Piatek Caroline I, Vergara-Lluri Maria E, Pullarkat Vinod, Siddiqi Imran N, O'Connell Casey, Brynes Russell K, Feinstein Donald I
Division of Hematology, Department of Medicine, Los Angeles County-University of Southern California Medical Center and Jane Anne Nohl Division of Hematology, University of Southern California Norris Comprehensive Cancer Center, Los Angeles, CA, USA.
Acta Haematol. 2017;138(3):129-137. doi: 10.1159/000479103. Epub 2017 Sep 2.
Autoimmune myelofibrosis (AIMF) is an underrecognized cause of nonmalignant bone marrow fibrosis which occurs in the presence or absence of a defined systemic autoimmune disease. Patients with AIMF present with cytopenias and autoantibodies, and have a distinctive nonclonal myelofibrosis on bone marrow examination. AIMF is distinguished from primary myelofibrosis by the absence of splenomegaly, eosinophilia, or basophilia, and the absence of abnormal myeloid, erythroid, or megakaryocytic morphology.
The objective of the study was to describe the clinical presentation and outcomes of patients with AIMF.
We conducted a single-institution, retrospective chart review of patients diagnosed with AIMF to investigate clinical presentations, therapies, and outcomes.
Twelve patients with AIMF were identified with a mean follow-up of 5.8 years. All patients had detectable autoantibodies and the majority had concomitant autoimmune disorders. Four patients experienced a complete response of cytopenias and 3 patients experienced a partial response (PR) of cytopenias with immunosuppressive therapy. One patient achieved a PR following splenectomy. No patients were diagnosed with myeloproliferative neoplasms during the follow-up period.
AIMF contributes to cytopenias in the subset of patients with various autoimmune disorders. The majority of patients with AIMF experience an improvement in cytopenias with immunosuppressive therapy.
自身免疫性骨髓纤维化(AIMF)是一种未被充分认识的非恶性骨髓纤维化病因,可在存在或不存在明确系统性自身免疫性疾病的情况下发生。AIMF患者表现为血细胞减少和自身抗体,骨髓检查显示有独特的非克隆性骨髓纤维化。AIMF与原发性骨髓纤维化的区别在于无脾肿大、嗜酸性粒细胞增多或嗜碱性粒细胞增多,且无异常的髓系、红系或巨核细胞形态。
本研究的目的是描述AIMF患者的临床表现和预后。
我们对一家机构诊断为AIMF的患者进行了回顾性病历审查,以调查临床表现、治疗方法和预后。
共识别出12例AIMF患者,平均随访5.8年。所有患者均可检测到自身抗体,大多数患者伴有自身免疫性疾病。4例患者血细胞减少完全缓解,3例患者经免疫抑制治疗后血细胞减少部分缓解(PR)。1例患者脾切除术后达到PR。随访期间无患者被诊断为骨髓增殖性肿瘤。
AIMF导致各种自身免疫性疾病患者出现血细胞减少。大多数AIMF患者经免疫抑制治疗后血细胞减少情况有所改善。