Copyright © 2021 Wolters Kluwer Health, Inc. All rights reserved.Introduction: Myofascial pain syndrome is a painful local or regional disease caused by myofascial trigger points. Trigger point injection (TPI) is a frequently used method for the treatment of myofascial pain. Major complications associated with TPI have rarely been reported in the literature. Case Report: A 24-year-old woman, without medical history of any disease, was diagnosed with myofascial syndrome based on the presence of long-standing neck and right arm pain, and TPI with lidocaine was applied to the right trapezius region. During the procedure, blurred vision and loss of strength in the left arm occurred. Magnetic resonance and computed tomography imaging of the brain revealed findings that were consistent with an ischemic stroke in the right capsular interna and right occipital region. Conclusion: The reported patient is the first in the literature who suffered from ischemic stroke after TPI. The use of ultrasound for injections into the neck muscles could avoid serious complications.