浆细胞定向治疗成功治愈单克隆胰岛素自身免疫综合征
Monoclonal Insulin Autoimmune Syndrome Successfully Treated With Plasma Cell Directed Therapy.
作者信息
Askeland Frida Bugge, Frøen Hege M, Bolstad Nils, Thorsby Per Medbøe, Schjesvold Fredrik, Wammer Anne Cathrine Parelius, Følling Ivar, Tjønnfjord Geir E
机构信息
Department of Haematology, Oslo Myeloma Center, Oslo University Hospital, Nydalen, Oslo, Norway; Institute of Clinical Medicine, University of Oslo, Blindern, Oslo, Norway.
Department of Haematology, Oslo Myeloma Center, Oslo University Hospital, Nydalen, Oslo, Norway.
出版信息
Clin Lymphoma Myeloma Leuk. 2025 Mar;25(3):e127-e130. doi: 10.1016/j.clml.2024.10.005. Epub 2024 Oct 11.
BACKGROUND
Monoclonal insulin autoimmune syndrome (IAS) is a very rare disease characterized by severe attacks of hypoglycemia caused by circulating anti-insulin antibodies produced by a B-cell clone, usually clonal plasma cells.
METHOD
We present 2 female Norwegian patients with monoclonal IAS. The anti-insulin antibodies were quantified by immune precipitation and characterized using a 3-step manual in-house assay. Both patients received plasma cell directed therapy.
RESULT
The first patient received plasma cell directed therapy for a time-limited period and achieved a sustained clinical remission without detectable anti-insulin antibodies. The second patient receives continuous plasma cell directed therapy and is in clinical remission with low values of detectable anti-insulin antibodies.
CONCLUSION
Plasma cell directed therapy was effective and safe in our 2 cases of monoclonal IAS. We recommend considering plasma cell directed therapy for these patients.
背景
单克隆胰岛素自身免疫综合征（IAS）是一种非常罕见的疾病，其特征是由B细胞克隆（通常是克隆性浆细胞）产生的循环抗胰岛素抗体引起严重低血糖发作。
方法
我们报告了2例挪威女性单克隆IAS患者。通过免疫沉淀法定量抗胰岛素抗体，并使用三步手工内部检测法对其进行鉴定。两名患者均接受了针对浆细胞的治疗。
结果
第一名患者接受了为期有限的针对浆细胞的治疗，并实现了持续的临床缓解，且未检测到抗胰岛素抗体。第二名患者接受持续的针对浆细胞的治疗，处于临床缓解状态，可检测到的抗胰岛素抗体水平较低。
结论
针对浆细胞的治疗在我们的2例单克隆IAS病例中有效且安全。我们建议考虑对这些患者进行针对浆细胞的治疗。
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