Case report: Allogeneic stem cell transplantation for type B insulin resistance

Thomas Ebert; Gerhard Behre; Lorenz Weidhase; Vladan Vucinic; Cornelia Gewert; Robert K Semple; Michael Stumvoll; Anke Tönjes

doi:10.3389/fmed.2023.1200037

Case report: Allogeneic stem cell transplantation for type B insulin resistance

Front Med (Lausanne). 2023 Aug 29:10:1200037. doi: 10.3389/fmed.2023.1200037. eCollection 2023.

Authors

Thomas Ebert¹, Gerhard Behre^{2

3}, Lorenz Weidhase⁴, Vladan Vucinic², Cornelia Gewert^{5

6}, Robert K Semple^{5

6

7}, Michael Stumvoll¹, Anke Tönjes¹

Affiliations

¹ Medical Department III-Endocrinology, Nephrology, Rheumatology, University of Leipzig Medical Center, Leipzig, Germany.
² University of Leipzig Medical Center, Medical Clinic and Policlinic 1, Hematology, Cellular Therapy and Hemostaseology, Leipzig, Germany.
³ Clinic for Internal Medicine I, Community Hospital Dessau, Dessau, Germany.
⁴ University of Leipzig Medical Center, Medical Intensive Care Unit, Leipzig, Germany.
⁵ Metabolic Research Laboratories, Wellcome Trust-Medical Research Council Institute of Metabolic Science, University of Cambridge, Cambridge, United Kingdom.
⁶ Cambridge Biomedical Research Centre, National Institute for Health Research, Cambridge, United Kingdom.
⁷ Centre for Cardiovascular Science, Queen's Medical Research Institute, University of Edinburgh, Edinburgh, United Kingdom.

Abstract

Type B insulin resistance (TBIR) is a rare, often fulminant form of insulin resistance caused by autoantibodies against the insulin receptor. If left untreated, its mortality is high. Various immunosuppressive regimens have shown efficacy, but treatment effects are variable and time-delayed, and drug-induced complications may arise. We report a patient with TBIR arising as a complication of Wiskott-Aldrich syndrome. Stable remission of TBIR was achieved through allogeneic peripheral blood stem cell transplantation (PBSCT) over a follow-up period of more than 1.5 years. We thus demonstrate that PBSCT can be considered a treatment option in TBIR where conventional immunosuppressive therapy is ineffective or contraindicated.

Keywords: Wiskott-Aldrich syndrome; allogeneic peripheral blood stem cell transplantation; case report; diabetes; rescue therapy; type B insulin resistance.

Publication types

Case Reports

Grants and funding

This study was funded by the German Research Foundation (DFG)—through SFB 1052, project number 209933838, subproject SFB 1052/2, A01 to MS, SFB 1052/3, C06, as well as SPP 1629 TO 718/2-1 to AT. AT and TE were supported by the German Diabetes Association (DDG). TE was supported by a Novo Nordisk postdoctoral fellowship run in partnership with Karolinska Institutet, Stockholm, Sweden, a Karolinska Institutet Research Foundation grant, as well as by the Swedish Kidney Foundation (Njurfonden) and by the Stiftelsen Stig och Gunborg Westman. TE was further funded through the EFSD Mentorship Programme supported by AstraZeneca. RS was funded by the Wellcome Trust (Grant 210752/Z/18/Z). The authors acknowledge support from the Open Access Publishing Fund of Leipzig University supported by the German Research Foundation (DFG) within the program of Open Access Publication Funding.