SEVERE INSULIN-RESISTANCE IN A PATIENT WITH TYPE-1 DIABETES AND STIFF-MAN SYNDROME TREATED WITH INSULIN LISPRO

We describe a patient with type 1 diabetes with recurrent diabetic ket oacidosis and severe insulin resistance. Extensive evaluation of the e tiology of the insulin resistance did not reveal an etiology, and well over 1000 U of daily insulin did not prevent the ketoacidosis. Her bl ood glucose and insulin requirements were improved with glucocorticoid s and octreotide, but the effects of both of these agents were short-l ived. She was given a trial of insulin lispro with immediate and drama tic effects, lowering her HbA(1c) from 14.6 to 5.1% in 7 months with a decrease in insulin requirements of 1600-100 U per day. Besides her d iabetes, she had a history of pain and stiffness affecting numerous mu scle groups, and hospitalization was required for pain control. The di agnosis of stiff-man syndrome (SMS) was confirmed with high titers of glutamic acid decarboxylase 65 antibodies in both serum and cerebral s pinal fluid. In summary, we describe the first patient with type 1 dia betes, SMS, and severe insulin resistance. Although the etiology of th e insulin resistance is unknown, due to the efficacious response to in sulin lispro, hydrocortisone, and perhaps octreotide, we propose an im mune-mediated etiology. Although rare, this syndrome needs to be consi dered as an etiology of insulin resistance. (C) 1998 Elsevier Science Ireland Ltd. All rights reserved.