Rouvière Bénédicte, Chasset Francois, Abisror Noémie, Hirsch Pierre, Fain Olivier, Mékinian Arsène
Department of Internal Medicine and Pneumology, Brest University Hospital, Brest, France.
Faculty of Medicine, AP-HP, Dermatology and Allergology Department, Tenon Hospital, Sorbonne University, Paris, 75020, France.
BMC Rheumatol. 2025 Apr 14;9(1):42. doi: 10.1186/s41927-025-00470-6.
IgA vasculitis is a predominantly pediatric autoimmune disease characterized by IgA deposit in small vessels. Chronic myelomonocytic leukemia (CMML) is a rare hematological malignancy classified within myelodysplastic syndromes. Here, we present a previously unrecognized case of CMML associated with IgA vasculitis. A 62-year-old woman presented with necrotic and infiltrated purpura and mild arthralgia, primarily affecting the knees and wrists, without gastrointestinal or kidney involvement. A comprehensive screening for other etiologies was unremarkable. Blood tests showed an increase of monocyte count and circulating monocyte phenotyping was consistent with CMML. Bone marrow analysis showed no blast cells or karyotypic abnormalities. Genetic testing identified an NRAS mutation. Autoantibody screening and viral serologies were negative. A skin biopsy revealed small-vessel vasculitis with IgA immune deposits. CMML can be associated with autoimmune diseases, such as polyarteritis nodosa and cutaneous leukocytoclastic vasculitis. However, this is the first report of IgA vasculitis occurring in the context of low risk CMML.
IgA血管炎是一种主要发生于儿童的自身免疫性疾病,其特征为小血管中有IgA沉积。慢性粒单核细胞白血病(CMML)是一种罕见的血液系统恶性肿瘤,归类于骨髓增生异常综合征。在此,我们报告一例此前未被认识到的与IgA血管炎相关的CMML病例。一名62岁女性出现坏死性和浸润性紫癜以及轻度关节痛,主要累及膝关节和腕关节,无胃肠道或肾脏受累。对其他病因进行的全面筛查无异常发现。血液检查显示单核细胞计数升高,循环单核细胞表型分析与CMML相符。骨髓分析未发现原始细胞或核型异常。基因检测发现NRAS突变。自身抗体筛查和病毒血清学检查均为阴性。皮肤活检显示为伴有IgA免疫沉积的小血管血管炎。CMML可与自身免疫性疾病相关,如结节性多动脉炎和皮肤白细胞破碎性血管炎。然而,这是首例在低风险CMML背景下发生IgA血管炎的报告。