Lee Joonyeop, Yoon Jae-Ho, Kwag Daehun, Lee Jong-Hyuk, Kim Tong Yoon, Min Gi June, Park Sung-Soo, Park Silvia, Lee Sung-Eun, Cho Byung-Sik, Eom Ki-Seong, Kim Yoo-Jin, Kim Hee-Je, Min Chang-Ki, Cho Seok-Goo, Wook Lee Jong, Hak Lee Sung, Lee Seok
Department of Hematology, Catholic Hematology Hospital and Leukemia Research Institute, Seoul St. Mary's Hospital, College of Medicine, The Catholic University of Korea, Seoul, Republic of Korea.
Department of Hospital Pathology, The Catholic University of Korea, Seoul, Republic of Korea.
Ther Adv Hematol. 2021 Dec 21;12:20406207211066176. doi: 10.1177/20406207211066176. eCollection 2021.
Inotuzumab ozogamicin (INO) showed improved treatment outcomes for relapsed or refractory B-cell precursor acute lymphoblastic leukemia (BCP-ALL) but can induce hepatotoxic adverse events. Hepatic venoocclusive disease/sinusoidal obstruction syndrome (VOD/SOS) frequently develops after allogeneic hematopoietic cell transplantation (allo-HCT), and INO is a strong pretransplant risk factor. However, VOD/SOS can occur just after INO therapy. Here, we describe a BCP-ALL patient treated with INO for isolated extramedullary relapse after allo-HCT. The patient experienced elevated liver enzymes with ascites at 21 days from the last INO dose. Although she met the criteria for VOD/SOS, the diagnosis was challenging because of her ongoing hepatic graft--host disease (GVHD) and normal portal vein flow on Doppler sonogram. The radiologist suggested liver cirrhosis based on computed tomography, with VOD/SOS, liver cirrhosis, and GVHD assumed to be differential diagnoses. She received supportive care with GVHD management; however, due to progressive hepatic failure, we conducted emergent deceased-donor liver transplantation, and the pathologic findings indicated VOD/SOS. Her leukemia was stable, but she died of sepsis after 3 months. INO use is a high-risk factor for VOD/SOS, but an accurate diagnosis can be challenging due to various hepatic complications. Early diagnosis and proper management for VOD/SOS is important for improved outcomes.
伊尼妥单抗奥佐米星(INO)对复发或难治性B细胞前体急性淋巴细胞白血病(BCP-ALL)显示出改善的治疗效果,但可诱发肝毒性不良事件。肝静脉闭塞病/窦性阻塞综合征(VOD/SOS)常在异基因造血细胞移植(allo-HCT)后频繁发生,而INO是移植前的一个重要危险因素。然而,VOD/SOS也可能在INO治疗后立即出现。在此,我们描述了一名在allo-HCT后因孤立性髓外复发接受INO治疗的BCP-ALL患者。该患者在最后一剂INO后21天出现肝酶升高并伴有腹水。尽管她符合VOD/SOS的标准,但由于其持续存在的肝移植物抗宿主病(GVHD)以及多普勒超声检查显示门静脉血流正常,诊断颇具挑战性。放射科医生基于计算机断层扫描提示肝硬化,将VOD/SOS、肝硬化和GVHD视为鉴别诊断。她接受了针对GVHD的支持性治疗;然而,由于进行性肝衰竭,我们进行了紧急的尸体供肝肝移植,病理结果显示为VOD/SOS。她的白血病病情稳定,但3个月后死于败血症。使用INO是发生VOD/SOS的高危因素,但由于各种肝脏并发症,准确诊断可能具有挑战性。早期诊断和对VOD/SOS的恰当管理对于改善预后很重要。