Takahashi Nobuhisa, Sano Hideki, Mochizuki Kazuhiro, Kobayashi Shogo, Ohara Yoshihiro, Kikuta Atsushi
Department of Pediatric Oncology, Fukushima Medical University, Fukushima, Japan.
Case Rep Oncol. 2021 Mar 22;14(1):525-530. doi: 10.1159/000515011. eCollection 2021 Jan-Apr.
Chronic myeloid leukemia (CML) is an uncommon entity in pediatric patients. CML in chronic phase (CML-CP) has a relatively favorable outcome. Leukostasis occurs in 9.7% of patients with CML. One of the most serious leukostasis-related complications is intracranial hemorrhage (ICH). However, this is very rare in patients with CML-CP, and few early mortalities have been reported in CML patients with leukostasis. We report the case of a 14-year-old female patient with CML-CP who developed ICH 8 days after admission. A 14-year-old girl developed symptoms of fatigue and slight fever and was diagnosed with CML-CP. She was treated with imatinib and received low-molecular-weight heparin owing to coagulation abnormalities. However, 6 days later, she developed sensorineural hearing loss, which is a symptom of leukostasis. She received hydroxyurea to reduce her white blood cell (WBC) count, and her treatment was changed from imatinib to nilotinib. The WBC and platelet counts remained unchanged, blast counts did not increase, and mild coagulation abnormality persisted. Eight days after admission, she suddenly lost consciousness and experienced respiratory arrest. Cranial computed tomography revealed multiple ICH lesions and brain hernia. She received intensive care but was diagnosed with brain death by electroencephalography and died 14 days after hospitalization. ICH is very rare in patients with CML-CP; however, patients with leukostasis and coagulation abnormalities can develop severe hemorrhage, even in the chronic phase. Thus, it is necessary to accurately estimate the cause and provide appropriate treatment for these patients.
慢性髓性白血病（CML）在儿科患者中并不常见。慢性期慢性髓性白血病（CML-CP）的预后相对较好。白细胞淤滞发生在9.7%的CML患者中。与白细胞淤滞相关的最严重并发症之一是颅内出血（ICH）。然而，这在CML-CP患者中非常罕见，并且很少有关于白细胞淤滞的CML患者早期死亡的报道。我们报告了一例14岁女性CML-CP患者，该患者在入院8天后发生了ICH。一名14岁女孩出现疲劳和低热症状，被诊断为CML-CP。她接受了伊马替尼治疗，并因凝血异常接受了低分子量肝素治疗。然而，6天后，她出现了感音神经性听力损失，这是白细胞淤滞的一种症状。她接受了羟基脲以降低白细胞（WBC）计数，治疗从伊马替尼改为尼洛替尼。WBC和血小板计数保持不变，原始细胞计数未增加，轻度凝血异常持续存在。入院8天后，她突然失去意识并出现呼吸骤停。头颅计算机断层扫描显示多处ICH病灶和脑疝。她接受了重症监护，但脑电图诊断为脑死亡，住院14天后死亡。ICH在CML-CP患者中非常罕见；然而，有白细胞淤滞和凝血异常的患者即使在慢性期也可能发生严重出血。因此，有必要准确评估病因并为这些患者提供适当的治疗。
