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Here, we describe a case of GT-related T-cell acute lymphoblastic leukemia (T-ALL) that developed 4.7 years after treatment. The patient underwent chemotherapy and haploidentical transplantation and is currently in remission. Blast cells contain a single vector insertion activating the LIM-only protein 2 (<jats:italic>LMO2)<\/jats:italic> proto-oncogene, confirmed by physical interaction, and low Adenosine Deaminase (ADA) activity resulting from methylation of viral promoter. The insertion is detected years before T-ALL in multiple lineages, suggesting that further hits occurred in a thymic progenitor. Blast cells contain known and novel somatic mutations as well as germline mutations which may have contributed to transformation. Before T-ALL onset, the insertion profile is similar to those of other ADA-deficient patients. 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GT for ADA-SCID was developed at SR-TIGET and licensed to GlaxoSmithKline (GSK) in 2010. The treatments under Named Patient Program and Hospital Exemption were provided free of charge by GSK. Strimvelis Marketing Authorization in Europe occurred in 2016 (under GSK holding) and then transferred to Orchard Therapeutics (Netherlands) B.V. in 2018. Following disinvestment by Orchard, Fondazione Telethon became the Strimvelis Marketing Authorization Holder in July 2023. The product is also currently licensed in Iceland, Norway, Liechtenstein, and UK. A.A: was the PI of pilot and pivotal SR-TIGET clinical trial of GT for ADA SCID. A.A. is a member of the Committee for Advanced Therapies (CAT) and his views are personal and may not be understood or quoted as being made on behalf of the European Medicine Agency (EMA). M.P. Cicalese is the PI of the Strimvelis Registry, RIS and RMMs studies. 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