Tanaka Yasuhiro, Ikunari Ryo, Take Shusaku, Inoue Eiki
Department of Hematology, Shinko Hospital, Kobe, JPN.
Department of Diagnostic Pathology, Kobe University Graduate School of Medicine, Kobe, JPN.
Cureus. 2025 Aug 11;17(8):e89791. doi: 10.7759/cureus.89791. eCollection 2025 Aug.
Burkitt lymphoma (BL) usually involves extranodal regions such as the bone marrow and central nervous system (CNS), but dural involvement is rarely reported. We report a case of the intracranial recurrence of BL involving the CNS, leptomeninges, and dura mater. The patient was a 76-year-old woman who presented with systemic lymphadenopathies, bilateral pleural effusion, and ascites. Laboratory examination showed elevated white blood counts with circulating abnormal cells and thrombocytopenia. Bone marrow examination revealed the presence of abnormal medium-sized lymphocytes with cytoplasmic vacuoles, and IGH::MYC fusion gene was detected in these abnormal lymphocytes. Thus, the diagnosis of BL was made. No CNS lesions were identified at diagnosis. A complete metabolic response was achieved by rituximab combination chemotherapy with prophylactic intrathecal chemotherapy, but isolated intracranial recurrence of BL occurred. In intracranial recurrence, dural involvement was observed in addition to the leptomeninges. High-dose chemotherapy was not effective for relapsed BL, and she died because of disease progression. At autopsy, subdural hematoma (SDH) and dural thickening were pathologically found. This is a rare case of isolated intracranial BL recurrence with dural involvement and nontraumatic SDH as a complication, and we reviewed previous reports of secondary dural involvement in BL. Our case showed the important viewpoint that in the case of dural involvement, clinicians should be aware of the nontraumatic SDH as a complication, and whole-brain radiotherapy may be the optimal treatment for isolated intracranial BL recurrence with dural involvement after rituximab combination chemotherapy.
伯基特淋巴瘤（BL）通常累及骨髓和中枢神经系统（CNS）等结外区域，但硬脑膜受累情况鲜有报道。我们报告一例BL颅内复发，累及CNS、软脑膜和硬脑膜。患者为一名76岁女性，表现为全身淋巴结肿大、双侧胸腔积液和腹水。实验室检查显示白细胞计数升高，伴有循环异常细胞和血小板减少。骨髓检查发现存在有细胞质空泡的异常中等大小淋巴细胞，且在这些异常淋巴细胞中检测到IGH::MYC融合基因。因此，确诊为BL。诊断时未发现CNS病变。通过利妥昔单抗联合化疗及预防性鞘内化疗实现了完全代谢缓解，但出现了孤立性BL颅内复发。在颅内复发时，除软脑膜外还观察到硬脑膜受累。大剂量化疗对复发的BL无效，患者因疾病进展死亡。尸检时，病理检查发现硬脑膜下血肿（SDH）和硬脑膜增厚。这是一例罕见的孤立性颅内BL复发伴硬脑膜受累及非创伤性SDH并发症的病例，我们回顾了既往关于BL继发性硬脑膜受累的报道。我们的病例表明了一个重要观点，即在硬脑膜受累的情况下，临床医生应意识到非创伤性SDH作为一种并发症，全脑放疗可能是利妥昔单抗联合化疗后孤立性颅内BL复发伴硬脑膜受累的最佳治疗方法。
