Monoclonal Insulin Autoimmune Syndrome Successfully Treated With Plasma Cell Directed Therapy

Frida Bugge Askeland; Hege M Frøen; Nils Bolstad; Per Medbøe Thorsby; Fredrik Schjesvold; Anne Cathrine Parelius Wammer; Ivar Følling; Geir E Tjønnfjord

doi:10.1016/j.clml.2024.10.005

Monoclonal Insulin Autoimmune Syndrome Successfully Treated With Plasma Cell Directed Therapy

Clin Lymphoma Myeloma Leuk. 2024 Oct 11:S2152-2650(24)02360-7. doi: 10.1016/j.clml.2024.10.005. Online ahead of print.

Authors

Frida Bugge Askeland¹, Hege M Frøen², Nils Bolstad³, Per Medbøe Thorsby⁴, Fredrik Schjesvold², Anne Cathrine Parelius Wammer⁵, Ivar Følling⁶, Geir E Tjønnfjord⁷

Affiliations

¹ Department of Haematology, Oslo Myeloma Center, Oslo University Hospital, Nydalen, Oslo, Norway; Institute of Clinical Medicine, University of Oslo, Blindern, Oslo, Norway. Electronic address: friask@ous-hf.no.
² Department of Haematology, Oslo Myeloma Center, Oslo University Hospital, Nydalen, Oslo, Norway.
³ Department of Medical Biochemistry, Oslo University Hospital, Nydalen, Oslo, Norway.
⁴ Department of Medical Biochemistry, Oslo University Hospital, Nydalen, Oslo, Norway; Hormone Laboratory, Biochemical Endocrinology and Metabolism Research Group, Oslo University Hospital, Nydalen, Oslo, Norway.
⁵ Department of Endocrinology, Akershus University Hospital, Lørenskog, Norway.
⁶ Institute of Clinical Medicine, University of Oslo, Blindern, Oslo, Norway; Department of Endocrinology, Akershus University Hospital, Lørenskog, Norway.
⁷ Department of Haematology, Oslo Myeloma Center, Oslo University Hospital, Nydalen, Oslo, Norway; Institute of Clinical Medicine, University of Oslo, Blindern, Oslo, Norway. Electronic address: gtjonnfj@ous-hf.no.

PMID: 39500643
DOI: 10.1016/j.clml.2024.10.005

Abstract

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.

Keywords: Anti-insulin antibodies; Hypoglycemia; IAS; Monoclonal gammopathy of clinical significance; Rare.

Publication types

Editorial