Rojas Joselyn, Villalobos Marjorie, Martínez María Sofía, Chávez-Castillo Mervin, Torres Wheeler, Mejías José Carlos, Miquilena Edgar, Bermúdez Valmore
Endocrine and Metabolic Diseases Research Center, School of Medicine The University of Zulia, Maracaibo 4004, Venezuela ; Endocrinology Department, Maracaibo University Hospital (SAHUM), Maracaibo 4004, Venezuela.
Endocrine and Metabolic Diseases Research Center, School of Medicine The University of Zulia, Maracaibo 4004, Venezuela.
Case Reports Immunol. 2014;2014:394754. doi: 10.1155/2014/394754. Epub 2014 Nov 19.
Introduction. Insulin allergy is a rare complication of insulin therapy, especially in type 1 diabetes mellitus (T1DM). Key manifestations are hypersensitivity-related symptoms and poor metabolic control. T1DM, as well as insulin allergy, may develop in the context of autoimmune polyendocrine syndrome (APS), further complicating management. Case Report. A 17-year-old male patient, diagnosed with T1DM, was treated with various insulin therapy schemes over several months, which resulted in recurrent anaphylactoid reactions and poor glycemic control, after which he was referred to our Endocrinology and Immunology Department. A prick test was carried out for all commercially available insulin presentations and another insulin scheme was designed but proved unsuccessful. A desensitization protocol was started with Glargine alongside administration of Prednisone, which successfully induced tolerance. Observation of skin lesions typical of vitiligo prompted laboratory workup for other autoimmune disorders, which returned positive for autoimmune gastritis/pernicious anemia. These findings are compatible with APS type 4. Discussion. To our knowledge, this is the first documented case of insulin allergy in type 4 APS, as well as this particular combination in APS. Etiopathogenic components shared by insulin allergy and APS beg for further research in immunogenetics to further comprehend pathophysiologic aspects of these diseases.
引言。胰岛素过敏是胰岛素治疗的一种罕见并发症,尤其在1型糖尿病(T1DM)患者中。主要表现为与超敏反应相关的症状以及代谢控制不佳。T1DM以及胰岛素过敏可能在自身免疫性多内分泌综合征(APS)的背景下发生,这进一步使治疗变得复杂。病例报告。一名17岁男性患者,被诊断为T1DM,在数月内接受了多种胰岛素治疗方案,结果反复出现类过敏反应且血糖控制不佳,之后他被转诊至我们的内分泌与免疫科。对所有市售胰岛素制剂进行了点刺试验,并设计了另一种胰岛素方案,但未成功。开始使用甘精胰岛素进行脱敏方案并同时给予泼尼松,成功诱导了耐受性。观察到典型的白癜风皮肤损害促使对其他自身免疫性疾病进行实验室检查,结果显示自身免疫性胃炎/恶性贫血呈阳性。这些发现与4型APS相符。讨论。据我们所知,这是4型APS中胰岛素过敏的首例记录病例,也是APS中这种特殊组合的首例记录病例。胰岛素过敏和APS共有的病因学成分有待免疫遗传学进一步研究,以进一步理解这些疾病的病理生理方面。