Supplementary Material for: Disseminated Marginal Zone Lymphoma in a Patient with Lyme Neuroborreliosis, a case report

Background: Lyme borreliosis has been associated with lymphoma, particularly cutaneous lymphomas. The literature is conflicted regarding the effect of antibiotic therapy in cutaneous marginal zone lymphomas (MZL) in individuals with Lyme borreliosis. We present a patient diagnosed with Lyme neuroborreliosis (LNB) and disseminated MZL. Case: A 67-year-old man was seen due to 6 weeks of neuropathic pain with nightly worsening, headache, and 5 kg weight loss. Two weeks prior to symptom debut he had a tick bite in the left groin, no subsequent rash. A lumbar puncture revealed mononuclear pleocytosis and elevated CSF-protein. The patient was admitted and started on ceftriaxone. The B. burgdorferi intrathecal test showed intrathecally produced Borrelia antibodies, and treatment was changed to doxycycline with a total treatment duration of 21 days. A PET/CT revealed enlarged lymph nodes with increased FDG uptake. On pathological examination, the CSF showed 62% clonal B-cells – compatible with low-grade B-cell lymphoma. Examination of bone marrow and an inguinal lymph node confirmed disseminated MZL. A control lumbar puncture eight weeks later showed declining pleocytosis and clonal B-cells. At last follow-up 20 months later, he was still asymptomatic and had not required antineoplastic treatment. Conclusion: To our knowledge, this is the first published case of LNB with non-cutaneous B-cell lymphoma treated and remitting on antibiotics alone. Antibiotic treatment for Borrelia-positive lymphomas has yet to be investigated with high-evidence study designs, so clinicians are encouraged to publish both positive and negative findings relevant to this. We believe this case brings new perspectives to future diagnosis and treatment of lymphomas in patients with verified Lyme borreliosis.

背景：莱姆疏螺旋体病（Lyme borreliosis）与淋巴瘤存在关联，尤以皮肤淋巴瘤为甚。现有文献针对莱姆疏螺旋体病患者合并皮肤边缘区淋巴瘤（cutaneous marginal zone lymphomas, MZL）时的抗生素治疗效果，结论尚存争议。本文报告1例确诊为莱姆神经疏螺旋体病（Lyme neuroborreliosis, LNB）伴播散性MZL的病例。
病例：患者为67岁男性，因6周来的神经性疼痛（夜间加剧）、头痛及5kg体重下降就诊。症状发作前2周，其左侧腹股沟曾遭蜱虫叮咬，未出现后续皮疹。腰椎穿刺结果显示单核细胞增多及脑脊液（CSF）蛋白水平升高。患者入院后接受头孢曲松治疗。伯氏疏螺旋体（B. burgdorferi）鞘内检测提示鞘内产生疏螺旋体抗体，遂将治疗方案调整为多西环素，总疗程为21天。PET/CT检查发现淋巴结肿大伴FDG摄取增高。病理检查显示脑脊液中62%为克隆性B细胞，符合低度恶性B细胞淋巴瘤的特征。骨髓及腹股沟淋巴结活检证实为播散性MZL。8周后复查腰椎穿刺，结果显示细胞增多及克隆性B细胞比例均有所下降。末次随访在20个月后，患者仍无相关症状，且未接受抗肿瘤治疗。
结论：据我们所知，本病例为首例经单纯抗生素治疗后获得缓解的LNB合并非皮肤性B细胞淋巴瘤的已发表病例。针对伯氏疏螺旋体阳性淋巴瘤的抗生素治疗，仍需采用高证据等级的研究设计开展探索，因此我们鼓励临床医生发表与此相关的阳性及阴性研究结果。我们认为本病例可为确诊莱姆疏螺旋体病患者的淋巴瘤诊疗提供全新视角。