Case of fulminant type 1 diabetes with coronary microcirculatory dysfunction.

A 29-year-old man was transferred to the Jichi Medical University Saitama Medical Center, Saitama, Japan, because of disturbance of consciousness and was admitted. A total of 6 days before the admission, he felt fatigue and fever. He first visited another clinic and was diagnosed with acute gastroenteritis. However, his symptoms remained unchanged and his consciousness level gradually worsened. On the admission day, his blood pressure was 122/46 mmHg, his pulse rate was 106 b.p.m. and he was afebrile. Laboratory data showed plasma glucose of 2,348 mg/dL and ketonuria. The patient had coexisting severe rhabdomyolysis and acute kidney injury (maximum CK 26,852 mU/mL, CK-MB 699 mU/mL and creatinine 5.6 mg/dL). Arterial blood gas analysis showed a pH of 7.03. The patient's glycated hemoglobin level was 6.0%, but serum and urinary C-peptide levels were undetectable. Islet-specific autoantibodies (anti-GAD, anti-IA2 antibodies and ICA) were undetectable. These suggested fulminant type 1 diabetes. Electrocardiography (ECG) was normal on admission. Insulin and saline intravenous infusion were started. After 14 h, ST-T elevation in II, III, aVF, V5 and V6 were observed, and ST-T depression in aVL (Figure1) and plasma glucose level was 976 mg/dL. Echocardiography showed diffuse severe hypokinesis, especially of the inferior wall of the left ventricle. We carried out coronary angiography (CAG) to rule out acute myocardial infarction (AMI). CAG showed normal coronary arteries, but a contrast medium staining in capillaries was seen through all coronary lesions. 123I-MIBG and 201TlCl scintigraphy on the third hospital day showed less perfusion in the posteroinferior wall. Viral antibodies, such as Coxsackie virus (A9, B1-6), Echovirus 9, Herpes simplex virus, Cytomegalovirus and Epstein–Barr virus, were negative. These results suggested myocardial microvascular dysfunction. ECG and echocardiography findings were normalized on the eighth hospital day. Myocardial contrast echocardiography was normal on the 15th hospital day. The no-reflow phenomenon is known as microvascular dysfunction as a result of myocardial ischemia, typically it is described as reperfusion injury after percutaneous coronary intervention of AMI. However, there were no abnormalities detected by coronary angiography. Other than AMI, acute myocarditis, acute pericarditis, stress-induced cardiomyopathy, hyperkalemia, hypophosphatemia and hypomagnesemia could be made into a differential diagnosis. Echocardiography, CAG and scintigraphic findings showed that both acute pericarditis and stress induced cardiomyopathy are unlikely. In addition, the electrolyte disorders were not present. Myocarditis was ruled out from ECG reciprocal change. Eventually, we considered this patient was likely to have impaired microvascular function elicited by hyperglycemia. Cohen et al.1 described a case with ST-T segment change, and hyperkalemia and ketoacidosis. Typical cases of AMI like ST-T segment change in the case with DKA were reported as pseudomyocardial infarction. Although the present patient had no hyperkalemia, he presumably had a pathophysiology similar to the pseudomyocardial infarction pattern with hyperglycemia. Acute hyperglycemia increases oxidative stress, and leads to elevation of inflammatory cytokines and platelet activation2–4. The present case is the first report of fulminant type 1 diabetes in which CAG was carried out just during the occurrence of ST-T elevation. We suggest that hyperglycemia is likely a cause of microcirculation abnormalities and acute myocardial damage. However, there is no clear evidence that hyperglycemia directly causes myocardial damage, although further studies are required.

Figure 1

(a) normal electrocardiography on arrival. (b) ST elevations in II, III, aVF, V5 and V6, and ST-T depression in aVL 14 h after hospitalization.