapsed/refractory extramedullary B acute lymphoblastic leukemia 11 Medical and Health Sciences 1112 Oncology and Carcinogenesis 11 Medical and Health Sciences 1102 Cardiorespiratory Medicine and Haematology

Luca Bertamini, Jacopo Nanni, Giovanni Marconi, Mariachiara Abbenante, Valentina Robustelli, Francesco Bacci, Antonella Matti, Stefania Paolini, Chiara Sartor, Silvia Lo Monaco, Maria Chiara Fontana, Stefano De Polo, Michele Cavo, Antonio Curti, Giovanni Martinelli, Cristina Papayannidis

Research output: Contribution to journalArticle

Abstract

Background: Extramedullary involvement of B-cell Acute Lymphoblastic Leukemia (EM-ALL) is a rare occurrence, characterized by dismal outcome and the absence of a defined and shared therapeutic approach. In the landscape of innovative compounds, inotuzumab ozogamicin (IO) is a promising drug, whose mechanism of action relies on the killing of CD22 positive leukemic cells, through the delivery, after cell binding, of a molecule of calicheamicin. Case presentation: We report two cases of CD22 positive relapsed EM-ALL treated with IO, obtained as compassionate use. Case 1, a 66 years old woman, affected by Philadelphia (Ph) negative B-ALL, relapsed with extramedullary involvement after 6 standard chemotherapy courses, who reached a complete metabolic response with IO treatment. Case 2, a 67 years old man with Ph positive B-ALL, initially treated with ponatinib, a third generation tyrosine-kinase inhibitor (TKI), obtaining a prolonged deep molecular remission. Nevertheless, for skin relapse during TKI treatment, the patient received local radiotherapy and, shortly after, standard chemotherapy, as multiple abdominal sites of relapse were detected too, with no response. The patient then received IO, obtained as compassionate use, with a good metabolic response. Conclusions: These two cases suggest a possible key role of IO in the setting of advanced CD22 positive ALL, and underline its potential activity also in patients with EM involvement, relapsed after or refractory to conventional chemotherapy. Despite the well known hepatotoxic effect of the compound (Sinusoid Occlusive Syndrome), neither of them had such adverse event, moreover the second patient safely underwent allogeneic bone marrow transplantation.

Original languageEnglish
Article number1117
JournalBMC Cancer
Volume18
Issue number1
DOIs
Publication statusPublished - Nov 15 2018

Keywords

  • Acute lymphoblastic leukemia
  • Extramedullary
  • Inotuzumab ozogamicin
  • PET-CT scan

ASJC Scopus subject areas

  • Oncology
  • Genetics
  • Cancer Research

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    Bertamini, L., Nanni, J., Marconi, G., Abbenante, M., Robustelli, V., Bacci, F., Matti, A., Paolini, S., Sartor, C., Monaco, S. L., Fontana, M. C., De Polo, S., Cavo, M., Curti, A., Martinelli, G., & Papayannidis, C. (2018). Inotuzumab ozogamicin is effective in relapsed/refractory extramedullary B acute lymphoblastic leukemia 11 Medical and Health Sciences 1112 Oncology and Carcinogenesis 11 Medical and Health Sciences 1102 Cardiorespiratory Medicine and Haematology. BMC Cancer, 18(1), [1117]. https://doi.org/10.1186/s12885-018-5026-x