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Abstract Background Takotsubo cardiomyopathy can present itself in the most varied clinical forms, with extremely variable electrocardiogram anomalies and presence of comorbidities with a significant systemic commitment. Guillain–Barré Syndrome concomitant with isolated right ventricular Takotsubo cardiomyopathy is a rare entity. Here we present a patient with Guillain–Barré syndrome who had electrocardiogram abnormalities consistent with isolated right ventricular Takotsubo cardiomyopathy which have not been described in literature. This case report may prompt early identification of right ventricular involvement in neurological comorbidities, especially if the electrocardiogram is not frankly suggestive of an acute ischemic condition linked to coronary artery disease. Case presentation A 37-year-old woman was misdiagnosed as acute coronary syndrome because of abnormally elevated troponin T level and electrocardiogram findings in the Emergency Department. Due to absence of any significant stenosis in the main coronary artery, the primary diagnosis was ruled out. Based on reanalysis of the ECG abnormalities, the patient was diagnosed as a case of isolated right ventricular Takotsubo cardiomyopathy in Guillain–Barré Syndrome. This case demonstrates the importance of electrocardiogram as a critical tool to identify isolated right ventricular Takotsubo cardiomyopathy in Guillain–Barré Syndrome. Indeed, in this case, the electrocardiogram abnormalities were distributed beyond the territory of a single coronary artery distribution. Conclusions The described electrocardiogram findings of isolated right ventricular Takotsubo cardiomyopathy in Guillain–Barré Syndrome may facilitate identification of right ventricular involvement in neurological diseases.