Aburisheh Khaled, Al Farsi Yousuf, Moyeen Fauzia, Mehmood Faryal
University Diabetes Center, College of Medicine, King Saud University, Riyadh, Saudi Arabia.
Diabetes Wellness Center, Lahore, Pakistan.
Oman Med J. 2020 Jun 30;35(3):e134. doi: 10.5001/omj.2020.52. eCollection 2020 May.
Type B insulin resistance syndrome (TBIRS) has not been previously reported in Arab populations. We report a case of TBIRS in an Arab patient with mixed connective tissue disease (MCTD). Investigations revealed a clinical condition marked by positive anti-insulin receptor and ribonucleoprotein antibodies. The patient presented with severe hyperglycemia, weight loss, arthralgia, and acanthosis nigricans. He was managed successfully with an intensive pulsed combination regimen of cyclophosphamide and plasmapheresis augmented with high doses of prednisolone, which normalized his blood sugar levels without insulin therapy. MCTD was controlled by hydroxychloroquine. During the course of his treatment, the patient developed diabetic ketoacidosis secondary to the gluteal abscess, leukopenia, and recurrent hypoglycemia. Here, we showed that immunosuppressive agents and plasmapheresis can induce remission of TBIRS and can be used to normalize the blood sugar levels of Arab patients with this condition.
B型胰岛素抵抗综合征(TBIRS)此前在阿拉伯人群中尚未有报道。我们报告了1例患有混合性结缔组织病(MCTD)的阿拉伯患者的TBIRS病例。检查发现该临床病例以抗胰岛素受体抗体和核糖核蛋白抗体阳性为特征。患者表现为严重高血糖、体重减轻、关节痛和黑棘皮病。通过环磷酰胺强化脉冲联合方案及血浆置换,并加大剂量泼尼松龙进行治疗,患者获得成功管理,无需胰岛素治疗血糖水平即恢复正常。MCTD通过羟氯喹得到控制。在治疗过程中,患者因臀肌脓肿继发糖尿病酮症酸中毒、白细胞减少症及反复低血糖。在此,我们表明免疫抑制剂和血浆置换可诱导TBIRS缓解,并可用于使患有该疾病的阿拉伯患者血糖水平恢复正常。