Saif Muhammad Wasif, Hopkins Jon L, Gore Steven D
Division of Hematology/Oncology, Wallace Tumour Institute, University of Alabama, Room 262-A, 1824 6th Avenue South, Birmingham, AL 35294-3300, USA.
Leuk Lymphoma. 2002 Nov;43(11):2083-92. doi: 10.1080/1042819021000016186.
Autoimmune paraneoplastic syndromes are commonly encountered in patients with myelodysplastic syndromes (MDS). A review of case reports and small series suggest as many as 10% of MDS patients may experience various autoimmune syndromes. Clinical manifestations of such phenomena may include an acute systemic vasculitic syndrome, skin vasculitis, fever, arthritis, pulmonary infiltrates, peripheral polyneuropathy, inflammatory bowel disease, glomerulonephritis, and even classical connective tissue disorders, such as relapsing polychondritis. On the other hand, asymptomatic immunologic abnormalities have also been reported in these patients. These autoimmune manifestations frequently respond to immunosuppressive agents including steroids and occasional hematologic responses to steroid therapy have also been reported. We report five patients with history of MDS who manifested different spectrums of autoimmune phenomena including: pyoderma gangrenosum (PG), vasculitis, Coombs negative hemolytic anemia, idiopathic thrombocytopenia, and chronic inflammatory demyelinating polyneuropathy (CIDP). We also review the incidence, nature, course and response to therapy of these manifestations and discuss potential pathogenic mechanisms.
自身免疫性副肿瘤综合征在骨髓增生异常综合征(MDS)患者中较为常见。对病例报告和小系列研究的回顾表明,多达10%的MDS患者可能会出现各种自身免疫综合征。此类现象的临床表现可能包括急性系统性血管炎综合征、皮肤血管炎、发热、关节炎、肺部浸润、周围性多发性神经病、炎症性肠病、肾小球肾炎,甚至经典的结缔组织疾病,如复发性多软骨炎。另一方面,这些患者中也有无症状免疫异常的报道。这些自身免疫表现通常对包括类固醇在内的免疫抑制剂有反应,也有报道称偶尔对类固醇治疗有血液学反应。我们报告了5例有MDS病史的患者,他们表现出不同类型的自身免疫现象,包括坏疽性脓皮病(PG)、血管炎、抗人球蛋白试验阴性的溶血性贫血、特发性血小板减少症和慢性炎症性脱髓鞘性多发性神经病(CIDP)。我们还回顾了这些表现的发生率、性质、病程及对治疗的反应,并讨论了潜在的致病机制。