• Mei-Xiao Shen, Fu-Ling Li, Xian-Sheng Luo, and Zhi-Ming Wang.
• Department of Hematology, Affiliated Haikou Hospital of Xiangya Medical College, Central South University, Haikou, China.
• Medicine (Baltimore). 2023 Dec 29; 102 (52): e36022e36022.

RationaleSplenic B-cell lymphoma/leukemia with prominent nucleoli (SBLPN) is a new classification, which is so rare that it lacks clinical data.Patient ConcernsAn increased proportion of prolymphocytes (84%) in the bone marrow smear. Whole exon sequence analysis revealed a TP53 mutation.DiagnosesCombining the clinical features with laboratory test results led to a diagnosis of SBLPN which was made according to the 5th edition of the WHO classification of hematolymphoid tumors, although the patient was diagnosed with B-PLL when guided by the 4th edition of the WHO classification.InterventionsThe use of Ibrutinib as an effective treatment.OutcomesThe patient was in complete remission after 5 months of Ibrutinib and then died of sudden aortic dissection.LessonsIbrutinib was an effective regimen for SBLPN. Aortic dissection might be considered as a suspicious adverse reaction to Ibrutinib.Copyright © 2023 the Author(s). Published by Wolters Kluwer Health, Inc.

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