{Reference Type}: Case Reports {Title}: 6-Mercaptopurine-associated Sinusoidal Obstructive Syndrome During Interim Maintenance I: A Case Report. {Author}: Voelz K;Miller G;Lee-Miller C; {Journal}: J Pediatr Hematol Oncol {Volume}: 46 {Issue}: 5 {Year}: 2024 07 1 {Factor}: 1.17 {DOI}: 10.1097/MPH.0000000000002883 {Abstract}: Thiopurine-methyltransferase (TPMT) and nudix-hydrolase-15 (NUDT15) are enzymes relevant to the metabolism of thiopurine medications, used to treat immunologic disorders and malignancies. Standard dosing administered in the setting of TPMT/NUDT15 dysfunction can cause excessive cytotoxic metabolites and life-threatening complications. We describe an adolescent with high-risk B-cell acute lymphoblastic leukemia (ALL) whose TPMT/NUDT15 status was unknown due to lack of insurance approval for genetic testing. He subsequently developed myelosuppression and severe veno-occlusive disease (VOD) after receiving 6-mercaptopurine (6-MP). Our patient provides an example of a very rare 6-MP-related toxicity and the potential benefit of TPMT/NUDT15 screening before initiating thiopurine therapy.